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Antibiotics May Eradicate Gastrointestinal Immunodeficiency.8 1122 Letters to the Editor J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.54.12.1122 on 1 December 1991. Downloaded from sitivity.? We present a case of OMM without pseudobulbar palsy. The association of vitro.8 Whipple's disease is associated with symptoms of Whipple's disease and with supranuclear gaze paresis with oculomas- immunodeficiency.8 Thus intestinal wall negative peroral jejunoileal biopsies, which ticatory myorhythmia, however, seems to be macrophages are ineffective in phagocytosing indicates the usefulness of laparotomy for pathognomonic of the effects of Whipple's intracellular gram positive bacilli, resulting in jejunoileal biopsies as an alternative to brain disease on the CNS.'2 This led us to perform inability to eliminate chronic infection.9 This biopsy to confirm Whipple's disease. surgical jejunal and mesenteric lymph node suggests that Whipple's disease may be con- A 47 year old woman was admitted in May biopsies rather than a brain biopsy despite sidered as a disease of macrophages.8 The 1988 in a depressed state. In June 1987 she negative endoscopic and numerous peroral periventricular and periaqueductal distribu- had noted progressive visual disturbance. distal jejunal and ileal biopsies. The nor- tion of the CNS involvement in Whipple's Rhythmic elevations of her right upper lip mality ofthe CT and MRI scans also suppor- disease consists of macrophagic infiltration appeared, and later, paroxysmal hypersomnia ted this decision. and subependymal nodules. Such a and considerable weight gain (10 kg). A nine The possibility of brain involvement with- "tumoural" involvement may explain why month course oftreatment for depression was out systemic manifestation in Whipple's dis- antibiotics with good BBB diffusion are not ineffective and her symptoms and signs ease should be kept in mind. This necessitates effective in this CNS disease. progressively deteriorated. Examination on a search for the disease by endoscopic and PIERRE AMARENCO ETIENNE ROULLET admission showed complete vertical and peroral jejunoileal biopsies in all patients with LAURENT HANNOUN horizontal ophthalmoplegia with sparing of unexplained supranuclear oculomotor palsy. RENE MARTEAU oculocephalic reflexes (which appeared spon- Despite two single reports of reversible CNS Service de Neurologie et Service de Chirurgie, the doll's eyes phenomenon the treatment of Hbpital Saint-Antoine taneously, giving involvement,4 5 however, 184, rue du Fg St-Antoine, with all movements of the head). Her eyes, CNS Whipple's disease has so far been dis- 75571 Paris Cedex 12, France. part of her limbs were mild in face, and proximal appointing.6 The improvement Correspondence to: Dr Amarenco, Service de affected by myorhythmia, which consisted of neurological states on pefloxacine was impor- Neurologie, Hopital Saint-Antoine. masticatory movements with synchronous tant in our patient but needs further confir- adduction of her eyes (convergence) and is a quinolone which mation. Pefloxacine 1 Schwartz MA, Selhorst JB, Ochs AL, et al. limbs at about 1 cps. Her soft palate was not readily diffuses across the blood-brain barrier Oculomasticatory myorhythmia: a unique involved. Movements also occurred during (BBB) and is effective against intracellular movement disorder occurring in Whipple's sleep. Gait disturbance and a slight facial micro-organisms such as those supposed to disease. Ann Neuroll986;20:677-83. Her 2 Hausser-Hauw C, Roullet E, Robert R, Marteau akinesia was noted but no neck rigidity. be the cause of Whipple's disease. Neverthe- R. Oculo-facio-skeletal myorhythmia as a mental state was normal. There was a less, taking into account the specificity of cerebral complication of systemic Whipple's pseudobulbar type dysarthria but no OMM in the diagnosis of Whipple's disease disease. Mov Disord 1988;3:179-84. difficulty in swallowing. No other systemic the of treatment6 were we 3 Adams M, Rhyner PA, Day J, DeArmond S, and poor efficacy Smuckler EA. Whipple's disease confined to sign or symptom was recorded; there was no justified in performing a laparotomy to con- the central nervous system. Ann Neurol diarrhoea. Her CSF was normal with no PAS firm the diagnosis? We think that this condi- 1987;21:104-8. positive cells present. EEG, CT, and Tl- tion needs to be formally recognised and 4 Ryser RJ, Locksley RM, Eng SC, Dobbins WO, on such a Schoenknecht FD, Rubin CE. Reversal of weighted (DTPA-Gadolinium enhanced) confirmed before embarking long dementia associated with Whipple's disease and T2-weighted MRI scans yielded normal and difficult treatment. by trimethoprim-sulfamethoxazole, drugs results, as did endoscopy and 30 peroral distal Riggs has suggested that incidental use of that penetrate