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Pathological Case of the Month
Gail D. Cawkwell, MD, CM, PhD;
Atilano G. Lacson, MD, FRCPC;
Tony Kriseman, MB, B, Ch
From the Divisions of Allergy and Clinical Immunology (Dr Cawkwell) and Pulmonology (Dr Kriseman), Department of Pediatrics, and the Department of Pathology and Laboratory Medicine (Dr Lacson), All Children's Hospital, St Petersburg, Fla. Dr Cawkwell is now with Pfizer Inc, New York, NY.
Arch Pediatr Adolesc Med. 2001;155:853-854.
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A PREVIOUSLY healthy female infant developed vomiting, lethargy, poor oral intake, gray coloring, and decreased lower extremity movement at age 8 weeks. Respiratory acidosis prompted intubation. An electromyogram (EMG) showed disorganized muscle fibrillations consistent with inflammatory myopathy. Trials of intravenous immunoglobulin and methotrexate proved ineffective. Muscle weakness and stiffness progressed for the next 2 years (Figure 1), and creatine kinase levels were increased. At age 2 years, she lost all motion of the knees and elbows and developed spontaneous dislocation of the hips with preserved mobility of ankles and wrists. Impaired swallowing and decreased diaphragmatic and thoracic cage compliance resulted in a need for mechanical ventilation. The EMG of the right deltoid, vastus medialis, and tibialis anterior muscles at rest showed spontaneous fibrillations and sharp waves superimposed on normal-appearing motor unit action . . . [Full Text of this Article]
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