J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.52.5.681 on 1 May 1989. Downloaded from

Letters 681 to the sink. She consumed 1-2 pitchers of did not include CT and MR studies or 2 Fitzsimmons JT. The hypothalamus and drink- water. Interictal EEG studies revealed inter- describe the pathological anatomy of this ing. Br Med Bull 1966;22:231-7. mittent epileptiform activity in the right ictal syndrome.' The neuroimaging studies 3 Martin JB, Reichlin S. The Neurohypophysis: Physiology and Disorders of Secretion. In: anterior region. Ictal EEG studies confirmed may have demonstrated an extratemporal Martin JB, Reichlin S: Clinical Neuro- the right anterior temporal localisation. Pos- structural abnormality associated with an endocrinology. Philadelphia, FA Davis tictally she appeared confused and did not aberration of water intake, such as a hypo- Company, 1987:79-81. recall test items presented to her during the thalamic lesion.' 4 Andersson B. Polydipsia, antidiuresis and milk ictus. She did, however, admit to her thirst The anterior and ventromedial hypo- ejection caused by hypothalamic stimula- and consumption of water. thalamus has an important role in thirst tion. In: Heller H, ed: Neurohypophysis. Electrolytes, serum and urine osmolality and water regulation.26 In addition, efferent London, Butterworth, 1957:131-140. and fasting blood glucose were normal. A from the amyg- 5 Stevenson JAF. Neural control of food and pathways hippocampus and water intake. In: Haymaker W et al(editors): computed tomography (CT) scan was nor- dala that project to the hypothalamus have The Hypothalamus. Springfield, Ill., CC mal. A magnetic resonance (MR) scan also been implicated in water balance and Thomas, 1969:524-621. showed an enlargement ofthe right temporal drinking behaviour.3" Stimulation and lesion 6 Andersson B, McCann SM. Drinking, horn of the lateral ventricle and suggested studies in the dog have implicated a role for antidiuresis and milk ejection from electrical atrophy of the right parahippocampal gyrus the anteroventral amygdala in thirst and stimulation within the hypothalamus of the (fig). The patient underwent an en bloc right water drinking behaviour.3 Amygdala goat. Acta Physiol Scand 1955;35:191-7. anterior temporal lobectomy. Hippocampal efferent fibres in the stria terminalis and 7 Brodal A. The Olfactory Pathways. In: Brodal sclerosis The neuronal converge A (editor): Neurological Anatomy, New was demonstrated. hippocampal efferents in the fornix York, Oxford University Press, 1981:658 loss and gliosis were widespread and prom- on the hypothalamus.' and 679. inent in the hippocampus, subiculum, and Thus, the hypothalamus is linked with dentate gyrus. The amygdala and the lateral limbic structures by a variety of multisynap- temporal neocortex were unremarkable. She tic pathways.' Epileptiform activity gen- has been asymptomatic and seizure free since erated in the temporal lobe may propogate surgery. into the hypothalamus and other structures The urge to pour and drink water has been implicated in thirst and water regulation, Single fibre and quantitative EMG study in documented rarely to occur as an ictal producing ictal manifestations of abnormal acute stage of human trichinosis Protected by copyright. behaviour in epilepsy. Remillard et al des- water seeking behaviour. cribed 20 patients with complex partial This study demonstrates that complex Sir: Human trichinosis is a parasitic infection epilepsy and ictal water drinking.' In these partial seizures, accompanied by mesiobasal now rarely reported, although the disease patients scalp recorded EEG demonstrated temporal lobe pathology, may indeed be has not been eradicated. Electro- electrographic seizure activity in the tem- associated with an ictal disturbance of water physiological information is scanty and poral lobe, and depth electrode studies drinking. The clinical and neuropathological usually only reports a myopathic pattern.'13 revealed the onset of seizure activity in the observations suggest that epileptiform We report the clinical findings, laboratory amygdala, hippocampus and parahippo- activity may propogate into regions that are manifestations and, particularly, the results campal gyrus in two patients.' This report synaptically remote from hippocampal path- ofa careful electrophysiological examination ology and produce ictal manifestations of in 10 patients with only mild neuromuscular abnormal water-seeking behaviour. This involvement