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另一种综合征——导致双肺移植的进行性肺病的家族性表现:一例病例报告及文献综述

ANOTHER syndrome-Familial presentations of progressive lung disease leading to double lung transplantation: A case report and literature review.

作者信息

Thornton Christina S, Puttagunta Lakshmi, Helmersen Douglas, Thakrar Mitesh V, Nagendran Jayan, Lien Dale, Varughese Rhea A

机构信息

Division of Respirology, Department of Medicine Cumming School of Medicine, University of Calgary Calgary Alberta Canada.

Department of Laboratory Medicine and Pathology University of Alberta Edmonton Alberta Canada.

出版信息

Respirol Case Rep. 2021 Nov 5;9(12):e0872. doi: 10.1002/rcr2.872. eCollection 2021 Dec.

Abstract

Ectodermal dysplasias (EDs) are a heterogeneous rare group of disorders with an incidence at 1/100,000 live births. Currently, there are limited case reports of patients requiring lung transplantation. Here, we report two brothers who present with a constellation of features including alopecia, nail dystrophy, ophthalmic complications, thyroid disease, hypohidrosis, ephelides, enteropathy and recurrent respiratory tract infections, known as ANOTHER syndrome, a rare autosomal recessive variant of ED. Both presented in early childhood with progressive respiratory decline and eventual failure. Chronic respiratory decline was refractory to standard therapy. Both patients required lung transplantation for sequelae of end-stage lung disease. Pathology demonstrated multifocal bronchiectasis with areas of fibrosis and small airway obstruction. ANOTHER syndrome is rare with a paucity of data in the literature. Given the limited therapeutic options available with natural progression towards respiratory failure, lung transplantation may be considered.

摘要

外胚层发育不良(EDs)是一组异质性罕见疾病,活产发病率为1/100,000。目前,需要肺移植的患者病例报告有限。在此,我们报告两兄弟,他们表现出一系列特征,包括脱发、甲营养不良、眼科并发症、甲状腺疾病、少汗、雀斑、肠病和反复呼吸道感染,即ANOTHER综合征,这是一种罕见的常染色体隐性遗传的ED变异型。两人均在幼儿期出现进行性呼吸功能衰退并最终呼吸衰竭。慢性呼吸功能衰退对标准治疗无效。两名患者均因终末期肺病后遗症需要进行肺移植。病理显示多灶性支气管扩张,伴有纤维化区域和小气道阻塞。ANOTHER综合征很罕见,文献资料匮乏。鉴于针对自然进展为呼吸衰竭的可用治疗选择有限,可考虑进行肺移植。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0dc4/8569409/5dcc56504c5d/RCR2-9-e0872-g004.jpg

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