Buryk Melissa A, Picarsic Jennifer L, Creary Susan E, Shaw Peter H, Simons Jeffrey P, Deutsch Melvin, Monaco Sara E, Nikiforov Yuri E, Witchel Selma Feldman
1 Division of Endocrinology, Department of Pediatrics, Children's Hospital of Pittsburgh, UPMC, 4401 Penn Ave, Pittsburgh, PA, USA.
2 Division of Pediatric Pathology, Department of Pathology, University of Pittsburgh School of Medicine, 4401 Penn Ave, Pittsburgh, PA, USA.
Pediatr Dev Pathol. 2015 Jul-Aug;18(4):318-23. doi: 10.2350/15-01-1597-CR.1. Epub 2015 Mar 9.
Papillary thyroid carcinoma (PTC) is rare in children, although it is a known secondary malignancy after treatment for neuroblastoma (NB). The interval between NB treatment completion and PTC is usually more than 5 years. A 4-year-old, female patient with a high risk adrenal NB was found to have a 2.9-cm, right thyroid nodule on surveillance chest computed tomography (CT) 6 months after completion of her NB treatment (induction chemotherapy, tumor resection, autologous stem cell transplantation, external beam radiation to the abdominal tumor site, immunotherapy, and retinoic acid). Posttreatment surveillance included iodine-123-metaiodobenzylguanidine scans and CT scans. Fine-needle aspiration of the thyroid nodule diagnosed a follicular neoplasm, which was negative for BRAF, NRAS, KRAS, HRAS, PAX8/PPARg, and RET/PTC mutations, without evidence of metastatic NB. Nodule histology demonstrated an encapsulated follicular variant of PTC (FVPTC). Next-generation sequence analysis for a 46 cancer-gene profile was performed on both tumors with subsequent peripheral blood DNA testing. A heterozygous missense mutation in STK11 (F354L) was identified in both the NB and FVPTC. This mutation was also detected in peripheral blood mononuclear cells. Two additional heterozygous somatic missense mutations of uncertain significance were identified: KDR/VEGF receptor 2 (Q472H) on chromosome 4 and MET (N375S) on chromosome 7. To our knowledge, this is the shortest reported duration from completion of NB treatment to detection of thyroid cancer. The association of the STK11 gene with Peutz-Jeghers syndrome, lung adenocarcinomas, and medullary thyroid cancer leads to a possible association between this genetic variant and our patient's tumors.
甲状腺乳头状癌(PTC)在儿童中较为罕见,尽管它是神经母细胞瘤(NB)治疗后已知的继发性恶性肿瘤。NB治疗完成与PTC之间的间隔通常超过5年。一名4岁女性高危肾上腺NB患者在完成NB治疗(诱导化疗、肿瘤切除、自体干细胞移植、腹部肿瘤部位外照射、免疫治疗和维甲酸)6个月后的胸部计算机断层扫描(CT)监测中发现右甲状腺有一个2.9厘米的结节。治疗后的监测包括碘-123-间碘苄胍扫描和CT扫描。甲状腺结节细针穿刺诊断为滤泡性肿瘤,BRAF、NRAS、KRAS、HRAS、PAX8/PPARg和RET/PTC突变均为阴性,无NB转移证据。结节组织学显示为PTC的包膜性滤泡变体(FVPTC)。对两个肿瘤以及随后的外周血DNA进行了46个癌症基因谱的下一代测序分析。在NB和FVPTC中均鉴定出STK11(F354L)的杂合错义突变。在外周血单个核细胞中也检测到了该突变。还鉴定出另外两个意义不确定的杂合体细胞错义突变:4号染色体上的KDR/VEGF受体2(Q472H)和7号染色体上的MET(N375S)。据我们所知,这是报道的从NB治疗完成到检测出甲状腺癌最短的持续时间。STK11基因与黑斑息肉综合征、肺腺癌和甲状腺髓样癌的关联导致这种基因变异与我们患者的肿瘤之间可能存在关联。