# Long-term outcomes of paediatric Guillain–Barré syndrome

Bastien Estublier, Hélène Colineaux, Catherine Arnaud, Pascal Cintas, Eloise

Baudou, Yves Chaix, Francois Rivier, Maelle Biotteau, Pierre Meyer,

Emmanuel Cheuret

► To cite this version:

Bastien Estublier, Hélène Colineaux, Catherine Arnaud, Pascal Cintas, Eloise Baudou, et al. Long-term outcomes of paediatric Guillain–Barré syndrome. Developmental Medicine and Child Neurology, In press, 10.1111/dmcn.15693. hal-04174339

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# Satisfaction with health care services in young people with cerebral palsy in the transition period: results from a European multicenter study

Holger Muehlan, Joaquim Alvarelhao, Catherine Arnaud, Chirine Cytera, Jerome Fauconnier, Kate Himmelmann, Marco Marcelli, Henriette Markwart, Marion Rapp, Silke Schmidt, et al.

<figure><img src="image_1.png" /></figure>

## To cite this version:

Holger Muehlan, Joaquim Alvarelhao, Catherine Arnaud, Chirine Cytera, Jerome Fauconnier, et al. Satisfaction with health care services in young people with cerebral palsy in the transition period: results from a European multicenter study. Developmental Medicine and Child Neurology - Developmental Medicine & Child Neurology, 2024, 11, pp.1306504. 10.3389/fmed.2024.1306504. hal-04174339

HAL Id: hal-04174339

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Submitted on 27 Nov 2025

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entific research documents, whether they are pub-
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abroad, or from public or private research centers.

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<figure><img src="image_2.png" /></figure>

Distributed under a Creative Commons Attribution 4.0 International License
Long-term outcomes of paediatric Guillain–Barré
syndrome

Bastien Estublier¹ | Hélène Colineaux²,³ | Catherine Arnaud²,³ | Pascal Cintas⁴ | Eloise Baudou¹,⁵ | Yves Chaix¹,⁵ | François Rivier⁶ | Maelle Biotteau¹,⁵ | Pierre Meyer⁶ | Emmanuel Cheuret¹

1 Neuropediatric Department, Toulouse-Purpan University Hospital, Toulouse, France

2Clinical Epidemiology Unit, Toulouse University Hospital, Toulouse, France

3 CERPOP, Centre for Epidemiology and Research in POPulation Health, UMR 1295, INSERM, Toulouse III University, Toulouse, France

4Neurophysiological Exploration Department, Toulouse-Purpan University Hospital,
Toulouse, France

5 ToNIC, Toulouse NeuroImaging Center, University of Toulouse, Inserm, UPS,
Toulouse, France

6 Department of Pediatric Neurology, CHU Montpellier, PhyMedExp, University of Montpellier, Inserm, CNRS, Montpellier, France

**Correspondence**

Bastien Estublier, Neuropediatric Department, Hopital des enfants, CHU Purpan Toulouse,
330, Avenue de Grande Bretagne, 31300 Toulouse, France.

Email:

estublier.b@chu-toulouse.fr

Abbreviations:

BDI, Beck Depression Inventory fast screen; CDI, Child Depression Inventory short version; FSS, Fatigue Severity Scale; GBS, Guillain–Barre syndrome; GBSDS, Guillain–Barre Syndrome Disability Score; INCATSSS, Inflammatory Neuropathy Cause and Treatment Sensory Sum Score; MSWS, Multiple Sclerosis Walking Scale; ONLS, Overall Neuropathy Limitations Scale.
<figure><img src="image_3.png" /></figure>

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OPEN ACCESS

EDITED BY

Olaf Kraus de Camargo,
McMaster University, Canada

REVIEWED BY

Janelle Noel-MacDonnell,
Children's Mercy Kansas City, United States
Pilar Marqués-Sánchez,
University of León, Spain

*CORRESPONDENCE

Holger Muehlan

✉ holger.muehlan@uni-greifswald.de

Ute Thyen

✉ ute.thyen@uni-luebeck.de

RECEIVED 10 October 2023

ACCEPTED 08 January 2024

PUBLISHED 30 January 2024

CITATION

Muehlan H, Alvarelhao J, Arnaud C, Cytera C, Fauconnier J, Himmelmann K, Marcelli M, Markwart H, Rapp M, Schmidt S and Thyen U (2024) Satisfaction with health care services in young people with cerebral palsy in the transition period: results from a European multicenter study.

Front. Med. 11:1306504.

doi: 10.3389/fmed.2024.1306504

COPYRIGHT

© 2024 Muehlan, Alvarelhao, Arnaud, Cytera, Fauconnier, Himmelmann, Marcelli, Markwart, Rapp, Schmidt and Thyen. This is an open-access article distributed under the terms of the [Creative Commons Attribution License (CC BY)](https://creativecommons.org/licenses/by/4.0/). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

# Satisfaction with health care services in young people with cerebral palsy in the transition period: results from a European multicenter study

Holger Muehlan¹*, Joaquim Alvarelhao², Catherine Arnaud³,⁴, Chirine Cytera⁵, Jerome Fauconnier⁶, Kate Himmelmann⁷, Marco Marcelli⁸, Henriette Markwart⁹, Marion Rapp⁵, Silke Schmidt¹ and Ute Thyen⁵* on behalf of the SPARCLE group

¹Department of Health and Prevention, University of Greifswald, Greifswald, Germany, ²School of Health Sciences, University of Aveiro, Aveiro, Portugal, ³UMR 1295 CERPOP Centre for Epidemiology and Research in POPulation Health, Inserm, Université Toulouse III - Paul Sabatier, Team SPHERE, Toulouse, France, ⁴Clinical Epidemiology Unit, University Hospital, Toulouse, France, ⁵Hospital for Child and Adolescent Medicine, University of Luebeck, Luebeck, Germany, ⁶Laboratoire TIMC-IMAG Equipe ThEMAS, Pavillon Taillefer, Université Joseph Fournier, Grenoble, France, ⁷Department of Pediatrics, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden, ⁸Azienda Sanitaria Locale Viterbo, Child and Adolescent Neuropsychiatric Unit—Adult Disability Unit, Viterbo, Italy, ⁹Institute for Community Medicine, University Medicine Greifswald, Greifswald, Germany

**Background:** Young people with chronic health conditions and disabilities rely on the healthcare system to maintain their best possible health. The appropriate delivery and utilization of healthcare services are key to improve their autonomy, self-efficacy and employment outcomes. The research question of our study is directed toward investigating if poor availability and accessibility of healthcare services in general, as identified by unmet needs in healthcare, are associated with dissatisfaction with healthcare.

**Methods:** Within a European multicenter observational study, 357 young adults with cerebral palsy aged 19–28 were included. We assessed special healthcare needs, utilization of healthcare services, and satisfaction with healthcare applying the short-form of the YHC-SUN-SF, environmental and social variables (EAEQ) as well as indicators for severity of condition and functionality (e.g., GMFCS) of these participants based on a self-, assisted self- or proxy-reports. We used correlation analyses to explore associations between satisfaction with healthcare and respective indicators related to availability and accessibility of healthcare services as well as severity of the condition. In addition, we included reference values for satisfaction with health care from young adults with various chronic conditions assessed within population-based surveys from some of the European countries included in the study.

**Results:** We identified several unmet healthcare needs, especially for widely used and established services (e.g., physical therapy). Satisfaction with healthcare (YHC-SUN-SF general and subscale scores) was moderate to high and almost consistently better for the sample of young adults with cerebral palsy as compared to reference values for young adults with various chronic conditions assessed within general population surveys). Correlation coefficients between
# Abstract

**Aim:**

To study long-term sequelae in children with Guillain–Barre syndrome (GBS).

**Method:**

This was a prospective observational study with children from two French tertiary centres. Data were from clinical and several standardized scales or questionnaires.

**Results:**

Fifty-one patients were included with a median follow-up of 6 years 4 months (range 3–20 years) after the acute phase. The sequelae rate was 67% (95% confidence interval [CI] 53–78) and did not vary with time. Most children had minor sequelae (Guillain–Barre Syndrome Disability Score [GBSDS] = 1); only one was unable to run (GBSDS = 2). The most frequent complaints were paraesthesia (43%), pain (35%), and fatigue (31%). The neurological examination was abnormal in 18% of children, autonomy was compromised in 14%, and symptoms of depression occurred in 34%. The factors associated with late-onset sequelae were correlated with severity during the initial phase (i.e. initial GBSDS >4, odds ratio 6.6, 95% CI 1.8–33; *p* = 0.009). The predictive factors of more severe late-onset conditions were initial severity (*p* = 0.002) and sex (female patients; *p* = 0.01).

**Interpretation:**

Two-thirds of children with GBS had late-onset sequelae following an episode, often minor, but sometimes with continuing effects on their everyday lives. Particularly affected were those who had severe GBS during the acute phase and who lost the ability to walk.
satisfaction with healthcare and utilization of services and (unmet) healthcare needs were low, also with different indicators for severity of the condition or functionality.

**Conclusion:** Young adults with cerebral palsy reports of unmet healthcare needs varied largely but showed substantial deficits in some aspects. This seems to have no impact on the satisfaction with healthcare those patients currently receive. We conclude that these are two different constructs and somewhat independent indicators to evaluate the quality of healthcare. Clinicians and other practitioners should consider this distinction when monitoring patient needs in their daily practice.

KEYWORDS

cerebral palsy, chronic condition, emerging adulthood, young adults, transition, special needs, healthcare utilization, satisfaction with care

Introduction

Over the last two decades, birth prevalence for cerebral
palsy (CP) caused in the prenatal or perinatal period decreased
substantially to 1.5 per 1000 live births in high income countries,
i.e., Europe and Australia, while in regions of the global south
it remained or increased to 3.4 per 1000 live births (1, 2).
The reasons for the decline in more affluent regions are not
fully understood, but better healthcare may play a role (3).
Thus, access to healthcare services play an important role
in preventing CP. However, the course of the condition and
functional outcomes also depends on good quality healthcare.
The impact of this lifelong condition often associated with
severe disability makes CP a significant condition from a public
health perspective (4). People with chronic health conditions and
disabilities rely on the healthcare system to maintain their best
possible health. Generally, they require interdisciplinary services
within the healthcare system and multisectoral cooperation with
social services, employment, housing, education and community
resources for communication and mobility. Healthcare services can
play an important part to improve, e.g., autonomy, self-efficacy, and
employment outcomes (5).

The transitioning from pediatric care to adult medicine is
associated with challenges for young people with special healthcare
needs (6). This is accompanied by risks of a deterioration of
care, the underutilization of services and ultimately poor health
outcomes. Gaps in transitional care had been identified and
recommendations for best practice were established (7, 8). Also, for
young patients with widespread chronic diseases, care structures

to address the obstacles associated with the transition process
were established, e.g., transition programs (9). Such programs have
also been introduced for young people with CP (10), but their
availability and accessibility is far from being comprehensive, so
does the fulfillment of the care needs of these patients, which are
diversified and special (11, 12). The transition process must be
individualized to the developmental needs of the adolescent/young
adult and gradually work toward more autonomy. A recent
qualitative study reported that the transition to adulthood for
young adults with CP was far from gradual and was perceived as
being “thrust into adulthood” (13).

Our research question is directed toward investigating if poor
availability and accessibility of healthcare services are associated
with dissatisfaction with healthcare. Given that limited access
is known to be associated with impaired healthcare satisfaction,
we assume that poor access to healthcare services, identified
by unmet needs in healthcare, is associated with dissatisfaction
with healthcare. To account for the patient's perspective, we
use satisfaction with healthcare as a surrogate marker for
good and adequate healthcare provision, given that patient-
reported experience measures have been widely established in
interdisciplinary healthcare and implementation studies.

