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在一名携带迄今已知最小微缺失的患者中明确临床可识别的17q22连续基因缺失综合征。

Delineation of the clinically recognizable 17q22 contiguous gene deletion syndrome in a patient carrying the smallest microdeletion known to date.

作者信息

Martínez-Fernández María Luisa, Fernández-Toral Joaquin, Llano-Rivas Isabel, Bermejo-Sánchez Eva, MacDonald Alexandra, Martínez-Frías María Luisa

机构信息

Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Instituto de Salud Carlos III, Ministerio de Economía y Competitividad, Madrid, Spain.

Centro de Investigación sobre Anomalías Congénitas (CIAC), Estudio Colaborativo Español de Malformaciones Congénitas (ECEMC), Instituto de Salud Carlos III, Ministerio de Economía y Competitividad, Madrid, Spain.

出版信息

Am J Med Genet A. 2015 Sep;167A(9):2034-41. doi: 10.1002/ajmg.a.37117. Epub 2015 Apr 21.

Abstract

We describe a patient with a 1.34 Mb microdeletion at chromosome band 17q22, which is also present in his affected mother. To better delineate this microdeletion syndrome, we compare the clinical and molecular characteristics of 10 previously reported cases and our patient. Of these, the present patient has the smallest deletion which includes five genes: MMD, TMEM100, PCTP, ANKFN1, and NOG. We compare the clinical manifestations described in relation to NOG, since this is the only gene whose loss is shared by our patient and the other eight patients. Previously, the clinical patterns associated with NOG mutations have been included under the general term "NOG-related symphalangism spectrum disorder (NOG-SSD)." Based on our analyses, and considering that there is a clinical correlation observed in cases with a "17q22 microdeletion including NOG" of which the main characteristics can be contributed to loss of this gene, we propose that the clinical patterns observed in these patients should be named as NOG-spectrum disorder-contiguous gene syndrome (NOGSD-CGS). This designation is important for clinicians because when a patient has defects concordant with alterations of NOG but also presents other anomalies not related to this gene, they would be able to suspect the existence of a microdeletion affecting 17q22, therefore, allowing an early diagnosis. This will also enable the clinician to provide the family with adequate information about the prognosis and the risk of reoccurrence in future potential offspring.

摘要

我们描述了一名患者,其17号染色体q22带存在1.34 Mb的微缺失,其患病母亲也有该微缺失。为了更好地描述这种微缺失综合征,我们比较了10例先前报道的病例和我们这位患者的临床及分子特征。其中,本患者的缺失最小,包含五个基因:MMD、TMEM100、PCTP、ANKFN1和NOG。我们比较了与NOG相关的临床表现,因为这是我们的患者和其他八名患者共有的唯一缺失基因。此前,与NOG突变相关的临床模式被统称为“NOG相关的指关节融合谱系障碍(NOG-SSD)”。基于我们的分析,并考虑到在“包含NOG的17q22微缺失”病例中观察到临床相关性,其主要特征可能归因于该基因的缺失,我们建议将这些患者中观察到的临床模式命名为NOG谱系障碍-邻接基因综合征(NOGSD-CGS)。这一命名对临床医生很重要,因为当患者出现与NOG改变一致的缺陷,但也存在与该基因无关的其他异常时,他们将能够怀疑存在影响17q22的微缺失,从而实现早期诊断。这也将使临床医生能够为家庭提供有关预后以及未来潜在后代复发风险的充分信息。

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