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15号染色体近端重复:临床难题

Proximal duplications of chromosome 15: clinical dilemmas.

作者信息

Hood O J, Rouse B M, Lockhart L H, Bodensteiner J B

出版信息

Clin Genet. 1986 Mar;29(3):234-40. doi: 10.1111/j.1399-0004.1986.tb00817.x.

Abstract

The apparently rare cytogenetic abnormality of partial trisomy 15 was diagnosed by the authors in a patient presenting with developmental retardation, macrocephaly with ventricular enlargement and prominent subarachnoid spaces, hypotonia, low-set ears, hyperextensible wrists and hands, high arched palate, tapering fingers, right esotropia, and bilateral metatarsus adductus. Clinical findings in this case are similar to previously reported cases of proximal duplications of chromosome 15 and bear some similarity to the Prader-Willi syndrome. However, our patient did not have the severe hypotonia, early failure to thrive, or genital abnormalities seen in classical Prader-Willi syndrome. This case supports the theory that a variety of cytogenetic aberrations in proximal 15q can cause a "Prader-Willi-like" syndrome. Increased clinical suspicion is needed when patients are seen with hypotonia, retarded development and mild dysmorphism if the variety of phenotypes are to be delineated.

摘要

作者诊断出一名患有发育迟缓、伴有脑室扩大和蛛网膜下腔增宽的巨头畸形、肌张力减退、低位耳、手腕和手部过度伸展、高拱腭、手指变细、右眼内斜视以及双侧内收跖骨的患者存在明显罕见的15号染色体部分三体细胞遗传学异常。该病例的临床发现与先前报道的15号染色体近端重复病例相似,并且与普拉德-威利综合征有一些相似之处。然而,我们的患者没有典型普拉德-威利综合征中出现的严重肌张力减退、早期生长发育不良或生殖器异常。该病例支持这样一种理论,即15q近端的各种细胞遗传学畸变可导致“普拉德-威利样”综合征。如果要明确各种表型,当患者出现肌张力减退、发育迟缓以及轻度畸形时,需要提高临床怀疑度。

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