書誌事項
- タイトル別名
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- Anesthesia-induced rhabdomyolysis in a patient with Fukuyama-type congenital muscular dystrophy.
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抄録
Malignant hyperthermia (MH) is one of the most serious complications during general anesthesia, representing high fever, cardiovascular insufficiency, acidosis and muscle contracture. Since there has been no reported case with MH in Fukuyama type congenital muscular dystrophy (FCMD), we report a case in details to draw an attention on this subject.<BR>Case Report: A 3-month-old boy was admitted to our hospital because of fever and clonic convulsion. Streptcoccus pneumoniae was isolated from the CSF, and penicillin G was started. A subdural tap was performed because of persistent fever, positive CRP and bilateral subdural effusion revealed by CT scan. About two months later, a surgical treatment was performed.<BR>Under N2O-O2 and halothane inhalation, the jaw muscles became rigid shortly after intravenous administration of succinylcholine chloride, followed by cardiac arrest and apnea. After immediate resuscitation he recovered without fever or acidosis. The serum CK and urine myoglobin were elevated, but serum electrolyte values remained normal. The urine became clear approximately 48 hours after the accident. The biopsied muscle showed characteristic fibers with a definite borderline susceptibility to caffeine by the skined fiber method, suggesting that the present case was a probable candidate for malignant hyperthermia. Although the present case had no elevated body temperature during anesthesia, he developed cardiac arrest and rhabdomyolysis, and highly sensitive myofibers to caffeine by the skinned fiver examination. With the clinical symptoms and an in vitro study, it is little doubt that he had a forme fruste of the MH syndrome. Since the present case had clinical and pathological characteristics of FCMD, a careful attention should be paid during general anesthesia not only to the Duchenne dystrophy patients but also to FCMD infants, because they may be at hight risk to develop anesthetic complication.
収録刊行物
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- 脳と発達
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脳と発達 16 (5), 386-392, 1984
THE JAPANESE SOCIETY OF CHILD NEUROLOGY
