J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.18.2.103 on 1 May 1955. Downloaded from J. Neurol. Neurosurg. Psychiat., 1955, 18, 103. I 'I 95b ENCEPHALITIS IN LOA-LOA FILARIASIS BY L. VAN BOGAERT, A. DUBOIS, P. G. JANSSENS, J. RADERMECKER, G. TVERDY, and M. WANSON From the Medical Clinic and the Laboratory ofParasitology of the Institute of Tropical Medicine, and the Laboratories ofPathological Anatomy and Electroencephalography of the Institute Bunge, Antwerp "L'etude des filarioses offre encore maints aspects In 1919, Robles noted in acute onchocercosis at inconnus dont l'examen reserve a ceux qui s'y Guatemala the possibility of profound prostration, consacreront des observations captivantes." delirium, convulsions, trigeminal involvement, and J. RODHAIN (1953) intermittent sweating. He also described a case of chronic onchocercosis with epilepsy resulting from Filariasis is usually regarded as a benign disorder perforation of the skull by a tumour. and its neurological manifestations have rarely Anderson's (1924) report from British Guiana attracted attention. This may be due to the fre- was more complete. His case concerned a coloured quency with which the malady is superimposed upon girl aged 7 years affected by filariasis, who presented other diseases with meningo-vascular involvement, at necropsy a marked cerebral congestion with fatal to which the complications have been attributed. pericapillary leucocytic infiltration. He found Protected by copyright. Viewing the scene from a greater distance, we have numerous microfilariae (M. bancrofti) of normal felt that these complications might be related to appearance in the cerebral and cerebellar vessels. filariasis itself. The subject is of some importance This was the only case of the six he reported in in view of the increasing availability of highly potent which he was able to make a post-mortem examina- anti-filarial drugs. tion. He classes the frequency of microfilariae in the different organs in this order: kidneys; lungs, REVIEW OF THE LITERATURE spleen, brain, liver, cerebellum, and myocardium. The first Loa loa was extracted from the eye of a Tha Mya (1928) reported a case with hemiplegia negress in 1770 by Mongin at San Domingo. The and contracture of the arm following a coma lasting disease was described in West Africa by Guyot in four hours. There were unidentified microfilariae 1777 as quoted by Lorenz (1890) and by Manson in the cerebrospinal fluid, which also contained later (1904). blood. Microfilariae have also been recovered from In 1908, Broden and Rodhain reported that in the cerebrospinal fluid of an advanced case of exceptional cases they had found a filarial embryo trypanosomiasis (Chambon, 1933, M. perstans) in the the and cerebrospinal fluid, admitting possibility from the sheath of the optic nerve in a young http://jnnp.bmj.com/ that a blood vessel had been injured by the needle. Mexican Indian woman (Giaquinto Mira, 1934). The same year, Kulz (1908) reported that in the Rodhain (1937) recalled that in Hissette's case he Cameroons he had observed for four months a had observed M. perstans in the capillaries of the patient showing all the motor and psychiatric mani- choroid plexus. (Chalgren and Baker in 1946 refer festations of sleeping sickness, but in whom it had wrongly to this case as one of onchocercosis with not been possible to demonstrate the trypanosome meningeal involvement.) in the blood or cerebrospinal fluid, or by lymph Meanwhile Peruzzi (1928) had observed cerebral node puncture. The results of inoculation were filariasis in five monkeys suffering from trypano- on September 30, 2021 by guest. negative and treatment was without effect. The somiasis, and Hashimoto (1939) found a micro- blood of Kulz's patient (not the cerebrospinal fluid filaria in the vessels of the anterior lobe of the as quoted by Chalgren and Baker, 1946 and 1947) pituitary of a dog. was swarming with microfilariae. There exists, however, experimental work which Brunetiere (1913) recorded a case with paralysis is little known, that of Wail, Popow, and Prjadko of the tongue and hand followed, seven months (1926). It concerns guinea-pigs infected with later, by paralysis of the entire left side of the body. Dioptholriaena tricupsis Fedschenko 1874. This He envisaged the possibility of a terminal filarial parasite was found in the capillaries which were (mf. Loa) embolism of the sylvian artery. permeable though filled with the filariae. They 103 c J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.18.2.103 on 1 May 1955. Downloaded from 104 L. VAN BOGAERT described acute neuronal lesions, with neuron In his book, Napier (1946) mentioned "the aphasia, phantom cells, and proliferation of the possibility of psychoneurotic disorders". The pericapillary glia. The lesions were thought to be possibility of a cerebral invasion was also mentioned due to disturbance of the capillary circulation by Chalgren and Baker (1946). dependent in turn upon the mechanical and obstruc- Bertrand-Fontaine, Schneider, Wolfromm, and tive effects of the microfilariae and to the toxic Cagnard (1948) reported a case with double hemi- effect of the metabolic products of the parasites. plegia and a progressive course, reacting to treatment This work is the first in which neuropathological with " hetrazan ". The patient was a man aged 30 studies of the disease were carried out with modern years without previous illness, who developed a techniques. progressive hemiplegia, though in the course of its The possible appearance of neurological and development there were several remissions. It was psychiatric symptoms in microfilariasis is now well accompanied by periods of confusion and agitation recognized and several reports have appeared in with variable and transient ocular signs. There recent years. That of Bonnet (1943) concerned a was a partial regression of symptoms spontaneously man of 42 years whose illness had begun five days and then an improvement with treatment. The before coming under observation, with rigors, fever, patient had an old loasis with oedema, marked headache, and vomiting; this was accompanied, eosinophilia in the blood, subjective ocular symp- two days before admission, by torpor interrupted by toms, a positive intradermal reaction, and living periods of agitation. On admission, his respiration M. boa in the peripheral blood. was rapid and there was a marked meningeal syn- During the first few hours of treatment the drome without any apparent motor defect, but with symptoms increased, but after three courses of absent knee and ankle jerks and a right-sided Babin- 3799 R.P. there was a considerable improvement ski response. Lumbar puncture yielded a fluid with suggesting that the symptoms arose from the filari- Protected by copyright. 26 cells -and 40 mg. of protein. There were no asis. The cerebrospinal fluid was normal. As for trypanosomes but 5 to 6 M. loa per field. A thick the mechanism of the neurological signs, the authors drop of blood showed M. loa. The blood urea was were undecided as to whether there was an adult 170 mg. and the urine contained 0 25 g. of albumin filaria in the subarachnoid spaces at the base of the per 100 ml. He died three days later with hyper- brain (localization of choice) or whether there had pyrexia. He showed an aortitis probably due to been a migration of microfilariae in the vascular old syphilis, of which, however, there was no further bed, causing obstruction with an oedematous evidence. In this case the unilateral Babinski reaction. response probably indicated an accentuation of the In their work of 1950, Kenney and Hewitt give as lesions in the one hemisphere. psychoneurotic manifestations of loasis, insomnia, Bonnet related the meningeal syndrome in his headache, nervous depression, and abnormal patient to the filariasis but believed that the finding irritability. The report is based on four observa- of microfilariae in the cerebrospinal fluid was fairly tions. The first concerned a man aged 28 years common. It is admitted, he said, that they are too with a change in personality accompanied by torpor. bulky to pass the meningeal barrier and that it is These symptoms were sufficiently severe for him to the needle that introduces them into the sub- return to Europe. During this leave he again http://jnnp.bmj.com/ cutaneous tissues or into a small vessel. The suffered from headache with acute mental symptoms frequent finding of red blood cells in the cerebro- and an eosinophilia varying from 50 to 56%. The spinal fluid and the small number of microfilariae removal of a worm (M. boa) from the eye led to an that one finds there is in favour of this interpre- amelioration of all his symptoms and a fall in the tation. However, contamination by a needle is eosinophilia to 3%. In the second case the symp- improbable even in the case of M. loa and perstans toms were less marked and were also improved by which invade the circulation, since the blood con- extraction of the filaria. These two cases in on September 30, 2021 by guest. tains few of these parasites: three or four is the Europeans were caused by Loa loa. The third case maximum in a thick drop. In any case injury to a concerned an Indian suffering only from depression. vessel is rare during lumbar puncture and Bonnet The fourth was a negro in whom irritability and believes that the passage of the parasites is the abnormal aggression, accompanied by a right hemi- result of damage to the vessels by another disease. plegia, appeared suddenly. These two cases were He emphasizes only the meningeal syndrome in his caused by M. bancrofti. A state of apathy followed article; he leaves in the background the severe the initial symptoms. With " hetrazan " treatment involvement of the central nervous system revealed the headache increased, and there was mental by a symptomatology that we shall meet again later.