{"title":"A Case of Postoperative Pouch-Vaginal Fistula of Ulcerative Colitis Successfully Treated with Estriol Vaginal Tablet.","authors":"Yuhei Hashimoto, Fumikazu Koyama, Yosuke Iwasa, Tadataka Takagi, Kosuke Fujimoto, Takashi Tamura, Goki Ejiri, Chihiro Yoshikawa, Masayuki Sho","doi":"10.1159/000546361","DOIUrl":"10.1159/000546361","url":null,"abstract":"<p><strong>Introduction: </strong>Ileal pouch-vaginal fistula (PVF) is a severe complication that can occur following surgery for ulcerative colitis (UC). Most cases of PVF are managed surgically, and reports on successful closure through conservative treatment alone are limited. We report the first documented case of PVF closure without stoma formation, successfully treated with antibiotics and estriol vaginal tablets.</p><p><strong>Case presentation: </strong>A 65-year-old woman was diagnosed in her 50s with total colitis-type UC. She developed steroid-dependent, refractory disease, prompting the indication of infliximab therapy. However, infliximab failed to maintain remission, necessitating restorative proctocolectomy with ileal pouch-anal anastomosis followed by loop ileostomy. The postoperative course was uneventful, and the ileostomy was closed 6 months later. Two years after surgery, she developed fever, diarrhea, and vaginal discharge containing fecal fluid. The endoscopic evaluation identified a PVF secondary to pouchitis. She was admitted to the hospital, placed on fasting, and treated with antibiotics and estriol vaginal tablets. Endoscopy 18 days after initiating of estriol therapy revealed vaginal wall thickening and PVF closure, and she was subsequently discharged. Estriol vaginal tablet administration continued for 3 months, and no recurrence has been observed 9 years following surgery.</p><p><strong>Conclusion: </strong>Estriol vaginal tablets may be serve as an effective conservative treatment option for PVF following surgery for UC.</p>","PeriodicalId":13605,"journal":{"name":"Inflammatory Intestinal Diseases","volume":"10 1","pages":"151-154"},"PeriodicalIF":0.0,"publicationDate":"2025-05-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12173437/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144316841","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Eosinophilic Esophagitis Pathogenesis: All Clear?","authors":"Jan Hendrik Niess, Tanay Kaymak","doi":"10.1159/000546241","DOIUrl":"10.1159/000546241","url":null,"abstract":"<p><strong>Background: </strong>Eosinophilic esophagitis (EoE) is a food- and aeroallergen-driven, type 2-mediated chronic inflammation that develops in genetically predisposed individuals with an impaired esophageal epithelial barrier. How pollutants, including detergents, the esophageal microbiome, immunity, and genetics trigger the multifaceted pathophysiology of EoE is not clear.</p><p><strong>Summary: </strong>This review summarizes and discusses recent findings concerning the possible contribution of the environment/exposome, the esophageal microbiome, genetics, immunity, and epithelial barrier integrity to developing esophageal type 2 inflammation and fibrosis in EoE. After summarizing the current literature, we formulate research questions that we consider relevant to EoE.</p><p><strong>Key messages: </strong>The anticipated progress in preclinical EoE animal models, primary cell culture technologies, sequencing technologies, and clinical trials, driven by academic research and the pharmaceutical industry, is poised to revolutionize our understanding of EoE. These advancements may uncover novel pathways that can be targeted for EoE treatment, inspiring hope for improved patient quality of life.</p>","PeriodicalId":13605,"journal":{"name":"Inflammatory Intestinal Diseases","volume":"10 1","pages":"135-150"},"PeriodicalIF":0.0,"publicationDate":"2025-05-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12165646/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144301995","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Eosinophilic Esophagitis without Eosinophils: Do You Want to Mock Me?","authors":"Elena González de Béthencourt, Thomas Greuter","doi":"10.1159/000546069","DOIUrl":"10.1159/000546069","url":null,"abstract":"<p><strong>Background: </strong>Eosinophils and eosinophil infiltration are the hallmark for the diagnosis of eosinophilic esophagitis (EoE), which represents the most common cause of solid food dysphagia in young adults. However, the role of eosinophils in the pathogenesis of EoE has been increasingly questioned.