好色先生

好色先生

Explore the latest content from across our publications
Join The AAN

Log In

Forgot Password?
Create New Account

Loading... please wait

Abstract Details

Oromandibular dystonia in a patient with herpes simplex virus encephalitis (HSVE)
General Neurology
P1 - Poster Session 1 (5:30 PM-6:30 PM)
4-037

To expand the differential diagnosis of isolated oromandibular dystonia.

A 77 year old man presented to the hospital due to increased frequency of spells consisting of right arm shaking, starring and speech arrest. Similar spells were reported 3 months prior to presentation, for which he was on zonisamide.  His prior medical history included CAD, hypertension, hyperlipidemia, type 2 diabetes, CKD, and neuroendocrine carcinoma with metastases to the lungs, liver and brain, treated with chemotherapy with etoposide and carboplatin, and whole brain radiation.

Case report.
Initial brain MRI with contrast showed decreased size of the known brain metastases. On admission, started IV fosphenytoin. Laboratory studies showed pancytopenia and he was treated for suspected pneumonia. One day later, he had mental status deterioration, fever and required intubation. Soon after, he developed dyskinetic rhythmic oromandibular movements resulting in forceful jaw opening and closure, 1 Hz frequency, without lingual or palatal component. There was no electrophysiological correlation on continuous EEG.  The patient’s mental status failed to improve despite weaning off sedation. Repeat MRI showed interval development of multiple restricted DWI foci with no ADC correlate, and bilateral hippocampal hyperintensities. The patient was started on empiric acyclovir. A lumbar puncture was not obtained due to severe thrombocytopenia.  The patient’s care was transitioned to comfort measures only.   At autopsy the brain showed scattered foci of softening primarily involving the cortex of both cerebral hemispheres.  Histology showed numerous necrotic neurons and glia with Cowdry A inclusions, confirmed as HSV by immunohistochemistry.
HSVE can present with a variety of symptoms and signs.  High clinical suspicion is needed to identify some of the rare ones, such as the isolated oromandibular jaw opening dystonia evidenced in our patient.  A similar presentation has only once been previously reported (Meyer JS et al, Arch Neurol 1970:23(5),438-50).
Authors/Disclosures
Sylvia Josephy, MD (Caja Costarricense de Seguro Social)
PRESENTER 		Dr. Josephy has nothing to disclose.
Thomas W. Smith, MD (Univ of Massachusetts Med Ctr) No disclosure on file
No disclosure on file
Anas Hannoun, MD (Dartmouth-Hitchcock Manchester) No disclosure on file
Dalia Abou Zeki, MD No disclosure on file
Raphael A. Carandang, MD, FAAN (University of Massachusetts Medical School/UMASS Memorial Medical Group) Dr. Carandang has nothing to disclose.