Impact of COVID-19 on the well-being of children with epilepsy including nodding syndrome in Uganda: A qualitative study.

OBJECTIVE: To investigate the impact of the COVID-19 pandemic and related restrictions on the access and use of health services by children with epilepsy including nodding syndrome in Uganda. METHODS: Four focus group discussions (FGD) with parents/caregivers of children with epilepsy and five in-depth interviews with key informants were conducted between April and May 2021 at Butabika National Mental Referral Hospital and Kitgum General Hospital. RESULTS: COVID-19-related restrictions, including the halting of non-essential services and activities, and suspension of public transport, created several challenges not only for children with epilepsy and their parents/caregivers but also for their healthcare providers. Study participants described extreme transport restrictions that reduced their access to healthcare care services, increased food insecurity and shortage or inability to afford essential medicines as consequences of COVID-19-related restrictions. However, parents/caregivers and healthcare workers adopted several coping strategies for these challenges. Parents/caregivers mentioned taking on casual work to earn an income to buy food, medicines, and other necessities. Healthcare workers intensified outreach services to affected communities. A positive impact of lockdown measures described by some FGD participants was that most family members stayed at home and were able to care for children with epilepsy in turn. CONCLUSIONS: Our study highlights the significant negative impact of the COVID-19 pandemic and related restrictions on access to health services and the general well-being of children with epilepsy. Decentralized epilepsy treatment services and nutritional support could reduce the suffering of children with epilepsy and their families during the ongoing COVID-19 pandemic and similar future emergencies.

The Pathogenesis of Nodding Syndrome.

Nodding syndrome is a rare, enigmatic form of pediatric epilepsy that has occurred in an epidemic fashion beginning in the early 2000s in geographically distinct regions of Africa. Despite extensive investigation, the etiology of nodding syndrome remains unclear, although much progress has been made in understanding the pathogenesis of the disease, as well as in treatment and prevention. Nodding syndrome is recognized as a defined disease entity, but it is likely one manifestation along a continuum of Onchocerca volvulus-associated neurological complications. This review examines the epidemiology of nodding syndrome and its association with environmental factors. It provides a critical analysis of the data that support or contradict the leading hypotheses of the etiologies underlying the pathogenesis of the syndrome. It also highlights the important progress made in treating and preventing this devastating neurological disease and prioritizes important areas for future research.

"Those who died are the ones that are cured". Walking the political tightrope of Nodding Syndrome in northern Uganda: Emerging challenges for research and policy.

BACKGROUND: Nodding Syndrome was first reported from Tanzania in the 1960s but appeared as an epidemic in Northern Uganda in the 1990s during the LRA civil war. It is characterized by repetitive head nodding, often followed by other types of seizures, developmental retardation and growth faltering with onset occurring in children aged 5-15 years. More than 50 years after the first reports, the aetiology remains unknown and there is still no cure. The recent hypothesis that Nodding Syndrome is caused by onchocerciasis also increases the relevance of onchocerciasis control. Northern Uganda, with its unique socio-political history, adds challenges to the prevention and treatment for Nodding Syndrome. This article aims to show how and why Nodding Syndrome has been politicised in Uganda; how this politicisation has affected health interventions including research and dissemination; and, the possible implications this can have for disease prevention and treatment. METHODOLOGY: Ethnographic research methods were used triangulating in-depth interviews, focus group discussions, informal conversations and participant observation, for an understanding of the various stakeholders' perceptions of Nodding Syndrome and how these perceptions impact future interventions for prevention, treatment and disease control. PRINCIPAL FINDINGS: Distrust towards the government was a sentiment that had developed in Northern Uganda over several decades of war and was particularly linked to the political control and ethnic divisions between the north and south. This coincided with the sudden appearance of Nodding Syndrome, an unknown epidemic disease of which the cause could not be clearly identified and optimal treatment had not clearly been established. Additionally, the dissemination of the inconclusive results of research conducted in the area lacked sufficient community involvement which further fueled this political distrust. Disease perceptions revolved around rumours that the entire Acholi ethnic group of the north would be annihilated, or that international researchers were making money by stealing study samples. This discouraged some community members from participating in research or from accepting the mass drug administration of ivermectin for prevention and treatment of onchocerciasis. Such rumour and distrust led to suspicions concerning the integrity of the disseminated results, which may negatively impact future disease management and control interventions. CONCLUSIONS AND RECOMMENDATIONS: Trust must be built up gradually through transparency and by de-politicising interventions. This can be done by engaging the community at regular intervals during research and data collection and the dissemination of results in addition to involvement during service delivery for prevention and treatment. Maintaining a regular feedback loop with the community will help control rumours, build trust, and improve the preparations for adequate dissemination.

