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Epidemic Spastic Paraparesis in Bandundu (Zaire)

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Epidemic Spastic Paraparesis in Bandundu (Zaire) J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.49.6.620 on 1 June 1986. Downloaded from Journal'of Neurology, Neurosurgery, and Psychiatry 1986;49:620-627 Epidemic spastic paraparesis in Bandundu (Zaire) H CARTON,* KAZADI KAYEMBE,t KABEYA,t ODIO, A BILLIAU§ K MAERTENSII From the Department ofNeurology, Catholic University ofLeuven, Belgium,* Centre Neuropsychopathologique University ofKinshasa, Zaire,t Hopital de Panzi, Bandundu, Zaire, Rega Institute, Catholic University of Leuven, Belgium,§ Department ofOphthalmology, University ofKinshasa, Zaire 11 SUMMARY Epidemiological findings of twenty sporadic cases of epidemic spastic paraparesis (buka-buka) in three areas of Bandundu (Zaire) are reported. These findings suggest the in- volvement of an infectious agent and do not support the hypothesis of a dietary cyanide intoxi- cation, which has been advanced to explain the outbreak of a very similar disease (Mantakassa) in Mozambique. Acute as well as chronic spastic paraparesis of un- covered essentially by a savanna highland of 900 m altitude, known origin is much more common in tropical coun- cut by forest galleries along the multiple rivers running in a tries than in temperate climates. Deficiencies or south-north direction. toxicities due to primitive diets as well as infectious Twenty patients residing in three localities more than agents have been implicated.' Sometimes these dis- 200 km apart, and belonging to three different tribes in eases occur in epidemics. The earliest documented Southern Bandundu were examined by two neurologists and one ophthalmologist. The histories were taken either in oneProtected by copyright. cases of epidemic spastic paraparesis in the Bandundu of the major languages of Zaire (Lingala) or in the local region of Zaire occurred in 1928 and were reported by languages: Kiyaka with the aid of a Muyaka physician, Trolli.2 Ever since there have been new outbreaks Tshitshokwe with the aid of the local schoolhead and Ki- affecting hundreds of cases in several areas of Band- suku with the aid of a local health worker. These patients undu, the latest ones in 1978 and 198 .3 Between epi- were not the victims ofa major outbreak but rather sporadic demics, there have been new sporadic cases every cases. In the village of Kidima, the local health worker indi- year. The disease is characterised by a sudden onset of cated all cases suspected of having or having presented with bilateral spastic paresis of the lower limbs, often asso- ciated with speech disorders and visual complaints. Recovery may be complete but some patients are left with a typical spastic gait, and more rarely with dys- arthria. The aetiology remains unknown. However, the clinical features and the disease course are very similar to those of an illness occurring in Mozambique, named Mantakassa.4 An outbreak in was to be the 1981 of more than 1,000 cases suspected http://jnnp.bmj.com/ consequence of chronic cyanide intoxication due to consumption of insufficiently detoxificated cassava.' No viruses were found. The epidemiological findings in Bandundu described here fail to support the intox- ication hypothesis but rather suggest participation of an infectious agent. Material and methods on September 25, 2021 by guest. Bandundu is one of the eight administrative regions of the republic of Zaire (fig 1). It is situated east of Kinshasa, be- tween 20 and 80 south and 160 and 200 east. The region is Address for reprint requests: H Carton, Dept of Neurology, Akade- misch Ziekenhuis Gasthuisberg, B 3000 Leuven, Belgium. Fig 1 Map ofthe Bandundu Region in Zaire. The Received 30 August 1985. neuroepidemiological study was conducted in Kidima, Accepted 28 September 1985 Kapemba and Kambangu and in Kimbongo (circles). 620 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.49.6.620 on 1 June 1986. Downloaded from Epidemic spastic paraparesis in Bandundu (Zaire) 621 acute spastic paraparesis. All these patients were screened by 900 m height. Drinking water is supplied by several wells the physician in charge of the primary care in that adminis- situated not more than 600 m outside the village. The trative zone (Dr Kabeya). Typical as well as doubtful cases women's work consists of carrying water, cultivating the were finally examined in the village by the neurologists and fields, preparing cassava (manioc) and cooking. The men's the ophthalmologist, and the diagnosis was either confirmed task is mainly hunting and burning wooded areas to prepare or discarded. The criteria for a case to be included were a fields for crop raising. Famine has not been present during typical history with sudden onset, complete or incomplete recent years. Food is abundantly available, mainly consis- recovery, andl the presence of pyramidal signs in the lower ting of