Suoh H, Sahashi K, Ibi T, Tashiro M, Mitsuma T
Fourth Department of Medicine, Aichi Medical University.
Rinsho Shinkeigaku. 1991 Jul;31(7):746-9.
A 65-year-old house-wife developed dirty erythematous rash on her face in April, 1989. Almost simultaneously, she complained of muscle soreness and weakness on both lower extremities. Pathological findings of the skin biopsy at that time was consistent with those of sarcoidosis with moderate inflammatory cell infiltration. In December, 1989, when she was admitted to our hospital, her lower extremities were paretic with marked spasticity, and mild bladder dysfunction was noted. HTLV-I antibody titers in serum and cerebrospinal fluid were significantly elevated. Biopsied limb skeletal muscle revealed the findings of the sarcoid myopathy with small inflammatory cell infiltration in endomysium. HLA haplotypes showed A24, B7, BW61, CW7, CW8, DR1 and DR4 which show relatively common types of those in HAM. Corticosteroid treatments including the methylprednisolone pulse therapy healed the skin lesion, but did not improve her neurological signs. Paraplegia and urinary disturbance were progressive. It is concluded that the inflammatory sarcoid myopathy with HAM in this patient may be caused by a common abnormal immunological background.
一名65岁的家庭主妇于1989年4月面部出现暗红色皮疹。几乎与此同时,她诉说双下肢肌肉酸痛和无力。当时皮肤活检的病理结果与结节病相符,有中度炎症细胞浸润。1989年12月,她入住我院时,双下肢瘫痪,伴有明显痉挛,且存在轻度膀胱功能障碍。血清和脑脊液中的HTLV-I抗体滴度显著升高。活检的肢体骨骼肌显示为结节病性肌病,肌内膜有小炎症细胞浸润。HLA单倍型显示为A24、B7、BW61、CW7、CW8、DR1和DR4,这些在热带痉挛性截瘫中相对常见。包括甲基强的松龙冲击疗法在内的皮质类固醇治疗使皮肤病变愈合,但未改善其神经体征。截瘫和排尿障碍呈进行性发展。结论是该患者的炎症性结节病性肌病合并热带痉挛性截瘫可能由共同的异常免疫背景引起。