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一名中国患者的类脂蛋白沉积症

Lipoid proteinosis in a Chinese patient.

作者信息

Xu Xian-Ting, Chen Qiping, Siong-See Lee Joyce

机构信息

*Department of Dermatology, Wenzhou Central Hospital, Wenzhou, China; and †National Skin Centre, Singapore, Singapore.

出版信息

Am J Dermatopathol. 2014 Jun;36(6):e108-13. doi: 10.1097/DAD.0000000000000006.

Abstract

Lipoid proteinosis is a rare autosomal recessive deposition disorder due to loss-of-function mutations in the gene encoding extracellular matrix protein 1 on chromosome 1q21. There are limited case reports of lipoid proteinosis in the Chinese population. The authors report 1 case of lipoid proteinosis in a Chinese patient with typical clinical and histopathological manifestations. Physical examination in this patient demonstrated hoarse voice, hypertrophy of tongue and lips, inability to fully protrude the tongue, and cutaneous features including moniliform blepharosis, verrucous plaques, and scarring. Biopsies from the eyelid, pharyngeal mucosa, and elbow lesions revealed diffuse amorphous deposits of hyaline material within the dermis and around blood vessels, which stained positively for periodic acid-Schiff, was diastase resistant and stained negatively on Congo red.

摘要

类脂蛋白沉积症是一种罕见的常染色体隐性遗传性沉积障碍疾病,由位于1号染色体长臂21区的细胞外基质蛋白1编码基因突变导致功能丧失引起。关于中国人群类脂蛋白沉积症的病例报道有限。本文作者报告了1例具有典型临床和组织病理学表现的中国类脂蛋白沉积症患者。该患者体格检查显示声音嘶哑、舌和嘴唇肥大、舌头不能完全伸出,皮肤表现包括串珠状睑裂狭小、疣状斑块和瘢痕形成。对眼睑、咽黏膜和肘部病变进行活检,结果显示真皮层和血管周围有弥漫性透明物质无定形沉积,过碘酸-希夫染色呈阳性,抗淀粉酶,刚果红染色呈阴性。

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