PDF(Pubmed)
Abstract：
BACKGROUND: Metastatic Crohn\'s disease is a rare disorder characterized by various granulomatous skin lesions that occur independently of gastrointestinal tract involvement. However, currently there is no standardized care or specific treatment. Therapeutic approaches include immunosuppressive agents, such as corticosteroids, azathioprine, and monoclonal antibodies targeting inflammatory cytokines like tumor necrosis factor (TNF).
METHODS: We present a case of a 29-year-old western European woman with significant blind ending abdominal subcutaneous fistulas and abscesses, who sought evaluation in the dermatology department. Histological examination revealed multiple epithelioid cell granulomas. There was no evidence of infectious or rheumatologic diseases such as sarcoidosis. The tentative diagnosis was metastatic Crohn\'s disease, which was not related to an intestinal manifestation of the disease. The patient responded to infliximab but had to discontinue it due to an allergic reaction. Subsequent adalimumab treatment failed to induce clinical remission; thus, therapy was switched to ustekinumab, resulting in a positive response. Written informed consent for publication of their clinical details and clinical images was obtained from the patient. For our study more than 1600 publications were screened for cases of metastatic Crohn\'s disease on PubMed database. 59 case reports with 171 patients were included in the analysis and evaluated for localization, diagnostic and therapeutic approaches, and complications and were summarized in this review.
CONCLUSIONS: The successful ustekinumab treatment of a patient with metastatic Crohn\'s disease underscores the potential of this minimally investigated therapeutic option, highlighting the need for future treatment guidelines given the increasing prevalence of such cases.
METHODS: We present a case of a 29-year-old western European woman with significant blind ending abdominal subcutaneous fistulas and abscesses, who sought evaluation in the dermatology department. Histological examination revealed multiple epithelioid cell granulomas. There was no evidence of infectious or rheumatologic diseases such as sarcoidosis. The tentative diagnosis was metastatic Crohn\'s disease, which was not related to an intestinal manifestation of the disease. The patient responded to infliximab but had to discontinue it due to an allergic reaction. Subsequent adalimumab treatment failed to induce clinical remission; thus, therapy was switched to ustekinumab, resulting in a positive response. Written informed consent for publication of their clinical details and clinical images was obtained from the patient. For our study more than 1600 publications were screened for cases of metastatic Crohn\'s disease on PubMed database. 59 case reports with 171 patients were included in the analysis and evaluated for localization, diagnostic and therapeutic approaches, and complications and were summarized in this review.
CONCLUSIONS: The successful ustekinumab treatment of a patient with metastatic Crohn\'s disease underscores the potential of this minimally investigated therapeutic option, highlighting the need for future treatment guidelines given the increasing prevalence of such cases.
摘要：
背景：转移性克罗恩病是一种罕见的疾病，其特征是各种肉芽肿性皮肤病变独立于胃肠道受累而发生。然而,目前没有标准化的护理或特定的治疗。治疗方法包括免疫抑制剂，比如皮质类固醇,硫唑嘌呤，和靶向炎症细胞因子如肿瘤坏死因子（TNF）的单克隆抗体。
方法：我们介绍了一例29岁的西欧妇女，其腹部皮下瘘和脓肿明显失明，他在皮肤科寻求评估。组织学检查发现多发性上皮样细胞肉芽肿。没有证据表明有传染性或风湿病，如结节病。初步诊断为转移性克罗恩病，这与疾病的肠道表现无关。患者对英夫利昔单抗有反应，但由于过敏反应而不得不停止治疗。随后的阿达木单抗治疗未能引起临床缓解；因此，治疗改用ustekinumab，产生积极的回应。从患者获得书面知情同意书，以发表其临床细节和临床图像。在我们的研究中，在PubMed数据库中筛选了1600多篇出版物中的转移性克罗恩病病例。59例病例报告，171例患者纳入分析，并进行定位评估，诊断和治疗方法，和并发症，并在这篇综述中进行总结。
结论：ustekinumab对转移性克罗恩病患者的成功治疗强调了这种最低限度的治疗选择的潜力，强调鉴于此类病例的患病率越来越高，未来需要制定治疗指南。
方法：我们介绍了一例29岁的西欧妇女，其腹部皮下瘘和脓肿明显失明，他在皮肤科寻求评估。组织学检查发现多发性上皮样细胞肉芽肿。没有证据表明有传染性或风湿病，如结节病。初步诊断为转移性克罗恩病，这与疾病的肠道表现无关。患者对英夫利昔单抗有反应，但由于过敏反应而不得不停止治疗。随后的阿达木单抗治疗未能引起临床缓解；因此，治疗改用ustekinumab，产生积极的回应。从患者获得书面知情同意书，以发表其临床细节和临床图像。在我们的研究中，在PubMed数据库中筛选了1600多篇出版物中的转移性克罗恩病病例。59例病例报告，171例患者纳入分析，并进行定位评估，诊断和治疗方法，和并发症，并在这篇综述中进行总结。
结论：ustekinumab对转移性克罗恩病患者的成功治疗强调了这种最低限度的治疗选择的潜力，强调鉴于此类病例的患病率越来越高，未来需要制定治疗指南。
