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范可尼贫血症并单侧拇指多指畸形——不容错过。

Fanconi's anaemia and unilateral thumb polydactyly--don't miss it.

机构信息

Department of Plastic and Reconstructive Surgery, Leeds General Infirmary, Leeds LS1 3EX, United Kingdom.

出版信息

J Plast Reconstr Aesthet Surg. 2012 Aug;65(8):1083-6. doi: 10.1016/j.bjps.2012.02.015. Epub 2012 Mar 3.

Abstract

Fanconi's anaemia (FA) is a rare, life threatening inherited syndrome. Patients usually present late in the first decade of life with aplastic anaemia or acute myeloid leukaemia. FA children are also at high risk of solid organ tumours, anogenital squamous cancers, and endocrinopathies. These patients can present with unilateral radial abnormalities including thumb duplication. Hand surgeons can help achieve early diagnosis and improved survival in this group by early referral for screening. In a retrospective study of 202 children with radial ray anomalies seen over a 20 year period seven children had FA. Of these seven with FA, four had bilateral thumb hypoplasia and three had unilateral thumb anomalies--two unilateral thumb hypoplasias and one thumb duplication. The three children with unilateral anomalies were diagnosed late, presenting with bone marrow failure. All three have subsequently died following late bone marrow transplants. This study highlights the link between unilateral radial anomalies, including thumb duplication and FA and the importance of early genetic referral for diagnosis and surveillance.

摘要

范可尼贫血(FA)是一种罕见的、危及生命的遗传性综合征。患者通常在生命的第一个十年晚期出现再生障碍性贫血或急性髓系白血病。FA 患儿也有很高的罹患实体器官肿瘤、肛门生殖器鳞状细胞癌和内分泌疾病的风险。这些患者可能会出现单侧桡骨异常,包括拇指重复。手部外科医生可以通过早期转介进行筛查,帮助这组患者实现早期诊断和提高生存率。在一项回顾性研究中,对 20 年间出现的 202 例桡骨射线异常的儿童进行了研究,其中 7 例患有 FA。在这 7 例 FA 患儿中,有 4 例双侧拇指发育不全,3 例单侧拇指异常,其中 2 例单侧拇指发育不全,1 例拇指重复。3 例单侧异常患儿诊断较晚,表现为骨髓衰竭。这 3 例患儿均在骨髓移植后因晚期移植而死亡。本研究强调了单侧桡骨异常,包括拇指重复与 FA 之间的联系,以及早期进行遗传转介以进行诊断和监测的重要性。

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