Materials and methods

Design and samples

Cerebral palsy sample (CP)

We used data from SPARCLE3, a multicenter European
observational population-based study combining the follow-up of
the SPARCLE cohort to young adulthood (19–28 years) and a cross-
sectional part allowing the recruitment of a larger sample of young
people with CP. SPARCLE3 was implemented in five of the nine
European regions originally investigated: South West and South
East France (Haute-Garonne and Isère counties, respectively),
North West Germany, Western Sweden (region of Goteborg),
Central Italy (Viterbo area). The region of Porto (Portugal)

<footnote>Abbreviations: BFMF, bimanual fine motor function; CP, cerebral palsy; EDACS, eating and drinking ability classification system; EAEQ, European Adolescent Environment Questionnaire; ECEQ, European Child Environment Questionnaire; FCCS, functional communication classification system; GMFCS, gross motor function classification system; HRQoL, health-related quality of life; MS, mean score; QoL, quality of life; SCPE, surveillance of cerebral palsy in Europe; SD, standard deviation; SPARCLE, study of PARTicipation of children with cerebral palsy living in Europe; WHO, World Health Organization; YHC-SUN-SF, Youth Health Care - Satisfaction, Utilization and Needs - Short Form.</footnote>
Guillain–Barre syndrome (GBS) is an acute post-infectious polyradiculoneuropathy with an incidence of 0.34 and 1.34 per 100 000, and is slightly rarer in children than in adults.¹ It has been the primary cause of flaccid paralysis in children since the introduction of the poliomyelitis vaccine.² The mortality rate remains high in the adult population at 3% to 7%,³ whereas it is only 0.2% in the paediatric population.⁴ Motor recovery also seems better, as nearly all children regain the ability to walk, regardless of the electrophysiological form, although axonal forms exhibit a longer recovery time.⁵

In the long term, despite seemingly good motor recovery, approximately 10% of adult patients have severe sequelae,⁶ and nearly two-thirds report different symptoms.⁷ Fatigue is among the most disabling of symptoms in immunological neuropathies.⁸ Some patients also report pain, paraesthesia, feelings of imbalance, etc., which significantly affect their everyday lives. A Dutch study⁹ found that nearly half of patients made negative comments about their psychosocial situation, 38% were forced to change jobs, and 44% had difficulties pursuing recreational activities. In children, motor recovery seems to be better and faster,¹⁰ but only two studies¹¹,¹² have specifically addressed long-term sequelae following the acute phase of GBS. In these studies, sequelae were frequent and had an impact on patients' everyday lives. The preliminary study identified a rate of highly visible sequelae of approximately 10%, 1 year after the acute phase of GBS in children.

The aim of this study was twofold: first, to estimate the frequency and describe long-term sequelae (at least 3 years after the episode) following GBS where the acute phase occurred during childhood; and second, to identify the factors during the acute phase that predicted an unfavourable long-term development.

# METHOD

## Participants

In this prospective observational study, we included patients who received care for GBS between January 2000 and June 2016 at the neuropediatric departments of Toulouse and Montpellier University Hospitals. They had all been included in a published study on early GBS sequelae.¹³ In our present study, inclusion criteria were (1) a confirmed clinical diagnosis of GBS determined by the Asbury criteria¹⁴ or a diagnosis of variant forms in the GBS spectrum;¹⁵ (2) a minimum period of 3 years between the acute phase of GBS and the follow-up consultation to ensure an examination of the stabilized sequelae since it is now well known that recovery from GBS can take up to 2 years after the acute period;¹³ (3) age between 0 and 18 years at the time of the acute phase; (4) the absence of an intercurrent event (major neurological or orthopaedic pathologies occurring after GBS); and (5) the absence of chronic polyradiculoneuropathy. Of the 110 families in the previous team's cohort,¹³ 106 matched the criteria and were contacted by two physicians at Toulouse University Hospital (the first and last authors). Fifty-five patients could not be included (lost to follow-up, refusal, etc.) and 51 patients were finally included (Figure S1).

## Ethics statement

All parents and children gave their informed oral and written consent after the nature and objectives of the study were thoroughly explained. The local ethics committee (Comité de Protection des Personnes Est III) and the committee on human research Est-III granted authorization to perform this study (sponsor code RC31/19/0311) in accordance with the Declaration of Helsinki.
participated in the cross-sectional part. Further information is
provided in the study protocol (14). The SPARCLE3 sample
included young adults born between 31/07/1991 and 01/04/1997
with confirmed diagnosis of CP as defined by the SCPE (15). Out
of 357 participants, 110 came from Germany, 105 from Portugal,
88 from France, 30 from Sweden, and 24 from Italy.

General population sample (GP)

Both in France and Germany, a population-based comparison
group of young adults of the same age group was recruited with
young adults participating in an online survey. Participants of the
survey received incentives by a panelist who recruited males and
females (in 50%/50% ratio) aged 19–29 years living in one of both
participating countries (N = 4.051; France: n = 2080; Germany:
n = 1964). In addition, comparison data from the other countries
were available as well, but not on a population-wide level (Sweden:
n = 987; Portugal: n = 105; Italy: n = 24).

Instruments

Youth Health Care - Satisfaction, Utilization &
Needs - Short-Form measure (YHC-SUN-SF)

Specifically developed for adolescent self-report (age 15–25)
on their satisfaction with their care provision (16), the YHC-
SUN was derived from the CHC-SUN, reported by parents (17).
This version was shortened to 30 items in SPARCLE3. The first
module "Receipt of services" comprises 7 items plus the list of unmet
needs (16 items) and the second module "Satisfaction with care"
comprises 7 items (18). Answering options included: (a) services
both needed and fully or partly used, (b) services not needed and
not used, and (c) services needed but not used. We labeled the
latter category as “unmet need” from the perspective of the user.
Written in German, this questionnaire had no official translation
available. The SPARCLE3 version has been translated with forward-
backward procedure from German to English (in Lübeck) and
afterwards by every country on their own in their language. Three
following subscale-scores were calculated: “diagnosis/information,”
“doctors’ behavior,” and “patient centered care.” The domains
represented by these subscales were originally chosen for the short-
form measure derived from the model of the original long-form
measure, because all relate to the interaction between physician and
patient. Item content covers satisfaction with information given
about the condition and treatment choices, about the way doctors
listened and explained as well about time spent and efforts made for
the consultation. The YHC-SUN-SF measure was applied in both
samples (CP, GP), except for the GP subsample from France.

European Adolescent Environment Questionnaire
(EAEQ)

This instrument was developed in the SPARCLE project
as an adaptation of the ECEQ [European Child Environment
Questionnaire, (19)]. Lindsay Pennington (Newcastle) and
Joaquim Alvarelhao (Porto) developed a new version specific for
adulthood, comprising 61 items related to the physical, social and
attitudinal environments. The questionnaire is divided into 8 parts:
Physical environment at home (6 items), Physical environment at
work/university/in day placement (5 items), Physical environment
in public places (6 items), Access to transport (7 items), Access to

health services and carer (9 items), Financial support (5 items),
Attitude and support (20 items), Access to information (4 items).
For this paper we used items from the Dimension “Access to
health services and carer.” The question “Do you need…?” offers
the answer categories “no” and “yes,” if yes a question follows
whether the service is available or not. The EAEQ was assessed in
the CP sample only.

Severity assessment

We also collected data on gross motor function (GMFCS)
(20-22), hand function [Bimanual Fine Motor Function (BFMF)]
(23), communication [Functional Communication Classification
System (FCCS)] (24), and feeding (Eating and Drinking Ability
Classification System (EDACS) (25), and vision and hearing
impairments. Cognitive level was estimated by combining:
information from the CP register at age five or later years,
neuropsychological assessment if available, current school
performance, and ability to self-report. Seizure frequency in the
last year and the use of anticonvulsants were recorded. Severity
assessments were applied in the CP sample only.

General health

The standard question “How is your health in general? Would
you say it is . . .” was used in the European Social Survey (ESS) from
round 1 (2002) to round 9 (2018). Respondents were asked to rate
their current health on a five-step ladder ranging from very bad (1)
to very good (5). Translations exist for SPARCLE countries (France,
Germany, Italy, Portugal, Sweden). In 2016, 66.7% of the EU-28
population aged 18 and over reported that their health status was
good or very good. At the other end of the spectrum, almost 1 in 10
(9.1%) persons perceived their health status to be bad or very bad
(26). General health was assessed in both samples (CP, GP).

Data analysis

Descriptive statistics were used to describe the respondents'
socio-demographic as well as clinical characteristics. Statistical tests
(ANOVA/χ²-test) as well as corresponding effect size measures and
p-values were calculated to provide indicators of group differences.
Spearman correlation analysis was used to investigate statistical
associations between YHC-SUN-SF scores for satisfaction with care
and different indicators for severity of condition and functioning.
All quantitative statistical analyses were conducted using IBM SPSS
Statistics 28.0.

Results

Sample

Among the 357 participants from the CP sample, 157 were
female (44%), 216 lived with parents (60.5%), about 10% in
facilities and 18% independently (Table 1A). A total of 117
responded by proxy-report by a carer (32.8%), less than a fifth had
achieved more than secondary education. A total of 319 (89.4%)
had a general practitioner and 236 (66.1%) a specialist for the
condition. Young adults with CP had a mean age of 24.0 years
(SD = 1.6 years) at the time of interview. Educational attainment
<table><caption>TABLE 1 Guillain–Barré Syndrome Disability Score (GBS Trial Group 1978).²⁰</caption><thead><tr><th colspan="2">Most commonly used measure of levels of activity and participation</th></tr></thead><tbody><tr><td>0</td><td>Healthy</td></tr><tr><td>1</td><td>Minor symptoms or sign of neuropathy but capable of manual work/capable of running</td></tr><tr><td>2</td><td>Able to walk without support of a stick (5 m across an open space) but incapable of manual work/running</td></tr><tr><td>3</td><td>Able to walk with a stick, appliance, or support (5 m across an open space)</td></tr><tr><td>4</td><td>Confined to bed or chair bound</td></tr><tr><td>5</td><td>Requiring assisted ventilation (for any part of the day or night)</td></tr><tr><td>6</td><td>Death</td></tr></tbody></table>

## Procedure and measurements

Parents were contacted by telephone by a paediatric neurologist and were asked to participate in the study. They were sent a leaflet describing the characteristics of the study, a recruitment letter, and a consent form. When the patients agreed to participate in the study, two paediatric neurologists (BE and EC) gathered information on the patients' clinical and administrative characteristics in the initial phase, using their medical records. All the patients then came for a standard consultation with one of the two paediatric neurologists (BE or EC). They underwent a medical examination to research clinical anomalies, especially cranial nerve impairments, residual ataxia, or pathological deep tendon reflexes. Their complaints were recorded during a semi-structured individual interview. All the patients then underwent the same complete individual evaluation using a comprehensive extensive protocol designed to assess (1) direct GBS sequelae (motor, sensory, and fatigue) and (2) the consequences of sequelae on quality of life. Each area was assessed using one or more self-administered questionnaires, which are listed as follows.

### Direct GBS sequelae (motor, sensory, fatigue)

The following tests were used to assess direct GBS sequelae: (1) The Guillain–Barré Syndrome Disability Score (GBSDS)¹⁶ to evaluate overall handicap (Table 1). (2) The Medical Research Council Sum Score¹⁷ to evaluate muscular strength (considered pathological if a muscle group is deficient [Medical Research Council Sum Score < 60]). (3) The Inflammatory Neuropathy Cause and Treatment Sensory Sum Score (INCATSSS)¹⁸ to evaluate sensory disorders (considered abnormal if the score is positive [INCATSSS >0]). (4) The DN4 questionnaire¹⁹ to evaluate the neuropathic aspect of the pain (considered to be positive if the DN4 score ≥4). (5) The Fatigue Severity Scale (FSS)²⁰ to evaluate fatigue (considered to be ‘significant’ if the FSS score ≥4 and ‘severe’ if the FSS score ≥5). (6) The Multiple Sclerosis Walking Scale (MSWS),²¹ a self-evaluation questionnaire validated for GBS (considered pathological if the MSWS score >5%) to evaluate walking, cumulated with a 6-minute walking test²² (considered abnormal at –2 standard deviations [SD] from the norm).
was much lower in the CP group. Noteworthy, self-rated health status (range 1–5) does not differ between the CP sample (M = 2.08, SD = 0.82) and the GP sample (M = 2.08, SD = 0.81; *p* = 0.40, η = 0.01).

Subtypes of CP represented in the sample were spastic unilateral (26.3%), spastic bilateral (54.9%), dyskinetic (10.5%) and ataxic (7.0%). Gross motor function (GMFCS) indicates that for nearly 40% of the participants with CP moving was either limited (18.2%) or even severely limited (20.2%), whereas more than one third is able to walk and climb stairs (35.3%). Around two third are able to communicate effectively in most situations on their own (58.8%) or with some help (7.6%), as indicated by the BFMFS. Further clinical characteristics may be found in Table 1B.

## Unmet healthcare needs

We found a substantial number of unmet healthcare needs (Table 2): Subjectively reported unmet healthcare needs exceeded 10% (up to 17%) in most areas (12 out of 16, 75%), except for physical aids (5%) and communication aids (6%), home nursing (6%) as well as nutrition counseling (8%). Noteworthy, from those who reported not to get physiotherapy (33%, *n* = 118), almost a half stated they would need it (47%, *n* = 55); among those without support with filling applications (58%, *n* = 208), approximately a quarter would need it (28%, *n* = 58), a similar proportion appears (24%, *n* = 61) among those who reported not to get occupational therapy (69%; *n* = 245).

