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解开谜团:解决一名患有扭转J形储袋的溃疡性结肠炎患者克罗恩病的误诊——视频案例

Untwisting the plot: Resolving a misdiagnosis of Crohn's disease in an ulcerative colitis patient with a twisted J-pouch-A video vignette.

作者信息

Erozkan Kamil, Khan Imran, Almadi Faris S, Holubar Stefan D, Steele Scott R, Hull Tracy L, Lavryk Olga

机构信息

Department of Colorectal Surgery, Digestive Disease and Surgery Institute, Cleveland Clinic, Cleveland, Ohio, USA.

出版信息

Colorectal Dis. 2025 Jun;27(6):e70117. doi: 10.1111/codi.70117.

DOI:10.1111/codi.70117
PMID:40462349
Abstract

BACKGROUND

Crohn's disease (CD) and ulcerative colitis (UC) are two primary forms of inflammatory bowel disease, both characterized by chronic, relapsing inflammation of the gastrointestinal tract. Clinicians differentiate between CD and UC using a combination of clinical, endoscopic and histological findings. However, there are instances where the initial pathology is indeterminate, or a diagnosis of CD or UC may change over time. Patients with UC are often considered candidates for proctectomy with ileal pouch-anal anastomosis (IPAA). Despite this, UC patients undergoing IPAA are still at risk of pouch failure. Additionally, pouch twisting can mimic symptoms of CD, further complicating the diagnosis. In such cases, detailed clinical examination and advanced imaging are essential for accurate diagnosis. This video presents a case of a UC patient who initially underwent proctocolectomy with IPAA and was later treated for what was believed to be misdiagnosed CD, which was, in fact, a twisted pouch requiring a redo IPAA.

INTERVENTION

We present the case of a 48-year-old woman with a history of UC who underwent a robotic total proctocolectomy with ileoanal J-pouch creation followed by diverting loop ileostomy closure in 2019. The patient later experienced abdominal pain, an inability to empty the J-pouch, and food avoidance shortly after the operation, initially suspected to be due to misdiagnosed CD and pouch failure. Various treatments, including biological and immunosuppressive treatments, were used in the patient's treatment. In January 2023, the patient underwent ileostomy creation and was referred to our centre. Flexible sigmoidoscopy and pouchography revealed mucosal congestion consistent with chronic ischaemia and a partial obstruction in the prepouch ileum. During laparotomy, a twisted pouch in the efferent limb was confirmed. Additionally, severe fibrosis was noted in the existing J-pouch, extending from the rectal cuff downward around the vagina, suggestive of either chronic subclinical leakage or pouch dysfunction associated with an elongated anterior rectal cuff. Based on these findings, we proceeded with pouch excision and redo J-pouch reconstruction with diverting loop ileostomy. The procedural details are described in Video 1.

OUTCOME

The surgical procedure was completed in 316 min with an estimated blood loss of 150 mL. No intraoperative complications were observed. The patient was discharged without postoperative complications. The diverting loop ileostomy was successfully reversed after 3 months. At follow-up, the patient had gained 10 kg and was tolerating a regular diet. No biological therapy was initiated.

CONCLUSION

This case highlights the critical importance of thorough diagnostic evaluation in patients with suspected pouch complications. Misdiagnosis can lead to unnecessary and ineffective interventions. Timely recognition and correction of the twisted pouch is crucial to improve symptoms and overall outcome in patients with twisted pouch.

摘要

背景

克罗恩病(CD)和溃疡性结肠炎(UC)是炎症性肠病的两种主要形式,均以胃肠道的慢性复发性炎症为特征。临床医生通过综合临床、内镜和组织学检查结果来区分CD和UC。然而,在某些情况下,初始病理结果不明确,或者CD或UC的诊断可能随时间而改变。UC患者通常被认为是行直肠切除回肠储袋肛管吻合术(IPAA)的候选者。尽管如此,接受IPAA的UC患者仍有储袋功能衰竭的风险。此外,储袋扭转可模拟CD的症状,使诊断更加复杂。在这种情况下,详细的临床检查和先进的影像学检查对于准确诊断至关重要。本视频展示了一例UC患者的病例,该患者最初接受了IPAA直肠结肠切除术,后来因被认为是误诊的CD而接受治疗,而实际上是一个扭转的储袋,需要再次进行IPAA手术。

干预措施

我们介绍了一例48岁有UC病史的女性病例,她于2019年接受了机器人辅助全直肠结肠切除术并创建了回肠J形储袋,随后进行了转流性袢式回肠造口关闭术。该患者术后不久出现腹痛、无法排空J形储袋以及避免进食,最初怀疑是由于误诊的CD和储袋功能衰竭所致。患者接受了包括生物治疗和免疫抑制治疗在内的各种治疗。2023年1月,该患者接受了回肠造口术,并被转诊至我们中心。乙状结肠镜检查和储袋造影显示黏膜充血,符合慢性缺血表现,并在储袋前回肠存在部分梗阻。在剖腹手术中,证实了传出袢中的储袋扭转。此外,在现有的J形储袋中发现严重纤维化,从直肠袖口向下延伸至阴道周围,提示慢性亚临床渗漏或与延长的直肠前袖口相关的储袋功能障碍。基于这些发现,我们进行了储袋切除并再次进行J形储袋重建及转流性袢式回肠造口术。手术细节见视频1。

结果

手术过程耗时316分钟,估计失血量为150毫升。未观察到术中并发症。患者出院时无术后并发症。转流性袢式回肠造口在3个月后成功还纳。随访时,患者体重增加了10公斤,能耐受正常饮食。未开始生物治疗。

结论

本病例突出了对疑似储袋并发症患者进行全面诊断评估的至关重要性。误诊可能导致不必要且无效的干预。及时识别并纠正扭转的储袋对于改善扭转储袋患者的症状和总体结局至关重要。

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