Longitudinal Evaluation of Patient Outcomes and Impact Assessment on Family Members of Home Parenteral Nutrition: New Directions for Research
Conditions: Parenteral Nutrition
Sponsor: University of Manchester

        IRAS Project ID: 270162                                   
Longitudinal PROMs for HPN V2: 16/12/2019                                                                       Page | 1  
 
 
 
 
 
 
Research Protocol Protocol  
 
Longitudinal Evaluation of Patient Outcomes and Impact 
Assessment on Family Members of Home Parenteral Nutrition: 
New Directions for Research 
IRAS 270162 
 
July 2019 
 
Dr Sorrel Burden, Senior Clinical Lecturer in Dietetics, University of Manchester & Salford 
Royal Foundation Trust 
Professor Simon Lal, Lead Physician, Intestinal Failure Unit, Salford Royal Foundation 
Trust & University of Manchester 
 
 

        IRAS Project ID: 270162                                   
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Contents 
Research team and key contacts 
4 
Introduction 
5 
Background 
5 
Quality of life in people with intestinal failure 
5 
Family member’s research 
6 
New innovation in treatments 
7 
Study objectives 
8 
Study design and protocol 
8 
Study 1: Systematic Review 
8 
Study 2: Longitudinal assessment of quality of life in people receiving HPN 
8 
Objective: 
8 
Patient reported outcome: 
8 
Method: 
9 
Study 3: Assessing the impact of HPN on families and partners. 
9 
Objectives: 
9 
Recruitment: 
9 
Timelines 
10 
Timelines for longitudinal survey and study of family members’ experiences. 
10 
Study participants 
11 
Study 2: Longitudinal assessment of quality of life in people receiving HPN 
11 
Recruitment: 
11 
Inclusion criteria: 
11 
Exclusion criteria: 
11 
Sample size: 
11 
Study 3: Assessing the impact of HPN on families and partners. 
12 
Recruitment: Family members will be nominated by eligible patient participants, who will 
receive both the patient survey and family member survey. 
12 
Inclusion criteria: 
12 
Exclusion criteria: 
12 
Sample size: 
12 
Outcome measures 
12 

        IRAS Project ID: 270162                                   
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Data Collection, Source data and Confidentiality 
12 
Statistical considerations 
13 
Data monitoring and quality assurance 
14 
Safety considerations and adverse events 
14 
Peer review 
14 
Ethical Considerations 
14 
Approvals 
14 
Risks 
14 
Statement of indemnity 
15 
Funding 
15 
Publication policy 
15 
References 
15 
 
 
 
 
 
 
 
 
 

        IRAS Project ID: 270162                                   
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Research team and key contacts 
 
Chief Investigator: 
 
Name:     Dr Sorrel Burden 
 
Address:  Rm 5.324, Jean Mcfarlane 
building, 
University of Manchester 
Oxford Road, 
Manchester 
M13 9PL 
 
Email: Sorrel.burden@manchester.ac.uk 
 
Telephone: 0161 306  
 
Co-investigator(s): 
 
Name:     Mrs Debra Jones 
 
Address:  Rm 5.327, Jean Mcfarlane 
building, 
University of Manchester 
Oxford Road, 
Manchester 
M13 9PL 
 
Email: Debra.jones@manchester.ac.uk 
 
Telephone: 0161 306 1508 
Sponsor(s): 
 
Name: The University of Manchester  
 
Sponsor contact: Ms Lynne Macrae, 
Faculty Research Practice Co-ordinator 
 
Address:  
Faculty of Medical and Human Sciences 
Research Office 
3.53 Simon Building 
University of Manchester 
M13 9PL 
 
Email: fmhsethics@manchester.ac.uk 
 
Telephone: 0161 275 5436 
Lead R&D Trust contact(s): 
 
Name:     Prof. Simon Lal 
 
Address:   Intestinal Failure Unit 
Salford Royal Hospital 
Stott Lane 
Salford 
M6 8HD 
 
Email: Simon.lal@srft.nhs.uk 
 
Telephone:  
 
 

