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巨大肠系膜炎性肌纤维母细胞瘤作为肠套叠的罕见病因并在10个月后复发:病例报告及文献复习

Huge mesenteric inflammatory myofibroblastic tumor as a rare cause of intussusception with recurrence after 10 months: Case report and literature review.

作者信息

Asbah Malvina, Shrateh Oadi N, Musleh Asil, Abbadi Khaled, Amro Wael, Shaltaf Ahmad

机构信息

Department of General Surgery, Palestinian Medical Complex (PMC), Ramallah, Palestine.

Faculty of Medicine, Al-Quds University, Jerusalem, Palestine.

出版信息

Int J Surg Case Rep. 2023 Oct;111:108871. doi: 10.1016/j.ijscr.2023.108871. Epub 2023 Sep 26.

Abstract

INTRODUCTION AND IMPORTANCE

Inflammatory myofibroblastic tumor (IMT) is an infrequent, generally non-cancerous mesenchymal growth. IMT can affect individuals across various age groups, with a higher prevalence in children and adolescents. While it can emerge in any bodily region, it has a tendency to develop more often in the lungs and mesentery. IMT occurrence in the small bowel is exceptionally uncommon. It's a rare cause of intussusception and has unpredictable recurrence rate.

CASE PRESENTATION

This report highlights a unique clinical presentation involving a mesenteric IMT, which presented as small intestine intussusception in a 2-year-old child. Additionally, the patient was found to have an asymptomatic mass in the right upper quadrant, later identified as a recurrent IMT 10 months after surgical intervention for intussusception caused by the same tumor.

CLINICAL DISCUSSION

IMTs originate from mesenchymal tissues and encompass a blend of fibroinflammatory conditions. They exhibit a diverse combination of inflammatory and spindle cells. Diagnosing IMTs prior to surgery is intricate, as they can mimic malignant growths. Histopathology following surgery is usually needed for confirmation. Complete removal with a clear margin is the favored treatment approach.

CONCLUSION

Intestinal IMT is an infrequent and often overlooked condition, but it should be taken into account when diagnosing small bowel intussusception. The best chance of preventing recurrence in cases of intestinal IMT is through complete surgical removal with a negative margin. Nonetheless, the most effective approach for managing local recurrence and metastasis is still uncertain and necessitates ongoing long-term observation.

摘要

引言与重要性

炎性肌纤维母细胞瘤(IMT)是一种罕见的、通常为非癌性的间充质生长物。IMT可影响各个年龄组的个体,在儿童和青少年中更为常见。虽然它可出现在身体的任何部位,但更倾向于在肺部和肠系膜中发生。小肠发生IMT极为罕见。它是肠套叠的罕见原因,且复发率不可预测。

病例介绍

本报告重点介绍了一例独特的临床表现,涉及一名肠系膜IMT,该病例表现为一名2岁儿童的小肠肠套叠。此外,该患者右上腹发现有无症状肿块,在因同一肿瘤导致的肠套叠手术干预10个月后,该肿块被确诊为复发性IMT。

临床讨论

IMT起源于间充质组织,包含多种纤维炎性病变。它们表现出炎性细胞和梭形细胞的多种组合。术前诊断IMT很复杂,因为它们可能类似恶性肿瘤。通常需要术后组织病理学检查来确诊。以清晰切缘完整切除是首选的治疗方法。

结论

肠道IMT是一种罕见且常被忽视的疾病,但在诊断小肠肠套叠时应予以考虑。肠道IMT预防复发的最佳机会是通过完整的手术切除且切缘阴性。然而,处理局部复发和转移的最有效方法仍不确定,需要持续进行长期观察。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a551/10539857/35dabdb68c94/gr1.jpg

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