Persistence of Tropical Ataxic Neuropathy in a Nigerian Community
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96 J Neurol Neurosurg Psychiatry 2000;69:96–101 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.69.1.96 on 1 July 2000. Downloaded from Persistence of tropical ataxic neuropathy in a Nigerian community O S A Oluwole, A O Onabolu, Hans Link, Hans Rosling Abstract as TAN diVer widely in clinical presentation, Objectives—The term tropical ataxic neu- natural history, and response to treatment. Two ropathy (TAN) is currently used to de- of the neurological syndromes grouped as scribe several neurological syndromes tropical ataxic neuropathy were described in attributed to toxiconutritional causes. Nigeria in the past 60 years. However, TAN was initially proposed to In 1930, a syndrome that aVected predomi- describe a specific neurological syndrome nantly adolescent school pupils in several seen predominantly among the Ijebu boarding schools was reported from some com- speaking Yorubas in south western Ni- munities in eastern and western parts of geria. In this study, the prevalence of TAN Nigeria.6–8 The main clinical features were sore was determined in Ososa, a semiurban tongue, angular stomatitis, and skin desquama- community in south western Nigeria de- tions followed by optic atrophy in a large scribed as endemic for TAN in 1969, and proportion of cases. Recovery of the lesions its neurological features were compared occurred during holidays, but many aVected with Strachan’s syndrome, prisoners of pupils had a recurrence on returning to school.9 war neuropathy, the epidemic neuropathy The school diet, dominated by cassava food in Cuba, and konzo. products, was implicated as causal and almost Methods—A census of Ososa was followed 100% of the patients responded to improvement by door to door screening of all subjects of diet and supplements with autoclaved dried aged 10 years and above with a newly yeast.9 The second syndrome, unlike the syn- designed screening instrument. Subjects drome of the 1930s, aVected middle aged and who screened positive had a neurological elderly people predominantly and its geographi- examination, and the diagnosis of TAN was made if any two or more of bilateral cal distribution was limited to some communi- ties in south western Nigeria.410It was variously optic atrophy, bilateral neurosensory 4 deafness, sensory gait ataxia, or distal named endemic neuropathy degenerative neuropathy,11 tropical nutrition neuropathy,12 symmetric sensory polyneuropathy were 13 present. and tropical ataxic neuropathy. DiVerent com- Neurology Unit, Results—A total of 4583 inhabitants were binations of sensory polyneuropathy, bilateral Department of registered in the census. Of these, 3428 optic atrophy, bilateral neurosensory deafness, Medicine, University 413 of Ibadan, Nigeria subjects aged 10 years and above were and sensory gait ataxia were found. Its causa- http://jnnp.bmj.com/ O S A Oluwole screened. The diagnosis of TAN was made tion was attributed to dietary cyanide from the 13–15 in 206 of 323 subjects who screened monotonous consumption of foods proc- International Institute positive for TAN. The prevalence of TAN essed from the starchy roots of cassava (Manihot for Tropical esculenta esculenta).16 In contradistinction to the Agriculture, Ibadan, was 6.0%, 3.9% in males and 7.7% in Nigeria females. The highest age specific preva- first syndrome, cases did not respond to O S A Oluwole lence was 24% in the 60–69 years age group improvement in diet or vitamin supplementa- A O Onabolu in women. tion.17 18 Conclusion—The occurrence of TAN in In the 1980s, the occurrence of TAN was on September 26, 2021 by guest. Protected copyright. Division of Ososa continues at a higher prevalence reported to have subsided because cases were International Health, IHCAR, Department than was reported 30 years ago. Its neuro- no longer registered in the teaching hospitals. It of Public Health logical features and natural history do not was speculated that improvement in diet, Sciences, Karolinska resemble those described for Strachan which followed the oil boom economic recov- Institute, SE-171 76 syndrome, epidemic neuropathy in Cuba, ery period of the 1970s in Nigeria, caused the Stockholm, Sweden or konzo. The increasing consumption of disappearance of the syndrome.19 However, a O S A Oluwole A O Onabolu cassava foods linked to its causation makes recent food survey in Ososa (fig 1), a H Rosling TAN of public health importance in Ni- semiurban community of Ijebu speaking Yoru- geria, the most populous African country. bas in south western Nigeria previously sur- Division of Neurology, (J Neurol Neurosurg Psychiatry 2000;69:96–101) veyed for TAN in 1969,11 showed that about Huddinge Hospital, 80% of the population consumed foods Karolinska Institute, Keywords: tropical myeloneuropathy; nutritional processed from the starchy roots of cassava Sweden polyneuropathy; ataxia 20 H Link thrice daily. The possibility that TAN may still be occurring in this community motivated Correspondence to: Several neurological syndromes described this study. Dr O S A Oluwole [email