        IRAS Project ID: 270162                                   
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Introduction 
Providing a person with food through a vein is known as parenteral nutrition (PN) or 
artificial tube feeding. This process is used when nutrients from food can't be taken in by 
the intestine (intestinal failure). Patients with type 3 intestinal failure are completely reliant 
on artificial feeding and often manage this at home; home parenteral nutrition (HPN). HPN 
therapy is life saving for these patients. 
It is important to assess patients quality of life and their own reported effects of the HPN. 
The Parenteral Nutrition Impact Questionnaire (PNIQ) is a proven tool for measuring 
quality of life when receiving HPN. 
A recent study involving the use of the PNIQ in multiple hospitals in the UK, showed that 
those on fewer nights of HPN had better quality of life than those on more nights of HPN. 
Whilst this was useful for looking at quality of life at one time point, it is now important to 
assess change in quality life over time and any impact on family members quality of life.  
This study will recruit HPN patients across the UK and ask them to complete the PNIQ 
survey at several different time points. This will assess the impact of HPN over time. 
Family members involved in the participants HPN care will also be asked to complete a 
carer burden survey (at one time point) to assess the impact of HPN on carers.  
The study is being funded by Shire Pharmaceuticals Ltd. 
Background 
Quality of life in people with intestinal failure 
Patients with Type 3 intestinal failure (IF) are dependent on parenteral support which may 
include fluids, electrolytes or nutrients [1].  From a recent national survey in the U.K., the 
point prevalence of patients dependent on home parenteral nutrition (HPN) was 1144 and 
34% of these patients had a diagnosis of short bowel syndrome (SBS) [2]. Although HPN is 
recognised as a lifesaving therapy for these patients, it is not without risks. The 
complications of parenteral nutrition (PN) are serious and include catheter related blood 
stream infections leading to septicaemia, and the presence of a central venous catheter can 
cause thrombosis and embolism [3]. There are also more long-term adverse complications 
including hepatic failure and osteoporosis [4].   

        IRAS Project ID: 270162                                   
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It is also well documented that patients who require HPN have a decreased quality of life 
(QoL) [5; 6]. Moreover, it has been established that people dependent on PN have higher 
rates of depression compared to the general population, suffer a higher rate of anxiety and 
are unlikely to ever return to meaningful employment [5; 7].  Other factors that affect QoL 
include use of opioids, nocturia, the presence of a stoma, age and the number of infusions 
required per week [8; 9; 10].   
The ability to be able to assess patient reported outcomes for different treatments is now 
essential for patients with chronic IF; the Parenteral Nutrition Impact Questionnaire (PNIQ) 
provides a validated tool to evaluate differences in needs based QoL.   
From a recent large multi-centre U.K. survey, we have now established face validity of the 
PNIQ and also reported a clinically meaningful difference between patients with different 
underlying diseases as well as between patients requiring different numbers of nights of PN. 
The survey demonstrated PNIQ was sensitive to change in treatment delivery and there was 
an increase in PNIQ score (reflecting improved QoL) in those individuals dependent on 
fewer nights of HPN [11]. While this survey assessed QoL at one time point, face validity 
addressing within person variation over time is now required. It is important for clinicians and 
patients to evaluate the impact of changes in HPN burden over time, as well as any impact 
of changes in treatment modalities. New technologies within intestinal failure, including the 
introduction of growth factors to patients with SBS, have led to the increased demand for 
tools with comprehensive validity assessments to be able to measure patient reported 
outcomes. 
The ability to be able to assess patient reported outcomes overtime in a large cohort of 
patients with IF-SBS is therefore very timely with new innovations imminent within the UK 
landscape. The assessment tool used will be able to show any changes that occur over time 
in relation to modifications in HPN provision. Treatment changes may be due to 
gastrointestinal adaption, small bowel transplant or use of new growth factors.       
Family member’s research  
In contrast to patients on HPN there is a paucity of research on QoL experienced by family 
members of people dependent on long-term HPN. The impact on family members of people 
needing HPN with advanced cancer has been evaluated and family members reported 
feeling very responsible for their relative [12]. A survey of 178 families, with a family member 
with advanced cancer dependent on HPN, demonstrated that family members provided daily 