Special healthcare needs were assessed using two different assessments (YHC-SUN-SF, EAEQ). The overlap between corresponding responses of both measures is printed in bold in Table 3, ranging between 80 and 98% for physical therapy, occupational therapy and speech therapy. For the assessment of healthcare needs of other services, the terms used vary between both assessments and the range of corresponding responses is substantially impaired (self-help/support groups: 63–82%; social/counseling services: 43–70%) as compared to the aforementioned services.

## Satisfaction with care

YHC-SUN-SF total and subscale (unweighted mean) scores as well as the single-item measure score (range 1–5) indicated moderate to high levels of satisfaction for the three different dimensions of healthcare with scores almost consistently higher on a significant level for the sample of young adults with CP as compared with data of young adults from the general population (Table 4).

## Associations between satisfaction with care and unmet needs and severity of condition

Associations between satisfaction with care and the status of the chronic health condition in the CP sample: With a few exceptions, there were very low negative associations between healthcare

satisfaction (YHC-SUN-SF) with different indicators of severity of the condition and functioning, respective correlations coefficients ranging from *r* = -0.17 to *r* = 0.08 (Table 5).

Moreover, except for provision of physical aids, rehabilitation measures, and nutritional/counseling therapy, low correlations coefficients were detected between satisfaction with care (YHC-SUN-SF) and utilization of healthcare services or healthcare needs for 3 of the 16 services assessed by the first module of the YHC-SUN-SF module (for a comprehensive list of these services see Table 3).

## Discussion

Our study explored unmet needs in healthcare in young adults with CP and how these are associated with dissatisfaction with healthcare. We assumed that poor access to healthcare, identified by unmet needs in healthcare, is associated with dissatisfaction with healthcare.

We found a substantial number of unmet healthcare needs, especially for widely used and established services such as physical therapy and occupational therapy. One third of participants reported that they did not receive physical therapy; 17.3% stated that they did not perceive the need for such service and 15.4% reported unmet needs. Two thirds of the participants did not receive occupational therapy, 42% of respondents perceived no need for such services; however, the rate of unmet needs was high with 17.1%. This result confirms results provided by a recent study from Ireland (27). The authors report similar rates of unmet needs in physical therapy (23%) and occupational therapy (13%). A study with a convenience sample recruited online in France reported that finding an available physiotherapist was very difficult for 47% of the children compared to 58% of the adolescents and adults. Finding a physiotherapist trained in CP rehabilitation was reported as very difficult for 61% of children and adolescents and 66% of adults. Physiotherapy was provided in a private outpatient practice for 27% in adolescents, 41% in young adults and 57% in adults over 25 years. Regular communication between health professionals was less common in adults compared to adolescents, indicating a sharp decline in access to multiprofessional services. Generally, the access to and satisfaction with physical therapy in adults is much lower compared to young people with CP (28).

Occupational therapists can play a key-role in the transition process of young people with disabilities fostering self-determination but their services are often limited to school-based services or are discontinued at age 18 (29). A study with young people age 18–22 with CP in Ireland compared those who were discharged from pediatric services and those remaining. Especially the rate in occupational therapy dropped to 37.5% post discharge compared to 80.0% pre-discharge (30).

In this study, 16.5% of participants reported to receive speech and language therapy, but 14.8% reported unmet needs. A study from France compared health service use in different age groups and found a frequency of speech and language therapy in 17% of the ambulatory 18–24 year old participants and only 6% in non-ambulatory participants (31).

In psychosocial services, we found high rates of unmet needs: support with filling applications (16.2%), social services (14.8%),
## Repercussion on everyday life

The following tests were used to assess repercussions on everyday life: (1) the Overall Neuropathy Limitations Scale (ONLS)23 to evaluate functional limitation in everyday activities (loss of autonomy was considered if ONLS score ≥1), (2) the Child Depression Inventory short version (CDI)24 for children older than 7 years, and (3) the Beck Depression Inventory fast screen (BDI)25 for adults were used to screen for depressive symptoms (considered pathological if CDI score >3 or BDI score >4 respectively).

## Pathological cut-off for the study

A patient was considered to have late-onset sequelae from the acute phase if the GBSDS was greater than or equal to 1.

The severity of sequelae was evaluated according to the number of domains of complaints: only one motor disorder or sensory disorder complaint or only a complaint of fatigue, or complaints in two or all areas. This classification allowed us to better characterize the sequelae, in particular the heterogenous group of minor symptoms (GBSDS = 1). Motor disorders were defined as a motor impairment (sensation of muscular weakness noted on the questioning or Medical Research Council Sum Score < 60 or significant infirmity experienced upon walking noted on the MSWS questionnaire or a 6-minute walking test < -2 SD). Sensory disorders were defined as the presence of pain, paraesthesia, cramps, or sensory impairment determined by the INCATSSS. Fatigue was defined by a complaint of fatigue during the medical interview or significant fatigue on the questionnaire (FSS ≥4).

The impact of these sequelae on everyday life was defined by the inability to practise sports or receive a normal education or limited autonomy with an abnormal ONLS (score ≥1). These sequelae on everyday life were analysed by screening for depression symptoms with a pathological score on the CDI or BDI (CDI >3 or BDI >4).

## Statistical analysis

The primary analysis was performed on all patients on complete data (no imputation for missing data). The rate of late-onset sequelae (primary objective) was estimated on the basis of the proportion of patients with a GBSDS ≥1, 3 or more years after the acute phase, with a 95% confidence interval (CI). The characteristics of sequelae were described with numbers and percentages for qualitative variables, and median and first/third quartiles for quantitative variables.

For bivariate comparative analyses, we used Pearson's $\chi^2$ test for qualitative variables (or Fisher's exact test when the conditions for application were not met) and Wilcoxon's non-parametric test for quantitative variables.

To identify predictive factors of late-onset conditions, the judgement criterion was modelled on exposures of interest, one by one, using bivariate logistic regression models after verification of the application conditions. To identify predictive factors of the severity of late-onset conditions, the judgement criterion was modelled on exposures of interest, one by one, using bivariate ordinal logistic regression models after verification of the application conditions.
<table><caption>TABLE 1A Characteristics of both samples.</caption><thead><tr><th rowspan="2"></th><th colspan="2">Cerebral palsy (CP)</th><th colspan="2">General population (GP)</th><th rowspan="2">ANOVA/χ<sup>2</sup>-test</th></tr><tr><th>N</th><th>%</th><th>N</th><th>%</th></tr></thead><tbody><tr><td>Country</td><td></td><td></td><td></td><td></td><td>χ<sup>2</sup>(1,4) = 515.87,<br/>p &lt; 0.001</td></tr><tr><td>France</td><td>88</td><td>24.6%</td><td>2080</td><td>40.8</td><td></td></tr><tr><td>Germany</td><td>110</td><td>30.8%</td><td>1964</td><td>38.5</td><td></td></tr><tr><td>Italy</td><td>24</td><td>6.7%</td><td>24</td><td>0.5</td><td></td></tr><tr><td>Portugal</td><td>105</td><td>29.4%</td><td>47</td><td>0.9</td><td></td></tr><tr><td>Sweden</td><td>30</td><td>8.4%</td><td>987</td><td>19.3</td><td></td></tr><tr><td>Sex</td><td></td><td></td><td></td><td></td><td>χ<sup>2</sup>(1,1) = 9.60, p &lt; 0.002</td></tr><tr><td>Male</td><td>200</td><td>56.0%</td><td>2426</td><td>47.5%</td><td></td></tr><tr><td>Female</td><td>157</td><td>44.0%</td><td>2676</td><td>52.5%</td><td></td></tr><tr><td>Age</td><td></td><td></td><td></td><td></td><td>F<sub>(1,5457)</sub> = 5.66, p &lt; 0.02,<br/>η = 0.03</td></tr><tr><td>Mean (SD), years</td><td>24.03</td><td>(2.01)</td><td>24.45</td><td>(3.29)</td><td>χ<sup>2</sup>(1,4) = 16.50, p = 0.002</td></tr><tr><td>Living environment</td><td></td><td></td><td></td><td></td><td></td></tr><tr><td>A big city (population over 200,000)</td><td>96</td><td>26.9%</td><td>1629</td><td>31.9%</td><td></td></tr><tr><td>The suburbs or outskirts of a big city<br/>(population over 200,000)</td><td>30</td><td>8.4%</td><td>668</td><td>45.0%</td><td></td></tr><tr><td>A town or a small city (population<br/>between 3,000 and 200,000)</td><td>159</td><td>44.5%</td><td>2070</td><td>40.6%</td><td></td></tr><tr><td>A country village (population less than<br/>3,000)</td><td>61</td><td>17.1%</td><td>621</td><td>12.2%</td><td></td></tr><tr><td>A farm or home in the country</td><td>9</td><td>2.5%</td><td>113</td><td>2.2%</td><td></td></tr><tr><td>(Missing)</td><td>2</td><td>0.6%</td><td>1</td><td>0%</td><td></td></tr><tr><td>Education</td><td></td><td></td><td></td><td></td><td>χ<sup>2</sup>(1,8) = 578.65,<br/>p = 0.001</td></tr><tr><td>Early childhood educational<br/>development</td><td>111</td><td>31.1%</td><td>91</td><td>1.8%</td><td></td></tr><tr><td>Primary education</td><td>33</td><td>9.2%</td><td>45</td><td>0.9%</td><td></td></tr><tr><td>Lower secondary education</td><td>65</td><td>18.2%</td><td>559</td><td>11.0%</td><td></td></tr><tr><td>Upper secondary education</td><td>83</td><td>23.2%</td><td>1448</td><td>28.4%</td><td></td></tr><tr><td>Post-secondary non-tertiary education</td><td>12</td><td>3.4%</td><td>745</td><td>14.6%</td><td></td></tr><tr><td>Short cycle tertiary education</td><td>11</td><td>3.1%</td><td>612</td><td>12.0%</td><td></td></tr><tr><td>Bachelor or equivalent level</td><td>21</td><td>5.9%</td><td>927</td><td>18.2%</td><td></td></tr><tr><td>Master or equivalent level</td><td>16</td><td>4.5%</td><td>611</td><td>12.0%</td><td></td></tr><tr><td>Doctoral or equivalent level</td><td>-</td><td>-</td><td>61</td><td>1.2%</td><td></td></tr><tr><td>(Missing)</td><td>5</td><td>1.4%</td><td>3</td><td>0.1%</td><td></td></tr><tr><td>Years of education</td><td></td><td></td><td></td><td></td><td>F<sub>(1,5405)</sub> = 26.06,<br/>p = 0.001, η = 0.07</td></tr><tr><td>Mean (SD)</td><td>11.75</td><td>(4.95)</td><td>13.14</td><td>4.93</td><td></td></tr><tr><td>Type of report</td><td></td><td></td><td></td><td></td><td>(n.a.)</td></tr><tr><td>Self-report without assistance</td><td>134</td><td>37.5%</td><td>5102</td><td>100%</td><td></td></tr><tr><td>Self-report with assistance</td><td>105</td><td>29.4%</td><td>-</td><td>-</td><td></td></tr><tr><td>Proxy-report</td><td>117</td><td>32.8%</td><td>-</td><td>-</td><td></td></tr><tr><td>(Missing)</td><td>1</td><td>0.3%</td><td>-</td><td>-</td><td></td></tr><tr><td>General health</td><td></td><td></td><td></td><td></td><td></td></tr><tr><td>Mean (SD)</td><td>2.08</td><td>(0.82)</td><td>2.04</td><td>0.81</td><td>F<sub>(1,5456)</sub> = 0.71, p = 0.40,<br/>η = 0.01</td></tr><tr><td>Very good</td><td>84</td><td>23.5%</td><td>1267</td><td>24.8%</td><td>χ<sup>2</sup>(1,4) = 1.66, p = 0.80</td></tr><tr><td>Good</td><td>181</td><td>50.7%</td><td>2618</td><td>51.3</td><td></td></tr><tr><td>Fair</td><td>75</td><td>21.0%</td><td>978</td><td>19.2%</td><td></td></tr><tr><td>Bad</td><td>14</td><td>3.9%</td><td>214</td><td>4.2%</td><td></td></tr><tr><td>Very bad</td><td>3</td><td>0.8%</td><td>24</td><td>0.5%</td><td></td></tr><tr><td>(Missing)</td><td>-</td><td>-</td><td>1</td><td>0%</td><td></td></tr></tbody></table>
# RESULTS

## Population description

One hundred and ten families were contacted. Fifty-five patients could not be included: 44 could not be reached, two refused to participate, and nine did not come to the consultation. Four more patients were not included because they did not meet the inclusion criteria: they had an intercurrent event (serious orthopaedic anomalies not related to GBS [$n = 2$], acute transverse myelitis [$n = 1$], and white matter lesions on brain imagery under exploration with a secondary onset in a child with epilepsy and in utero exposure to sodium valproate [$n = 1$]). Finally, 51 patients were included and analysed (Table 2 and Figure S1).

