Senff H, Köllner A, Engelmann L, Woort-Menker M, Mensing H, Kunze J
Klinik für Dermatologie und Allergologie, St. Barbara-Hospital, Duisburg.
Hautarzt. 1990 Oct;41(10):578-82.
Two female patients developed localized scleroderma on the trunk and the thighs after oral ingestion of L-tryptophan for some years. Both patients reported acute progressive myalgia and weakness of the proximal parts of the extremities. On laboratory evaluation, the leucocyte count was approximately 20,000/mm3, with 38% blood eosinophils in one patient and 53% in the other. The ESR was slightly elevated; electrophoresis and muscle enzymes were normal. Skin and muscle biopsies revealed characteristic features of diffuse fasciitis with eosinophilia. High-dose glucocorticoid therapy resulted in a rapid normalization of the ESR and blood eosinophilia, whereas the scleroderma showed little improvement. The diffuse edema observed in one patient receded within a few days. A correlation between oral ingestion of L-tryptophan and the eosinophilia-myalgia syndrome has been reported recently, and the present case reports must be discussed in the light of this observation. Both patients developed a tryptophan-induced scleroderma-like illness resembling diffuse fasciitis with eosinophilia (Shulman's syndrome).
两名女性患者在口服L-色氨酸数年之后,于躯干和大腿出现局限性硬皮病。两名患者均报告有急性进行性肌痛以及四肢近端无力。实验室检查发现,白细胞计数约为20,000/mm³,其中一名患者血液嗜酸性粒细胞占38%,另一名患者占53%。血沉略有升高;电泳和肌肉酶检查结果正常。皮肤和肌肉活检显示出伴有嗜酸性粒细胞增多的弥漫性筋膜炎的特征性表现。大剂量糖皮质激素治疗使血沉和血液嗜酸性粒细胞增多迅速恢复正常,而硬皮病几乎没有改善。在一名患者身上观察到的弥漫性水肿在数天内消退。最近有报道称口服L-色氨酸与嗜酸性粒细胞增多性肌痛综合征之间存在关联,必须根据这一观察结果对本病例报告进行讨论。两名患者均患上了色氨酸诱导的硬皮病样疾病,类似于伴有嗜酸性粒细胞增多的弥漫性筋膜炎(舒尔曼综合征)。