        IRAS Project ID: 270162                                   
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support for patients on HPN but were faced with challenges including loss of friends, loss of 
employment and depression [13].  However, while these studies describe the impact of HPN 
burden on the family of patients with advanced cancer and palliative needs, there are no 
comparable data detailing the impact of living with a family member on HPN with a chronic, 
long term, benign underlying condition. Prior to investigating methods of providing support 
for family members of people on HPN it is essential to investigate the experiences of family 
members and carers. Once this has been determined then the necessity to intervene to 
provide support can be evaluated.   
New innovation in treatments  
Recently, the development of growth factors, such as glucagon-like peptide 2 analog 
(Teduglutide), aimed at enhancing small bowel absorption, has changed the landscape of 
therapeutic options available for people with SBS [14]. Teduglutide (Revestive® Shire 
Pharmaceuticals) enhances fluid, electrolyte, and nutrient absorption. It is indicated in the 
treatment and management of SBS-associated IF (SBS-IF).  Teduglutide is an analogue of 
glucagon like peptide 2, a naturally-occurring hormone which promotes the growth of 
nutrient absorbing cells on the surface of the intestine.  
Teduglutide reduces HPN requirements by enhancing absorption requirements; hence, 
PNIQ is an ideal measurement tool to assess this and other treatment modalities.  The ability 
to assess outcomes in patients on HPN and their family members is required so new 
innovations in treatments and service provision can be fully evaluated. The introduction of 
new technological innovations in the UK is imminent so this research is timely as it will 
assess patient reported outcomes prospectively allowing for any changes to be reported pre 
and post the introduction of innovations within the UK population 
Future direction of research that is required for patients and their families can be explored 
by implementing a James Lind Priority Setting Partnership between healthcare 
professionals, patients and family members.    
The intestinal Failure Unit (IFU) at Salford Royal Foundation Trust (SRFT) currently 
manages around 280 patients on long-term PN throughout the UK. Each year there are 63 
new patients discharged from the Unit at Salford whilst on PN.        
The proposed project is therefore a logical development in assessing the validity of PNIQ by 
addressing changes over time in a national cohort of HPN patients. It is also necessary to 

        IRAS Project ID: 270162                                   
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determine the impact on families of long-term HPN. However, to develop research for people 
receiving HPN, this proposal also incorporates a Patient and Professionals Priority Setting 
Partnership to be registered with the James Lind Alliance. This is a process whereby 
research priorities are set independently with health care professionals and patients.     
Study objectives 
1. Systematic review of the literature of quality of life in patients and their families on HPN 
with intestinal failure.   
2. To determine patient reported outcomes using PNIQ longitudinally in people receiving 
HPN.   
3. Assess the impact of living with a family member dependent on HPN.  
Study design and protocol 
Study 1: Systematic Review  
Undertake a systematic review of research on QoL in patients receiving HPN with full quality 
assessment.  
Study 2: Longitudinal assessment of quality of life in people receiving HPN 
Objective: To measure patient reported outcomes longitudinally in people receiving HPN.  
Patient reported outcome: The Parenteral Nutrition Impact Questionnaire (PNIQ)© was 
developed from undertaking  semi-structured qualitative interviews with people on HPN to 
identify ways in which need fulfilment was impaired by HPN. The interviews were used to 
develop a questionnaire for a postal survey undertaken in two NHS Trusts. The survey had 
the purpose of developing items for scale, scaling properties including unidimensional 
properties, construct validity and reproducibility.  The survey was sent out twice to a 
proportion of respondents to determine intra-rater reliability. Cognitive debriefing interviews 
were then performed to check the relevance of the scale, clarity and ease of use [15]. This 
enabled ratification of themes relevant to people living with HPN. The tool was also validated 
against the Nottingham Health Profile [16]. 
There is currently a paucity of longitudinal data for patients on HPN using any tool. This 
proposed study now looks at determining longitudinal face validity and aims to determine if 
PNIQ score is sensitive to length of treatment and treatment changes, if any, over time. The 

        IRAS Project ID: 270162                                   
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frequency of administration of PN, and other changes in QoL including Short version 36 
questionnaire (SF-36) and EQ-5D   
Method: Design of the proposed study is a longitudinal observational cohort study and data 
will be collected using a questionnaire sent to participants as a national postal survey. 
Patients may also receive the questionnaire via a clinic appointment or via email. The PNIQ 
will be used along with a brief questionnaire to collect participant’s characteristics and 
demographics. Questions will also be included on recent changes in treatment, length of 
time on PN, frequency of PN infusions per week, re-admissions to hospital and any changes 
in medications. We will also include EQ-5D-5L generic health questionnaire and a 
questionnaire that measures Activities of Daily Living Questionnaire, to assess generic QoL.  
PNIQ Tool: The PNIQ is made up of 20 questions. The questions are specifically related to 
HPN and aim to assess QoL and the overall impact of treatment in those who receive HPN. 
Each statement on PNIQ has a dichotomous response ‘True’ or ‘Not true’. Each response 
is allocated a score of 1 for ‘True’ and a score or 0 for ‘Not true’. All item scores are summed 
up to give a total score ranging from 0 (good quality of life) to 20 (very poor quality of life).  
Study 3: Assessing the impact of HPN on families and partners.  
This study will be run in conjunction with the patient survey above to assess family members’ 
quality of life.  
Objectives:  Assess the impact of living with a family member on HPN. Explore family 
members’ and caregivers’ experiences of living with a patient with IF on HPN. 
Recruitment: We will aim to recruit 300 hundred family members or caregivers of patients 
who are having HPN. We will ask each patient recruited for Study 2 to nominate a family 
member to this survey. The family members’/care givers’ survey will be sent to the patient 
along with the survey that they are to complete themselves. Both surveys can be returned 
in the freepost envelope that will be provided. A receipt of a completed family member survey 
will be taken as consent.   
 

