Micturitional Disturbance in Herpetic Brainstem Encephalitis; Contribution of the Pontine Micturition Centre
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J Neurol Neurosurg Psychiatry 1998;64:269–272 269 SHORT REPORT Micturitional disturbance in herpetic brainstem encephalitis; contribution of the pontine micturition centre Ryuji Sakakibara, Takamichi Hattori, Toshio Fukutake, Masahiro Mori, Tomonori Yamanishi, Kosaku Yasuda Abstract coeruleus14 and lateral dorsal tegmental Micturitional disturbance is rarely men- nucleus.5 A pontine storage centre also exists tioned in human herpetic brainstem en- just ventromedial or lateral to the pontine mic- cephalitis although the pontine turition centre. Recently, we found micturi- tegmentum, called the pontine micturi- tional disturbance in patients with brainstem tion centre, seems to regulate the lower stroke.6 Their MRI showed that the responsible urinary tract in experimental animals. sites are comparable with those reported in The case of a 45 year old man, who devel- experimental studies.1–3 Herpes simplex virus oped subacute coma and hiccup-like dys- type 1 (HSV-1) infection also causes brainstem rhythmic breathing, and needed assisted lesions78characterised by acute onset of multi- ventilation is reported. Examination of ple cranial nerve palsies, ataxia, and pyramidal CSF showed mononuclear pleocytosis and tract involvement. Disturbances of conscious- antibody against herpes simplex virus ness and respiration are not uncommon. type 1, but the opening pressure was 90 cm Micturitional disturbance is rarely reported in this disease. We here describe the micturitional H2O. Brain CT showed brain swelling, predominantly in the posterior fossa, and disturbance of a patient with herpetic brain- bilateral subdural eVusion. Herpetic stem encephalitis who showed bilateral pontine brainstem encephalitis was diagnosed, tegmental lesions on MRI. and he received 900 mg/day vidarabine. On regaining consciousness, he had left Case report trochlear nerve palsy, left corectopia, A 45 year old, previously healthy man devel- ageusia, and urinary retention. Brain MRI oped band-like headaches around the bilateral Department of showed right side dominant, bilateral Neurology occipitotemporal area which responded par- R Sakakibara pontine segmental lesions extending tially to analgesia but continued for five weeks, T Hattori slightly to the midbrain and medulla. changing to a persistent throbbing headache in T Fukutake After two weeks he was able to urinate but the bilateral frontal area. A week after onset he M Mori showed nocturnal urinary frequency, uri- was admitted to our hospital. On admission his nary incontinence, and voiding diYculty. body temperature was normal, and he was alert Department of Urodynamic studies showed a residual Urology, Chiba and responded well, although his neck was stiff University School of urine volume of 350 ml and detrusor and the Kernig sign was positive. The ocular Medicine, Chiba, hyporeflexia on voiding. Micturitional fundi, pupils, extraocular muscles, and other Japan disturbance gradually disappeared to- cranial nerves were intact, and coordination of T Yamanishi gether with the neurological signs. The the limbs was normal. Tendon reflexes were K Yasuda bilateral pontine tegmental lesions in this active and symmetric with no extensor planter patient are similar to those in previous Department of responses. Sensations to pin prick and position Neurology, Kashima findings on brainstem strokes, evidence of were normal. The second day after admission Rosai Hospital, the presence of a pontine micturition cen- he gradually became somnolent. On the fourth Kashima, Japan tre in humans. day he was comatose and had no oculocephalic R Sakakibara (J Neurol Neurosurg Psychiatry 1998;64:269–272) reflex, hiccup-like dysrhythmic breathing, and T Fukutake ° M Mori Keywords: brainstem encephalitis; herpes simplex virus a fever of 38.5 C. An indwelling urinary cath- type 1 (HSV-1); urinary retention; urodynamic study; eter was used to monitor urinary volume, and Correspondence to: pontine micturition centre he was placed on assisted ventilation. Periph- Dr R Sakakibara, eral blood analysis showed normal findings. An Department of Neurology, Chiba University EEG showed diVuse slowing, without periodic 1–8–1 Inohana, Chuo-Ku, The pontine tegmentum, also called the synchronous discharge. Brain CT detected dif- Chiba 260 Japan. Telephone pontine micturition centre, has an essential fuse brain swelling, particularly in the posterior 0081 43 226 2129; fax 0081 43 226 2160; email function in urinary evacuation. Lesioning and fossa, and bilateral subdural eVusion with [email protected] electrical or chemical stimulations in animals1–3 niveau. Examination of CSF showed a low Received 30 May 1997 show that the pontine micturition centre is opening