Social and financial barriers may contribute to a “hidden mortality” in Uganda for children with congenital anomalies

Commander, Sarah Jane; Ellis, Danielle; Williamson, Hannah; Oyania, Felix; Ruhigwa, Comfart; Situma, Martin; Fitzgerald, Tamara N.

doi:10.1016/j.surg.2020.09.018

Cited by 9 publications

(10 citation statements)

References 30 publications

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“…One paper [ 134 ] evaluated self-reported quality of life of children living with spina bifida compared to unaffected children. A few papers investigated the impact on the quality of life for mothers (e.g., [ 30 , 195 , 256 ]) and caregivers of affected children (e.g., [ 25 , 31 ]). One paper [ 214 ] derived disability weights, used to calculate Years Lived with Disability (YLD).…”

Section: Resultsmentioning

confidence: 99%

“…For the 11 papers identified for this category, the focus was upon cost of care in relation to financial challenges influencing access to care and poor outcomes (e.g., [ 25 , 31 , 89 , 236 ]), alongside social challenges [ 31 , 175 ]. Papers on cost of care primarily focused on CHD (45.5%) and OFC (27.3%), relating to the financial burden of individuals accessing care (and the impact on care seeking) rather than the cost to the health system.…”

Section: Resultsmentioning

confidence: 99%

“…Papers on stigma and related cultural issues (n = 12) were often based on surveys administered to care givers (mostly mothers) of children with CAs and addressed issues such as the influence of stigma on the quality of care (e.g., [ 30 , 34 ]), social/cultural barriers to uptake of preventive measures [ 31 ], prenatal screening [ 116 ] and treatment [ 31 ], genital reconstruction in disorders of sex development [ 120 , 128 ], plastic surgery [ 102 ], community rejection and abandonment by spouse [ 31 , 175 ], as well as health system responsiveness [ 175 ]. Some papers documented family and community beliefs about the causes of CA, e.g., witchcraft, contraception, and the guilt of the mother [ 31 , 116 , 241 ]. Beliefs and perceptions about the efficacy of medical care, preference for traditional medicine [ 241 ], and parental/community believes on whether treatment should be sought [ 31 , 241 ] were also included.…”

Section: Resultsmentioning

confidence: 99%

“…Some papers documented family and community beliefs about the causes of CA, e.g., witchcraft, contraception, and the guilt of the mother [ 31 , 116 , 241 ]. Beliefs and perceptions about the efficacy of medical care, preference for traditional medicine [ 241 ], and parental/community believes on whether treatment should be sought [ 31 , 241 ] were also included.…”

Section: Resultsmentioning

confidence: 99%

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The burden, prevention and care of infants and children with congenital anomalies in sub-Saharan Africa: A scoping review

Leke,

Malherbe,

Kalk

et al. 2023

PLOS Glob Public Health

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The aim of this scoping review was to determine the scope, objectives and methodology of contemporary published research on congenital anomalies (CAs) in sub-Saharan Africa (SSA), to inform activities of the newly established sub-Saharan African Congenital Anomaly Network (sSCAN). MEDLINE was searched for CA-related articles published between January 2016 and June 2021. Articles were classified into four main areas (public health burden, surveillance, prevention, care) and their objectives and methodologies summarized. Of the 532 articles identified, 255 were included. The articles originated from 22 of the 49 SSA countries, with four countries contributing 60% of the articles: Nigeria (22.0%), Ethiopia (14.1%), Uganda (11.7%) and South Africa (11.7%). Only 5.5% of studies involved multiple countries within the region. Most articles included CA as their primary focus (85%), investigated a single CA (88%), focused on CA burden (56.9%) and care (54.1%), with less coverage of surveillance (3.5%) and prevention (13.3%). The most common study designs were case studies/case series (26.6%), followed by cross-sectional surveys (17.6%), retrospective record reviews (17.3%), and cohort studies (17.2%). Studies were mainly derived from single hospitals (60.4%), with only 9% being population-based studies. Most data were obtained from retrospective review of clinical records (56.1%) or via caregiver interviews (34.9%). Few papers included stillbirths (7.5%), prenatally diagnosed CAs (3.5%) or terminations of pregnancy for CA (2.4%).This first-of-a-kind-scoping review on CA in SSA demonstrated an increasing level of awareness and recognition among researchers in SSA of the contribution of CAs to under-5 mortality and morbidity in the region. The review also highlighted the need to address diagnosis, prevention, surveillance and care to meet Sustainable Development Goals 3.2 and 3.8. The SSA sub-region faces unique challenges, including fragmentation of efforts that we hope to surmount through sSCAN via a multidisciplinary and multi-stakeholder approach.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

