Nontraumatic Bilateral Diaphragmatic Myopathy: An Unusual Disturbance
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© Springer-Verlag New York Inc. 1997
Nontraumatic Bilateral Diaphragmatic Myopathy: An Unusual Disturbance P. Morales,1 E. Cases,1 and J. Gudı´n2 1
Servicio de Neumologia and 2Unidad de Cuidados Intensivos, Hospital Universitario La Fe, Valencia 46009, Espan˜a
Abstract. Bilateral diaphragmatic paralysis (BDP) is an uncommon entity among neuromuscular diseases. We present a young female with no triggering factors who developed progressive dyspnea with respiratory failure. Further studies led to the diagnosis of BDP. Key words: Diaphragmatic myopathy—Diaphragmatic paralysis—Idiopathic diaphragmatic paralysis. Introduction Bilateral diaphragmatic paralysis (BDP) is an uncommon entity among neuromuscular diseases involving the thoracic cage [4]. It is mainly the result of myopathies and muscular dystrophies [6]. Since BDP impairs diaphragmatic mobilization during the respiratory cycle, it results in a severe ventilatory limitation [5]. We present a young female patient with no triggering factors who developed progressive dyspnea with respiratory failure requiring mechanical ventilation. Case Report A 37-year-old female secretary, nonsmoker, reported dyspnea after her third delivery at term, which had progressively worsened for a year, occurring after minimal effort. Dyspnea occurred together with somnolence, unintentional tremors, and use of the accessory muscles during inspiration. At first, the patient attached no importance to these symptoms. She was also diagnosed with functional vomiting (by esophageal endoscopy) and was given gastric motility regulators (metoclopramide) and sedative drugs (5 mg of diazepam per day if required).
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Fig. 1. Posteroanterior chest X-ray showing no movement of both diaphagms during inspiration (A) and expiration (B). Two months after taking this medicine, the patient was hospitalized because of hypercapnic coma during bronchopneumonia. The only pertinent data were that she had been operated on at the age of 11 because of cervical malposition (the type of surgery is unknown), and she had undergone three full term pregnancies with difficult deliveries. The patient presented asthenic characteristics, pale skin and sweating, using her accessory neck muscles to breathe. Routine laboratory test results, including creatine phosphokinase serum levels, were within normal values. Arterial blood gases (inspired fraction of O2,FIO2, 0.21) were as follows: PaO2, 8.6 Kpa (65 torr); PaCO2, 8.1 Kpa (61 torr); pH 7.27; HCO−3, 24.6 mmol/liter. Her chest X-ray upon admission showed right basal infiltrate and elevation of both diaphragms. Because of her clinical condition, she required a tracheostomy and continuous mechanical ventilation for 40 days. She then tolerated daytime spontaneous ventilation with nocturnal controlled mechanical ventilation through the tracheostomy. X-ray showed no movement of either hemidiaphragm during fluoroscopy (‘‘sniff test’’) (Fig. 1, a, and b,). Functional tests showed a very severe ventilatory limitation [1]: vita
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