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系统性红斑狼疮的一种罕见表现:狼疮性肠炎伴肠道假性梗阻和侵袭性念珠菌病。

An Uncommon Manifestation of Systemic Lupus Erythematosus as Lupus Enteritis With Intestinal Pseudo-Obstruction and Invasive Candidiasis.

作者信息

Patnaik Rohit, Chawla Atul, Ramakrishnan Ajay B, Jain Nibha, Molazadeh Negin, Pillai Anbalagan, Pillai Deepak S, Remya Rajan, Parmar Hardik, Karan Nupur

机构信息

Critical Care Medicine, Medeor 24x7 Hospital, Abu Dhabi, ARE.

Gastroenterology, Medeor 24x7 Hospital, Abu Dhabi, ARE.

出版信息

Cureus. 2024 Sep 30;16(9):e70485. doi: 10.7759/cureus.70485. eCollection 2024 Sep.

DOI:10.7759/cureus.70485
PMID:39479075
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11522948/
Abstract

Systemic lupus erythematosus (SLE) is a multifaceted autoimmune disorder, occasionally presenting with rare complications like lupus enteritis (LE) and intestinal pseudo-obstruction (IPO). We present a unique case of a 32-year-old woman with LE and IPO, complicated by invasive candidiasis, as an initial manifestation of SLE. The patient presented with a 15-day history of abdominal pain, vomiting, and poor oral intake, and was initially misdiagnosed with infective enterocolitis. Examination revealed abdominal distension and absent bowel sounds due to IPO, alongside severe hypokalemia and signs of intra-abdominal hypertension (IAH), necessitating ventilator support. Subsequent workup confirmed SLE with LE and associated lupus nephritis (LN). The patient's condition was further complicated by disseminated invasive candidiasis involving multiple organs, including the bloodstream, chorioretinitis, and endocarditis. Despite her critical state, intensive multidisciplinary care, including high-dose steroids, antifungal therapy, and supportive measures, led to her recovery and discharge after a 51-day ICU stay. This case underscores the complexity of diagnosing SLE when it presents with non-specific symptoms. The concomitant occurrence of LE, IPO, and invasive candidiasis is particularly rare, highlighting the need for high clinical suspicion in the presence of SLE serological activity. The presence of invasive candidiasis was likely secondary to gut translocation due to LE-associated inflammation, a phenomenon not previously well-documented. LE can manifest as the primary and sole presentation of SLE, even in the absence of typical lupus features. Prompt immunomodulatory treatment and comprehensive care are essential for a favorable outcome. Clinicians should consider invasive candidiasis in SLE patients with acute GI involvement, particularly in the presence of LE.

摘要

系统性红斑狼疮(SLE)是一种多方面的自身免疫性疾病,偶尔会出现狼疮性肠炎(LE)和肠道假性梗阻(IPO)等罕见并发症。我们报告了一例独特的32岁女性病例,以LE和IPO为表现,并并发侵袭性念珠菌病,这是SLE的初始表现。患者有15天的腹痛、呕吐和进食减少病史,最初被误诊为感染性肠炎。检查发现由于IPO导致腹胀和肠鸣音消失,同时伴有严重低钾血症和腹腔内高压(IAH)体征,需要呼吸机支持。随后的检查确诊为SLE合并LE及相关狼疮性肾炎(LN)。患者病情因累及包括血液、脉络膜视网膜炎和心内膜炎在内的多个器官的播散性侵袭性念珠菌病而进一步复杂化。尽管患者病情危急,但包括大剂量类固醇、抗真菌治疗和支持措施在内的强化多学科护理使她在重症监护病房住院51天后康复出院。该病例强调了SLE以非特异性症状表现时诊断的复杂性。LE、IPO和侵袭性念珠菌病同时发生极为罕见,突出了在存在SLE血清学活动时需要高度临床怀疑。侵袭性念珠菌病的发生可能继发于LE相关炎症导致的肠道细菌移位,这一现象此前记录较少。LE可表现为SLE的主要和唯一表现,即使没有典型的狼疮特征。及时的免疫调节治疗和综合护理对于取得良好预后至关重要。临床医生应考虑SLE患者急性胃肠道受累时的侵袭性念珠菌病,尤其是在存在LE的情况下。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/53aaf8466cb3/cureus-0016-00000070485-i08.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/5988e99a2803/cureus-0016-00000070485-i05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/b51bd264e181/cureus-0016-00000070485-i06.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/45f784118572/cureus-0016-00000070485-i07.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/53aaf8466cb3/cureus-0016-00000070485-i08.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/89a4e74dbca2/cureus-0016-00000070485-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/945d3e5c0ebe/cureus-0016-00000070485-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/359f9cc3f494/cureus-0016-00000070485-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/b81427806c3c/cureus-0016-00000070485-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/5988e99a2803/cureus-0016-00000070485-i05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/b51bd264e181/cureus-0016-00000070485-i06.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/45f784118572/cureus-0016-00000070485-i07.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/24de/11522948/53aaf8466cb3/cureus-0016-00000070485-i08.jpg

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本文引用的文献

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Cureus. 2023 Dec 16;15(12):e50628. doi: 10.7759/cureus.50628. eCollection 2023 Dec.
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Fungal gut microbiota dysbiosis in systemic lupus erythematosus.系统性红斑狼疮中的真菌肠道微生物群失调
Front Microbiol. 2023 Apr 5;14:1149311. doi: 10.3389/fmicb.2023.1149311. eCollection 2023.
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Meta-analysis of clinical risk factors of patients with systemic lupus erythematosus complicated with invasive fungal infection.
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Medicine (Baltimore). 2023 Mar 17;102(11):e29652. doi: 10.1097/MD.0000000000029652.
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