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戈谢病中的系统性 AL 淀粉样变性。一例病例报告及文献复习。

Systemic AL amyloidosis in Gaucher disease. A case report and review of the literature.

作者信息

Kaloterakis A, Filiotou A, Koskinas J, Raptis I, Zouboulis C, Michelakakis H, Hadziyannis S

机构信息

Academic Department of Internal Medicine and Department of Pathology, Hippokration Hospital, Athens, Greece.

出版信息

J Intern Med. 1999 Dec;246(6):587-90. doi: 10.1046/j.1365-2796.1999.00607.x.

Abstract

Chronic Gaucher disease [GD] in association with systemic AL amyloidosis is extremely rare. We describe a 46-year-old Greek male with chronic GD confirmed by low glucocerebroside activity in fibroblasts and N370S/L444P mutations at the cerebrosidase gene, who also had systemic AL amyloidosis diagnosed by bone marrow diffuse plasmacytosis, serum monoclonal IgA-lambda, severe total proteinuria with monoclonal IgA-lambda, Bence-Jones-lambda and amyloid deposits in bone marrow, liver, spleen and kidney biopsy specimens. Treatment with melphalan and prednizolon has dramatically decreased both levels of serum M-IgA and proteinuria and also improved the clinical symptoms of amyloidosis. He died from restrictive cardiac disease 30 months after the diagnosis of amyloidosis. Previously reported cases of GD in association with AL amyloidosis are reviewed.

摘要

慢性戈谢病(GD)合并系统性AL淀粉样变性极为罕见。我们描述了一名46岁的希腊男性,其成纤维细胞中葡萄糖脑苷脂活性降低以及脑苷脂酶基因存在N370S/L444P突变,确诊为慢性GD,同时通过骨髓弥漫性浆细胞增多、血清单克隆IgA-λ、伴有单克隆IgA-λ的严重总蛋白尿、本周氏蛋白-λ以及骨髓、肝脏、脾脏和肾脏活检标本中的淀粉样沉积诊断为系统性AL淀粉样变性。美法仑和泼尼松龙治疗显著降低了血清M-IgA水平和蛋白尿水平,也改善了淀粉样变性的临床症状。他在淀粉样变性诊断30个月后死于限制性心脏病。本文对先前报道的GD合并AL淀粉样变性的病例进行了综述。

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