tailieunhanh - Báo cáo y học: "Status dystonicus resembling the intrathecal baclofen withdrawal syndrome: a case report and review of the literature"

Tuyển tập báo cáo các nghiên cứu khoa học quốc tế ngành y học dành cho các bạn tham khảo đề tài: Status dystonicus resembling the intrathecal baclofen withdrawal syndrome: a case report and review of the literature. | Muirhead et al. Journal of Medical Case Reports 2010 4 294 http content 4 1 294 jAg JOURNALOF medical ÌỤr case REPORTS CASE REPORT Open Access Status dystonicus resembling the intrathecal baclofen withdrawal syndrome a case report and review of the literature William Muirhead1 Ibrahim Jalloh2 Michael Vloeberghs2 Abstract Introduction Status dystonicus is a rare but life-threatening disorder characterized by increasingly frequent and severe episodes of generalized dystonia that may occur in patients with primary or secondary dystonia. Painful and repetitive spasms interfere with respiration and may cause metabolic disturbances such as hyperpyrexia dehydration respiratory insufficiency and acute renal failure secondary to rhabdomyolysis. Intrathecally administered baclofen delivered by an implantable pump system is widely used for the treatment of refractory spasticity. Abrupt cessation of intrathecal baclofen infusion has been associated with a severe withdrawal syndrome comprised of dystonia autonomic dysfunction hyperthermia end-organ failure and sometimes death. The aetiology of this syndrome is not well understood. Status dystonicus describes the episodes of acute and lifethreatening generalized dystonia which occasionally manifest themselves in patients with dystonic syndromes. Case presentation We present the case of a nine-year-old Caucasian boy who experienced a severe episode of status dystonicus with no known cause and clinical features resembling those described in intrathecal baclofen withdrawal. Our patient subsequently underwent the placement of an intrathecal baclofen pump without incident. Conclusion The similarity between the clinical features of the case we present and those reported in connection to abrupt withdrawal of intrathecal baclofen is emphasized. Several drugs although not intrathecal baclofen withdrawal have previously been associated with status dystonicus. The similarity between the life-threatening dystonic