</p><p><strong>Summary: </strong>It is now well accepted that EoE is a Th2-mediated disorder with a myriad of inflammatory processes being involved rather than a single cell disease. In recent years, several nuances of EoE, so-called EoE variants, have been described, among which are EoE-like esophagitis, nonspecific esophagitis, lymphocytic esophagitis, and potentially also mast-cell esophagitis. These variants appear to have distinct molecular fingerprints sharing pronounced traits of EoE. Of note, there is a considerable flux between the variants (with frequent transitions) and eventual progression to EoE over time. Thus, EoE variants and EoE appear to be a spectrum disorder, where EoE only represents the most extreme phenotype.</p><p><strong>Key messages: </strong>This review summarizes current knowledge about these different variants and discusses future directions and open questions.</p>","PeriodicalId":13605,"journal":{"name":"Inflammatory Intestinal Diseases","volume":"10 1","pages":"126-134"},"PeriodicalIF":0.0,"publicationDate":"2025-04-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12162120/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144283777","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Plasma Concentration of Calprotectin, but Not Serum Concentration, Is a Predictive Biomarker for Clinical Remission in Ulcerative Colitis.","authors":"Sailish Honap, Isabelle Aimone-Gastin, Sylvain Salignac, Justine Flayac, Justine Paoli, Laurent Peyrin-Biroulet, Abderrahim Oussalah","doi":"10.1159/000545722","DOIUrl":"10.1159/000545722","url":null,"abstract":"<p><strong>Introduction: </strong>Fecal calprotectin is a validated biomarker for assessing disease activity in patients with inflammatory bowel disease (IBD). Blood calprotectin concentrations are correlated with disease activity in numerous immune-mediated inflammatory diseases. The aim of this study was to prospectively assess the diagnostic accuracy of plasma calprotectin as a potential biomarker of remission in IBD patients.</p><p><strong>Methods: </strong>This prospective observational study enrolled 131 patients at the time of infliximab administration alongside clinical assessment and blood analyses on the same day. The primary endpoint was to assess the diagnostic accuracy of plasma calprotectin for predicting remission in patients with IBD.</p><p><strong>Results: </strong>Plasma calprotectin concentration ≤10.5 ng/mL had a sensitivity of 98.6%, specificity of 100%, positive predictive value of 100%, negative predictive value of 96.3%, and an area under the receiver operating characteristic (AUROC) curve of 0.999 for diagnosing remission in patients with ulcerative colitis (UC). Plasma calprotectin had poor diagnostic accuracy for diagnosing remission in Crohn's disease. In UC, plasma calprotectin had significantly greater diagnostic accuracy than C-reactive protein for diagnosing remission (absolute difference between AUROCs, 0.06; 95% CI: 0.008 to 0.113; <i>p</i> = 0.03). Plasma calprotectin concentrations were not correlated with those measured in serum samples. The median serum-to-plasma calprotectin concentration ratio was 12-fold.</p><p><strong>Conclusion: </strong>Plasma calprotectin is a promising biomarker for predicting remission in UC patients treated with infliximab.