by trichinosis. Correlation bet- case confirms the previous report regarding ween electrophysiological and laboratory the temporal lobe localisation of ictal thirst, findings was made. and in addition identified a pathological The symptoms began 4 to 16 days after lesion associated with this unusual ictal consuming home prepared wild boar behaviour.' sausages (table). A large number ofmembers of several families in the same small town GREGORY D CASCINO*t were affected, although most cases were http://jnnp.bmj.com/ THOMAS P SUTULAt almost asymptomatic. Ten patients (table) Francis M. Forster Epilepsy Center,* had diffuse myalgias of variable duration, William S. Middleton Memorial Veterans' and some of them had subjective weakness. Hospital, These 10 cases were electrophysiologically 2500 Overlook Terrace, evaluated early, in the second or third week Madison, Wisconsin 53705 after onset of the symptoms (mean = 17, SD University of Wisconsin Comprehensive 3 days). A second electrophysiological Epilepsy Program,t examination was performed in two patients, 600 Highland Avenue, 2 months (case 5) and 6 weeks (case 8) after on October 2, 2021 by guest. Madison, Wisconsin 53792, USA the first exploration, when the patients were Fig A coronal Ti weighted image magnetic already asymptomatic. resonance scan shows atrophy ofthe right The first manifestation of the disease was parahippocampalgyrus (arrow) and Referenmes often characterised by nausea, vomiting, enlargement ofthe right temporal horn ofthe abdominal cramp and diarrhoea, always I Remillard GM, Andermann F, Gloor P, Olivier had a vari- lateral ventricle (arrow). (Note the right A, Martin JB. Water-drinking as ictal followed by myalgias. Myalgias temporal lobe is on the left side ofthe behaviour in complex partial seizures. able duration, from 7 to 48 days. Subjective photograph.) Neurology 1981;31:1 17-24. weakness was a common symptom (seven J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.52.5.681 on 1 May 1989. Downloaded from

682 Letters Table Findings in 10 patients with mild trichinosis

Mean % end- Mean Incuba- Mean Percentage amplitude Ratio plates jitter tion duration polyphasic T (m V) TIA (p V) nFD increased (ps) CK level Number Age period Fibril- MUP (% potentials (N= 215- (N=0 24- (N= 0 71- (N= jitter (N< (N<260 eosinophils No Sex (yr) (days) lation decreased) (N< 15%) 457) 044) 123) <2) (N< 10%) 395) IU) (N<500) I M 47 6 + 10 33 391 028 1 39 NP NP NP 1033 1008 2 M 22 8 + 20 25 394 0-27 145 NP NP NP 1351 1808 3 M 30 4 + 24 25 400 0-27 1 48 5 0 30 58 5 167 3200 4 M 27 10 + 15 20 517 0-27 1 91 NP NP NP 446 1230 5 F 61 9 + 45 45 NP NP NP 5 3 20 52 0 121 1410 6 M 31 7 + NP NP 479 0-29 1-65 4-2 15 48-3 173 2880 7 F 28 10 + 28 20 NP NP NP 3-8 20 39 9 483 1370 8 M 21 7 + 10 15 NP NP NP 2-6 10 43-8 1420 1130 9 F 48 14 + 35 30 536 034 1-57 3-1 10 37-8 1176 660 10 F 37 16 - NP NP 630 0-32 1-96 3-8 0 33-0 121 700 M =male; F=female. Fibrillation potentials (+ )= present; (- )=absent. Mean duration of the MUP (% decrease from normal mean) and percentage of polyphasic potentials in biceps brachii muscle. Automatic analysis of the EMG in biceps brachii muscle (T =number of turns; mean amplitude in mV. and T/A (yV) ratio). Single fibre EMG study in extensor digitorum communis muscle with data of nFD, percentage of end-plates with increased jitter and mean jitter values in ps. N = normal values; NP = not performed. cases), although examination only revealed it decreased from 52-0 to 38-4 ps and from 43-8 transmission of the reinnervating complex in one case and in another was doubtful. to 31-7 ps respectively. The remaining was still uncertain. The follow-up in two Clinical examination suggested that weak- patient refused a new EMG study. cases is consistent with this hypothesis, ness was mainly secondary to muscle pain. The motor and sensory conduction because it indicated a large rearrangement of Sensation and tendon reflexes were normal. velocities performed in upper and lower the motor unit (progressive increment in Other signs and symptoms were: periorbital limbs were within the normal range for age. nFD) and more stable transmission of Protected by copyright. the and facial oedema in all patients, fever in The laboratory and electrophysiological impulse, perhaps owing to the maturation of nine subjects, conjunctivitis in four cases, data were correlated (Spearman's correla- the sproutings. headache in three and paraesthesiae in only tion coefficient). Decrement