Qualitative evaluation of the outcomes of care and treatment for children and adolescents with nodding syndrome and other epilepsies in Uganda.

BACKGROUND: In 2012, the Ugandan Government declared an epidemic of Nodding Syndrome (NS) in the Northern districts of Gulu, Kitgum, Lamwo and Pader. Treatment guidelines were developed and NS treatment centres were established to provide symptomatic control and rehabilitation. However, a wide gap remained between the pre-defined care standards and the quality of routine care provided to those affected. This study is to qualitatively assess adherence to accepted clinical care standards for NS; identify gaps in the care of affected children and offer Clinical Support Supervision (CSS) to Primary Health Care (PHC) staff at the treatment centres; and identify psychosocial challenges faced by affected children and their caregivers. METHODS: This case study was carried out in the districts of Gulu, Kitgum, Lamwo and Pader in Uganda from September to December in 2015. Employing the 5-stage approach of Clinical Audit, data were collected through direct observations and interviews with PHC providers working in public and private-not-for-profit health facilities, as well as with caregivers and political leaders. The qualitative data was analysed using Seidel model of data processing. RESULTS: Clinical Audit and CSS revealed poor adherence to treatment guidelines. Many affected children had sub-optimal NS management resulting in poor seizure control and complications including severe burns. Root causes of these outcomes were frequent antiepileptic drugs stock outs, migration of health workers from their work stations and psychosocial issues. There was hardly any specialized multidisciplinary team (MDT) to provide for the complex rehabilitation needs of the patients and a task shifting model with inadequate support supervision was employed, leading to loss of skills learnt. Reported psychosocial and psychosexual issues associated with NS included early pregnancies, public display of sexual behaviours and child abuse. CONCLUSIONS: Despite involvement of relevant MDT members in the development of multidisciplinary NS guidelines, multidisciplinary care was not implemented in practice. There is urgent need to review the NS clinical guidelines. Quarterly CSS and consistent anticonvulsant medication are needed at health facilities in affected communities. Implementation of the existing policies and programs to deal with the psychosocial and psychosexual issues that affect children with NS and other chronic conditions is needed.

Treatment and rehabilitation outcomes of children affected with nodding syndrome in Northern Uganda: a descriptive case series.

INTRODUCTION: Nodding Syndrome (NS) is a neurological disorder affecting children 5-15 years at onset in East Africa. A major criterion for diagnosis is atonic seizure with dorso-ventral "nodding" of the head. Comorbidities include psychological and behavioral abnormalities, malnutrition, cognitive decline, school dropout and other seizure types. We aimed to describe the presentations and rehabilitation outcomes of NS children at Hope for HumaNs (HfH) centre in Gulu from September 2012 to October 2013. METHODS: Data was obtained from a retrospective review of 32 NS children's medical records at HfH center. Ethical approval was obtained from Gulu University IRB. Data analysis was conducted using WHO AnthroPlus, SPSS and Excel software. RESULTS: Growth statistics showed steady improvement over time using local nutrition and multivitamin supplementation. Severe and moderate stunting was reduced from a combined total of 54.8% to 7.7% and 12.8% respectively. Severe and moderate wasting was reduced from 29.1% to 2.6% and 5.1% respectively. Three groups of NS children were identified and compared in the review; Low seizure occurrence averaging <2 seizures/month (28.1%); Moderate averaging 2-4 seizures/month (34.4%) and High averaging >4 seizures/month (37.5%). CONCLUSION: NS is a neurological disorder of unknown etiology. Treatment with regular high quality local nutrition, multivitamin supplementation, anti-seizures, regular follow up and illness prevention; children's seizures can be reduced or stopped completely. The debilitating malnutrition and stunting of NS children in Uganda could be partially independent of the syndrome but attributable to poor nutrition. NS as observed is not "invariably fatal" but rather a treatable neurological disorder.

Contextualization of psychological treatments for government health systems in low-resource settings: group interpersonal psychotherapy for caregivers of children with nodding syndrome in Uganda.