Among the patients who were included, the median age at diagnosis was 5 years 4 months (IQR 3–10 years, range 1–16 years). There were slightly more males than females (sex ratio 1.3). The initial characteristics at the acute phase of GBS for included patients were compared with those not included to ensure the representativeness of the follow-up cohort in relation to the initial cohort (Table S1 and Figure S1). The individuals who could not be included in the follow-up study were more often the older patients in the cohort ($p < 0.001$). Overall, the other characteristics of those included and those excluded were similar: hospital stay, initial severity, clinical symptoms, demyelination, frequency of transfers to intensive care, and the frequency of intubation. There were more axonal forms in the excluded patients (29.2%) than in the included children (15.6%), but this criterion was not significant ($p = 0.117$).

## Description of residual symptoms after 3 years

At the time of the consultation, follow-up after the acute phase of GBS ranged from 3 years to 19 years 6 months, with a median of 6 years 4 months (IQR 5–8 years). The median age at the follow-up consultation was 11 years 6 months (IQR 9–17 years, range 5–31 years). The rate of long-term sequelae, defined by a GBSDS score of at least 1, was 67% (95% CI 53–78%). No correlation was found between the time since the episode and the sequelae rate ($p = 0.795$). The sequelae therefore seemed to be stable after 3 years and no longer varied with time.

## Late-onset characteristics

The late-onset characteristics of the patients are described in Table 3. All patients had regained the ability to walk autonomously. Among those with sequelae ($n = 34, 67\%$), most (98%) exhibited minor symptoms but were able to run (GBSDS score = 1). Only one patient was more severely affected and could not run, although he was still able to walk autonomously for at least 10 metres (GBSDS score = 2).

With a detailed medical interview, the most common functional signs were paraesthesia (43%), pain (35%), and fatigue (31%). The DN4 questionnaire helped verify the neuropathic nature of pain only in half of the individuals who had pain ($n = 9$ out of 18). Nevertheless, we identified the diagnosis of neuropathic pain thanks to the medical interview in most patients with pain (89% of patients with pain had neuropathic pains such as painful paraesthesia). When measured ($n = 14$), the average pain intensity was 4.9 (SD 1.8) on the Numeric Pain Rating Scale.
<table><caption>TABLE 1B Clinical and functional characteristics of sample with young adults with cerebral palsy (CP sample, n = 357).</caption><thead><tr><th>Type of CP</th><th>N</th><th>%</th></tr></thead><tbody><tr><td>Spastic unilateral</td><td>94</td><td>26.3%</td></tr><tr><td>Spastic bilateral</td><td>196</td><td>54.9%</td></tr><tr><td>Dyskinetic</td><td>35</td><td>9.8%</td></tr><tr><td>Dyskinetic chorea-athetotic</td><td>6</td><td>1.7%</td></tr><tr><td>Ataxic</td><td>25</td><td>7.0%</td></tr><tr><td>(Missing)</td><td>1</td><td>0.3%</td></tr><tr><td>GMFCS</td><td>Mean (SD)</td><td>2.74</td><td>1.58</td></tr><tr><td>I</td><td>Child walks and climbs stairs</td><td>126</td><td>35.3%</td></tr><tr><td>II</td><td>Child walks inside</td><td>50</td><td>14.0%</td></tr><tr><td>III</td><td>Child walks with limitations</td><td>44</td><td>12.3%</td></tr><tr><td>IV</td><td>Moving about is limited</td><td>65</td><td>18.2%</td></tr><tr><td>V</td><td>Moving about is severely limited</td><td>72</td><td>20.2%</td></tr><tr><td>BFMF</td><td>Mean (SD)</td><td>2.58</td><td>1.43</td></tr><tr><td>I</td><td>Without limitation</td><td>114</td><td>31.9%</td></tr><tr><td>II</td><td>Both hands limited in fine skills</td><td>80</td><td>22.4%</td></tr><tr><td>III</td><td>Child needs help with tasks</td><td>55</td><td>15.4%</td></tr><tr><td>IV</td><td>Child needs help and adapted equipment</td><td>59</td><td>16.5%</td></tr><tr><td>V</td><td>Child needs total human assistance</td><td>49</td><td>13.7%</td></tr><tr><td>FCCS</td><td>Mean (SD)</td><td>2.12</td><td>1.50</td></tr><tr><td>I</td><td>Effective communicator in most situations</td><td>210</td><td>58.8%</td></tr><tr><td>II</td><td>Effective communicator in most situations, but does need some help</td><td>27</td><td>7.6%</td></tr><tr><td>III</td><td>Effective communicator in some situations, small range of messages/topics to most familiar people</td><td>26</td><td>7.3%</td></tr><tr><td>IV</td><td>Assistance required in most situations, especially with unfamiliar people and communicates daily/routine needs and wants</td><td>56</td><td>15.7%</td></tr><tr><td>V</td><td>Communicates unintentionally with others, using movements and behavior</td><td>38</td><td>10.6%</td></tr><tr><td>EDACS</td><td>Mean (SD)</td><td>1.84</td><td>1.24</td></tr><tr><td>I</td><td>Safely and efficiently</td><td>215</td><td>60.2%</td></tr><tr><td>II</td><td>Safely but with some limitations to efficiency</td><td>51</td><td>14.3%</td></tr><tr><td>III</td><td>With some limitations to safety, maybe limitations to efficiency</td><td>45</td><td>12.6%</td></tr><tr><td>IV</td><td>With significant limitations to safety</td><td>24</td><td>6.7%</td></tr><tr><td>V</td><td>Unable to eat or drink safely/tube feeding</td><td>22</td><td>6.2%</td></tr></tbody></table>

CP, cerebral palsy; GMFCS, gross motor function classification system; BFMF, bimanual fine motor function; FCCS, functional communication classification system; EDACS, eating and drinking ability classification system; SD, standard deviation.

information (16.3%), and counseling about sexuality (11.3%). In our study 19.2% received psychotherapy but 16.3% reported unmet needs in this area. Given the high prevalence of mental health problems in young people with CP the provision of services appears to be poor (32, 33). In sexual counseling we would like to address the issue, that participants may not be aware of such service or sexuality may be considered a private issue not relevant to medical care. A Dutch study with young adults with CP described high levels of sexual problems mostly related to the fact of having CP and many young people wanted more information; however, in 90% the issue had never been discussed during rehabilitation treatment (34).

Unmet needs in rehabilitation (16.5%) and access to self-help groups (15.2%) indicate barriers to services aiming at fostering

independence and self-efficacy. The need for home nursing was comparatively low in our sample. A study with a sample with CP aged 4–27 years demonstrated a considerable decrease of specialized rehabilitation services in the young adults compared to children and low levels of formal respite services and support groups/youth clubs (35).

The often abrupt transition from mostly well-equipped centers for integrated multiprofessional healthcare available for children and adolescents to a much more fragmented care in adulthood appears counter-intuitive as health in people with CP decreases over time and more but not less healthcare is required to prevent especially chronic pain, fatigue and declining walking ability (36). A systematic review of observational studies showed transition-associated poor outcomes including housing instability,
<table><caption>TABLE 2 Initial-phase characteristics of the patients included in the follow-up cohort.</caption><thead><tr><th></th><th colspan="2">Included (n=51)</th></tr></thead><tbody><tr><td>Centre (n, %)</td><td></td><td></td></tr><tr><td>Toulouse</td><td>33</td><td>65%</td></tr><tr><td>Montpellier</td><td>18</td><td>35%</td></tr><tr><td>Demographic variables</td><td></td><td></td></tr><tr><td>Age at diagnosis<sup>a</sup> (years:months)</td><td>5:4</td><td>2:11; 9:7</td></tr><tr><td>Sex (n, %)</td><td></td><td></td></tr><tr><td>Female</td><td>22</td><td>43%</td></tr><tr><td>Male</td><td>29</td><td>57%</td></tr><tr><td>Initial episode</td><td></td><td></td></tr><tr><td>Year of the episode (n, %)</td><td></td><td></td></tr><tr><td>2000-2007</td><td>5</td><td>10%</td></tr><tr><td>2007-2012</td><td>17</td><td>33%</td></tr><tr><td>2012-2014</td><td>15</td><td>29%</td></tr><tr><td>2014-2016</td><td>14</td><td>28%</td></tr><tr><td>GBSDS (n, %)</td><td></td><td></td></tr><tr><td>1</td><td>2</td><td>4%</td></tr><tr><td>2</td><td>14</td><td>28%</td></tr><tr><td>3</td><td>12</td><td>23%</td></tr><tr><td>4</td><td>20</td><td>39%</td></tr><tr><td>5</td><td>3</td><td>6%</td></tr><tr><td>Interval between onset of symptoms and hospitalisation<sup>a</sup> (days) (n=50)</td><td>5.0</td><td>3.0-11.5</td></tr><tr><td>Pain complaints (n, %)</td><td>39</td><td>77%</td></tr><tr><td>Paraesthesia complaints (n=45)</td><td>12</td><td>27%</td></tr><tr><td>Motor impairment complaints (n, %)</td><td>45</td><td>88%</td></tr><tr><td>Sensory impairment complaints (n=44)</td><td>6</td><td>14%</td></tr><tr><td>Facial paralysis (n, %)</td><td>12</td><td>24%</td></tr><tr><td>Cranial nerve impairments (n, %)</td><td>24</td><td>47%</td></tr><tr><td>Difficulty walking (n, %)</td><td>50</td><td>98%</td></tr><tr><td>Loss of ability to walk (n, %)</td><td>22</td><td>43%</td></tr><tr><td>Asthenia (n, %)</td><td>13</td><td>26%</td></tr><tr><td>Autonomic dysfunction (n, %)</td><td>20</td><td>39%</td></tr><tr><td>Ataxia (n, %)</td><td>21</td><td>41%</td></tr><tr><td>Impairment of consciousness (n, %)</td><td>1</td><td>2%</td></tr><tr><td>Aetiology (n, %)</td><td></td><td></td></tr><tr><td>Anti-neuronal antibodies (n=39)</td><td>13</td><td>33%</td></tr><tr><td>Campylobacter (n=38)</td><td>5</td><td>13%</td></tr><tr><td>Axonal Guillain-Barré syndrome (n=45)</td><td>7</td><td>16%</td></tr><tr><td>Initial care</td><td></td><td></td></tr><tr><td>Treatment (n, %)</td><td>43</td><td>84%</td></tr><tr><td>Intubation (n, %)</td><td>3</td><td>6%</td></tr><tr><td>Transfer to intensive care (n, %)</td><td>11</td><td>22%</td></tr><tr><td>Major sequelae following the episode (n=50)</td><td>2</td><td>4%</td></tr><tr><td>Length of hospitalization<sup>a</sup> (days)</td><td>7.0</td><td>6.0-14.5</td></tr></tbody></table>

Total number is indicated if data are missing.