        IRAS Project ID: 270162                                   
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Timelines 
Timelines for longitudinal survey and study of family members’ experiences.    
 
Year 1  
Year 2 
Year 3 
Systematic Review: Study 1  
Undertake and complete review 
 
 
 
 
 
 
 
 
 
 
Longitudinal QoL: Study 2  
Study set up 
 
 
 
 
 
 
 
 
 
 
Patient recruitment  
 
 
 
 
 
 
 
 
 
 
Follow up  
 
 
 
 
 
 
 
 
 
 
Data inputting  
 
 
 
 
 
 
 
 
 
Data Analysis  
 
 
 
 
 
 
 
 
 
 
 
Write up for longitudinal study  
 
 
 
 
 
 
 
 
 
 
 
Family members:  Study 3  
Study set up 
 
 
 
 
 
 
 
 
 
 
Participant  recruitment/survey   
 
 
 
 
 
 
 
 
 
 
Data inputting  
 
 
 
 
 
 
 
 
 
 
Data Analysis  
 
 
 
 
 
 
 
 
 
 
Write up for family members/care 
givers survey   
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 

        IRAS Project ID: 270162                                   
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Study participants 
Study 2: Longitudinal assessment of quality of life in people receiving HPN 
Recruitment: Each hospital will who agrees to take part will recruit participants from 
National Health Service (NHS) locations throughout England. The direct clinical care team 
at each NHS location will identify and recruit patients with type 3 intestinal failure requiring 
HPN.  
Inclusion criteria: All people in receipt of HPN due to type 3 intestinal failure including new 
patients and those 18 years and over. 
Exclusion criteria: Those who cannot give informed consent and cannot read or write in 
English. 
Upon identification, participants will be sent a pack which includes all explanatory study 
documentation and questionnaires.  Completion and return of the questionnaires will be 
taken as consent. As this is a longitudinal survey we will ask participants if they are happy 
to be contacted directly by the research team and disclose their names and addresses 
(consent to contact), thus enabling the researchers to send out (post or email) follow up 
questionnaires at 5 monthly intervals. The first of the follow up questionnaires will be sent 
along with a participant information sheet and the participant will be asked to complete and 
return written consent in order to formally consented onto the study. Data from follow up 
surveys will only be entered into the study if written consent is provided.   
Sample size: Given the study design, a formal comparison test suitable for a sample size 
calculation is not available for the main survey proposed. Hospitals with substantial HPN 
populations will be identified and considered as potential sites for this study. At least 15 sites 
will be identified with an overall estimated total of 1400 patients receiving HPN.  A previous 
survey in patients with HPN, conducted  at Salford Royal Foundation Trust had a response 
rate of 45% across two study sites, indicating an expected sample size of 630 could be 
achieved if all 1400 patients were approached for this study. 
Given the number of subjects approached in the previous study (n=506) an 80% binomial 
confidence interval (CI) of 43% to 49% for the 45% response rate indicated that we had an 
80% confidence in a response between 447 and 510, or 90% confidence of a sample size 
greater than 447. 

        IRAS Project ID: 270162                                   
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Study 3: Assessing the impact of HPN on families and partners.  
Recruitment: Family members will be nominated by eligible patient participants, who will 
receive both the patient survey and family member survey. 
Inclusion criteria: A family member or person involved in the healthcare of the participating 
patient. We will ask patients to nominate their closest family member or friend who in their 
opinion is potentially most effected by the parenteral feeding.    
Exclusion criteria:  Family members not directly involved with caring for participants.  
The study pack sent to patients will also contain the family member survey and extra study 
information will be provided within this survey for the family member to read. Completion 
and return of the survey will be taken as family member consent. No personal or contact 
details will be asked for in the family survey. 
Sample size: 
Based on the sample size from the patient survey and assuming that we receive 50% 
response rate from family members we expect a sample size of 300. 
Outcome measures 
For the patient survey the primary outcome will be patient reported PNIQ score. Other 
outcomes of interest will include the participants’ age, gender, disease state and socio-
economic group. Secondary outcome measures relating to PN will include; frequency and 
duration of time on PN; treatment changes; medication alterations, diagnosis and changes 
in social circumstances. 
For the Family member survey the primary outcome will be carer burden score and quality 
of life. Other outcomes of interest will include: Employment status, educational attainment, 
income category, recent medication or hospital care received, and how supported they feel 
in the carer role. 
Data Collection, Source data and Confidentiality 
The initial patient survey sent out by the clinical team will be allocated an identification 
number, which will be noted by each centre and a recruitment log will be completed. This 
will allow for linkage back to the participant without sharing identifiable participant details 