BACKGROUND: Evidence for the effectiveness of psychological treatments in low- and middle-income countries is increasing. However, there is a lack of systematic approaches to guide implementation in government health systems. The objective of this study was to address this gap by employing the Replicating Effective Programs (REP) framework to guide contextualization of a psychological treatment in the Uganda public health system for caregivers of children affected by nodding syndrome, a neuropsychiatric disorder endemic to Sub-Saharan Africa associated with high morbidity and disability. METHODS: To contextualize a psychological treatment, we followed the four components of the REP framework: pre-conditions, pre-implementation, implementation, and maintenance and evolution. A three-step process involved reviewing health services available for nodding syndrome-affected families and current evidence for psychological treatments, qualitative formative research, and analysis and documentation of implementation activities. Stakeholders included members of affected communities, health care workers, therapists, local government leaders, and Ministry of Health officials. Detailed written, audio, and video documentation of the implementation activities was used for content analysis. RESULTS: During the pre-condition component of REP, we selected group interpersonal therapy (IPT-G) because of its feasibility, acceptability, effectiveness in the local setting, and availability of locally developed training materials. During the pre-implementation component, we adapted the training, logistics, and technical assistance strategies in conjunction with government and stakeholder working groups. Adaptations included content modification based on qualitative research with caregivers of children with nodding syndrome. During the implementation component, training was shortened for feasibility with government health workers. Peer-to-peer supervision was selected as a sustainable quality assurance method. IPT-G delivered by community health workers was evaluated for fidelity, patient outcomes, and other process-level variables. More than 90% of beneficiaries completed the treatment program, which was effective in reducing caregiver and child mental health problems. With the Ministry of Health, we conducted preparatory activities for the maintenance and evolution component for scale-up throughout the country. CONCLUSIONS: The REP framework provides a systematic approach to guide contextualization of psychological treatments for delivery in low-resource public health systems. Specific recommendations are provided for REP's application in global mental health. TRIAL REGISTRATION: ISRCTN11382067 ; 08/06/2016; retrospectively registered.

Modelling optimal allocation of resources in the context of an incurable disease.

Nodding syndrome has affected and led to the deaths of children between the ages of 5 and 15 in Northern Uganda since 2009. There is no reliable explanation of the disease, and currently the only treatment is through a nutritional programme of vitamins, combined with medication to prevent symptoms. In the absence of a proper medical treatment, we develop a dynamic compartmental model to plan the management of the syndrome and to curb its effects. We use incidence data from 2012 and 2013 from Pader, Lamwo and Kitgum regions of Uganda to parameterize the model. The model is then used to look at how to best plan the nutritional programme in terms of first getting children on to the programme through outreach, and then making sure they remain on the programme, through follow-up. For the current outbreak of nodding disease, we estimate that about half of available resources should be put into outreach. We show how to optimize the balance between outreach and follow-up in this particular example, and provide a general methodology for allocating resources in similar situations. Given the uncertainty of parameter estimates in such situations, we perform a robustness analysis to identify the best investment strategy. Our analysis offers a way of using available data to determine the best investment strategy of controlling nodding syndrome.

Health Seeking Behaviours among Caretakers of Children with Nodding Syndrome in Pader District - Northern Uganda: A Mixed Methods Study.

BACKGROUND: Nodding syndrome is a neurological disorder which had affected about 3000 children with over 170 deaths in northern Uganda by 2012. With limited data on health seeking, the study aimed to assess the health seeking behavior and associated factors among caretakers of children with nodding syndrome in Pader district. METHODS: A mixed methods cross sectional study was conducted in July 2013 among 249 caretakers of children with nodding syndrome in three sub-counties of Pader. Respondents were consecutively interviewed using semi-structured questionnaires. Eleven key informants were additionally interviewed. We determined the associations of various factors with health care seeking and obtained adjusted odds ratios and 95% confidence intervals using logistic regression model. Quantitative data was analysed using Stata version 12 while qualitative data was analysed manually and quotes reported. RESULTS: Most caretakers, 78.3% (195/249) sought care first from a health facility, 12.9% (32/249) visited traditional healers and 8.8% (22/249) self-medicated. Of those who sought care from a health facility, 50% sought care after a month. Factors associated with improved care seeking included: Time taken to reach care 1-3 hours; adjusted odds ratio = 6.4 (95% CI = 2.96-14.03), time spent in care above five years; adjusted odds ratio = 12.0 (95% CI: 1.24-117.73) and changed care seeking place; adjusted odds ratio = 17.2 (95% CI: 3.64-81.67). CONCLUSION/ RECOMMENDATION: Caretakers sought care from multiple places. One in five caretakers still sought care outside a formal health facility. Many respondents who sought care first from health facilities went late, at least one month after symptoms onset. Factors associated with health seeking included distance, duration in treatment and not having changing care provider. There is need for massive sensitization of community to enhance prompt care seeking. More research is needed to elucidate the cause, thus finding the treatment for nodding syndrome, to prevent "wandering in hope".

Is nodding syndrome an Onchocerca volvulus-induced neuroinflammatory disorder? Uganda's story of research in understanding the disease.