<footnote><sup>a</sup>Quantitative variables by median and first and third quartiles.</footnote>
<table><caption>TABLE 2 Healthcare needs as assessed with the YHC-SUN-SF (CP sample, n = 357).</caption><thead><tr><th rowspan="2">Do you get...</th><th colspan="2">Missing</th><th colspan="2">Yes</th><th colspan="2">Yes, partly</th><th colspan="2">No and no need</th><th colspan="2">No, but need</th><th colspan="2">No receipt (total)</th></tr><tr><th>n</th><th>%</th><th>n</th><th>%</th><th>n</th><th>%</th><th>n</th><th>%</th><th>n</th><th>%</th><th>n</th><th>%</th></tr></thead><tbody><tr><td>Physiotherapy</td><td>0</td><td>0.0</td><td>211</td><td>58.9</td><td>29</td><td>8.1</td><td>62</td><td>17.3</td><td>55</td><td>15.4</td><td>117</td><td>33.0</td></tr><tr><td>Physical aids</td><td>1</td><td>0.3</td><td>172</td><td>48.2</td><td>54</td><td>15.1</td><td>113</td><td>31.7</td><td>17</td><td>4.8</td><td>130</td><td>36.4</td></tr><tr><td>Support with filling applications</td><td>1</td><td>0.3</td><td>116</td><td>32.5</td><td>32</td><td>9.0</td><td>150</td><td>42.0</td><td>58</td><td>16.2</td><td>208</td><td>58.3</td></tr><tr><td>Occupational therapy</td><td>1</td><td>0.3</td><td>83</td><td>23.2</td><td>28</td><td>7.8</td><td>184</td><td>51.5</td><td>61</td><td>17.1</td><td>245</td><td>68.6</td></tr><tr><td>Social services</td><td>0</td><td>0.0</td><td>77</td><td>21.5</td><td>31</td><td>8.7</td><td>196</td><td>54.7</td><td>53</td><td>14.8</td><td>249</td><td>69.6</td></tr><tr><td>Health services at work or school</td><td>5</td><td>1.4</td><td>63</td><td>17.8</td><td>25</td><td>7.1</td><td>228</td><td>64.6</td><td>36</td><td>10.2</td><td>264</td><td>74.8</td></tr><tr><td>Psychotherapy</td><td>2</td><td>0.6</td><td>68</td><td>19.1</td><td>16</td><td>4.5</td><td>213</td><td>59.8</td><td>58</td><td>16.3</td><td>271</td><td>76.1</td></tr><tr><td>Nutrition counseling</td><td>1</td><td>0.3</td><td>63</td><td>17.6</td><td>9</td><td>2.5</td><td>256</td><td>71.7</td><td>28</td><td>7.8</td><td>284</td><td>79.6</td></tr><tr><td>Speech therapy</td><td>0</td><td>0.0</td><td>47</td><td>13.1</td><td>12</td><td>3.4</td><td>245</td><td>68.4</td><td>53</td><td>14.8</td><td>298</td><td>83.2</td></tr><tr><td>Information</td><td>2</td><td>0.6</td><td>32</td><td>9.0</td><td>23</td><td>6.5</td><td>242</td><td>68.0</td><td>58</td><td>16.3</td><td>300</td><td>84.3</td></tr><tr><td>Communication aids</td><td>2</td><td>0.6</td><td>36</td><td>10.1</td><td>19</td><td>5.3</td><td>279</td><td>78.2</td><td>22</td><td>6.2</td><td>301</td><td>84.3</td></tr><tr><td>Home nursing</td><td>1</td><td>0.3</td><td>41</td><td>11.5</td><td>11</td><td>3.1</td><td>283</td><td>79.3</td><td>21</td><td>5.9</td><td>304</td><td>85.2</td></tr><tr><td>Rehabilitation</td><td>1</td><td>0.3</td><td>58</td><td>16.2</td><td>14</td><td>3.9</td><td>255</td><td>71.4</td><td>59</td><td>16.5</td><td>314</td><td>88.0</td></tr><tr><td>Self-help group</td><td>2</td><td>0.6</td><td>19</td><td>5.3</td><td>9</td><td>2.5</td><td>273</td><td>76.7</td><td>54</td><td>15.2</td><td>327</td><td>91.9</td></tr><tr><td>Telephone advice by medical professional</td><td>0</td><td>0.0</td><td>27</td><td>7.5</td><td>18</td><td>5.0</td><td>292</td><td>81.6</td><td>40</td><td>11.2</td><td>332</td><td>92.7</td></tr><tr><td>Counseling about sexuality</td><td>3</td><td>0.8</td><td>7</td><td>2.0</td><td>15</td><td>4.2</td><td>292</td><td>82.3</td><td>40</td><td>11.3</td><td>332</td><td>93.5</td></tr></tbody></table>
The detection of a deep tendon reflex anomaly (28%) was not considered as a sequela because it was not clinically relevant. However, the rest of the neurological examination found disabling sequelae (18%) such as ataxia, motor deficit, or cranial nerve impairments.

The FSS questionnaire identified significant fatigue in 20% of patients and severe fatigue in 4%. Infirmity experienced upon walking, measured with the MSWS questionnaire, was significant for 26% of patients. The 6-minute walking test, performed by 46 patients, was below 2 SD for 15% of them. On average, patients had performances of approximately -1.1 SD (1.4 SD) and the distance travelled during this walking test was correlated with the infirmity experienced upon walking measured with the MSWS questionnaire (correlation coefficient -0.6; *p* < 0.001).

These symptoms had a significant impact on the everyday lives of the patients in our cohort. In fact, autonomy in everyday activities, as evaluated by the ONLS, was abnormal for 14% of patients. Most children (96%) had been able to receive a normal education, although adaptation to the disability was sometimes needed in the first months after the acute phase of GBS. Three patients (6%) were unable to practise sports because of motor impairment or effort-related susceptibility to fatigue. Symptoms of depression, screened by the CDI or BDI, were detected in 34% of patients (15% male, 57% female). The same patients who reported symptoms of depression also reported fatigue (*p* = 0.019), motor impairment (*p* = 0.019), and complaints (*p* = 0.043).

## Combination of complaints and severity score

A GBSDS equal to 1 represents a heterogeneous group of minor symptoms. To clarify this group, we classified the sequelae into three different areas (motor, sensory, and fatigue).

### Motor complaints

All patients with a motor impairment (complaints or abnormal motor strength during examination or ataxia) also had difficulty walking, as measured by the MSWS questionnaire or an abnormal 6-minute walk test. Therefore, the MSWS questionnaire and 6-minute walking test detected more patients than the interview or physical examination. These investigations seemed to be the most sensitive method for identifying motor complaints: 14 patients (45%) among patients with minor sequelae (GBSDS = 1).

### Sensory complaints

Twenty-seven patients (82%) with minor symptoms were identified as having a sensory disorder (complaint of pain and/or paraesthesia and/or cramps and/or abnormal INCATSSS). The semi-structured medical interview was more sensitive than questionnaires in identifying sensory complaints. In fact, only one patient had an abnormal sensory test (INCATSSS >0).
<table><caption>TABLE 3 Overlap of corresponding responses to YHC-SUN-SF and EAEQ items (CP sample, n = 357).</caption><thead><tr><th rowspan="3">YHC-SUN-SF question: "Do you get..."*</th><th colspan="8">EAEQ question: "Do you need..."*</th></tr><tr><th colspan="8">...Physiotherapy?</th></tr><tr><th>Total**</th><th colspan="2">No</th><th colspan="2">Yes, I have it</th><th colspan="2">Yes, but I don't have it</th></tr><tr><th>N</th><th>n</th><th>%</th><th>n</th><th>%</th><th>n</th><th>%</th></tr></thead><tbody><tr><td>Yes, fully or partly</td><td>240</td><td>2</td><td>0.8</td><td>228</td><td>95.0</td><td>10</td><td>4.2</td></tr><tr><td>No, not needed</td><td>62</td><td>58</td><td>93.5</td><td>2</td><td>3.2</td><td>2</td><td>3.2</td></tr><tr><td>No, but I need it</td><td>54</td><td>4</td><td>7.4</td><td>4</td><td>7.4</td><td>46</td><td>85.2</td></tr><tr><td>(Missing)***</td><td>1</td><td></td><td></td><td></td><td></td><td></td><td></td></tr><tr><th colspan="8">...Occupational therapy?</th></tr><tr><th></th><th colspan="2">No</th><th colspan="2">Yes, I have it</th><th colspan="2">Yes, but I don't have it</th></tr><tr><th></th><th>n</th><th>%</th><th>n</th><th>%</th><th>n</th><th>%</th></tr><tr><td>Yes, fully or partly</td><td>110</td><td>11</td><td>10.0</td><td>95</td><td>86.4</td><td>4</td><td>3.6</td></tr><tr><td>No, not needed</td><td>184</td><td>172</td><td>93.5</td><td>5</td><td>2.7</td><td>7</td><td>3.8</td></tr><tr><td>No, but I need it</td><td>60</td><td>9</td><td>15.0</td><td>1</td><td>1.7</td><td>50</td><td>83.3</td></tr><tr><td>(Missing)</td><td>3</td><td></td><td></td><td></td><td></td><td></td><td></td></tr><tr><th colspan="8">...Speech therapy?</th></tr><tr><th></th><th colspan="2">No</th><th colspan="2">Yes, I have it</th><th colspan="2">Yes, but I don't have it</th></tr><tr><th></th><th>n</th><th>%</th><th>n</th><th>%</th><th>n</th><th>%</th></tr><tr><td>Yes, fully or partly</td><td>59</td><td>6</td><td>10.2</td><td>47</td><td>79.7</td><td>6</td><td>10.2</td></tr><tr><td>No, not needed</td><td>245</td><td>239</td><td>97.6</td><td>0</td><td>0.0</td><td>6</td><td>2.4</td></tr><tr><td>No, but I need it</td><td>52</td><td>8</td><td>15.4</td><td>0</td><td>0.0</td><td>44</td><td>84.6</td></tr><tr><td>(Missing)</td><td>1</td><td></td><td></td><td></td><td></td><td></td><td></td></tr><tr><th colspan="8">Do you have access to self-help groups?</th></tr><tr><th colspan="8">Do you need support groups in your area?</th></tr><tr><th></th><th colspan="2">No</th><th colspan="2">Yes, I have it</th><th colspan="2">Yes, but I don't have it</th></tr><tr><th></th><th>n</th><th>%</th><th>n</th><th>%</th><th>n</th><th>%</th></tr><tr><td>Yes, fully or partly</td><td>28</td><td>5</td><td>17.9</td><td>20</td><td>71.4</td><td>3</td><td>10.7</td></tr><tr><td>No, not needed</td><td>271</td><td>223</td><td>82.3</td><td>20</td><td>7.4</td><td>28</td><td>10.3</td></tr><tr><td>No, but I need it</td><td>54</td><td>16</td><td>29.6</td><td>4</td><td>7.4</td><td>34</td><td>63.0</td></tr><tr><td>(Missing)</td><td>4</td><td></td><td></td><td></td><td></td><td></td><td></td></tr><tr><th colspan="8">Do get advice from social services?</th></tr><tr><th colspan="8">Do you need counseling services?</th></tr><tr><th></th><th colspan="2">No</th><th colspan="2">Yes, I have it</th><th colspan="2">Yes, but I don't have it</th></tr><tr><th></th><th>n</th><th>%</th><th>n</th><th>%</th><th>n</th><th>%</th></tr><tr><td>Yes, partly</td><td>107</td><td>44</td><td>41.1</td><td>46</td><td>43.0</td><td>16</td><td>15.0</td></tr><tr><td>No, not needed</td><td>196</td><td>138</td><td>70.4</td><td>39</td><td>19.9</td><td>19</td><td>9.7</td></tr><tr><td>No, but I need it</td><td>53</td><td>16</td><td>30.2</td><td>10</td><td>18.9</td><td>27</td><td>50.9</td></tr><tr><td>(Missing)</td><td>1</td><td></td><td></td><td></td><td></td><td></td><td></td></tr></tbody></table>

unemployment, difficulty forming relationships, increased hospital admission rates, and decreased use of rehabilitation services. Factors associated with improved outcomes included family participation, promotion of self-efficacy, and meeting the adult team before transition (7).

Moreover, as special healthcare needs were assessed using two different assessments in our study (YHC-SUN-SF, EACD)

the overlap between corresponding responses to both different measures ranging between 80 and 98% for physical therapy and occupational therapy, providing even stronger evidence for the reliability of this finding.

