A 10-year-old white female honors student with an uneventful past medical history except for congenital dislocated lenses underwent surgical repair of a slipped femoral epiphysis followed by the usual postoperative reduction in mobility. Three months later she was examined for left-sided spasmodic torticollis. No neurologic cause for these movements was ascertained, and the patient was hospitalized for 7 weeks on a psychiatric ward with a diagnosis of conversion reaction with secondary gains, without improvement.
Arbour, L., Rosenblatt, B., Clow, C., & Wilson, G. (1988). Postoperative dystonia in a female patient with homocystinuria. The Journal of Pediatrics., 113(5), 863-864.
A cranial computed tomography scan at the onset of movements was normal. Magnetic resonance imaging was delayed until dystonic movements had decreased after 9 months, and was also found to be normal. A positive cyanide nitroprusside urinary test for sulfhydryl compounds led to urinary and plasma amino acid quantification by ion exchange chromatography. Increased homocysteine and methionine levels in the proband and her sister confirmed the diagnosis of homocystinuria. Methylmalonic aciduria was ruled out by organic acid studies. Treatment of the dystonic movements with sequential trials of carbamazepine, lorazepam, and carbidopa/levodopa was unsuccessful. Trihexiphenidyl therapy was then instituted. The patient showed the first signs of improvement after 4 months of treatment. After 6 months the dystonie movements were no longer evident.