Division of Hematology/Oncology, Department of Pediatrics, The University of Texas Health Science Center, San Antonio, USA.
Am J Med Genet A. 2012 Oct;158A(10):2534-6. doi: 10.1002/ajmg.a.35284. Epub 2012 Aug 14.
We describe the case of a 12-year-old Hispanic male with a clinical and molecular diagnosis of Simpson-Golabi-Behmel Syndrome (SGBS) who subsequently developed metastatic medulloblastoma. While individuals with SGBS have been documented to have increased risk for intra-abdominal tumors such as Wilms tumor and neuroblastoma, medulloblastomas, or CNS tumors in general, have not been reported in patients with this syndrome. Our patient was clinically diagnosed with SGBS as an infant. He presented with many of the common features of the syndrome, such as cleft palate, macroglossia, post-axial polydactyly, "coarse" facial features, and ventricular septal defects (VSDs). Molecular testing performed in April 2009 confirmed the SGBS diagnosis. This testing detected a large intragenic deletion in the GPC3 gene (more than 500 kb, 8 exons) extending from intron 2, 37 kb downstream of exon 2, to the 5' end of the gene, deleting exons 1 and 2. However, subsequent testing by gene-centric high-density array comparative genomic hybridization (aCGH) detected a deletion encompassing only exon 2. Therefore, the exact 5' boundary of the deletion cannot currently be determined, due to an apparent complex rearrangement upstream of exon 1. We present this case of metastatic medulloblastoma as a unique malignancy in a patient with SGBS.
我们描述了一例 12 岁西班牙裔男性的病例,该患者临床和分子诊断为 Simpson-Golabi-Behmel 综合征(SGBS),随后发展为转移性髓母细胞瘤。虽然已有文献报道 SGBS 患者患腹部肿瘤(如肾母细胞瘤和神经母细胞瘤)的风险增加,但该综合征患者的髓母细胞瘤或中枢神经系统肿瘤尚未见报道。我们的患者在婴儿期被临床诊断为 SGBS。他表现出该综合征的许多常见特征,如腭裂、巨舌、后轴多指、“粗糙”的面部特征和室间隔缺损(VSD)。2009 年 4 月进行的分子检测证实了 SGBS 的诊断。该检测发现 GPC3 基因(超过 500kb,8 个外显子)内的一个大型内含子缺失,从内含子 2 开始,延伸至 2 号外显子下游 37kb 处,到基因的 5'端,缺失 1 号和 2 号外显子。然而,随后的基因靶向高密度阵列比较基因组杂交(aCGH)检测到仅包含外显子 2 的缺失。因此,由于 1 号外显子上游存在明显的复杂重排,目前无法确定缺失的精确 5'边界。我们提出了这例 SGBS 患者转移性髓母细胞瘤的独特恶性肿瘤病例。