See 2 more Smart Citations

The burden, prevention and care of infants and children with congenital anomalies in sub-Saharan Africa: A scoping review

Leke,

Malherbe,

Kalk

et al. 2023

PLOS Glob Public Health

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“…Stomas further limit interpersonal relations of children with their friends, relatives and family members. Most of these children delay starting school, returning to school or completely drop out of school [19][28] [29] and their fathers leave families [30]. As a result, any change in our standard of care for HD patients that reduces the time with stomas, or the number of operations required, can have a signi cant impact on patient outcomes, as well as quality of life.…”

Section: Introductionmentioning

confidence: 99%

Pull-through Procedure for Hirschsprung’s Disease with Simultaneous Stoma Closure: Insights for Limited-Resource Settings from Mbarara, Uganda

Oyania

Kotagal

Wesonga

et al. 2021

Preprint

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Background: In many resource-limited settings, patients with Hirschsprung’s Disease (HD) undergo initial diverting colostomy, followed by pull-through, and lastly, colostomy closure. This approach allows for decompression of dilated and thickened bowel and improved patient nutritional status. However, this 3-stage approach prolongs treatment duration, with significant stoma morbidity, costs, and impact on quality of life (QOL).Aim: To determine whether pull-through for HD can safely be performed with simultaneous stoma closure, reducing treatment approach from three to two stages.Patients and Methods: Children with HD and diverting colostomy were prospectively followed as they underwent pull-through with simultaneous stoma closure. Their in-hospital course, and 3-month outpatient course, were assessed for postoperative complications. Patients with total colonic HD, redo pull-through, and residual dilated colon were excluded from the study. Results: Of the 20 children, seventeen were male (n = 17, 85%). All patients had rectosigmoid HD. The median weight, age at colostomy formation, and age at pull-through were 11.05 kg (interquartile range [IQR] 10-12.75), 0.9 years (IQR 0.25-2.8), and 2.08 years (IQR 1.28-2.75) respectively. Mean duration with colostomy before pull-through was 1.1 years (SD 1.51). Median hospital length of stay was 6 days (IQR 5-7). Early complications included anastomotic leak (n=1), perianal skin excoriation (n=2), surgical site skin infection (n=3_, and burst abdomen (n=1). Longer-term complications included stricture (n=1, 5%) and enterocolitis (n=2, 10%).Conclusion: In this small case series, we have demonstrated that pull-through with simultaneous stoma closure can be safely performed in resource-constrained settings. Further studies are needed to understand the QOL and economic impact of this change in management for HD patients.

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Hirschsprung disease: a cost analysis study of the direct, indirect costs and financial coping strategies for the surgical management in Western Uganda

Binde,

Oyania,

Ullrich

et al. 2024

Pediatr Surg Int

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Background: Surgical management of Hirschsprung disease (HD) in low-and middle-income countries is typically a staged procedure, necessitating multiple hospitalizations and clinic visits increasing family nancial burden. Currently there is limited information on the costs borne by caretakers of children with Hirschsprung disease seeking surgical intervention. This study seeks to measure the costs and economic burden of surgical treatment for Hirschsprung Disease in western Uganda.Methods: A cross-sectional study using cost analysis was conducted among caretakers of patients who completed surgical treatment of HD between January 2017 to December 2021 at two hospitals in western Uganda. The average direct and indirect costs incurred by caretakers presenting at a public and private hospital were computed.Results: A total of 69 patients (M: F = 7:1) were enrolled in the study. The median age at diagnosis was 60.5 (IQR 3 -151.25) days for children and 2-staged pull through procedure was the common surgery performed. The mean overall cost for treatment was US $960 (SD = $720), with the majority of costs coming from direct medical costs. Nearly half (48%) of participants resorted to distress nancing to nance their child's surgical care. The overwhelming majority of patients (n = 64, 93%) incurred catastrophic expenditure from the total costs of surgery for HD, and 97% of participants fell below the international poverty line at the time treatment was completed.Conclusion:Despite the availability of 'free care' from government hospital and non-pro t services, this study found that surgical management of Hirschsprung disease imposed substantial cost burden on families with Hirschsprung disease patients.

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Social and financial barriers may contribute to a “hidden mortality” in Uganda for children with congenital anomalies

Cited by 9 publications

References 30 publications

The burden, prevention and care of infants and children with congenital anomalies in sub-Saharan Africa: A scoping review

The burden, prevention and care of infants and children with congenital anomalies in sub-Saharan Africa: A scoping review

Pull-through Procedure for Hirschsprung’s Disease with Simultaneous Stoma Closure: Insights for Limited-Resource Settings from Mbarara, Uganda

Hirschsprung disease: a cost analysis study of the direct, indirect costs and financial coping strategies for the surgical management in Western Uganda

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