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两种不同的源自母性 t(4;10)(q33;p15.1)易位的不平衡分离的结果。

Outcomes of two different unbalanced segregations from a maternal t(4;10)(q33;p15.1) translocation.

机构信息

Department of Medicine, University of California, 10833 Le Conte Avenue, Los Angeles, CA, 90095, USA.

Department of Pathology and Laboratory Medicine, UCLA Clinical Genomics Center, University of California, Los Angeles, USA.

出版信息

BMC Med Genomics. 2023 Mar 29;16(1):65. doi: 10.1186/s12920-023-01491-1.

Abstract

BACKGROUND

Unbalanced translocations can cause developmental delay (DD), intellectual disability (ID), growth problems, dysmorphic features, and congenital anomalies. They may arise de novo or may be inherited from a parent carrying a balanced rearrangement. It is estimated that 1/500 people is a balanced translocation carrier. The outcomes of different chromosomal rearrangements have the potential to reveal the functional consequences of partial trisomy or partial monosomy and can help guide genetic counseling for balanced carriers, and other young patients diagnosed with similar imbalances.

METHODS

We performed clinical phenotyping and cytogenetic analyses of two siblings with a history of developmental delay (DD), intellectual disability (ID) and dysmorphic features.

RESULTS

The proband, a 38-year-old female, has a history of short stature, dysmorphic features and aortic coarctation. She underwent chromosomal microarray analysis, which identified partial monosomy of 4q and partial trisomy of 10p. Her brother, a 37-year-old male, has a history of more severe DD, behavioral problems, dysmorphic features, and congenital anomalies. Subsequently, karyotype confirmed two different unbalanced translocations in the siblings: 46,XX,der(4)t(4;10)(q33;p15.1) and 46,XY,der(10)t(4;10)(q33;p15.1), respectively. These chromosomal rearrangements represent two possible outcomes from a parent who is a carrier for a balanced translocation 46,XX,t(4;10)(q33;p15.1).

CONCLUSION

To our knowledge, this 4q and 10p translocation has not been described in literature. In this report we compare clinical features due to the composite effects of partial monosomy 4q with partial trisomy 10p and partial trisomy 4q with partial monosomy 10p. These findings speak to the relevance of old and new genomic testing, the viability of these segregation outcomes, and need for genetic counseling.

摘要

背景

不平衡易位可导致发育迟缓(DD)、智力障碍(ID)、生长问题、发育异常和先天性异常。它们可能是新发的,也可能是从携带平衡重排的父母那里遗传而来的。据估计,每 500 人中就有 1 人是平衡易位携带者。不同染色体重排的结果有可能揭示部分三体或部分单体的功能后果,并有助于为平衡携带者和其他诊断出类似不平衡的年轻患者提供遗传咨询。

方法

我们对两名有发育迟缓(DD)、智力障碍(ID)和发育异常病史的同胞进行了临床表型和细胞遗传学分析。

结果

先证者,一名 38 岁女性,有身材矮小、发育异常和主动脉缩窄病史。她接受了染色体微阵列分析,发现 4q 部分单体和 10p 部分三体。她的哥哥,一名 37 岁男性,有更严重的 DD、行为问题、发育异常和先天性异常病史。随后,核型证实了这对同胞的两种不同的不平衡易位:46,XX,der(4)t(4;10)(q33;p15.1)和 46,XY,der(10)t(4;10)(q33;p15.1)。这些染色体重排代表了父母为平衡易位 46,XX,t(4;10)(q33;p15.1)携带者的两种可能结果。

结论

据我们所知,这种 4q 和 10p 易位在文献中尚未描述。在本报告中,我们比较了由于 4q 部分单体和 10p 部分三体以及 4q 部分单体和 10p 部分单体复合效应导致的临床特征。这些发现表明了新旧基因组检测的相关性、这些分离结果的可行性以及遗传咨询的必要性。

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