Department of Pathology, University of California, San Francisco, San Francisco, CA 91343, United States.
Kaiser Permanente, Woodland Hills, CA 91367, United States.
Hum Pathol. 2018 Jul;77:166-174. doi: 10.1016/j.humpath.2018.03.026. Epub 2018 Apr 7.
Goblet cell carcinoid (GCC) is a rare appendiceal tumor with unique morphologic features that shows glandular and neuroendocrine differentiation on immunohistochemistry. An additional component of adenocarcinoma (AC) can be present (GCC-AC). Both GCC and GCC-AC are staged and treated like AC. The histogenesis and genetic alterations underlying GCC and GCC-AC are unclear. Capture-based next-generation DNA sequencing targeting 479 cancer genes was performed on 19 appendiceal tumors: 4 GCC, 9 GCC-AC, 3 neuroendocrine tumors (NET), and 3 AC (2 conventional, 1 mucinous). Somatic coding mutations were not seen in any NET. Pathogenic (P)/likely pathogenic (LP) mutations were present in 1 GCC, 8 GCC-AC and all 3 AC cases. P/LP mutations in chromatin remodeling genes were seen in 4 (44.4%) GCC-AC cases, but not in NET, GCC or AC. In GCC-AC, P/LP mutations in ARID1A and RHOA were each present in 3 cases, and KDM6A and SOX9 mutations were each seen in 2 cases. APC and KRAS mutations were present in 1 conventional AC case, but were not observed in any GCC or GCC-AC. This limited series reveals mutations in SOX9, RHOA, and chromatin-modifier genes in goblet cell tumors, and shows that the mutational profile of GCC/GCC-AC is distinct from NET and conventional appendiceal AC.
杯状细胞类癌(GCC)是一种罕见的阑尾肿瘤,具有独特的形态学特征,在免疫组织化学上显示出腺体和神经内分泌分化。腺癌(AC)的附加成分也可能存在(GCC-AC)。GCC 和 GCC-AC 均按 AC 进行分期和治疗。GCC 和 GCC-AC 的组织发生和遗传改变尚不清楚。对 19 个阑尾肿瘤进行了靶向 479 个癌症基因的基于捕获的下一代 DNA 测序:4 个 GCC、9 个 GCC-AC、3 个神经内分泌肿瘤(NET)和 3 个 AC(2 个常规,1 个黏液性)。任何 NET 中均未发现体细胞编码突变。在 1 个 GCC、8 个 GCC-AC 和所有 3 个 AC 病例中均存在致病性(P)/可能致病性(LP)突变。在 44.4%的 GCC-AC 病例中观察到染色质重塑基因中的 P/LP 突变,但在 NET、GCC 或 AC 中未观察到。在 GCC-AC 中,ARID1A 和 RHOA 中的 P/LP 突变各存在于 3 例,KDM6A 和 SOX9 突变各存在于 2 例。APC 和 KRAS 突变存在于 1 例常规 AC 病例中,但在任何 GCC 或 GCC-AC 中均未观察到。这个有限的系列揭示了杯状细胞肿瘤中 SOX9、RHOA 和染色质修饰基因的突变,并表明 GCC/GCC-AC 的突变谱与 NET 和常规阑尾 AC 不同。