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先天性阑尾重复畸形合并阑尾-乙状结肠瘘1例罕见病例报告

A rare case of congenital appendiceal duplication complicated by an appendiceal-sigmoid fistula: A case report.

作者信息

Belay Suleiman Ayalew, Negussie Michael A, Kassie Melaku Tessema, Abdulsemed Yishak, Assefa Filimon Getaneh, Ebrahim Fuad Seid

机构信息

School of Medicine, College of Medicine and Health Sciences, University of Gondar, Gondar, Ethiopia.

School of Medicine, College of Health Sciences, Addis Ababa University, Addis Ababa, Ethiopia.

出版信息

Int J Surg Case Rep. 2025 Jun;131:111429. doi: 10.1016/j.ijscr.2025.111429. Epub 2025 May 13.

DOI:10.1016/j.ijscr.2025.111429
PMID:40367792
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12142545/
Abstract

INTRODUCTION

Duplication of the appendix is a rare congenital anomaly, occurring in approximately 0.004 % to 0.009 % of the population. In contrast, appendiceal-sigmoid fistulas are uncommon acquired conditions, usually associated with chronic inflammation or neoplastic processes. To our knowledge, the coexistence of these two entities has not been previously reported.

CASE PRESENTATION

A 26-year-old female presented with a one-month history of right lower quadrant abdominal pain, loss of appetite, and low-grade fever. Imaging findings initially suggested chronic appendicitis. During exploratory laparotomy, a Type A appendiceal duplication was discovered, with one of the appendices forming a fistulous connection to the sigmoid colon. Surgical treatment involved appendectomy, excision of the fistula, and primary repair of the colonic defect. Histopathological examination confirmed the diagnosis of duplicated appendix with chronic inflammatory changes. The patient had an uneventful postoperative recovery.

DISCUSSION

Appendiceal duplication typically remains an incidental finding, rarely causing clinical symptoms. The coexistence of appendiceal duplication and sigmoid fistula formation haven't been previously reported. Diagnosis can be challenging due to the limitations of preoperative imaging in identifying these anomalies. Therefore, surgical exploration remains crucial, particularly in symptomatic patients, to manage symptoms and prevent potential complications.

CONCLUSION

Clinicians should maintain a high index of suspicion for rare congenital anomalies like appendiceal duplication in patients who present with atypical symptoms of appendicitis.

摘要

引言

阑尾重复是一种罕见的先天性异常,在大约0.004%至0.009%的人群中发生。相比之下,阑尾-乙状结肠瘘是一种不常见的后天性疾病,通常与慢性炎症或肿瘤性病变相关。据我们所知,这两种情况的共存此前尚未见报道。

病例报告

一名26岁女性,有右下腹痛、食欲不振和低热1个月的病史。影像学检查结果最初提示慢性阑尾炎。在探查性剖腹手术中,发现了A型阑尾重复,其中一个阑尾与乙状结肠形成了瘘管连接。手术治疗包括阑尾切除术、瘘管切除术和结肠缺损的一期修复。组织病理学检查证实了重复阑尾伴有慢性炎症改变的诊断。患者术后恢复顺利。

讨论

阑尾重复通常为偶然发现,很少引起临床症状。阑尾重复与乙状结肠瘘形成的共存此前尚未见报道。由于术前影像学检查在识别这些异常方面存在局限性,诊断可能具有挑战性。因此,手术探查仍然至关重要,特别是对于有症状的患者,以控制症状并预防潜在并发症。

结论

对于出现阑尾炎非典型症状的患者,临床医生应高度怀疑阑尾重复等罕见先天性异常。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/07f3/12142545/55147e675455/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/07f3/12142545/4903123f7fff/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/07f3/12142545/55147e675455/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/07f3/12142545/4903123f7fff/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/07f3/12142545/55147e675455/gr2.jpg

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本文引用的文献

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A rare case report of an appendico-sigmoid fistula complicating untreated acute appendicitis: Unraveling complexity in diagnosis and treatment.一例未治疗的急性阑尾炎并发阑尾-乙状结肠瘘的罕见病例报告:解析诊断与治疗中的复杂性
Int J Surg Case Rep. 2024 Mar;116:109298. doi: 10.1016/j.ijscr.2024.109298. Epub 2024 Jan 26.
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Appendiceal duplication an unusual cause of abdominal pain: A case report.阑尾重复畸形:腹痛的罕见病因——病例报告
Clin Case Rep. 2023 Nov 30;11(12):e8279. doi: 10.1002/ccr3.8279. eCollection 2023 Dec.
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