pressure of 90 cm H2O, mild mononu- Accepted 9 July 1997 located adjacent to,3 or includes,2 the locus clear pleocytosis of 10 /mm3, and increased 270 Sakakibara, Hattori, Fukutake, et al Figure 1 MRI (A Axial plane R–L, B sagittal plane; T2 weighted image, TR 2500,TE100). Abnormally high signal intensities present in the right side dominant, bilateral pontine tegmentum, and extend slightly to the midbrain and medulla. total protein of 93 mg/dl. Antibody values protein of 44 g/dl. At this time, after removal of against HSV-1 in the CSF and the serum the urinary catheter, he had left trochlear nerve respectively were 0.583 and 1.864 in the IgG palsy, left corectopia, ageusia, and urinary enzyme immunoassay and 1:32 and 1:256 in retention, and needed clean, intermittent cath- the complement fixation test.9 The antibody eterisation. Brain MRI showed right side index was 8.5 (albumin 60 mg/dl in CSF and dominant, bilateral pontine segmental lesions 4100 mg/dl in serum). Herpetic encephalitis, extending slightly to the midbrain and medulla predominantly in the brainstem, was diag- (fig 1), but no apparent cerebral lesion. He nosed. gradually became able to urinate and to walk to Aciclovir (1200 mg/day) was started, but was the toilet. On the 44th day, as he still had noc- without benefit. On treatment with 900 mg turnal frequency, urinary incontinence and vidarabine/day, 24 mg dexamethasone/day, and voiding diYculty, urodynamic studies were 900 ml mannitol/day, he gradually regained made. normal consciousness on the 32nd day. Brain The methods and definitions used for the oedema and subdural eVusion had disappeared urodynamic studies conformed to the stand- on the follow up brain CT. A CSF examination ards proposed by the International Continence gave a normal opening pressure of 150 cm Society.10 Neither urinary tract infection nor H2O, no pleocytosis, and mildly increased total organic obstructive urological disease were Figure 2 Results of urodynamic studies. Simultaneous recordings of intravesical pressure (Pves) and external urethral sphincter EMG. Bladder volume at first desire to void (FDV) was 200 ml and at maximum desire to void (MDV) 500 ml. There is no detrusor hyperreflexia. During voluntary micturition (VOID), the detrusor pressure rise was insuYcient, evidence of hypocontractile bladder on voiding. EMG activity has disappeared, and there is no detrusor-sphincter dyssynergia (DSD). Micturitional disturbance in human herpetic brainstem encephalitis 271 Locus coeruleus petic encephalitis, particularly in the brainstem type,11 12 corresponds to a low CSF pressure.13 Superior cerebellar peduncle Micturitional disturbance has rarely been described in herpetic brainstem encephalitis, Medial parabrachial nucleus probably because serious clinical features, such Pontine reticular nucleus and as disturbed consciousness or respiratory reticular formation arrest, usually mask this disturbance. Antiviral chemotherapy with steroids and ventilatory Medial leminiscus support ameliorated the acute signs of our patient, but he showed urinary retention which changed to nocturnal urinary frequency, uri- nary incontinence and voiding diYculty. Uro- dynamic study results showed the presence of 350 ml residual urine volume, and detrusor hyporeflexia on voiding, evidence of a severe evacuating disorder. Detrusor areflexia occurs in peripheral nerve lesions. Our patient, however, had neither the decreased tendon Abducens nucleus reflexes nor disturbed sensation in the limbs Trigeminal sensory nucleus that indicate peripheral neuropathy. Detrusor areflexia also occurs in cerebral diseases within Reticular formation several months during the shock phase,14 and 6 Facial nucleus may persist for years. Detrusor hypoflexia in our patient indicates a supranuclear type of Medial leminiscus pelvic nerve dysfunction as has been reported in vascular diseases6 and tumours15 16 of the Spinothalamic tract brainstem. Other than micturitional disturbance, our patient had coma, hiccup-like dysrhythmic Figure 3 Suspected pontine micturition centre in humans. The dotted circle (pontine breathing, absence of the oculocephalic reflex, micturition centre) includes the pontine reticular nucleus, the reticular formation adjacent to and left trochlear palsy with corectopia after- 6 the medial parabrachial nucleus, and the locus coeruleus. (Cited from Sakakibara et al. ) wards, suggestive of brainstem tegmental found by digital examination of the prostate lesion. Ageusia reflects a lesion of the central and cystourethrography. He had a residual gustatory pathway, which travels in the medial urine volume of 350 ml. Water cystometry part of the medial leminiscus or of the reticular showed a bladder volume of 200 ml at first formation.17 18 MRI showed right sided domi- desire to void, and 500 ml at maximum desire nant, bilateral pontine tegmental