Epigraph Vol. 26 Issue 1, Winter 2024

Epilepsy research in low-resource areas: The complexities of data and consent

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By Nancy Volkers, ILAE communications officer

Cite this article: Volkers N. Epilepsy research in low-resource areas: The complexities of data and consent. Epigraph 2024; 26(1):47-51.

Although 80% of people with epilepsy live in low- and middle-income countries (LMICs), most epilepsy research originates in high-income regions. Initiating and perpetuating epilepsy research in LMICs requires addressing economic, ethical, human resource, and infrastructure challenges.

Generating representative data

Epidemiology studies are a first step toward establishing the burden of epilepsy in a given area: Without knowing how many people are affected by epilepsy, governments, academia, and other organizations cannot prioritize research and care.

In some countries, there are so few studies that existing data are often analyzed and applied in non-representative ways. Small or geographically focused epidemiology studies may be extrapolated to other populations within the same country — or the same continent.

A 2023 article on ethical and validity issues in low-income countries described the diversity of epilepsy-related factors in Africa. For example, infectious diseases are a significant contributor to epilepsy in rural Kenya and Tanzania, but neuroimaging studies have not shown the same associations in urban Kenya and South Africa. Metabolic causes of epilepsy are more prevalent in North Africa than sub-Saharan Africa.

Samson Awili Gwer
Samson Gwer

“Africa is a diverse continent; a study done in one part may have totally different data compared with a study done in another part,” said Samson Gwer, senior author of the study. “But because of the paucity of studies, the data that does exist is taken to be representative of the whole of Africa.” Gwer is a consultant pediatric neurologist and executive director of Afya Research Africa in Nairobi.

Gwer said that within Kenya, most health data is collected in the coastal region, which is home to a structured and funded research center. While this data may be extrapolated to the entire country, western Kenya is different from the coastal region in many ways.

“Just considering the role of malaria in epilepsy risk — malaria is almost non-existent in western Kenya,” he said. “So the etiological profile of epilepsy is not the same as on the coast. There also are big differences in access to care in urban versus rural areas, and those differences affect the risk of adverse experiences during pregnancy and delivery, which have an impact on epilepsy risk.”

Pauline Samia, chair of the department of pediatrics and child health at Aga Khan University, Nairobi, was first author of the 2023 study.

“If you say that research generated in one rural part of Kenya represents the whole of our country, then that’s not accurate,” she said. “And I believe the same can be said of various other countries in Africa, and parts of Asia. We need to generate a lot more research so the data are representative.”

Historical focus on communicable diseases

The historical focus on communicable diseases also has affected datasets in Africa and other lower-resource regions. “When you look at research in the 1980s, 1990s, and early 2000s, there was a huge focus on infection-related problems,” said Samia.

Gwer agreed. “The prioritization is toward diseases that are very visible and have the greatest burden in these settings, like malaria and HIV and tuberculosis,” he said.

Once global health initiatives began devoting resources to infectious diseases, the health system focused research there as well. This bias means that most digital health information systems also focus on infectious diseases.

“There is a bias against addressing and exploring solutions for other fairly common conditions, like epilepsy,” said Gwer. “These systems support a lopsided visibility of conditions, and that limits our capability to justify a research agenda.”

Adapted from Kwon et al, 2021 (Epilepsia)
Variable treatment gap estimates by study and country
Country # studies Treatment gap estimate range
China 6 58%-100%
USA 3 10%-48%
Kenya 2 61%-70%
India 4 15%-95%
Tanzania 2 70%-97%

Data infrastructure

Epilepsy researchers in high-income countries think nothing of using databases as research tools. In many areas of the world, however, there are few digital information systems, and there are no electronic medical records to populate them.

In Kenya, as in many lower-resource countries, people are their own personal medical databases, said Samia.

Pauline Samia
Pauline Samia

“The patient will go to the local dispensary or the health center, and they'll buy a [notebook], and they'll go to a visit and the clinical officer will write down information,” she said. “The lab will take a malaria test and bring back the result written in the same book. Then the gentleman will write the medication that is required and make out a prescription. The patient will take the book to the pharmacy, the pharmacy will dispense the medication and tick in the book that they have dispensed it.”

Even in large hospitals in some countries, said Samia, patients carry their own medical files. “How do we conduct research in these situations?”

Any databases that exist are largely populated with information from research consortia or specialized centers. “Those cover maybe one or two parts of the country,” said Samia. “And people may proactively create databases where they can look back and see retrospective findings from their patients. But apart from that, reporting how many epilepsy patients we have—it’s a challenge. And that’s not unique to Kenya.”

Epilepsy research efforts can make advocacy efforts more robust, she said.

“When we engage with government, governments sometimes want to know: How big is this problem? How much is it affecting our population? Who is actually affected? Having large databases that include diverse groups of patients would help move the epilepsy research agenda forward for everybody.”

Ethical principles, weak systems, vulnerable people

Protecting the privacy of people with epilepsy in low-resource areas, as well as ensuring they understand the implications of participating in research, can be fraught with difficulty. How can data privacy and protection policies cover medical records written in notebooks? Is it ethical to include people in epilepsy research who otherwise would not have access to treatment?

Headshot of Dr. Alessandra Nicoletti
Alessandra Nicoletti

“People with epilepsy may not always understand the implications of participating in research,” said Gwer. “They may not understand that it means they're giving out their personal information. Some patients may not be able to express themselves; they need a guardian who understands the adverse effects and how to opt out. They need another layer of protection.”

With limited access to health care services and even more limited access to specialized care, people with epilepsy in lower-resource regions may have only one opportunity to get the care they need: participate in research. “That introduces bias in patient selection, as well as the types of research conducted,” said Gwer.

In 1976, the US National Commission for the Protection of Human Subjects of Biomedical and Behavioral Research outlined ethical principles and provided guidelines that address ethical issues arising from the conduct of research with human subjects. Informally known as the Belmont Report, this publication is available in English and Spanish.

Compared with people with epilepsy in higher-resource countries, people in low- and middle-income countries may have less education and awareness about epilepsy. They may be affected by stigma, misinformation, and myths that make it less likely they will be open about their epilepsy or understand the importance of participating in research.

Informed consent and literacy

Alessandra Nicoletti has been researching epilepsy in remote areas of Bolivia for the past three decades. The research was carried out with the agreement of the Ministry of Social Welfare and Public Health and the support of the Asamblea del Pueblo Guaraní, the political party of the indigenous Guaraní people.

Gagandeep Singh
Gagandeep Singh

Nicoletti and colleagues completed extensive door-to-door surveys to estimate the incidence and prevalence of epilepsy in the Gran Chaco region of Bolivia and to shrink the diagnosis and treatment gap in the area. All study participants gave verbal consent to take part in the studies, which was witnessed and documented by study investigators as well as an anthropologist fluent in the native language. Due to illiteracy, researchers could not obtain written informed consent.

Low literacy levels also are faced in many areas of India, where Gagandeep Singh conducts research on epilepsy awareness, diagnosis, and treatment.

“We are very particular about consent in our research,” said Gagandeep. “However, many of the patients have low levels of literacy, which translates to poor treatment literacy—they have a poor understanding of treatment plans and an insufficient capacity to make independent decisions. For instance, many patients would not be able to read my prescriptions and are not interested in knowing more about the side effects of medications.”

The authors of the 2023 paper recommend that researchers deliberately consider individual protection, clear communication, direct benefits of research, and effective stakeholder participation, noting that establishing these practices in low- and middle-income countries can be more of a financial and time burden due to undeveloped or underdeveloped infrastructure.