Satisfaction with healthcare was moderate to good and almost consistently higher for the sample of young adults with CP as compared with data of young adults sampled from the general

<footnote>*Corresponding assessments of both questions are printed in bold. **Percentages are calculated based on row-wise frequency of cases. ***Number of missing values varies for each pair of variables (YHC-SUN-SF, EAEQ). EAEQ, European Adolescent Environment Questionnaire; YHC-SUN-SF, Youth Health Care - Satisfaction, Utilization and Needs - Short Form.</footnote>
<table><caption>TABLE 3 Late-onset sequelae.</caption><thead><tr><th></th><th colspan="2">Total (n=51)</th></tr></thead><tbody><tr><td>General characteristics (n, %)</td><td></td><td></td></tr><tr><td>Age<sup>a</sup> (years:months)</td><td>11:6</td><td>9:2-16:10</td></tr><tr><td>Follow-up<sup>a</sup> (years:months)</td><td>6:4</td><td>4:10-8:5</td></tr><tr><td>GBSDS</td><td></td><td></td></tr><tr><td>0</td><td>17</td><td>33%</td></tr><tr><td>1</td><td>33</td><td>65%</td></tr><tr><td>2</td><td>1</td><td>2%</td></tr><tr><td>Abnormal education</td><td>2</td><td>4%</td></tr><tr><td>Inability to do sports</td><td>3</td><td>6%</td></tr><tr><td>Patient complaints (n, %)</td><td></td><td></td></tr><tr><td>Fatigue</td><td>16</td><td>31%</td></tr><tr><td>Pain</td><td>18</td><td>35%</td></tr><tr><td>Paraesthesia</td><td>22</td><td>43%</td></tr><tr><td>Motor impairment</td><td>3</td><td>6%</td></tr><tr><td>Cramps (n=50)</td><td>5</td><td>10%</td></tr><tr><td>Tremors</td><td>5</td><td>10%</td></tr><tr><td>Falling</td><td>3</td><td>6%</td></tr><tr><td>Fine motor disorders</td><td>4</td><td>8%</td></tr><tr><td>Neurological examination (n, %)</td><td></td><td></td></tr><tr><td>Abnormal DTR</td><td>14</td><td>28%</td></tr><tr><td>Abnormal examination, except abnormal DTR</td><td>9</td><td>18%</td></tr><tr><td>Ataxia</td><td>2</td><td>4%</td></tr><tr><td>Cranial nerve anomalies</td><td>1</td><td>2%</td></tr><tr><td>Abnormal motor strength (MRCSS&lt;60)</td><td>4</td><td>8%</td></tr><tr><td>Abnormal sensory test (INCATSSS&gt;0)</td><td>1</td><td>2%</td></tr><tr><td>Standardized tests and questionnaires (n, %)</td><td></td><td></td></tr><tr><td>Significant fatigue (FSS ≥4)</td><td>10</td><td>20%</td></tr><tr><td>Severe fatigue (FSS ≥5)</td><td>2</td><td>4%</td></tr><tr><td>Neuropathic pain (DN4 + interview)</td><td>16</td><td>31%</td></tr><tr><td>Neurological disability with loss of autonomy (ONLS ≥1)</td><td>7</td><td>14%</td></tr><tr><td>Upper limb disability (ONLS upper limb ≥1)</td><td>4</td><td>8%</td></tr><tr><td>Lower limb disability (ONLS lower limb ≥1)</td><td>4</td><td>8%</td></tr><tr><td>Depression CDI or BDI (n=47)</td><td>16</td><td>34%</td></tr><tr><td>Inability to walk (MSWS&gt;5%)</td><td>13</td><td>26%</td></tr><tr><td>Distance walked &lt;-2 SD (n=46)</td><td>7</td><td>15%</td></tr></tbody></table>

<sup>a</sup>Quantitative variables by median and first and third quartiles.

Total number is indicated if data are missing.

Abbreviations: BDI, Beck Depression Inventory fast screen; CDI, Child Depression Inventory short version; DTR, deep tendon reflexes; FSS, Fatigue Severity Scale; GBSDS, Guillain-Barré Syndrome Disability Score; INCATSSS, Inflammatory Neuropathy Cause and Treatment Sensory Sum Score; MRCSS, Medical Research Council Sum Score; MSWS, Multiple Sclerosis Walking Scale; ONLS, Overall Neuropathy Limitations Scale; SD, standard deviation.
<table><caption>TABLE 4 Satisfaction with care (YHC-SUN-SF) for young adults with cerebral palsy from the SPARCLE study (n = 357) and young adults with different types of chronic health conditions from the general population in different European countries (GP sample, n = 14–703).</caption><thead><tr><th colspan="2">YHC-SUN-SF scores</th><th>N</th><th>Subscale<br>"Information/<br>Diagnosis"</th><th>Subscale<br>"Behavior<br>of the doctor"</th><th>Subscale<br>"Patient<br>centered care"</th><th>Short-Form<br>Global Score</th><th>Satisfaction with healthcare<br>(single item)</th></tr><tr><th></th><th></th><th></th><th>M (SD)</th><th>M (SD)</th><th>M (SD)</th><th>M (SD)</th><th>M (SD)</th></tr></thead><tbody><tr><td rowspan="5">Cerebral palsy</td><td>France</td><td>88</td><td>3.22 (0.93)</td><td>3.69 (1.00)</td><td>3.57 (1.07)</td><td>3.45 (0.85)</td><td>3.35 (0.91)</td></tr><tr><td>Germany</td><td>110</td><td>2.90 (1.01)</td><td>3.65 (1.05)</td><td>3.46 (1.01)</td><td>3.32 (0.87)</td><td>3.35 (1.02)</td></tr><tr><td>Italy</td><td>24</td><td>3.00 (0.96)</td><td>3.52 (1.13)</td><td>3.39 (1.14)</td><td>3.24 (0.97)</td><td>2.79 (0.93)</td></tr><tr><td>Portugal</td><td>105</td><td>3.37 (0.96)</td><td>3.57 (0.97)</td><td>3.42 (1.02)</td><td>3.45 (0.87)</td><td>3.34 (0.99)</td></tr><tr><td>Sweden</td><td>30</td><td>2.76 (1.08)</td><td>3.52 (1.07)</td><td>3.22 (1.11)</td><td>3.12 (0.99)</td><td>3.07 (1.02)</td></tr><tr><td></td><td>Total (ANOVA)</td><td>345</td><td>F<sub>(1,4)</sub> = 4.21, p = 0.002,<br>η = 0.05</td><td>F<sub>(1,4)</sub> = 0.33, p = 0.86,<br>η = 0.01</td><td>F<sub>(1,4)</sub> = 0.65, p = 0.63,<br>η = 0.01</td><td>F<sub>(1,4)</sub> = 1.14, p = 0.035,<br>η = 0.01</td><td>F<sub>(1,4)</sub> = 2.16, p = 0.07, η = 0.02</td></tr><tr><td rowspan="5">General population*</td><td>France**</td><td>–</td><td>–</td><td>–</td><td>–</td><td>–</td><td>–</td></tr><tr><td>Germany</td><td>703</td><td>2.82 (1.03)</td><td>2.98 (1.08)</td><td>3.11 (1.03)</td><td>2.96 (0.92)</td><td>3.04 (1.06)</td></tr><tr><td>Italy***</td><td>–</td><td>–</td><td>–</td><td>–</td><td>–</td><td>–</td></tr><tr><td>Portugal</td><td>14</td><td>2.61 (1.06)</td><td>3.43 (1.12)</td><td>3.68 (1.14)</td><td>3.24 (0.91)</td><td>3.14 (1.10)</td></tr><tr><td>Sweden</td><td>217</td><td>2.37 (1.02)</td><td>2.70 (1.21)</td><td>2.88 (1.22)</td><td>2.64 (0.98)</td><td>2.57 (1.09)</td></tr><tr><td></td><td>Total (ANOVA)</td><td>935</td><td>F<sub>(1,3)</sub> = 10.69,<br>p = 0.000, η = 0.03</td><td>F<sub>(1,3)</sub> = 4.57, p = 0.003,<br>η = 0.02</td><td>F<sub>(1,3)</sub> = 4.24, p = 0.005,<br>η = 0.01</td><td>F<sub>(1,3)</sub> = 7.22, p = 0.000,<br>η = 0.02</td><td>F<sub>(1,3)</sub> = 11.57, p = 0.000, η = 0.04</td></tr><tr><td colspan="2">ANOVA between both samples</td><td></td><td>F<sub>(1,1272)</sub> = 38.88,<br>p = 0.00, η = 0.03</td><td>F<sub>(1,1279)</sub> = 102.84, p = 0.00,<br>η = 0.07</td><td>F<sub>(1,1276)</sub> = 34.26,<br>p = 0.00, η = 0.03</td><td>F<sub>(1,1279)</sub> = 67.77,<br>p = 0.00, η = 0.05</td><td>F<sub>(1,1290)</sub> = 29.46, p = 0.00, η = 0.02</td></tr></tbody></table>

<footnote>*Subsamples of the general population surveys including a selection of young adults with chronic health conditions only. **The YHC-SUN-SF measure was not applied in the general population survey in France. ***Data from Italy are not reported, the subsample with a chronic condition is too small (n = 2). M, mean score; SD, standard deviation; YHC-SUN-SF, Youth Health Care - Satisfaction, Utilization and Needs - Short Form.</footnote>
## Fatigue complaints

Once again, the semi-structured medical interview seemed more sensitive than the FSS questionnaire in identifying patients with sequelae of fatigue: 16 patients (49% of those with minor symptoms) were considered to have symptoms of fatigue while the FSS score was only positive (≥4) for 12 out of 16 patients.

These different types of complaint were often interconnected. Sometimes, one patient presented with several types of complaint. The patient experiencing the most severe sequelae (GBSDS = 2) experienced fatigue and motor and sensory disorders.

Moreover, a complaint in one domain increased the likelihood of a complaint in another. For example, patients with fatigue more frequently also had a sensory complaint (odds ratio [OR] 4.1, 95% CI 1.1–17.1; *p* = 0.034) or a motor complaint (OR 22.4, 95% CI 5.2–129.9; *p* < 0.001).

We established a severity score based on the number of complaint domains affecting each patient (Table 4). Nine of the 10 patients (90%) with repercussions on their everyday lives (inability to practise sports or receive a normal education or limited autonomy with an abnormal ONLS) were affected in all three areas. On the contrary, 31 patients (78%) without repercussions in their everyday lives had no complaints or were affected in only one area. Therefore, it would seem that the impact on a patient's life increased (*p* < 0.001) with the number of areas affected.

Two patients had no affected domains, but a GBSDS score of 1 due to a ‘fine motor disorder’ and complaints of ‘tremors’ that were not considered in the calculation of the number of domains affected, but which nevertheless are minor complaints.

## Initial factors associated with severity of residual symptoms

The initial characteristics associated with the risk of late-onset sequelae (Table 5) are variables related to the severity of the initial episode: an initial GBSDS score of at least 4 (GBSDS ≥4, OR 6.7; *p* = 0.009) and loss of the ability to walk (*p* = 0.014).

The initial characteristics associated with the severity (number of areas of complaint affected) of the late-onset sequelae (Table S1 and Figure S1) are also variables related to the severity of the initial episode: GBSDS at least 4 (*p* = 0.002), loss of the ability to walk (*p* = 0.001), and length of hospitalization (*p* = 0.013). An association was also noted between female sex and a greater number of domains affected (*p* = 0.005).
<table><caption>TABLE 5 Correlation coefficients for association between YHC-SUN-SF scores for satisfaction with care and different indicators for severity of condition and functioning (CP sample, n = 357).</caption><thead><tr><th>Satisfaction with healthcare (YHC-SUN-SF)</th><th>GMFCS</th><th>BFMF</th><th>EDACS</th><th>FCCS</th><th>Visual impairment</th><th>Hearing impairment</th><th>Seizures</th><th>Speaking Ability</th></tr></thead><tbody><tr><td>Global score</td><td>-0.126*</td><td>-0.148**</td><td>-0.112</td><td>-0.156*</td><td>-0.116</td><td>0.054</td><td>-0.009</td><td>-0.115*</td></tr><tr><td>"Information/ Diagnosis"</td><td>-0.147*</td><td>-0.173**</td><td>-0.112</td><td>-0.128*</td><td>-0.100</td><td>0.047</td><td>-0.022</td><td>-0.092</td></tr><tr><td>"Behavior of the doctor"</td><td>-0.082</td><td>-0.117*</td><td>-0.078</td><td>-0.112*</td><td>-0.086</td><td>0.039</td><td>0.004</td><td>-0.090</td></tr><tr><td>"Patient centered care"</td><td>-0.093</td><td>-0.103*</td><td>-0.097</td><td>-0.133*</td><td>-0.053</td><td>0.079</td><td>0.029</td><td>-0.101*</td></tr></tbody></table>

Spearman correlation coefficients.

population. Likewise, in a recent study from our research group, we also found better quality of life for the environmental domain in young adults with CP as compared to reference values for young adults from a sample of the general population (37). The Transition Collaborative Group in UK used a different approach measuring satisfaction with healthcare: the Mind the Gap scale measures the difference or “gap” between a young person’s ideal service and the service they have received. The results show decreasing levels of satisfaction with age; appropriate parental involvement was significantly associated with higher satisfaction in all age groups (38).

In this study, satisfaction with healthcare services was similar to that of the general population sample of the same age. Several factors may play a role: the recruitment in the general population survey did not exclude any other chronic health conditions. We also speculate that young adults with less exposure and interaction with healthcare providers may be more critical while the young adults with CP are likely to experience long lasting positive relations with at least some professionals in their healthcare. Higher involvement may lead to a higher appreciation of the services received.

