Wu Mona Kay, Goudie Catherine, Druker Harriet, Thorner Paul, Traubici Jeffrey, Grant Ronald, Albrecht Steffen, Weber Evan, Charles Adrian, Priest Jack R, Fabian Marc Robert, Foulkes William D
Department of Medical Genetics, Lady Davis Institute, Jewish General Hospital, McGill University, Montreal, Quebec, Canada.
Department of Pediatric Hematology-Oncology, McGill University Health Center, Montreal, Quebec, Canada.
Pediatr Blood Cancer. 2016 Jul;63(7):1272-5. doi: 10.1002/pbc.25959. Epub 2016 Feb 29.
Anaplastic sarcoma of kidney (ASK) is a rare neoplasm recently associated with DICER1 mutations. We report a child with germline DICER1 mutation who developed ASK in preexisting septated renal cysts, which were likely cystic nephroma. From age 2.5 to 6 years, sonographic imaging illustrated changes in the size and number of renal cysts, followed at age 8.8 years by a mass, pathologically an ASK. Lung cysts resected in infancy were diagnosed retrospectively as pleuropulmonary blastoma. Both tumors had acquired somatic DICER1 mutations. Ultrasonographic evolution of renal cysts to ASK has not previously been documented. Children with both pulmonary and renal cysts are candidates for DICER1 mutation testing.
肾间变性肉瘤(ASK)是一种罕见肿瘤,最近发现与DICER1突变有关。我们报告了一名患有种系DICER1突变的儿童,其在先前存在的分隔性肾囊肿(可能为囊性肾瘤)中发生了ASK。从2.5岁到6岁,超声成像显示肾囊肿的大小和数量发生变化,8.8岁时出现一个肿块,病理检查为ASK。婴儿期切除的肺囊肿经回顾性诊断为胸膜肺母细胞瘤。两种肿瘤均获得了体细胞DICER1突变。肾囊肿向ASK的超声演变此前未见报道。患有肺囊肿和肾囊肿的儿童是DICER1突变检测的候选对象。