</p>","PeriodicalId":13605,"journal":{"name":"Inflammatory Intestinal Diseases","volume":"10 1","pages":"104-114"},"PeriodicalIF":0.0,"publicationDate":"2025-04-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12074619/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144006812","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Investigation of the Clinical Remission Course in Ulcerative Colitis from Tofacitinib Induction to Tapering or Withdrawal in Japanese Patients: A Single-Center Retrospective Study.","authors":"Miki Koroku, Teppei Omori, Shun Murasugi, Ayumi Ito, Maria Yonezawa, Shinichi Nakamura, Katsutoshi Tokushige, Yousuke Nakai","doi":"10.1159/000545704","DOIUrl":"10.1159/000545704","url":null,"abstract":"<p><strong>Introduction: </strong>Tofacitinib (TOF), a Janus kinase inhibitor, has emerged as an innovative treatment option for patients with moderate-to-severe ulcerative colitis (UC). However, the clinical course of patients who achieve induction and maintain remission followed by TOF tapering or withdrawal is unclear. We investigated the efficacy of TOF and the clinical course after TOF tapering or withdrawal in real-world clinical practice.</p><p><strong>Method: </strong>Thirty-two patients treated with TOF 20 mg/day for UC relapse between October 2018 and August 2023 were included in this single-center, retrospective observational study. Disease activity was defined by partial Mayo score (PMS), and remission was defined as PMS ≤2 and rectal bleeding score 0, other score ≤1. PMS before TOF 20 mg/day induction was compared with PMS at 8 weeks. Patients who achieved clinical remission were tapered to 10 mg/day, while those who requested for drug withdrawal were allowed. The relapse rate of the TOF 10 mg/day maintenance group and the TOF withdrawal group was compared. Both groups included patients who had maintained remission at 6 months after tapering TOF to 10 mg/day. In addition, the efficacy of TOF 20 mg/day reinduction therapy was also compared between patients who relapsed in the TOF 10 mg/day maintenance group and the TOF withdrawal group.</p><p><strong>Result: </strong>Twenty-three patients (71.9%) achieved induction of remission by 8 weeks after TOF 20 mg/day administration, with significantly lower PMS than before TOF (<i>p</i> < 0.0001). Ultimately, 27 patients (84.4%) achieved remission, 24 who achieved remission were tapered to 10 mg/day, whereas 18 were able to maintain remission for 6 months. Seven of the 18 eventually withdrew from TOF. There was no significant difference in relapse rates between the TOF 10 mg/day maintenance group (<i>n</i> = 11; follow-up, 525 [29-1,483] days) and the TOF withdrawal group (<i>n</i> = 7; follow-up, 284 [77-797] days) (5/11 [45.5%] vs. 3/7 [42.9%], log-rank test: <i>p</i> = 0.7091). All patients who received TOF 20 mg/day reintroduction therapy after relapse went into remission.</p><p><strong>Conclusion: </strong>In clinical practice, TOF 20 mg/day significantly induced induction of remission, and in patients who received 6 months of maintenance remission therapy with TOF 10 mg/day, the relapse rates between the TOF 10 mg/day maintenance group and the TOF withdrawal group were similar. After relapse, TOF 20 mg/day reintroduction therapy improved symptoms.</p>","PeriodicalId":13605,"journal":{"name":"Inflammatory Intestinal Diseases","volume":"10 1","pages":"115-124"},"PeriodicalIF":0.0,"publicationDate":"2025-04-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12083954/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144093528","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Robin Mona, Andreas Göldi, Tobias Schneider, Isabelle Panne, Andrea Egger, Jan Hendrik Niess, Petr Hrúz
{"title":"Fatigue Is Strongly Associated with Depressive Symptoms in Patients with Inflammatory Bowel Disease.","authors":"Robin Mona, Andreas Göldi, Tobias Schneider, Isabelle Panne, Andrea Egger, Jan Hendrik Niess, Petr Hrúz","doi":"10.1159/000545572","DOIUrl":"https://doi.org/10.1159/000545572","url":null,"abstract":"<p><strong>Introduction: </strong>Fatigue is an extraintestinal manifestation in patients with inflammatory bowel disease (IBD), such as Crohn's disease (CD) and ulcerative colitis (UC), with limited information on the underlying factors. This study aimed to determine the prevalence of fatigue and associated factors in IBD patients.