Associations of healthcare satisfaction with severity of the condition and functional abilities were not substantial. A large French study including 354 children, 145 adolescents and 511 adults with CP measured satisfaction with motor rehabilitation services identified significant factors that decreased satisfaction: being an adolescent, higher levels of motor impairment, frequent pain, receiving physical therapy in private practice and poor access to a physiotherapist with specific CP training (39). Because the outcome was limited to satisfaction with a specific service whereas our study probed for satisfaction with healthcare in general the different findings are difficult to interpret.

Also, satisfaction with healthcare was not associated with utilization of healthcare services or (unmet) healthcare needs. Thus, unmet needs and satisfaction with healthcare seem to be unrelated constructs. In the Dutch PERRIN-PiP study a similar lack of association between satisfaction with another outcome was noted: their study revealed a dissociation between participation accomplishment and satisfaction with participation among adolescents with CP. The authors recommend not only to focus on accomplishment but also, if not mainly, on satisfaction (40).

## Strengths and limitations

Our study reveals some *strong features*: We assessed a large sample of young adults with CP from different regional contexts and countries of Europe. In addition, a large proportion of this sample included patients that were longitudinally assessed throughout their childhood and adolescence (41–43). Moreover, we managed to collect reference data from young adults with various chronic health conditions, based on population-wide surveys in the respective countries. Finally, most of the constructs used in our analyses were assessed using different indicators, e.g., for (unmet) special healthcare needs (YHC-SUN-SF, EAEQ), for satisfaction with care (YHC-SUN-SF generic single item vs. total scale score) or severity of condition (e.g., GMFCS, BFMF).

One *limitation* of the study is that all results are based on self- or proxy-reported data only, thus we did not include any objective assessment for utilization of healthcare services. Thus, the validity of the subjective responses about individual behavior remains questionable. Furthermore, reference values were not eligible for all countries included and for some we collected only small samples. However, also the results from the countries with smaller sample sizes confirm the evidence provided by population-based surveys, indicating quite similar findings. Beside of this, we also missed to include CP and GP samples from Eastern European countries, thus our study is biased to Western Europe.

## Conclusion

Young adults with CP reports of unmet healthcare needs varied largely but showed substantial deficits in some aspects. During the transition period there must be sustained support and access to condition specific treatments such as physical therapy, occupational and speech and language therapy, psychosocial services and counseling and care coordination to allow growth of self-determination and autonomy.

Unmet needs do not have an impact on the satisfaction with healthcare these patients currently receive and we conclude that these are two different concepts and separate indicators to evaluate the quality of healthcare services delivery.

<footnote>*p ≤ 0.05; **p ≤ 0.01.</footnote>
<table><caption>TABLE 4 GBSDS and repercussions on everyday life based on the number of areas affected.</caption><thead><tr><th rowspan="2"></th><th rowspan="2">n</th><th colspan="6">Number of areas affected</th></tr><tr><th>0 (n=18)</th><th>1 (n=13)</th><th>2 (n=6)</th><th>3 (n=14)</th></tr></thead><tbody><tr><td>GBSDS</td><td></td><td></td><td></td><td></td><td></td><td></td></tr><tr><td>0</td><td>17</td><td>17</td><td>100.0</td><td>0</td><td>0.0</td><td>0</td><td>0.0</td></tr><tr><td>1</td><td>33</td><td>2</td><td>6.1</td><td>13</td><td>39.4</td><td>6</td><td>18.2</td></tr><tr><td>2</td><td>1</td><td>0</td><td>0.0</td><td>0</td><td>0.0</td><td>0</td><td>1</td></tr><tr><td>Repercussions on everyday life</td><td></td><td></td><td></td><td></td><td></td><td></td><td></td></tr><tr><td>No</td><td>41</td><td>18</td><td>43.9</td><td>13</td><td>31.7</td><td>5</td><td>12.2</td></tr><tr><td>Yes</td><td>10</td><td>0</td><td>0.0</td><td>0</td><td>0.0</td><td>1</td><td>10.0</td></tr></tbody></table>

Abbreviation: GBSDS, Guillain-Barré Syndrome Disability Score.

DISCUSSION

To our knowledge, this is the largest cohort with the acute phase of GBS occurring during childhood in which long-term sequelae have been examined. In this study, we aimed to estimate the frequency and describe the direct (motor, sensory, and fatigue) and indirect (repercussions on quality of life) long-term sequelae (at least 3 years after the acute phase). We found a rate of 67% of late-onset sequelae after the acute phase. Three areas of complaint were investigated: fatigue disorders, present in 33% of patients; sensory disorders, in 55%; and motor disorders, in 31%. In our study, we also aimed to highlight the characteristics of the acute phase that predicted unfavourable long-term development. It is interesting to understand that the population was derived from a first retrospective study in which the rate of important sequelae in the first year of these patients was estimated at 9%.¹³ Usually, the follow-up of these patients was quickly stopped because of a good overall recovery and the minor sequelae probably went unnoticed. We found that the factors associated with late-onset sequelae are related to the severity at the initial phase: GBSDS of at least 4 and loss of the ability to walk. Female sex seems to be a risk factor of more severe sequelae. Therefore, the predictive factors of the severity of sequelae were initial severity and female sex.

All the patients who were included survived, had recovered the ability to walk autonomously, and most could run (98%). Two-thirds of our patients experienced long-term sequelae. Our results highlighted ‘silent’ late-onset sequelae. In an adult population, 65% of minor sequelae were observed⁶ and comparable rates following childhood GBS, which was in line with previous studies.¹² The nature of complaints was in agreement with those in the literature, particularly paraesthesia, pain, and fatigue, which were the most frequent complaints. We compared the frequencies of the different sequelae in our cohort with previous cohorts (Table 6), especially with two paediatric studies¹¹,¹² and one mixed paediatric/ young adult study.⁷ Results of late-onset sequelae are fairly heterogeneous between the four studies because of the areas assessed in each. Our study goes deeper, and shows permanent residual sequelae because one-third of the patients exhibited pain and one-third fatigue. Fatigue is a common manifestation of GBS and has been most studied in adults: 60% of adult patients experience fatigue 1 year after the acute phase.²⁶ Our study extends these findings to the paediatric population, highlighting the importance of examining fatigue in children after GBS.
## Data availability statement

The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding authors.

## Ethics statement

The studies involving humans were approved by Germany: Ethikkommission der Universität zu Lübeck (AZ 18-172). Italy: 352 Comitato Etico Lazio 1c/o A.O. San Camillo Forlanini (2143/CE Lazio 1). Sweden: Regional Ethical 353 Review Board in Göteborg. The studies were conducted in accordance with the local legislation and institutional requirements. The participants provided their written informed consent to participate in this study.

## Author contributions

HoM: Methodology, Writing – original draft. JA: Conceptualization, Investigation, Methodology, Writing – review and editing. CA: Conceptualization, Funding acquisition, Investigation, Methodology, Project administration, Resources, Supervision, Writing – review and editing. CC: Data curation, Investigation, Writing – review and editing. JF: Conceptualization, Investigation, Methodology, Project administration, Writing – review and editing. KH: Conceptualization, Investigation, Methodology, Writing – review and editing. MM: Conceptualization, Investigation, Methodology, Writing – review and editing. HeM: Conceptualization, Data curation, Investigation, Methodology, – review and editing. MR: Conceptualization, Investigation, Methodology, Writing – review and editing. SS: Conceptualization, Funding acquisition, Investigation, Methodology, Project administration, Resources, Supervision, Writing – review and editing. UT: Conceptualization, Funding acquisition, Investigation, Methodology, Project administration, Resources, Supervision, Writing – original draft.

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## Funding

The author(s) declare financial support was received for the research, authorship, and/or publication of this article. This work was supported by the Deutsche Forschungsgemeinschaft (DFG) and the French Agence Nationale de la Recherche (ANR) (DFG-ANR No. 316684170) in Germany and France; the Sunnerdahls Handikappfond, the Swedish state under the agreement between Swedish government and the country councils, the ALF-agreement (ALFGBG-726001) in Sweden; and the Fondazione Carivit in Italy and the Federação das Associações Portuguesas de Paralisia Cerebral e Santa Casa da Misericórdia de Lisboa in Portugal. We acknowledge support for the Article Processing Charge from the Open Access Publication Fund of the University of Greifswald. The funders were not involved in any way in the preparation of this manuscript or the decision to submit it.

## Acknowledgments

We are grateful to the study's research associates who contributed to data collection in the different centers and provided data control and management, which resulted in a quality database. We thank all families for their generous participation.

## Conflict of interest

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

## Publisher's note

All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.

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We also noted a strong association between complaints. For example, fatigue was mainly associated with motor complaints. However, it seems difficult to determine the trend and the direction of the relation: does motor disorder induce fatigue or does a child have walking difficulties because of fatigue?

<table><caption>TABLE 5 Initial characteristics associated with late-onset sequelae (bivariate logistic regressions).</caption><thead><tr><th rowspan="2">Initial characteristics</th><th colspan="2">Presence of sequelae</th><th colspan="3">Bivariate models</th></tr><tr><th>No</th><th>Yes</th><th>OR</th><th>95% CI</th><th>p</th></tr></thead><tbody><tr><td>Male<sup>a</sup></td><td>n = 17, 65%</td><td>n = 34, 53%</td><td>0.6</td><td>0.2-2.0</td><td>0.426</td></tr><tr><td>Mean age at diagnosis (SD) (years:months)</td><td>6:2 (3:10)</td><td>6:7 (4:4)</td><td>1.0</td><td>0.9-1.2</td><td>0.797</td></tr><tr><td>Initial symptoms</td><td></td><td></td><td></td><td></td><td></td></tr><tr><td>Initial GBSDS ≥4</td><td>18%</td><td>59%</td><td>6.6</td><td>1.8-33.0</td><td>0.009*</td></tr><tr><td>Pain</td><td>82%</td><td>74%</td><td>0.6</td><td>0.1-2.4</td><td>0.487</td></tr><tr><td>Paraesthesia</td><td>40%</td><td>20%</td><td>0.4</td><td>0.1-1.5</td><td>0.160</td></tr><tr><td>Motor impairment</td><td>82%</td><td>91%</td><td>2.2</td><td>0.4-13.3</td><td>0.365</td></tr><tr><td>Sensory impairment</td><td>6%</td><td>18%</td><td>3.3</td><td>0.5-65.8</td><td>0.302</td></tr><tr><td>Facial paralysis</td><td>18%</td><td>27%</td><td>1.7</td><td>0.4-8.5</td><td>0.487</td></tr><tr><td>Cranial nerve impairment</td><td>47%</td><td>47%</td><td>1.0</td><td>0.3-3.3</td><td>0.100</td></tr><tr><td>Difficulty walking</td><td>100%</td><td>97%</td><td>—</td><td>—</td><td>—</td></tr><tr><td>Loss of the ability to walk</td><td>18%</td><td>56%</td><td>5.9</td><td>1.6-29.2</td><td>0.014*</td></tr><tr><td>Asthenia</td><td>18%</td><td>29%</td><td>1.9</td><td>0.5-9.8</td><td>0.368</td></tr><tr><td>Autonomic dysfunction</td><td>29%</td><td>44%</td><td>1.9</td><td>0.6-7.1</td><td>0.314</td></tr><tr><td>Ataxia</td><td>41%</td><td>41%</td><td>1.0</td><td>0.3-3.4</td><td>0.100</td></tr><tr><td>Loss of consciousness</td><td>0%</td><td>3%</td><td>—</td><td>—</td><td>—</td></tr><tr><td>Aetiology</td><td></td><td></td><td></td><td></td><td></td></tr><tr><td>Anti-neuronal antibodies</td><td>17%</td><td>41%</td><td>3.4</td><td>0.7-25.3</td><td>0.155</td></tr><tr><td>Campylobacter</td><td>7%</td><td>17%</td><td>2.6</td><td>0.3-53.9</td><td>0.415</td></tr><tr><td>Axonal Guillain-Barré syndrome</td><td>7%</td><td>20%</td><td>3.5</td><td>0.6-69.7</td><td>0.268</td></tr><tr><td>Management</td><td></td><td></td><td></td><td></td><td></td></tr><tr><td>Treatment</td><td>71%</td><td>90%</td><td>4.3</td><td>0.9-23.8</td><td>0.070</td></tr><tr><td>Intubation</td><td>0%</td><td>9%</td><td>—</td><td>—</td><td>—</td></tr><tr><td>Transfer to intensive care</td><td>12%</td><td>27%</td><td>2.7</td><td>0.6-19.3</td><td>0.241</td></tr><tr><td>Mean length of hospitalization (SD) (days)</td><td>7.9 (4.5)</td><td>15.4 (18.6)</td><td>1.1</td><td>1.0-1.3</td><td>0.077</td></tr></tbody></table>

Overall, although only one-third of the children were completely free of sequelae, most patients were considered cured, without sequelae, at the end of the initial follow-up. Our study highlights the need for long-term follow-up for children who have had GBS. Therefore, we recommend a routine consultation at a university centre for evaluation of long-term sequelae 3 years after the acute phase to assess fatigue, sensory impairment (especially pain or paraesthesia), and motor impairment (with the MSWS questionnaire, the most sensitive way of identifying motor disorder). Identifying these sequelae seems significant to effectively inform patients and their families after the acute phase because sequelae can lead to permanent repercussions on everyday life. It may also help to identify appropriate rehabilitation programmes for children. It is known that the symptoms related to GBS can be alleviated in adults after therapy targeting specific activities27 or bicycle exercises,28 with very good results for fatigue and functional scores. Follow-up with rehabilitation physicians seems essential to improve the autonomy and independence of all children who have GBS but there is no evidence-based medical trial about rehabilitation programmes for children after GBS.