protected] from diVerent tropical regions in the past The aim of this study was to determine the century1–4 have been classified as tropical prevalence of TAN in Ososa, and to compare Received 15 July 1999 and in myeloneuropathies and further grouped into its neurological features with those described revised form 1 29 December 1999 tropical ataxic neuropathy (TAN) and tropical for Strachan’s syndrome, prisoner of war Accepted 9 February 2000 spastic paraparesis.5 The syndromes grouped neuropathy,2 epidemic neuropathy of Cuba,21 Persistence of tropical ataxic neuropathy in a Nigerian community 97 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.69.1.96 on 1 July 2000. Downloaded from bination of the questionnaire and the tandem walk test. Only subjects 10 years of age and above were screened as previous reports showed TAN to be rare below the age of 10 years.10 15 Subjects who screened positive from either the questionnaire or the tandem walk Abuja test were invited for neurological examination • at the local health centre in Ososa. • Ibadan HISTORY AND EXAMINATION Ososa • Ijebu-Ode In two of the seven quarters in Ososa, Lagos• Idomowo, and Odolakoye, all subjects who screened positive had a detailed history taken, and a clinical and neurological examination, and those diagnosed with TAN had their urine Figure 1 Map of Nigeria showing Ososa in relation to some major towns. tested for sugar and protein. These two quarters were purposely sampled to represent and konzo, which is also attributed to exposure the known socioeconomic variations among all to dietary cyanide.22 quarters. In depth interviews to determine the history of illness and exposure to industrial chemicals, drugs, and pesticides were carried Subjects and methods out. Detailed neurological examination with STUDY SITE standard methods assessed higher cerebral Ososa is a semiurban community in the Odog- functions, cranial nerves, motor system, coor- bolu local government area of Ogun State, dination, gait, and somatosensory modalities of Nigeria, where TAN was endemic in the 24 25 11 pain, touch, joint position, and vibration. In 1960s. This community, divided into seven the remaining five quarters subjects who administrative quarters from historical ante- screened positive had an abbreviated neuro- cedents, has an indigenous population of logical examination more focused on specific Ijebus, an ethnic group of the Yoruba tribe. All modalities required for the diagnosis of TAN. houses in the town are cement faced but vary widely in size and quality. Small scale cassava farming is the source of livelihood of most of DIAGNOSIS The diagnosis of TAN was made when any two the population. Social amenities such as or more of bilateral optical atrophy, bilateral electricity, water, good road networks, and neurosensory deafness, distal symmetric sen- motor transport are within access of all inhab- sory polyneuropathy, or sensory gait ataxia itants. were present as defined in previous studies.10 13 15 The diagnostic criteria were CENSUS defined as follows: optic atrophy required All houses in Ososa were numbered, and every distinct pallor of at least half of the disc; neuro- member of each household had their names, sensory deafness required impairment of http://jnnp.bmj.com/ ages, and sex recorded. Subjects without birth Rinne’s test; sensory polyneuropathy required records had their ages estimated to the nearest impairment of pin prick at a level above the 5 years through comparison with peers whose malleoli and wrist on both extensor and flexor ages were known and belong to the same “age surfaces; and gait ataxia required impairment group society” peculiar to the local culture. of tandem walk. SCREENING CONSENT A screening questionnaire with items to assess on September 26, 2021 by guest. Protected copyright. Consent for this study was obtained from the visual, auditory, motor, gait, and somatosen- local government health authority, the mon- sory symptoms consistent with the clinical arch (Oba) of Ososa, chiefs of the quarters, and neurology of TAN as previously described in heads of families. studies from Nigeria23 was developed. The questionnaire was prepared in the English lan- guage and translated to the local language, Results Yoruba, by two independent translators, who EPIDEMIOLOGY subsequently agreed on consensus translation. A total of 4583 inhabitants was registered in The consensus translation was translated back the census. Of these, 3428 subjects aged 10 into English to check for accuracy before final years and above were screened and 323 were translation to the Yoruba language. The positive for TAN. Sixty six patients screened questionnaire was tested and piloted on 10 positive on all the five items of the screening subjects and then revised. The tandem walk instrument, 94 on all items of the questionnaire examination was added as a clinical test to only, and 15 on tandem walk test only. The improve the sensitivity of the questionnaire. remaining 148 were positive on various combi- The tandem walk test was standardised by nations of the questionnaire items and the tan- making the subjects walk heel to toe in a dem walk test.