</p><p><strong>Methods: </strong>This prospective observational study assessed 216 IBD patients treated with intravenous infliximab or vedolizumab. Clinically meaningful fatigue was defined using a visual analog scale with a score ≥4 (VAS-F, range 0-10). Further assessments included the patient health questionnaire (PHQ-8) for depressive symptoms, the IBD-control-8 questionnaire to evaluate subjective disease control and the fatigue impact scale (FIS) for patients' quality of life (QoL). Demographic, clinical and laboratory data of the study population were collected and compared to identify fatigue-associated factors.</p><p><strong>Results: </strong>Overall, 53.2% (<i>n</i> = 115) of the IBD patients reported clinically meaningful fatigue with a higher prevalence in UC (63.0%) versus CD (47.4%). Among patients with CD, disease activity was significantly associated with fatigue symptoms (<i>p</i> < 0.001), whereas no such correlation was observed in UC patients (<i>p</i> = 0.85). Clinically meaningful fatigue symptoms were reported in 90.9% of patients with depressive symptoms (PHQ-8 ≥10). Furthermore, patients with fatigue were younger (40 vs. 42 years, <i>p</i> = 0.04), reported more frequent use of concomitant psychoactive and/or sedative medication (<i>p</i> = 0.03) and had lower IBD-control-8 scores (median 12 vs. 16 points, <i>p</i> < 0.001). Only minor differences were observed when comparing serum and fecal laboratory values of patients with fatigue symptoms to those without.</p><p><strong>Conclusion: </strong>Fatigue is highly prevalent among IBD patients treated with vedolizumab or infliximab and has a substantial impact on patients' QoL. Fatigue and depressive symptoms were strongly associated, suggesting closer monitoring for depression and the use of psychoactive medication in patients with IBD.</p>","PeriodicalId":13605,"journal":{"name":"Inflammatory Intestinal Diseases","volume":"10 1","pages":"90-103"},"PeriodicalIF":0.0,"publicationDate":"2025-04-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12058115/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144013471","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Asymptomatic Wilson's Disease Diagnosed during the Course of Ulcerative Colitis: A Case Report and Review.","authors":"Megumi Kinjo, Shintaro Sagami, Akira Nogami, Kanade Serizawa, Shunsuke Shibui, Satoko Umeda, Kunio Asonuma, Hidetsugu Saito, Masaru Nakano, Toshifumi Hibi, Taku Kobayashi","doi":"10.1159/000545142","DOIUrl":"https://doi.org/10.1159/000545142","url":null,"abstract":"<p><strong>Introduction: </strong>While previous reports have suggested an association between Wilson's disease (WD) and ulcerative colitis (UC), we present the first case of asymptomatic WD diagnosed during the treatment course of UC.</p><p><strong>Case presentation: </strong>A 14-year-old male receiving treatment for UC developed elevated liver enzymes without any related symptoms. After ruling out drug-induced liver toxicity and other possible causes of hepatitis, further investigation was initiated due to his sister's subsequent diagnosis of WD. Tests revealed low serum ceruloplasmin and ATP7B gene variants, confirming WD. Following zinc therapy, liver enzymes have been normalized, and his previously refractory UC became under control.</p><p><strong>Conclusion: </strong>This case raises important questions about potential pathophysiological interactions between the two diseases.</p>","PeriodicalId":13605,"journal":{"name":"Inflammatory Intestinal Diseases","volume":"10 1","pages":"85-89"},"PeriodicalIF":0.0,"publicationDate":"2025-03-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12021379/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144013466","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Diagnostic Process and Applied Criteria for Crohn's Disease in Patients Presenting with Perianal Lesions in Japan: A Retrospective Observational Multicenter Cohort Study.","authors":"Naoto Saigusa, Naoki Hotta, Jun-Ichi Saigusa","doi":"10.1159/000545081","DOIUrl":"10.1159/000545081","url":null,"abstract":"<p><strong>Introduction: </strong>In Japan, the confirmed diagnosis of Crohn's disease (CD) is based on a single, historically established set of clinical criteria. However, for patients who present with a perianal lesion (PL), the diagnostic pattern actually applied is unclear.