of the MUPs The significant correlation between one patient. The number of eosinophils was duration was significantly (p < 0-01) decreased mean duration and raised CK raised in all cases, and CK values were correlated with the raised CK values. Sig- could be due to drop-out of muscle fibres in elevated in six (table) at the time of the first nificant correlation (p < 0-05) was also the acute phase ofthe disease. Early examin- electrophysiological examination. All cases found between nFD and mean jitter values. ation might explain the poor correlation were clinically asymptomatic 6-8 weeks after The main electrophysiological findings between enzymatic abnormalities, electro- the onset of the illness. The patients were reported here are consistent with myopathy physiological parameters and eosinophils treated with thiabendazole (25-50 mg/kg/ in all patients. EMG changes were mild to count. A larger follow-up study could day, for 5 days) and prednisone (0-3-0 5 mg/ moderate in eight cases, and moderate to provide more information ofthe evolution of kg/day, for I week). severe in two. Peripheral nerve abnormalities the electrophysiological parameters and Muscle biopsy was performed in case I were not found. Our findings in the conven- long-time effects of the trichinosis on the (biceps brachii), and showed Trichinella tional EMG were those typically reported in motor unit architecture. spiralis without encysted larvae within the inflammatory myopathies. Quantitative Our study proves that even in the acute muscle fibres. The diagnosis in the other EMG4 also showed myopathic features. stage of mild human trichinosis electro- patients was confirmed by positive trichinella The nFD and the jitter were abnormal in physiological study shows early changes, and serology. the patients with trichinosis reported here. A has a diagnostic yield for muscle involvementhttp://jnnp.bmj.com/ Conventional EMG was performed in slight to moderate increase in FD in all tested higher than raised CK values. distal and proximal muscles ofthe upper and cases and abnormal jitter in some of the lower limbs. Full interference pattern was patients have been previously reported in This study has been supported in part with a found in all tested muscles. Mean duration of polymyositis,' where abnormalities in grant from "Caja de Ahorros y Monte de the motor unit potentials (MUPs) and num- SFEMG were not correlated with clinical Piedad de Madrid". ber ofpolyphasic potentials are shown in the involvement and CK activity.7 The results of A CRUZ MARTiNEZ, table. Automatic analysis of the EMG4 SFEMG in our patients with trichinosis were J GOMEZ CEREZO showed increase of the Turns-Amplitude the same as those reported in polymyositis,' M T FERRER (T:A) ratio (table). The SFEMG was perfor- and might be due to reinnervation following J MEDRANO on October 2, 2021 by guest. med in extensor digitorum communis muscle denervation. Preceding denervation could be M C PtREZ CONDE (EDC)5 and fibre density (FD) was expressed caused by segmental necrosis of the muscle J COBO as normalised FD (nFD), which excludes the fibres.57 Reinnervation in polymyositis is F MOLINA changes due to age.6 The values ofnFD were consistent with the fibre type grouping found Section ofElectromyography increased in all examined patients (table). in biopsied muscles.7 Significant correlation and Service ofInternal Medicine, The SFEMG was newly performed in cases 5 between nFD and mean jitter in our patients Hospital "La Paz", and 8. The nFD increased from 5-3 to 9-7 and might be due to the early stage of the Po de la Castellana, 261, from 2-6 to 3-8, whereas the mean jitter regenerating activity, when the impulse 28046 Madrid, Spain. J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.52.5.681 on 1 May 1989. Downloaded from

Letters 683