Nodding syndrome is a devastating neurological disorder, mostly affecting children in eastern Africa. An estimated 10000 children are affected. Uganda, one of the most affected countries, set out to systematically investigate the disease and develop interventions for it. On December 21, 2015, the Ministry of Health held a meeting with community leaders from the affected areas to disseminate the results of the investigations made to date. This article summarizes the presentation and shares the story of studies into this peculiar disease. It also shares the results of preliminary studies on its pathogenesis and puts into perspective an upcoming treatment intervention. Clinical and electrophysiological studies have demonstrated nodding syndrome to be a complex epilepsy disorder. A definitive aetiological agent has not been established, but in agreement with other affected countries, a consistent epidemiological association has been demonstrated with infection by Onchocerca volvulus. Preliminary studies of its pathogenesis suggest that nodding syndrome may be a neuroinflammatory disorder, possibly induced by antibodies to O. volvulus cross-reacting with neuron proteins. Histological examination of post-mortem brains has shown some yet to be characterized polarizable material in the majority of specimens. Studies to confirm these observations and a clinical trial are planned for 2016.

Is the glass half full or half empty? A qualitative exploration on treatment practices and perceived barriers to biomedical care for patients with nodding syndrome in post-conflict northern Uganda.

BACKGROUND: Nodding syndrome has increasingly become an issue of public health concern internationally. The etiology of the disorder is still unknown and there are yet no curative treatments. We explored perceptions about treatment practices and barriers to health seeking for nodding syndrome in Pader and Kitgum districts in northern Uganda in order to provide data necessary for informing policy on treatment adherence and rehabilitations. METHODS: We used focus group discussions and individual interviews to gain deep insights into help-seeking and treatment practices for nodding syndrome. Purposive sampling was used to identify information-rich participants that included village health teams, community members not directly affected with nodding syndrome, district leaders, healthcare professionals, and caregivers of children affected with nodding syndrome. We used qualitative content analysis to analyze data and presented findings under distinct categories and themes. RESULTS: Caregivers and communities sought care from multiple sources including biomedical facilities, traditional healers, traditional rituals from shrines, and spiritual healing. Nodding syndrome affected children reportedly have showed no enduring improvement with traditional medicines, traditional rituals, and prayers. A substantial minority of participants reported minimal improvements in symptoms of convulsions with use of western medicines. Challenges involved in health seeking included; (1) health system factors e.g. long distances to facilities, frequent unavailability of medicines, few healthcare providers, and long waiting times; (2) contextual and societal challenges e.g. lack of money for transport and medical bills, overburdening nature of the illness that does not allow time for other activities, and practical difficulties involved in transporting the physically deformed and mentally retarded children to the health facilities. CONCLUSIONS: Help-seeking for nodding syndrome is pluralistic and include use of traditional and biomedical practices. Western medicines admittedly showed at least short term control on nodding syndrome symptoms, especially convulsions and led in a few cases to regain of functional abilities. However, multiple barriers hinder health seeking and interfere with adherence to biomedical treatments. Regarding cure, there are hitherto no treatments participants perceive cure nodding syndrome.

Patients with nodding syndrome in Uganda improve with symptomatic treatment: a cross-sectional study.

OBJECTIVES: Nodding syndrome (NS) is a poorly understood neurological disorder affecting thousands of children in Africa. In March 2012, we introduced a treatment intervention that aimed to provide symptomatic relief. This intervention included sodium valproate for seizures, management of behaviour and emotional difficulties, nutritional therapy and physical rehabilitation. We assessed the clinical and functional outcomes of this intervention after 12 months of implementation. DESIGN: This was a cross-sectional study of a cohort of patients with NS receiving the specified intervention. We abstracted preintervention features from records and compared these with the current clinical status. We performed similar assessments on a cohort of patients with other convulsive epilepsies (OCE) and compared the outcomes of the two groups. PARTICIPANTS: Participants were patients with WHO-defined NS and patients with OCE attending the same centres. OUTCOME MEASURES: The primary outcome was the proportion of patients with seizure freedom (≥1 month without seizures). Secondary outcome measures included a reduction in seizure frequency, resolution of behaviour and emotional difficulties, and independence in basic self-care. RESULTS: Patients with NS had had a longer duration of symptoms (median 5 (IQR 3, 6) years) compared with those with OCE (4 (IQR 2, 6) years), p<0.001. The intervention resulted in marked improvements in both groups; compared to the preintervention state, 121/484 (25%) patients with NS achieved seizure freedom and there was a >70% reduction in seizure frequency; behaviour and emotional difficulties resolved in 194/327 (59%) patients; 193/484 (40%) patients had enrolled in school including 17.7% who had earlier withdrawn due to severe seizures, and over 80% had achieved independence in basic self-care. These improvements were, however, less than that in patients with OCE of whom 243/476 (51.1%) patients were seizure free and in whom the seizure frequency had reduced by 86%. CONCLUSIONS: Ugandan children with NS show substantial clinical and functional improvements with symptomatic treatments suggesting that NS is probably a reversible encephalopathy.