Rodrigues K M, Puccioni-Sohler M, Castineiras T M, Pereira M T, Papi J A, Schechter M, Avila C, Duarte F, Novis S A
Serviços de Doenças Infecciosas e Parasitárias (DIP), Hospital Universitário Clementino Fraga Filho, Rio de Janeiro, Brasil.
Arq Neuropsiquiatr. 1991 Sep;49(3):352-6. doi: 10.1590/s0004-282x1991000300022.
Report of an unusual case of myopathy in an HIV infected patient, responsive only to the immunosuppressor drug methotrexate. The patient was a 39 year old homosexual male with no past history of HIV-related manifestations. One month prior to admission he noticed that his left thigh was swollen and painful. Two weeks later both arms became enlarged and tender. A few days before admission he noticed intermittent fever and progressive dyspnea. Upon admission, oral thrush, dyspnea and global enlargement of both arms was noted. There was no articular involvement. Fiberoptic bronchoscopy revealed Pneumocystis carinii pneumonia (PCP). Serology for HIV was positive. Tests for antinuclear antibodies were negative. Serum CPK level was 1019 IU. Capillaroscopy was compatible with vasculitis. Muscle biopsy revealed multifocal myonecrosis. PCP was successfully treated with standard doses of TMP/SMZ. Although indomethacin, prednisone and dexamethasone were administered in succession, there was relentless progression of myopathy and persistence of fever. Six days after administration of methotrexate, the patient defervesced, volume of arms and legs diminished. CPK levels returned to normal after a second course of methotrexate. Upon reduction of the dose thigh enlargement recrudesced. The patient remained asymptomatic on weekly doses of methotrexate. He died five months later of acute respiratory failure.
一名HIV感染患者出现罕见肌病病例报告,该病例仅对免疫抑制剂甲氨蝶呤有反应。患者为一名39岁同性恋男性,既往无HIV相关表现病史。入院前一个月,他注意到左大腿肿胀疼痛。两周后,双臂也变得肿大且触痛。入院前几天,他出现间歇性发热和进行性呼吸困难。入院时,发现有口腔念珠菌病、呼吸困难以及双臂全面肿大。无关节受累情况。纤维支气管镜检查显示卡氏肺孢子虫肺炎(PCP)。HIV血清学检测呈阳性。抗核抗体检测为阴性。血清肌酸磷酸激酶(CPK)水平为1019 IU。毛细血管镜检查结果符合血管炎表现。肌肉活检显示多灶性肌坏死。PCP用标准剂量的复方磺胺甲恶唑(TMP/SMZ)成功治疗。尽管先后给予了吲哚美辛、泼尼松和地塞米松,但肌病仍持续进展且发热持续存在。给予甲氨蝶呤六天后,患者体温下降,四肢体积缩小。在第二个疗程的甲氨蝶呤治疗后,CPK水平恢复正常。减少剂量后,大腿再次肿大。患者每周服用甲氨蝶呤时无症状。五个月后,他死于急性呼吸衰竭。