Refereomes 91 t '' b Yl b 1 Davis MJ, Cilo M, Plaitakis A, Yahr MD. ., +.X# _f w>). , : Trichinosis: severe myopathic involvement with recovery. Neurology 1976;26:37-40. *;:yO c, .. W - 2 Gross B, Ochoa J. Trichinosis: clinical report o b. . and histochemistry of muscle. Muscle Nerve 1979;2:394-8. 3 Banker BQ. Parasitic Myositis. In: Engel AG, b.'D'ty' f ,.''.. Banker BQ, eds Myology. Basic and clinical. : *. New York: McGraw-Hill, 1986:1467-99. 4 Rose AL, Willison RG. Quantitative elec- tromyography using automatic analysis: studies in healthy subjects and patients with w;iNd .s . 'w; .# ...... : primary muscle disease. J Neurol Neurosurg Psychiatry 1967;30:403-10. 5 StAlberg E, Trontelj J. Single Fibre Electro- ax myography. Old Woking, Surrey: Mirvalle 4 W: a Y < .; Press, 1979. 4pi u t {4 / - . a,^.. 6 Hilton-Brown P, Stalberg E, Osterman PO. Signs of reinnervation in myasthenia gravis. .e,fa, 2 < >~~~~~~~4 Muscle Nerve 1982;S:215-21. tX f : ,2t ..... 7 Henriksson KG, Stalberg E. The terminal innervation pattern in polymiositis: a histo- chemical and SFEMG study. Muscle Nerve Ws r * 'S 1978;1:3-13. ; 6 .-Z_ ...... ,'st ...... ' ":zS t Accepted 12 December 1988. Fig Cross-section ofthe middle cerebral artery with perivascular tumourfragments. The adventitial tissue is intimately connected with tumour stroma (arrow heads). A haemorrhage is situated underneath the internal elastic membrane. The large arrow Protected by copyright. indicates the defect in the internal elastic membrane. (Elastic van Gieson stain, bar = 50 gm.) Dissecting aneurysm ofmiddle cerebral artery following resection of meningioma hours after surgery, CT showed a slight cerebral artery. This is the first instance Sir: Dissecting aneurysms of the intracranial hypodensity in the right temporal lobe. reported after surgery for a brain tumour. arteries are rare. Spontaneous dissecting Three days after operation a large infarct in NORBERT HEYE aneurysms have been reported in patients the area of the right middle cerebral artery JOSE R IGLESIAS with arterial hypertension, cystic media was diagnosed by CT. The patient died on HANS HENKES degeneration, fibromuscular dysplasia, and day 6 after surgery. RON FERZST in cases of Marfan's syndrome.'2 Blunt skull A large, approximately 5 day old infarct of KLAUS MAEER-HAUFF trauma may also lead to defects of the the middle cerebral artery was found at Institute ofNeuropathology, internal elastic membrane of intracranial postmortem examination. Small tumour Neurosurgery, Psychiatry and Radiology, vessels, but there is always the possibility of fragments were still attached to the vessel Klinikum Steglitz and Klinikum Westend, preexisting vascular lesions.2 We present a wall. Histologically, an endotheliomatous Freie Universitat Berlin, case of post-traumatic dissecting aneurysm meningioma had invaded the wall of the Hindenburgdamm 30, following partial resection of a large sphen- middle cerebral artery. There were multiple 1000 Berlin, FRG. oid ridge meningioma. ruptures of the internal elastic membrane http://jnnp.bmj.com/ A 49 year old patient was admitted for the along a 5 mm segment, with underlying References removal of a large meningioma. CT showed haematoma (fig). Some granulation tissue a tumour ofthe right sphenoid wing measur- and iron-positive macrophages at the border I O'Connell BK, Towfighi J, Brennan RW, et al. 3-5 x 4 with a 0 5 cm shift of the of the haematoma were found. The other Dissecting aneurysms of the head and neck. ing cm, Neurology 1985;35:993-7. midline structures to the left. In preoperative arteries were free ofdegenerative changes. 2 Krauland W. Verletzungen der Intrakraniellen angiography there was distension of the Some cases of dissecting aneurysms of Schlagadern. Berlin: Springer, 1982:125-40. carotid siphon and the circle of Willis. The intracranial arteries had recent head trauma, 3 Farrell MA, Gilbert JJ, Kaufmann JCE. Fatal Ml sector of the ipsilateral middle cerebral in other cases a generalised vasculopathy was intracranial arterial dissection: clinical artery was stenosed. At operation, the observed. " In our case, this was clearly pathological correlation. J Neurol Neurosurg on October 2, 2021 by guest. tumour was found surrounding the M 1, M2, absent; all other vessels were free of path- Psychiatry 1985;48: 111-2. and M3 segment of the middle cerebral ological changes. The resection of a tumour 4 Morki B, Sundt TM, Houser OW, Piepgras connected to the vessel wall DG. Spontaneous dissection of the cervical artery and the proximal segments of the intimately internal carotid artery. Ann Neurol anterior and communicans posterior appears to be the only plausible explanation 1986;19:126-38. arteries. Partial resection of the tumour was for this dissecting aneurysm. In one previous 5 Bigelow NH. Intracranial dissecting performed, leaving some surrounding the case5 a dissecting aneurysm was considered aneurysms. Arch Pathol 1955;60:271-5. medial cerebral artery, which was seen pul- to be directly related to surgery and occurred sating during the operation. Twenty four -after resection of an aneurysm of the middle Accepted 5 November 1988