</p><p><strong>Methods: </strong>We conducted a retrospective observational multicenter study among patients who presented with a PL without synchronous abdominal symptoms and were subsequently diagnosed with confirmed or probable CD according to the Japanese diagnostic criteria from May 1996 to April 2024. In total, 100 patients with confirmed CD and 10 with probable CD were identified and enrolled.</p><p><strong>Results: </strong>Among the 100 patients with confirmed CD, 72% met the criterion for the category \"confirmed 1: main finding <i>A</i> (longitudinal ulcer) or <i>B</i> (cobblestone appearance).\" In the same cohort, 35% met the criterion for the category \"confirmed 2: main finding <i>C</i> (non-caseating epithelioid cell granuloma [NCEG]) with secondary finding <i>a</i> (extensive irregular-to-round ulcers or aphthae in the gastrointestinal tract) or <i>b</i> (characteristic anorectal lesions),\" including 24% without the main finding <i>A</i> or <i>B</i>. Finally, 4% met the criterion for the category \"confirmed 3: all secondary findings <i>a</i>, <i>b</i>, and <i>c</i> (characteristic gastric and duodenal lesions).\" All 10 patients with probable CD were diagnosed based on secondary finding <i>b</i> only or secondary findings <i>a</i> and <i>b</i>.</p><p><strong>Conclusion: </strong>In cases of suspected CD due to initial PLs, histological investigation of NCEG and precise total gastrointestinal inspection should be conducted to confirm the diagnosis.</p>","PeriodicalId":13605,"journal":{"name":"Inflammatory Intestinal Diseases","volume":"10 1","pages":"76-84"},"PeriodicalIF":0.0,"publicationDate":"2025-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11999661/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143978832","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Development of a Burden Scale for Colonoscopy Experienced by Patients with Inflammatory Bowel Disease.","authors":"Maya Nunotani, Miho Takahashi, Takuro Miyazaki","doi":"10.1159/000543686","DOIUrl":"https://doi.org/10.1159/000543686","url":null,"abstract":"<p><strong>Introduction: </strong>This study aimed to develop and validate a burden scale for colonoscopy-specific experiences among patients with inflammatory bowel disease (IBD) and to assess its reliability and validity.</p><p><strong>Methods: </strong>Building upon previous research on patient experiences and perceptions of colonoscopy, a 33-item pain scale was developed. Content validity was assessed to refine the questionnaire. An online survey was conducted through an IBD patient community. The reliability of the scale was evaluated using Cronbach's α coefficient and test-retest reliability. Validity was examined through factor analysis to assess construct validity and correlation coefficients with external criteria for criterion-related validity.</p><p><strong>Results: </strong>Of the 371 distributed questionnaires, 176 were returned, and data from 173 participants were included in the analysis. Item analysis and exploratory factor analysis yielded a 21-item scale with four distinct factors: pain during colonoscopy, burden with bowel preparation, anxiety and symptoms after colonoscopy, and difficulty in taking time off to receive colonoscopy. The scale demonstrated strong internal consistency (Cronbach's α = 0.875) and test-retest reliability (intraclass correlation coefficient = 0.879). Criterion-related validity was supported by correlations with external measures, including the cognitive appraisal rating scale (<i>r</i> = 0.615), anxiety related to colonoscopy (<i>r</i> = 0.582), pain during colonoscopy (<i>r</i> = 0.544), and satisfaction with colonoscopy (<i>r</i> = -0.333).</p><p><strong>Conclusion: </strong>The newly developed burden scale for colonoscopy in patients with IBD demonstrated robust reliability and validity, indicating its potential utility as a clinical instrument for assessing the burden in this patient population.</p>","PeriodicalId":13605,"journal":{"name":"Inflammatory Intestinal Diseases","volume":"10 1","pages":"61-75"},"PeriodicalIF":0.0,"publicationDate":"2025-02-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11908815/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143648439","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}