Contribution of perinatal conditions to cerebral palsy in Uganda
Authors: Jane Hassell, Cally Tann, Richard Idro, Nicola J Robertson
Journal: The Lancet Global Health (2018)

Worldwide, an estimated 93 million children are disabled, 80% of whom live in low-income countries.1WHOWorld report on disability.http://www.who.int/disabilities/world_report/2011/report.pdfDate: 2011Google Scholar Cohort studies in high-income countries attribute more than 50% of cerebral palsy cases to premature birth or prenatal causes.2Himmelmann K Uvebrant P The panorama of cerebral palsy in Sweden. XI. Changing patterns in the birth-year period 2003–06.Acta Paediatr. 2014; 103: 618-624Crossref PubMed Scopus (104) Google Scholar Equivalent data have been difficult to obtain in low-resource settings, limiting the ability to lobby for better service provision for these children. In October, 2017, Angelina Kakooza-Mwesige and colleagues3Kakooza-Mwesige A Andrews C Peterson S Wabwire Mangen F Eliasson AC Forssberg H Prevalence of cerebral palsy in Uganda: a population-based study.Lancet Glob Health. 2017; 5: e1275-e1282Summary Full Text Full Text PDF PubMed Scopus (80) Google Scholar published the largest population-based study of cerebral palsy in sub-Saharan Africa. This important work highlighted the higher prevalence of cerebral palsy in children in sub-Saharan Africa than in children in high-income countries, and that a greater proportion of cases are from potentially preventable causes. However, for diagnostic precision, consistent with all major international cerebral palsy registers, children younger than 2 years were not included. We suggest that further exploration of this younger group is important given the high proportion of full-term neonatal cases with severe cerebral palsy identified in this setting. We wish to add our experience of this important younger subgroup in an urban hospital setting in Kampala, Uganda. We investigated the causes and subtypes of cerebral palsy in 130 children younger than 18 years who presented to Mulago National Referral Hospital—the largest paediatric centre in Uganda—over an 8 week period in 2013. Children younger than 2 years were not excluded because of concern that those with severe cerebral palsy might not survive beyond early childhood. Children in whom there was concern about movement or posture were identified by doctors and therapists working in the paediatric in-patient wards and neurology, physiotherapy, and occupational therapy clinics. Children were recruited and assessed by the study lead (JH) or a local paediatrician trained in cerebral palsy assessment, assisted by a local language (Luganda) translator. Written consent was obtained. Cerebral palsy diagnosis was confirmed in cases of onset of non-progressive movement disorder before age 2 years. Children with neural tube defects or isolated hypotonia were excluded. Assessment involved obtaining detailed retrospective histories from primary caregivers, including any self-identified antecedents to the onset of motor impairment, and neurological examination. Cerebral palsy subtype was assigned according to the Surveillance of Cerebral Palsy in Europe hierarchical classification. Neuroimaging was not available. In this hospital-based cohort, 56% of patients were male, 9% were in-patients, and 63% were younger than 2 years (median age 17 months [IQR 9–29]). 78% of patients had bilateral spastic disease; 72% had four-limb spastic disease resulting in severe functional impairment. 68% of caregivers gave histories consistent with term intrapartum-related encephalopathy or neonatal sepsis, and 76% attributed their child's cerebral palsy to severe (full-term) neonatal illness, supported by a history of admission to neonatal care facilities (table). Two children were HIV positive.TableAttributed causes of cerebral palsyTotal (n=130)Subtype of cerebral palsyBilateral spastic (n=101)Unilateral spastic (n=15)Dyskinetic (n=14)Premature birth (<37 weeks)8 (6%)8/8 (100%)0/8 (0%)0/8 (0%)Term neonatal illness99 (76%)......Intrapartum-related encephalopathy, with or without infection75 (58%)66/75 (88%)5/75 (7%)4/75 (5%)Neonatal sepsis, with no evidence of encephalopathy14 (11%)13/14 (93%)1/14 (7%)0/14 (0%)Neonatal jaundice7 (5%)0/7 (0%)0/7 (0%)7/7 (100%)Other neonatal illness3 (2%)1/3 (33%)2/3 (67%)0/3 (0%)Post-neonatal event14 (11%)......CNS infection12 (9%)9/12 (75%)2/12 (17%)1/12 (8%)Other2 (2%)0/2 (0%)2/2 (100%)0/2 (0%)Unknown9 (7%)4/9 (44%)3/9 (33%)2/9 (22%)Data are n (%) or n/N (%). Open table in a new tab Data are n (%) or n/N (%). Key differences exist between our urban hospital-based cohort and the predominantly rural population-based cohort reported by Kakooza-Mwesige and colleagues.3Kakooza-Mwesige A Andrews C Peterson S Wabwire Mangen F Eliasson AC Forssberg H Prevalence of cerebral palsy in Uganda: a population-based study.Lancet Glob Health. 