Durieux Emeline, Descotes Françoise, Nguyen Anh-Minh, Grange Jean Daniel, Devouassoux-Shisheboran Mojgan
Department of Pathology, Hospices Civils de Lyon, Hôpital de la Croix Rousse, 69317 Lyon Cedex 04, France.
Biochimie Biologie Moléculaire, Hospices Civils de Lyon, Centre hospitalier Lyon Sud, 69495 Pierre Bénite, France.
Hum Pathol. 2015 May;46(5):783-7. doi: 10.1016/j.humpath.2015.01.020. Epub 2015 Feb 19.
Germline DICER1 gene mutation has been described in ocular medulloepithelioma associated with pleuropulmonary blastoma family tumor and dysplasia syndrome. We present a case of sporadic ocular medulloepithelioma in an 18-year-old woman with D1709N somatic mutation in DICER1 gene, which has not been previously described. This case highlights the potential use of DICER1 gene sequencing to resolve the diagnostic challenge in recurrent and metastatic malignant medulloepithelioma, when morphology and immunohistochemistry are inconclusive. Further studies in larger series of this type of tumor are needed to confirm the relevance of this molecular abnormality in the tumorigenesis of this embryonic-type ocular tumor.
种系DICER1基因突变已在与胸膜肺母细胞瘤家族性肿瘤和发育异常综合征相关的眼髓上皮瘤中被描述。我们报告了一例18岁女性散发型眼髓上皮瘤病例,该患者DICER1基因存在D1709N体细胞突变,此前尚未见相关报道。该病例突出了在形态学和免疫组织化学结果不明确时,DICER1基因测序在解决复发性和转移性恶性髓上皮瘤诊断难题方面的潜在应用价值。需要对更大系列的此类肿瘤进行进一步研究,以证实这种分子异常在这种胚胎型眼肿瘤发生中的相关性。