the Blood-brain barrier. Gas- and proximal jejunal and ileal biopsies. may eradicate gastrointestinal troenterology 1984;86:745-52. antibiotics 5 Alder CH, Galetta SL. Oculo-facial-skeletal Erythrocyte sedimentation rate was 35 mm involvement in presymptomatic Whipple's myorhythmia in Whipple Disease: Treatment per hour. Schilling's test was 5% (normal disease but not the meningeal involvement with ceftriaxone. Ann Intern Med range 12-30%), suggesting distal ileal because of their poor BBB diffusion.' This 1990;1 12:467-9. isolated and late 6 Keinath RD, Merrell DE, Vlietstra R, Dobbins involvement. may explain the apparently WO. Antibiotic treatment and relapse in A laparotomy was performed and surgical CNS involvement. Such a hypothesis con- Whipple's disease. Gastroenterology 1985; biopsy specimens of the jejunum and mesen- siders only the infectious mechanism and 88:1867-73. nodes were taken. Pathological not the poor effect of some 7 Riggs JE. The evolving natural history of teric lymph does explain neurologic involvement in Whipple's disease: examination showed no jejunal abnormality antibiotics which are well able to cross the a hypothesis. Arch Neurol 1988;45:830. but PAS positive cells with positive Gram BBB. Unlike most infectious diseases, there 8 Dobbins WO. Whipple's disease. In: Berk JD stain in the mesenteric lymph nodes were are no established cases ofdirect transmission ed. Gastroenterology. Philadelphia: WB Saun- to ders Company, 1985:1803-13. found, confirming Whipple's disease. Treat- of Whipple's disease from one patient and gastroin- 9 Webster ADB. Immunodeficiency http://jnnp.bmj.com/ ment with rifampicin and tetracycline for two another, no reproduction of the disease in testinal disease. In: Triger DR ed. Clinical months stabilised the disease. Trimetho- laboratory animals, and no convincingly immunology of the liver and gastrointestinal prim-sulfamethoxazole and chloamphenicol specific organisms isolated by culture in tract. Bristol: Wright, 1986:127-49. were then given for a further two months. The results of Schilling's test returned to normal but signs and symptoms did not improve. Chloramphenicol had to be with- Binswanger's clinical and neuropatho- their suggestions on current English and drawn and was replaced by pefloxacine. logical criteria for "Binswanger's dis- French literature. It might be of interest to Two and a half years after the onset of ease" compare these modern criteria to Binswan- treatment there was mild improvement of ger's original description,2 which had only on September 28, 2021 by guest. Protected copyright. signs. Masticatory movements remained the Bennett et al' have made a timely and critical been available in English in a grossly trun- same, but the adduction of the eyes had attempt to standardise the diagnostic criteria cated form.3 disappeared; the eyes could be displaced to for "Binswanger's disease". They have based The table shows the clinical hallmarks the left and right to 50% and slightly down- Table Binswanger's clinical and neuropathological criteriafor the diagnosis of "Encephalitis ward but not upward. Unsteadiness of gait, subcorticalis chronica progressiva"2 probably due to the supranuclear oculomotor palsy, persisted. Her mental state was largely Clinical criteria: (verbatimfrom reference 2, page 1184) status was unaffected (minimental 30/30). the disease begins at the onset of senility (early in the fifties) or in advanced old age (early in the sixties); Her full scale intellectual quotient on the slow impairment of intellectual capabilities mainfesting primarily by the progressive impairment and WAIS fell slightly from 92 to 86 then in- ultimate loss of the association between cortical sensory and motor areas; creased to 96 in 1990. An uncontrolled new most frequently observed are aphasic disturbances (as in the present case), hemiamblyopia or hemianopia, hemiparesis with loss of the sense of pressure, position or touch; synchronous movement of flexion extension these circumscribed deficits are of a stable character during the fully developed disease and they are of the right thigh appeared when she relaxed combined with the slow and relentless deterioration of intellectual performance; in bed. Worsening of hypersomnia and ap- .... until) the patients resemble decerebrate laboratory animals. doses of pearance of cataplexy required high criteria: (from reference 2, page 1137) imipramine which gave partial relief. Neuropathological a we find a pronounced atrophy of the hemispheric white matter, either restricted to one or more gyri in Progressive supranuclear palsy is feature one brain area or of several hemispheric regions affected with variable severity; of Steele Richardson Olszewski disease, a these changes are most clearly found in the area of the occipital and
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