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一例罕见的10q22.3q23微缺失新病例,伴有嵌合型克兰费尔特综合征,与面部畸形、房间隔和室间隔缺损以及运动发育迟缓相关。

A New Case of Rare Microdeletion 10q22.3q23 along with Mosaic Klinefelter Syndrome Associated with Facial Dysmorphic Finding, Atrial Ventricular Septal Defect, and Motor Retardation.

作者信息

Dincsoy Bir Firdevs, Silan Fatma, Velickovic Jelena, Berkay Akcan Mehmet, Ozdemir Ozturk

机构信息

Department of Medical Genetics, Erzurum Regional Training and Research Hospital, Erzurum, Turkey.

Department of Medical Genetics, Faculty of Medicine, Canakkale Onsekiz Mart University, Canakkale, Turkey.

出版信息

Mol Syndromol. 2022 May;13(3):254-260. doi: 10.1159/000519965. Epub 2022 Feb 7.

DOI:10.1159/000519965
PMID:35707596
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9149477/
Abstract

The chromosome 10q22.3q23.2 deletion syndrome is characterized by craniofacial dysmorphic features, developmental delay, congenital heart defect, and hand/foot abnormalities. In this study, we report a patient carrying a microdeletion of 7.5 Mb at 10q22.3q23.2 and in addition a mosaicism mos 47,XXY[47]/46,XY[23]. This male patient was 3 years and 3 months years old at the time of genetic evaluation. Atrial ventricular septal defect (AVSD), mild hypotonia, torticollis, and left-sided club foot were noticed after birth. The boy had surgical correction of the AVSD and the club foot. His dysmorphic features were frontal bossing, overfolded ear helix, hypertelorism, epicanthal folds, broad base of nose, flat nasal bridge, full cheeks, thick lips, micrognathia, and joint hyperextensibility. His speech/language development was delayed. Klinefelter syndrome is one of the most common congenital chromosomal abnormalities, but usually it is detected in puberty or in adulthood when reproductive failure occurs. Deletions in the 10q22.3q23.2 region are rare, and previously only a few numbers of cases were described with this microdeletion, but none of them together with Klinefelter syndrome and it could be associated with our case clinical features. The new case described will improve understanding the phenotype associated with 10q22.3q23.2 microdeletions. By presenting this case, we aimed to improve the understanding of the phenotype caused by the rare 10q22.3q23.2 deletion and to show the rare coexistence of this deletion with Klinefelter syndrome.

摘要

10q22.3q23.2染色体缺失综合征的特征为颅面畸形、发育迟缓、先天性心脏缺陷以及手足异常。在本研究中,我们报告了一名患者,其在10q22.3q23.2区域存在7.5 Mb的微缺失,此外还存在47,XXY[47]/46,XY[23]的嵌合体。该男性患者在进行基因评估时为3岁3个月大。出生后发现有房间隔缺损(AVSD)、轻度肌张力减退、斜颈和左侧马蹄内翻足。该男孩接受了AVSD和马蹄内翻足的手术矫正。他的畸形特征包括前额突出、耳轮过度折叠、眼距增宽、内眦赘皮、鼻根部宽阔、鼻梁扁平、脸颊丰满、嘴唇增厚、小颌畸形以及关节过度伸展。他的语言发育延迟。克兰费尔特综合征是最常见的先天性染色体异常之一,但通常在青春期或成年期出现生殖功能衰竭时才被检测到。10q22.3q23.2区域的缺失较为罕见,此前仅有少数几例这种微缺失的病例报道,但均未与克兰费尔特综合征同时出现,而这可能与我们病例的临床特征相关。所描述的新病例将有助于增进对与10q22.3q23.2微缺失相关表型的理解。通过展示该病例,我们旨在提高对由罕见的10q22.3q23.2缺失所导致表型的认识,并展示这种缺失与克兰费尔特综合征罕见的共存情况。

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