<footnote><sup>a</sup>All percentages are those in columns (frequency of the modality in each group defined by the presence or absence of sequelae).</footnote>

<footnote>*p < 0.05.</footnote>

<footnote>Abbreviations: CI, confidence interval; GBSDS, Guillain-Barré Syndrome Disability Score; OR, odds ratio; SD, standard deviation.</footnote>
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Sensory testing with the INCATSSS was almost always normal, which indicates proper large-fibre recovery (tactile discrimination, pallaesthesia, and proprioception). On the contrary, the frequency of paraesthesia and neuropathic pain is more indicative of residual small-fibre sequelae. This predominantly small-fibre complaint has been previously demonstrated in adult patients after GBS.29

We aimed to identify the link between direct late-onset sequelae and repercussion on daily life. Overall, we determined that, for a significant proportion of the children with GBS, residual sequelae are part of daily life and for some there is a great impact on quality of life as practising sports is inhibited (6%), and normal schooling (4%) and functional autonomy (in 14% of cases) are compromised.

Therefore, in our study, the severity of the impact on quality of life seemed to be related to the number of areas of complaint affected: sensory, motor, or fatigue. In most instances, patients whose daily lives were impacted were affected in the three domains.

It would be beneficial if future research were to delve further into this relation between these late-onset sequelae and the GBSDS. GBSDS stage 1, 'minor symptoms', is currently non-specific, and could be subdivided according to the type or number of areas of complaint affected (fatigue, sensory disorders, and motor disorders). These areas of complaint could be integrated into the GBSDS. This graduation would be interesting to improve the characterization of this heterogeneous group of patients with minor sequelae.

As many as one out of every three children who had been affected by GBS were at risk for depressive symptoms (34%). Female sex was a predictive factor and more females (57%) than males (15%) had depressive symptoms. Our results did not correspond with previous ones. Although it is well established that autoimmune diseases of the nervous system increase the risk of psychiatric illnesses,30 the rate of depressive symptoms in GBS was close to 7% to 10%.12,31 We can explain this by the scales used in our study. Both scales, the CDI24 for children and the BDI25 for adults, were used just for screening and to detect depressive symptoms. Therefore, these should be considered as screening scales not as diagnostic scales. In addition, both scales were short versions, identified to be more sensitive than long versions.24

We identified no association between the frequency of depressive symptoms and age at diagnosis or initial severity. However, we noted a significant association between depressive symptoms and fatigue ($p = 0.019$). The same trend was noted between depressive symptoms and motor complaints ($p = 0.019$). The number of complaints was also found to be associated with the risk of depressive symptoms ($p = 0.043$): the risk of depression increases as the number of complaints increases. We also detected an association between depressive symptoms and indirect complaints (repercussions on quality of life). At equal age, sex, and initial severity, children with GBS repercussions on quality of life were found to be most often depressed (OR = 8.2 [1.3–82.3]; $p = 0.041$). However, because our study was cross-sectional, it was difficult to specify the implication of one or the other, especially which was causing the other. A complementary study would be very important to examine the long-term psychiatric repercussions of GBS in children.
<table><caption>TABLE 6 Comparison of the results with data from the literature.</caption><thead><tr><th colspan="5">Population</th></tr><tr><th>Country</th><th>France (this study)</th><th>Netherlands<sup>17</sup></th><th>Canada<sup>16</sup></th><th>Bangladesh<sup>12</sup></th></tr></thead><tbody><tr><td>Number of patients</td><td>51</td><td>37</td><td>31</td><td>38</td></tr><tr><td>Median age at diagnosis (years:months)</td><td>6:6 (1–16)</td><td>9 (0–18)</td><td>9 (3–18)</td><td>20 (4–39)</td></tr><tr><td>Male children (%)</td><td>57</td><td>51</td><td>45</td><td>58</td></tr><tr><td>Median follow-up time (years)</td><td>6 (3–20)</td><td>11 (1–22)</td><td>5 (3–10)</td><td>7 (1–13)</td></tr><tr><td>Median age at evaluation (years:months)</td><td>11:6 (5–31)</td><td>20 (4–39)</td><td>—</td><td>30 (7–55)</td></tr><tr><td>Results</td><td></td><td></td><td></td><td></td></tr><tr><td>Residual complaints (GBS SD ≥1) (%)</td><td>68</td><td>65</td><td>27</td><td>63</td></tr><tr><td>Fatigue (%)</td><td></td><td></td><td></td><td></td></tr><tr><td>Complaints</td><td>34</td><td>—</td><td>12</td><td>—</td></tr><tr><td>Significant (FSS >4)</td><td>19</td><td>—</td><td>—</td><td>—</td></tr><tr><td>Severe (FSS >5)</td><td>5</td><td>14</td><td>—</td><td>13</td></tr><tr><td>Pain (%)</td><td>34</td><td>24</td><td>8</td><td>29</td></tr><tr><td>Muscular weakness (MRCSS <60) (%)</td><td>8</td><td>8</td><td>—</td><td>—</td></tr><tr><td>Muscular weakness (other tests) (%)</td><td>—</td><td>—</td><td>23</td><td>—</td></tr><tr><td>Sensory disorder (INCATSSS >0) (%)</td><td>2</td><td>14</td><td>—</td><td>—</td></tr><tr><td>Sensory disorder (other tests) (%)</td><td>—</td><td>—</td><td>15</td><td>—</td></tr><tr><td>Paraesthesia (%)</td><td>47</td><td>38</td><td>—</td><td>11</td></tr><tr><td>Tremors (%)</td><td>9</td><td>11</td><td>—</td><td>—</td></tr><tr><td>Cranial nerve impairments (%)</td><td>2</td><td>3</td><td>—</td><td>—</td></tr><tr><td>Pathological deep tendon reflexes (%)</td><td>27</td><td>5</td><td>27</td><td>—</td></tr><tr><td>Symptoms of depression (%)</td><td>32</td><td>7</td><td>—</td><td>16</td></tr></tbody></table>

Data are expressed as median (range) for quantitative data; otherwise, they are percentages.

Abbreviations: FSS, Fatigue Severity Scale; GBS, Guillain–Barré syndrome; INCATSSS, Inflammatory Neuropathy Cause and Treatment Sensory Sum Score; MRCSS; Medical Research Council Sum Score; SD, standard deviation.

In our study, the presence of late-onset sequelae after the critical phase was significantly related to two factors. First, we found that the initial severity during the acute phase of GBS was correlated with the presence of late-onset sequelae and more severe sequelae (with a larger number of areas of complaint present in the severity score). In adult GBS, the initial clinical severity has been well known to be a predictive factor for late-onset sequelae.32 However, to our knowledge, it is the first time that it has been attested in children. This supports the need for optimal treatment, especially for children with severe forms of GBS in the acute phase because it would seem that they will be the most affected in the long term. Although intravenous immunoglobulins seem to accelerate motor recovery in children, 33 this treatment may not influence the prognosis in the longer term.34,35

Second, in our study, female sex was a predictive factor for more serious sequelae on the severity score. We found only one study with adult patients that also identified female sex as a predictive factor for a poor long-term prognosis after GBS, regardless of the functional or psychological level.36 Several hypotheses could support this finding. It has been consistently demonstrated in many studies37,38 that the GBS incidence was higher among males than females (male: female ratio 1.59:1). Such male predominance is uncommon for an autoimmune disease. Furthermore, GBS incidence is modified during pregnancy,39 which supports the theory of a hormonal action or a genetic/ sex-determination influence. This raises
the question of whether there is also a difference in the sequelae between female and male
children.

Lastly, in our study, we detected no association between age at the time of the episode and the rate of sequelae. Therefore, age at the time of GBS is not an explanatory factor for sequelae, nor is it an aggravating or enhancing factor. Consequently, it should be noted that the silent residual sequelae of GBS are generally stable 2 years after the onset of GBS. However, this finding is very important to set the date for follow-up care.

To our knowledge, ours was the largest cohort with the acute phase of GBS occurring during childhood in which long-term sequelae have been examined. Although the number of patients lost to follow-up compared with the original cohort may raise concerns about selection bias, this bias is more likely to be in favour of underestimating the rate of distant sequelae, as it has been shown that initial severity is associated with the risk of sequelae, and the initial involvement of included patients was slightly less severe (non-significant) than that of excluded patients (Table S1 and Figure S1). However, we cannot exclude the possibility that the non-included patients, who had an episode earlier than the others, had different characteristics in other respects and that this influenced the result.

Patient assessment was performed by a single examiner, which helped to homogenize the
assessment across patients, but did not eliminate examiner-related measurement bias.
However, we used several tests and questionnaires that are validated and standardized. Three
types of complaint were investigated: fatigue, sensory disorders, and motor disorders, by
means of both a semi-structured interview and standardized scales. The semi-structured
interview, even if more subjective and examiner-relative, was more sensitive than the
standardized questionnaire scores in assessing fatigue and neuropathic pain. The standardized
questionnaires detected less than half of the children complaining of fatigue during a semi-
structured interview. Therefore, in clinical practice, for these two domains it seems that
identifying the complaint during a semi-structured interview may be preferable to using
standardized scales. Conversely, for motor impairment, the scores on the questionnaires
seemed more sensitive than recording the complaint in a semi-structured interview. The
MSWS questionnaire, especially, seemed to effectively detect patients with a motor and
walking disorder with a threshold set at 5%, beyond which the infirmity experienced seemed
significant. According to our observations, the DN4 questionnaire was found to be
inappropriate for paediatric patients. Indeed, most of the items were too complex for younger
patients. This observation is shared by other paediatricians and an adaptation of the DN4
questionnaire using illustrations for younger patients is currently being validated. The ONLS
questionnaire was useful in discovering physical functioning, but it could not take account of
patients' participation in activities.

In this study, we created a severity score based on the number of domains affected in sensory
impairment, motor impairment, and fatigue. This score is interesting because it allows us to
better characterize a heterogeneous group of patients with mild sequelae. We have shown that
more severe scores have more frequent impacts on activities of daily living. However, for
clinical use, these tests and questionnaires should first be validated specifically in other
populations, which was not the aim of this study. In terms of identifying predictive factors,
this study allowed us to prospectively identify initial characteristics associated with the
presence and severity of sequelae. It would be interesting to confirm the relevance and
evaluate the performance of these factors, as predictors that could potentially guide clinical
decisions, in other studies and other populations. However, from this study, we cannot draw
causal conclusions about the relation between these factors and subsequent disease progression, as neither the study design nor the analysis strategy was designed to do so.

## CONCLUSION

We highlight the fact that two-thirds of the children in our study had late-onset sequelae after childhood GBS. These did not improve over time. Late-onset sequelae are often underestimated in children who have recovered the ability to walk and, for the most part, to run after being paralysed in the acute phase of the disease.

The risk of sequelae is related to the initial severity (particularly the loss of the ability to walk during the acute phase of GBS) and to sex (females are more likely than males to have more severe sequelae).

Three areas of complaint are possible: fatigue, sensory disorders, and motor disorders. We have highlighted the fact that silent residual sequelae of GBS limit participation in sports, normal schooling, autonomy, etc., de facto hindering everyday life. The association of several areas of complaint leads to repercussions on everyday lives. Depressive symptoms are also especially common. These conditions could be integrated into the scale to evaluate sequelae after GBS (GBSDS). Stage 1, which is characterized by the rather imprecise term 'minor sequelae', could be subdivided. A full follow-up after 2 or 3 years from the acute phase seems essential once the situation has stabilized.

## CONFLICT OF INTEREST STATEMENT

The authors have stated that they had no interests that might be perceived as posing a conflict or bias.

## DATA AVAILABILITY STATEMENT

data are available

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