一名年轻的克罗恩病患者同时患有口腔丘疹性荨麻疹和单纯疱疹病毒感染:抗 TPN alpha 和免疫调节剂治疗期间口腔病变的罕见病例报告。

IF 2.5 3区 医学 Q2 GASTROENTEROLOGY & HEPATOLOGY
Danielle Nobre Lopes, Noêmia Pereira de Oliveira, Karla Cristina de Campos Augusto, Adrianna Milagres, Ana Luiza Miguez, Arley Silva Junior, Danielle Castex Conde, Karin Soares Cunha, Márcia Henriques Magalhães, Rafaela Elvira Rozza-de-Menezes
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引用次数: 0

摘要

背景:寻常性丘疹性荨麻疹(Pemphigus vulgaris,PV)是一种可能危及生命的粘膜皮肤自身免疫性疾病,它会影响去甲斑鸠蛋白-1(desmoglein-1)和去甲斑鸠蛋白-3(desmoglein-3),导致上皮内囊泡状病变。在口腔粘膜中,丘疹性荨麻疹病变可与其他疾病相似,如粘膜丘疹性荨麻疹、其他形式的丘疹性荨麻疹、复发性阿弗他口腔炎、多形性红斑、史蒂文斯-约翰逊综合征(Stevens-Johnson Syndrome)以及单纯疱疹病毒(HSV)等病毒引起的溃疡,这给诊断带来了挑战。丘疹性荨麻疹与克罗恩病并发的情况很少见,主要见于年轻患者。皮损和克罗恩病的主要治疗方法通常是全身使用皮质类固醇激素,并结合使用免疫抑制剂和免疫生物学药物。文献表明,使用这些药物(尤其是 TNF-α 抑制剂)治疗克罗恩病等自身免疫性疾病有可能诱发其他自身免疫性疾病,即自身免疫样综合征,其中包括狼疮样综合征和炎症性神经病。接受英夫利昔单抗治疗的克罗恩病患者出现皮损的文献报道很少:病例报告:一名患有严重克罗恩病的年轻女性在接受 TNF-α 抑制剂英夫利昔单抗治疗后,出现了易碎的假膜性口腔溃疡。组织病理学和免疫荧光分析证实这些溃疡为假膜溃疡。治疗包括丙酸氯倍他索和低水平光生物调节,结果部分好转。后来,患者出现了严重的肠道出血,需要静脉注射氢化可的松,这改善了她的全身状况和口腔溃疡。几周后,又发现了由疱疹病毒和念珠菌病引起的新溃疡,患者接受了口服阿昔洛韦治疗、为期21天的口服奈司他丁冲洗疗法和光动力疗法,最终治愈了口腔感染。为了控制病情,胃肠病专家在她的治疗方案中加入了甲氨蝶呤(25 毫克),以降低英夫利昔单抗的免疫原性,并尽量减少皮质类固醇的使用,因为患者的克罗恩病已得到缓解,口腔 PV 病变也得到了控制:结论:年轻的克罗恩病患者应转诊至口腔内科专科医生进行合并症调查,因为在免疫抑制治疗期间可能会出现口腔PV和机会性感染。应仔细评估TNF-α抑制剂对克罗恩病等炎症性肠病患者的潜在副作用,包括口腔PV。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Co-occurrence of oral pemphigus vulgaris and herpes simplex virus infection in a young patient with Crohn's disease: report of a rare case of oral lesions during anti-TFN alpha and immunomodulator therapy.

Co-occurrence of oral pemphigus vulgaris and herpes simplex virus infection in a young patient with Crohn's disease: report of a rare case of oral lesions during anti-TFN alpha and immunomodulator therapy.

Background: Pemphigus vulgaris (PV) is a potentially life-threatening mucocutaneous autoimmune disease that affects desmoglein-1 and desmoglein-3, leading to intraepithelial vesiculobullous lesions. In the oral mucosa, PV lesions can mimic other diseases such as mucous membrane pemphigoid, other forms of pemphigus, recurrent aphthous stomatitis, erythema multiforme, Stevens-Johnson syndrome, and virus-induced ulcers like herpes simplex virus (HSV), making diagnosis challenging. The co-occurrence of PV with Crohn's disease is rare and predominantly seen in younger patients. The therapeutic mainstay for both PV and Crohn's disease usually involves systemic corticosteroids combined with immunosuppressants and immunobiological drugs. Literature indicates that the use of these drugs, particularly TNF-alpha inhibitors, for managing autoimmune diseases like Crohn's can potentially induce other autoimmune diseases known as autoimmune-like syndromes, which include episodes of lupus-like syndrome and inflammatory neuropathies. There are few cases in the literature reporting the development of PV in individuals with CD undergoing infliximab therapy.

Case report: A young female with severe Crohn's disease, treated with the TNF-alpha inhibitor infliximab, developed friable pseudomembranous oral ulcerations. Histopathological and immunofluorescence analyses confirmed these as PV. The treatment included clobetasol propionate and low-level photobiomodulation, which resulted in partial improvement. The patient later experienced severe intestinal bleeding, requiring intravenous hydrocortisone therapy, which improved both her systemic condition and oral lesions. Weeks later, new ulcerations caused by herpes virus and candidiasis were identified, leading to treatment with oral acyclovir, a 21-day regimen of oral nystatin rinse, and photodynamic therapy, ultimately healing the oral infections. To manage her condition, the gastroenterologists included methotrexate (25 mg) in her regimen to reduce the immunogenicity of infliximab and minimize corticosteroid use, as the patient was in remission for Crohn's disease, and the oral PV lesions were under control.

Conclusion: Young patients with Crohn's disease should be referred to an oral medicine specialist for comorbidity investigation, as oral PV and opportunistic infections can arise during immunosuppressive therapy. The use of TNF-alpha inhibitors in patients treated for inflammatory bowel disease, such as Crohn's, should be carefully evaluated for potential side effects, including oral PV.

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来源期刊
CiteScore
4.90
自引率
3.60%
发文量
206
审稿时长
3-8 weeks
期刊介绍: The International Journal of Colorectal Disease, Clinical and Molecular Gastroenterology and Surgery aims to publish novel and state-of-the-art papers which deal with the physiology and pathophysiology of diseases involving the entire gastrointestinal tract. In addition to original research articles, the following categories will be included: reviews (usually commissioned but may also be submitted), case reports, letters to the editor, and protocols on clinical studies. The journal offers its readers an interdisciplinary forum for clinical science and molecular research related to gastrointestinal disease.
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