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穿孔性梅克尔憩室中的胃肠道间质瘤:一例报告及文献复习

Gastrointestinal stromal tumor in perforated Meckel's diverticulum: a case report and literature review.

作者信息

Hashizume Naoki, Sakamoto Saki, Fukahori Suguru, Ishii Shinji, Saikusa Nobuyuki, Koga Yoshinori, Higashidate Naruki, Tsuruhisa Shiori, Nakahara Hirotomo, Tanaka Yoshiaki, Yagi Minoru

机构信息

Department of Pediatric Surgery, Kurume University School of Medicine, 67 Asahimachi, Kurume, Fukuoka, 830-0011, Japan.

Division of Medical Safety Management, Kurume University Hospital, 67 Asahimachi, Kurume, 830-0011, Fukuoka, Japan.

出版信息

Surg Case Rep. 2020 Oct 7;6(1):265. doi: 10.1186/s40792-020-01038-x.

Abstract

INTRODUCTION

Gastrointestinal stromal tumor (GIST) is rare neoplasms of the gastrointestinal tract associated with high rates of malignant transformation. GIST has been found largely in the stomach, small bowel, colon and rectum, and esophagus, but about 5% are found in other locations. We herein report a 56-year-old woman with a GIST in perforated Meckel's diverticulum. After encountering this patient, a review of the literature found reports of 18 similar patients.

CASE PRESENTATION

A 56-year-old woman diagnosed with galactosialidosis (β-galactosidase-neuraminidase deficiency) presented with vomiting. On contrast-enhanced computed tomography, peritonitis due to perforation of the intestine was diagnosed based on the free air and dilated loop of the small bowel. Laparotomy revealed perforation of Meckel's diverticulitis located 50 cm from the ileocecal valve. Partial resection of the ileum, including the diverticulum, and end-to-end anastomosis of the small intestine were performed. Regarding the pathological findings, the edge of the diverticulum wall consisted of a solid mass measuring 1.0 cm in size, and the tumor cells were spindle-shaped with 1 mitosis present per 50 high-power fields. The diagnosis was established as GIST of the Meckel's diverticulum. The postoperative period was uneventful. Follow-up at two years revealed no evidence of recurrence.

CONCLUSION

GIST in perforated Meckel's diverticulum is very rare. The potential for the coexistence of GIST or other tumor should be considered in the treatment of perforated Meckel's diverticulum.

摘要

引言

胃肠道间质瘤(GIST)是胃肠道罕见的肿瘤,恶性转化率高。GIST主要见于胃、小肠、结肠、直肠和食管,但约5%见于其他部位。我们在此报告一名56岁女性,其梅克尔憩室穿孔处患有GIST。遇到该患者后,查阅文献发现有18例类似患者的报告。

病例介绍

一名56岁被诊断为半乳糖唾液酸贮积症(β-半乳糖苷酶-神经氨酸酶缺乏症)的女性出现呕吐症状。在增强计算机断层扫描中,根据游离气体和小肠扩张袢诊断为肠道穿孔引起的腹膜炎。剖腹探查发现距回盲瓣50 cm处的梅克尔憩室炎穿孔。进行了包括憩室在内的部分回肠切除术和小肠端端吻合术。关于病理结果,憩室壁边缘有一个大小为1.0 cm的实性肿块,肿瘤细胞呈梭形,每50个高倍视野有1个有丝分裂象。诊断为梅克尔憩室的GIST。术后恢复顺利。两年后的随访未发现复发迹象。

结论

梅克尔憩室穿孔处的GIST非常罕见。在治疗梅克尔憩室穿孔时应考虑GIST或其他肿瘤共存的可能性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4ec4/7541806/f58c417c8c7b/40792_2020_1038_Fig1_HTML.jpg

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