        IRAS Project ID: 270162                                   
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with research team members (pseudonymisation). If a participant provides their contact 
details on the first survey these details will be received by the research team. Upon receipt 
of personal details the research team will immediately separate these from the survey data 
and replace with the participants ID number. Personal details and survey details will be 
entered into separate electronic database and a secure pseudonymisation key will be 
created. All databases will be stored within the University of Manchester’s Research Data 
Storage (RDS) system. During the course of this study, if we have any concerns about the 
safety or safety of others we will need to break confidentiality and inform the patients 
hospital care team. We will adhere to the guidelines specified by General Data Protection 
Regulations (GDPR 2018).    
The family member survey will be sent out by the clinical team and will be allocated an 
identification number, which will be noted by each centre and a recruitment log will be 
completed. This will allow for linkage back to the participant without sharing identifiable 
participant details with research team members (pseudonymisation). The family member 
survey will not ask for consent to contact and so no identifiable information will be received 
by the research team.  
Statistical considerations 
The responses from patient study participants will be summarised and reported using 
standard descriptive statistics and appropriate graphical techniques. Factors influencing 
the primary outcome, of the PROMs PNIQ score, will be investigated in the form of an 
exploratory analysis. Factors of interest include the participants’ age, gender, disease 
state and socio-economic group. Secondary outcome measures relating to PN, including; 
frequency and duration of time on PN; treatment changes; medication alterations, 
diagnosis and changes in social circumstances will be investigated. In each case the 
appropriate regression model will be chosen given the structure of the outcome of interest 
(continuous, binary, ordinal). The factors of interest will be investigated, initially individually 
in an unadjusted model and then adjusted for potential confounders chosen a priori. 
Where possible, given the information recorded, measures will be taken to account for 
potential bias due to clustering of subjects within NHS sites, non-response characteristics, 
and sample specific characteristics. All appropriate goodness of fit and model assumptions 
will be checked and measures will be taken into account for any departures. The presence 

        IRAS Project ID: 270162                                   
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of multiple testing will be monitored and steps will be taken to adjust the p-value 
accordingly. 
The responses from family member study participants will be summarised and reported 
using standard descriptive statistics and appropriate graphical techniques. Difference 
between groups will be explored including disease states, age, gender, and frequency of 
HPN infusions per week whilst at home, length of function bowel, length of time on HPN, 
disease classification. 
Data monitoring and quality assurance 
The study will be subject to the audit and monitoring regime of the University of Manchester. 
Quarterly reports will be provided to the funder (Shire International) via an electronic portal. 
Safety considerations and adverse events 
As this study is survey based there will be no risks or adverse events. 
Peer review 
Clare Donnellan from Leeds Teaching Hospitals NHS Foundation Trust and Martyn Dibb from Royal 
Liverpool University Hospital have both reviewed and provided a letter of support.  
In addition, the nutrition research team at the University of Manchester have reviewed and 
approved the scientific quality of the research. 
Ethical Considerations 
Approvals 
All governance and ethical approvals will be obtained prior to starting the project. 
NHS Research Ethics Committee approval will be obtained before commencing research. 
The study will be conducted in full conformance with all relevant legal requirements and the 
principles of the Declaration of Helsinki, Good Clinical Practice (GCP) and the UK Policy 
Framework for Health and Social Care Research 2017.   
Risks 

        IRAS Project ID: 270162                                   
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There are no risks associated with completing a survey. The survey is not of a sensitive 
nature. However, all surveys will be monitored by the research team and any sensitive 
issues that arise will be passed onto the clinical team 
Statement of indemnity 
The University has insurance available in respect of research involving human subjects 
that provides cover for legal liabilities arising from its actions or those of its staff or 
supervised students.  The University also has insurance available that provides 
compensation for non-negligent harm to research subjects occasioned in circumstances 
that are under the control of the University. 
Funding  
This project is funded by an unrestricted research grant from Shire International GmbH. 
Publication policy 
Following completion of the study the results will be published in open access, peer reviewed 
journals. 
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adults. Clinical Nutrition. 2016, 35, 247-307. 
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4. Dibb M, Soop M, Teubner A et al. Survival and nutritional dependence on home 
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7. Carlson GL, Maguire G, Williams N et al. Quality of life on home parenteral nutrition: a 
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