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一名患有视网膜母细胞瘤且存在13号染色体长臂缺失的患者的病例报告:人类13号染色体上一个新基因(LCP1基因)的定位

A case report of a patient with retinoblastoma and chromosome 13q deletion: assignment of a new gene (gene for LCP1) on human chromosome 13.

作者信息

Kondo I, Shin K, Honmura S, Nakajima H, Yamamura E, Satoh H, Terauchi M, Usuki Y, Takita H, Hamaguchi H

出版信息

Hum Genet. 1985;71(3):263-6. doi: 10.1007/BF00284588.

Abstract

Retinoblastoma (Rb) occurs in hereditary, non-hereditary, and chromosomal deletion forms and the locus for the Rb gene (Rb-1) is closely linked to the locus for esterase D (ESD) assigned to the chromosome 13q14.11. We describe a patient who was predicted to have Rb from the genetic analysis of the chromosome and ESD phenotype. Furthermore, the gene for lymphocyte cytosol polypeptide with molecular weight of 64,000 (LCP1: McKusick catalogue No. 15343, 1983) was assigned to chromosome 13 by deletion mapping. A 3-month-old female had many characteristics of chromosome 13q-syndrome, including dolichocephaly, epicanthus, ptosis, depressed nasal bridge, micrognathia, short webbed neck, and short fifth fingers with clinodactyly and single crease. The karyotype of the patient was 46,XX,del(13) (q14.1-q32), though both the parents had normal karyotypes. As expected, the phenotype of ESD derived from one of the parents, the father in this case, was not detected in peripheral blood lymphocytes by two-dimensional gel electrophoresis (two-DE), indicating that ESD from the father was deleted in the abnormal chromosome 13. The possibility of paternity was calculated to be 0.996 based on the data using 22 genetic markers. Bilateral retinoblastomas could be diagnosed by ophthalmologic examinations before the manifestation of any clinical signs of the tumor and immediately intensive care was taken. In addition, the phenotype of LCP1 derived from the father was not expressed in the lymphocyte proteins from the patient. These data indicate that the gene for LCP1 (LCP1) is located in the region q14.1-q32 of chromosome 13 and may be a useful genetic marker for preclinical diagnosis of Rb.

摘要

视网膜母细胞瘤(Rb)以遗传性、非遗传性和染色体缺失形式出现,Rb基因(Rb-1)的位点与定位于13号染色体q14.11的酯酶D(ESD)位点紧密连锁。我们描述了一名通过染色体和ESD表型的基因分析被预测患有Rb的患者。此外,通过缺失定位将分子量为64,000的淋巴细胞胞质多肽基因(LCP1:麦库西克目录编号15343,1983)定位于13号染色体。一名3个月大的女性具有13号染色体q-综合征的许多特征,包括长头畸形、内眦赘皮、上睑下垂、鼻梁凹陷、小颌畸形、短蹼颈以及第五指短并向尺侧弯曲和单一掌纹。患者的核型为46,XX,del(13)(q14.1-q32),而其父母的核型均正常。正如预期的那样,通过二维凝胶电泳(双向电泳)在外周血淋巴细胞中未检测到来自父母一方(在本病例中为父亲)的ESD表型,这表明父亲的ESD在异常的13号染色体中缺失。基于使用22个遗传标记的数据计算出亲子关系的可能性为0.996。在肿瘤的任何临床体征出现之前,通过眼科检查可诊断出双侧视网膜母细胞瘤,并立即进行了重症监护。此外,患者淋巴细胞蛋白中未表达来自父亲的LCP1表型。这些数据表明LCP1基因(LCP1)位于13号染色体的q14.1-q32区域,可能是Rb临床前诊断的有用遗传标记。

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