2017; 5: e1275-e1282Summary Full Text Full Text PDF PubMed Scopus (80) Google Scholar Our cohort was younger (median age 17 months vs 4–5 years), with more cases of four-limb cerebral palsy, suggesting a higher prevalence of global brain injury that usually results from intrapartum or neonatal complications, as opposed to less disabling unilateral lesions. The younger age of patients and greater severity of disease seen in our hospital cohort might reflect increased care seeking or referral for these patients, as well as an increased incidence of comorbidities (eg, infection, malnutrition) precipitating hospital attendance. In another Mulago Hospital-based study of cerebral palsy, also by Kakooza-Mwesige and colleagues,4Kakooza-Mwesige A Forssberg H Eliasson AC Tumwine JK Cerebral palsy in children in Kampala, Uganda: clinical subtypes, motor function and comorbidities.BMC Res Notes. 2015; 8: 166Crossref PubMed Scopus (24) Google Scholar children older than 2 years recruited from the cerebral palsy outpatient service (n=135) were also younger (median age 3·5 years) and more had severe impairment (39% vs 20%) than in the recent population-based study.3Kakooza-Mwesige A Andrews C Peterson S Wabwire Mangen F Eliasson AC Forssberg H Prevalence of cerebral palsy in Uganda: a population-based study.Lancet Glob Health. 2017; 5: e1275-e1282Summary Full Text Full Text PDF PubMed Scopus (80) Google Scholar Difficulty confirming cerebral palsy subtype in children younger than 2 years might have affected our findings; neurodisability from severe brain injury is often apparent early, whereas milder impairments might be missed. The lower proportion of bilateral spastic disease in Kakooza-Mwesige and colleagues' hospital-based cohort4Kakooza-Mwesige A Forssberg H Eliasson AC Tumwine JK Cerebral palsy in children in Kampala, Uganda: clinical subtypes, motor function and comorbidities.BMC Res Notes. 2015; 8: 166Crossref PubMed Scopus (24) Google Scholar than in our cohort (46% vs 78%) might be explained by our inclusion of patients younger than 2 years and of in-patients, potentially with cerebral palsy-related comorbidities. However, the population-based study3Kakooza-Mwesige A Andrews C Peterson S Wabwire Mangen F Eliasson AC Forssberg H Prevalence of cerebral palsy in Uganda: a population-based study.Lancet Glob Health. 2017; 5: e1275-e1282Summary Full Text Full Text PDF PubMed Scopus (80) Google Scholar also showed a trend towards increased cerebral palsy prevalence and severity in younger children. The very high proportion of severely disabled young children in our hospital-based cohort indicates an increased early morbidity and mortality. Therefore, studies excluding young children might underestimate the preventable burden of severe neurodisability. Importantly, 73% of Kakooza-Mwesige and colleagues' population-based cohort3Kakooza-Mwesige A Andrews C Peterson S Wabwire Mangen F Eliasson AC Forssberg H Prevalence of cerebral palsy in Uganda: a population-based study.Lancet Glob Health. 2017; 5: e1275-e1282Summary Full Text Full Text PDF PubMed Scopus (80) Google Scholar were neonatal full-term cases; however, no further perinatal history was given. Our study suggests that intrapartum-related encephalopathy and sepsis are key contributors to this cerebral palsy burden and that severe cases attributed to these causes might die before reaching age 2 years, when they can be included in cerebral palsy registries. Newborn deaths due to premature birth, intrapartum-related events, and sepsis account for almost half of under-5 mortality worldwide,5Liu L Oza S Hogan D et al.Global, regional, and national causes of under-5 mortality in 2000–15: an updated systematic analysis with implications for the Sustainable Development Goals.Lancet. 2016; 388: 3027-3035Summary Full Text Full Text PDF PubMed Scopus (1723) Google Scholar with a high estimated burden of adverse neurodevelopmental outcomes in survivors.6Blencowe H Lee AC Cousens S et al.Preterm birth-associated neurodevelopmental impairment estimates at regional and global levels for 2010.Pediatr Res. 2013; 74: 17-34Crossref PubMed Scopus (264) Google Scholar, 7Lee AC Kozuki N Blencowe H et al.Intrapartum-related neonatal encephalopathy incidence and impairment at regional and global levels for 2010 with trends from 1990.Pediatr Res. 2013; 74: 50-72Crossref PubMed Scopus (356) Google Scholar, 8Kohli-Lynch M Russel NJ Seale AC et al.Neurodevelopmental impairment in children after group B streptococcal disease worldwide: systematic review and meta-analyses.Clin Infect Dis. 2017; 65: S190-S199Crossref PubMed Scopus (108) Google Scholar Together, these studies emphasise the substantial burden of severe neurodisability attributable to term neonatal morbidity. Intrapartum and newborn care must remain key priorities to ensure all children can survive and thrive. We declare no competing interests. Prevalence of cerebral palsy in Uganda: a population-based studyCerebral palsy prevalence was higher in rural Uganda than in high-income countries (HICs), where prevalence is about 1·8–2·3 cases per 1000 children. Children younger than 8 years were more likely to have severe cerebral palsy than older children. Fewer older children than younger children with cerebral palsy suggested a high mortality in severely affected children. The small number of preterm-born children probably resulted from low preterm survival. About five times more children with post-neonatal cerebral palsy in Uganda than in HICs suggested that cerebral malaria and seizures were prevalent risk factors in this population. Full-Text PDF Open Access