cassava, corn, and vegetables. The villagers own limbs. In Kapemba, a small village of fifteen families, in numerous chickens and goats, which move freely from one addition to history taking and examination of the patients, area to another. Chicken meat is consumed even if obtained medical information was obtained and the ties of kinship from birds that died from fowl plague. All members of a established of every inhabitant of the village. The huts oc- family eat their food out of a common jar, but the husband cupied by the patients at the onset of the disease could also is first served and left-overs of the day before are eaten by be localised. Several patients coming from the surroundings the wife and children. Children always accompany their of Kapemba were examined at the infirmary of Kambangu. mother in her different occupations. If a man has only one Finally in the region of Kimbongo, where an important epi- wife, they dwell and sleep in the same hut. However, poly- demic took place in 1978, a few recent cases were examined gamy is still prevalent, each wife then occupying with her in the local infirmary. children her own hut. The Bayaka adhere to the matriarchal The neurological examination was as complete as possi- system. Epidemic spastic paraparesis has not been noticed in ble, but hearing was examined with a watch and vibration this village for at least 25 years until the arrival at the village sense was not tested. A general inspection for der- of patient MM in 1977. matological abnormalities and a brief physical examination I Patient MM is a female born in 1954 in Kidima. In 1972 including lung and heart auscultation and blood pressure she moved with her husband to the neighbouring village of examination were also performed. The ophthalmological ex- Kasongo Bolumbu, where she fell sick in May 1977. Sub- amination was performed as follows: Visual acuity: outside, sequently she was brought back to Kidima, to be taken care with a decimal Snellen chart at 5 m; Colour vision: outside, of by her parents, with whom she has been living ever since. with the Ishihara test, plates 18 to 24 for illiterates; Direct Her disease started with a coldish sensation in the lower legs, and indirect pupillary reflexes: in a dark room (inside a hut) followed by pain in the feet and knees which lasted one Protected by copyright. with a torch lamp; Eye position: with a torch lamp, by study- week. This episode was followed by shaking and acute paral- ing the corneal light reflex at primary position and after ysis of the lower limbs (clonus?). During the days or weeks occlusion; Eye movements: with the torch lamp, at six preceding the illness no malaise, fever, headache or diar- positions; Convergence: by approaching the torch lamp; rhoea had been noticed. The parents as well as the two chil- Anterior segment: with a portable Kowa slitlamp, dren of MM, one born before and one after her return to magnification 15 x, working from a car battery; Fun- Kidima, are all in good health. duscopy: with a Welch-Allyn direct ophthalmoscope before Clinical examination: Cranial nerves were normal. Gait was and after pupil dilatation with Mydriacyl (R); Special atten- spastic. Muscle strength was slightly diminished in the mm. tion to the colour of the optic disc, the visibility of the lam- triceps, extensor carpi and abductor digiti of both upper ina cribrosa, the margins ofthe optic disc, the striation of the limbs, with hyperreflexia and positive Hoffmann-Tromner retina near the optic disc. reflex. Examination of the lower limbs revealed moderate Blood was drawn from 18 patients and three controls, paresis of the flexors of the hip, the hamstrings and the dor- cerebrospinal fluid (CSF) was obtained from 10 and stool sal flexors of the foot. Patellar reflexes were excessively brisk, from two patients. In the CSF albumin and gammaglobulins with ankle clonus. The abdominal reflexes were present and were determined by nephelometry. Protein gel electro- the plantar reflexes equivocal. No atrophy or fasciculations phoresis and isoelectrofocusing of the CSF were performed were observed. Touch, pin-prick and position senses as well according to the method described by Delmotte.6 Blood and as coordination were unaffected. Eve examination: Vision: I http://jnnp.bmj.com/ stool samples are being stored for possible future analysis. O.U.; Ishihara: normal; Fundus: the upper, lower and nasal So far only tests for the possible involvement of the virus margins of the optic disc were blurred. associated with AIDS (HTLV-III/LAV) have been done. 2 Patient MA is a female born in 1954 in Kidima and the Serum samples were tested for possible-associated reverse third living wife of the father of patient MM. Her disease transcriptase as described by Billiau et al.7 The supernatant started one month after the arrival of MM who came to live fluids were tested for antibody by radio-immuno- on the same parcel (but not in the same house) as MA.
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