Miettinen Markku, Felisiak-Golabek Anna, Wang Zengfeng, Inaguma Shingo, Lasota Jerzy
*Laboratory of Pathology, National Cancer Institute, Bethesda, MD †Aichi Medical University School of Medicine, Nagakute, Japan.
Am J Surg Pathol. 2017 May;41(5):577-585. doi: 10.1097/PAS.0000000000000807.
Most gastrointestinal stromal tumors (GISTs) occur in the tubular gastrointestinal (GI) tract, but some present apparently outside the GI tract. In this study, we analyzed 112 GISTs located in the retroperitoneum. These tumors occurred in 55 women and 57 men with a median age of 65 years (range: 21 to 89 y). On the basis of clinically or histologically detected connections to GI tract, 15 tumors were considered likely of gastric, 9 duodenal, and 13 of small intestinal origin. The remaining cases were categorized by location as peripancreatic (n=25), pelvic (n=11), mesenteric (n=4), and of unspecified/miscellaneous sites (n=35). The tumors varied in size 3 to 35 cm (median, 15 cm) and by mitotic rate per 5 mm, 0 to >100 (median, 10). Histologically the tumors apparently arising outside the GI tract had features of intestinal (n=41) and gastric GISTs (n=25); 9 cases had indeterminate histology. The histologic variants included spindled, epithelioid, vacuolated, nested, and myxoid potentially simulating other tumors such as liposarcoma and solitary fibrous tumor. Most GISTs were KIT-positive (106/112 cases), and the remaining 6 tumors were DOG1/Ano1-positive. Five cases showed focal nuclear positivity for MDM2. KIT mutations were detected in 42/59 cases, and PDGFRA mutations in 4/16 KIT wild-type and 3/5 of the KIT-negative tumors analyzed. One pelvic retroperitoneal GIST was succinate dehydrogenase deficient. All 79 patients were dead at last follow-up with a median survival of 14 months, with few survivals >5 years. Only operable versus inoperable tumor was a statistically favorable factor in univariate analysis (P<0.01). In multivariate analysis, mitotic rate >50/5 mm was significant for a shorter survival (hazard ratio, 5.25; 95% confidence interval, 1.65-16.8; P<0.01). Histologic and clinicopathologic similarity of extragastrointestinal retroperitoneal GISTs with GISTs of GI tract suggests their GI tract origin. Potentially overlapping features between GIST and other retroperitoneal tumors necessitate use of multiple diagnostic markers and molecular genetic studies.
大多数胃肠道间质瘤(GIST)发生于胃肠道(GI)管腔,但有些表现为明显位于胃肠道外。在本研究中,我们分析了112例位于腹膜后的GIST。这些肿瘤发生于55名女性和57名男性,中位年龄65岁(范围:21至89岁)。根据临床或组织学检测到的与胃肠道的联系,15例肿瘤被认为可能起源于胃,9例起源于十二指肠,13例起源于小肠。其余病例根据位置分类为胰周(n = 25)、盆腔(n = 11)、肠系膜(n = 4)以及未明确/其他部位(n = 35)。肿瘤大小从3至35 cm不等(中位值15 cm),每5 mm的有丝分裂率为0至>100(中位值10)。组织学上,明显起源于胃肠道外的肿瘤具有肠道型(n = 41)和胃型GIST(n = 25)的特征;9例组织学类型不确定。组织学变异包括梭形、上皮样、空泡状、巢状和黏液样,可能模拟其他肿瘤,如脂肪肉瘤和孤立性纤维瘤。大多数GIST为KIT阳性(106/112例),其余6例肿瘤为DOG1/Ano1阳性。5例显示MDM2局灶性核阳性。在59例中的42例检测到KIT突变,在分析的16例KIT野生型和5例KIT阴性肿瘤中的4例检测到PDGFRA突变。1例盆腔腹膜后GIST为琥珀酸脱氢酶缺陷型。在最后一次随访时,所有79例患者均死亡,中位生存期为14个月,很少有患者存活超过5年。在单因素分析中,仅可手术与不可手术的肿瘤是一个具有统计学意义的有利因素(P<0.01)。在多因素分析中,有丝分裂率>50/5 mm对生存期较短具有显著意义(风险比,5.25;95%置信区间,1.65 - 16.8;P<0.01)。胃肠道外腹膜后GIST与胃肠道GIST在组织学和临床病理上的相似性提示它们起源于胃肠道。GIST与其他腹膜后肿瘤之间可能重叠的特征需要使用多种诊断标志物和分子遗传学研究。