Cases reported "Dystonia"

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1/6. bupropion-induced acute dystonia.

    OBJECTIVE: To report a case of acute dystonia consisting of neck stiffness, trismus, and unilateral temporomandibular joint (TMJ) pain and subluxation secondary to an increase in sustained-release (SR) bupropion. CASE SUMMARY: A 44-year-old white man with a history of chronic low-back pain and tension headaches, taking no other medications, was started on bupropion SR 150 mg once a day for depression. The dosage was increased to 150 mg SR twice a day and eventually augmented with buspirone 15 mg 3 times a day. He developed bilateral trismus, inability to rotate his head laterally, and spontaneous left TMJ subluxation. Symptoms recessed with discontinuation of both medications and failed to reappear with a trial of buspirone 15 mg 3 times a day alone. A retrial of bupropion alone evidenced no adverse effects at a dosage of 150 mg SR once a day. However, when the dosage was increased to 150 mg SR twice a day, the patient reexperienced initial signs of neck stiffness, jaw muscle tightness, and left TMJ subluxation within 24-48 hours. Reduction of the bupropion dosage to 150 mg SR once daily stopped the symptoms; the patient has continued at this dosage without adverse effects for > 1 year. DISCUSSION: Medication-induced focal dystonias usually present with dramatic head (most frequently oral-buccal) and neck muscle spasm with occasional jaw clenching, bruxism, and TMJ syndrome. In this case, the rapid onset of neck and jaw symptoms within 24-48 hours of an increase of bupropion SR from 150 mg once a day to 150 mg twice a day suggest that the patient may have been sensitized by an initial trial of bupropion and buspirone, or by the increased dose of bupropion alone. Both agents are reported to interact with both the dopaminergic and serotonergic systems. Although buspirone has been implicated in inducing acute dystonia, it did not do so in this case when used alone at a dose of 45 mg a day. During a second trial of bupropion SR 150 mg a day, neck and jaw symptoms recurred within 24-48 hours of increasing the dose to 150 mg SR twice a day. The symptoms receded when the bupropion dose was returned to 150 mg SR once a day, suggesting a dose-response relationship. The Naranjo probability scale indicated that this untoward reaction was probable. CONCLUSIONS: This case suggests that selected patients may experience dose-related acute dystonic adverse reactions to bupropion with or without buspirone augmentation. Dystonias, which usually follow administration of antipsychotics, have been linked to acute dopamine depletion and basal ganglion-derived gamma synchronization dysfunction. Acute dystonia symptoms may begin within hours of starting or changing antipsychotic drug dosage; however, 90% of symptoms are observed during the first 3-5 days of starting or increasing dosage. To the best of our knowledge, there have been no reports of bupropion-induced dystonia.
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keywords = ganglion
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2/6. movement disorders associated with chronic GM2 gangliosidosis. Case report and review of the literature.

    A 24-year-old man presented with dystonia, dementia, amyotrophy, choreoathetosis, and ataxia. Partial hexosaminidase a deficiency was documented in serum and leukocytes and confirmed by rectal biopsy with ganglion cells containing membranous cytoplasmic bodies. A brief review of the literature reveals that tremor, dystonia and choreoathetosis are common but neglected symptoms associated with chronic GM2 gangliosidosis.
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3/6. Pure hemidystonia with basal ganglion abnormalities on positron emission tomography.

    We present a patient with hemidystonia and an abnormality of the contralateral basal ganglion seen only with positron emission tomography. A 50-year-old sinistral man suffered minor trauma to the right side of his head and neck. Within 20 minutes he developed paroxysmal intermittent dystonic posturing of his right face, forearm, hand, and foot, with weaker contractions of the left foot, lasting several seconds and recurring every few minutes. Neurological findings between spells were normal. The following were also normal: electrolyte, calcium, magnesium, and arterial blood gas levels, and findings of drug screen, cerebrospinal fluid examination, electroencephalography with nasopharyngeal leads, computed tomographic scanning (initially and four weeks later), and cerebral angiography. Positron emission tomographic scanning revealed abnormalities in the left basal ganglion region, including decreased oxygen metabolism, decreased oxygen extraction, increased blood volume, and increased blood flow.
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ranking = 6
keywords = ganglion
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4/6. Reversible parkinsonism and dystonia following probable mycoplasma pneumoniae infection.

    Infectious disease is a rare cause of parkinsonism. We report a 7-year-old boy who developed flu-like symptoms followed by parkinsonian features including hypophonia, hypomimia, bradykinesia, and dystonia. A T2-weighted brain magnetic resonance imaging showed high signal intensities in both basal ganglia. The results of serial serologic tests of Mycoplasma pneumonia antibody suggest that this movement disorder was associated with Mycoplasma pneumonia infection. The patient's symptoms gradually resolved, and the basal ganglionic lesions disappeared on follow-up magnetic resonance imaging.
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keywords = ganglion
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5/6. Basal ganglionic angioleiomyoma.

    A rare case of basal ganglionic angioleiomyoma in a 12-year-old girl is reported. She presented with features of raised intracranial tension, multifocal seizures, left hemidystonia and apraxia of eyelid closure. Cranial CT scan showed a large hypodense lesion with an enhancing mural nodule in the region of head of the caudate nucleus on the right side. The lesion was extending to the posterior limb of internal capsule postero-medially and to globus pallidus laterally. Another small, hyperdense, enhancing lesion was seen in the region of globus pallidus on the left side. The right basal ganglionic mass was excised through frontal craniotomy. Histopathological, Immunocytochemical and Ultrastructural examination of the right basal ganglionic mass showed features characteristic of angioleiomyoma. Following surgery the patient improved initially with respect to dystonia but worsened later on. Repeat cranial CT, 20 months after surgery, showed bilateral frontotemporal atrophy without any tumor recurrence while the lesion on the left side remained quiescent.
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ranking = 7
keywords = ganglion
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6/6. Acute dystonia with thalamic and brainstem lesions after initial penicillamine treatment in Wilson's disease.

    dystonia is a common manifestation in Wilson's disease (WD). The striatum, especially the putamen, has been considered to be responsible for dystonia. We reported 3 patients who developed acute generalized dystonia and akinetic rigid syndrome following an initial therapy with d-penicillamine 125-500 mg daily. brain MRI revealed lesions in the thalamus and the brainstem, particularly the tegmentum, and the basis pontis in addition to the basal ganglion lesions. After the episode, 1 patient continued to receive d-penicillamine therapy and 2 changed to zinc sulfate treatment. The generalized dystonia improved in the following 3 months and 3 years respectively in 2 patients. Follow-up brain MRI of these 2 patients revealed that the lesions in the thalamus and brainstem disappeared or resolved almost completely. From these data, acute generalized dystonia with brainstem and thalamic lesions may occur in WD patients after an initial d-penicillamine therapy. Furthermore, the dystonia may resolve following the disappearance of the brainstem and thalamic lesions.
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