26vzmnrw135 10.3205/26vzmnrw135 urn:nbn:de:0183-26vzmnrw1356 Meeting Abstract de Angelis de Angelis C. C

Universitätsklinikum Düsseldorf, Klinik für Gastroenterologie, Hepatologie und Infektiologie, Düsseldorf, Deutschland

author Yaqubi Yaqubi K. K

Universitätsklinikum Düsseldorf, Klinik für Gastroenterologie, Hepatologie und Infektiologie, Düsseldorf, Deutschland

author Störkmann Störkmann H. H

Universitätsklinikum Düsseldorf, Klinik für Gastroenterologie, Hepatologie und Infektiologie, Düsseldorf, Deutschland

author Seidl Seidl M. M

Universitätsklinikum Düsseldorf, Institut für Pathologie, Düsseldorf, Deutschland

author Esposito Esposito I. I

Universitätsklinikum Düsseldorf, Institut für Pathologie, Düsseldorf, Deutschland

author Luedde Luedde T. T

Universitätsklinikum Düsseldorf, Klinik für Gastroenterologie, Hepatologie und Infektiologie, Düsseldorf, Deutschland

author May May P. P

Universitätsklinikum Düsseldorf, Klinik für Gastroenterologie, Hepatologie und Infektiologie, Düsseldorf, Deutschland

author German Medical Science GMS Publishing House

Düsseldorf

610 20260618 engl This is an Open Access article distributed under the terms of the Creative Commons Attribution 4.0 License. Dieser Artikel ist ein Open-Access-Artikel und steht unter den Lizenzbedingungen der Creative Commons Attribution 4.0 License (Namensnennung). M0644 135 Niederrheinisch-Westfälische Gesellschaft für Chirurgie Gesellschaft für Gastroenterologie in Nordrhein-Westfalen e.V. 192. Jahrestagung der Niederrheinisch-Westfälischen Gesellschaft für Chirurgie, 34. Jahrestagung der Gesellschaft für Gastroenterologie Viszeralmedizin NRW 2026 Gastroenterologie Dortmund 20260618 20260619 135 TextBackground and objective: Collagenous sprue is a rare and severe small intestinal enteropathy characterized by a markedly thickened subepithelial collagen band, villous atrophy, crypt hyperplasia, and intraepithelial lymphocytosis. Clinically, patients present with chronic diarrhoea, malabsorption, protein-losing enteropathy, micronutrient deficiencies, and significant weight loss, often leading to cachexia. Although frequently associated with celiac disease, collagenous sprue typically demonstrates poor or absent response to a gluten-free diet . Given its frequently refractory clinical course, therapeutic management often necessitates prolonged immunosuppressive treatment.Method: We present a case of severe collagenous sprue achieving sustained clinical and histological remission under anti–tumor necrosis factor alpha (TNF-α) therapy with infliximab.Result: A 50-year-old woman diagnosed with refractory celiac disease was referred to our in-patient treatment due to intractable watery diarrhoea and life-threatening cachexia. Duodenal biopsies revealed complete villous atrophy, crypt hyperplasia, pronounced intraepithelial lymphocytosis, and a subepithelial collagen band consistent with collagenous sprue. T-cell clonality analyses of biopsies showed no signs of mono-/oligoclonality. After nutritional stabilization and substitution of human immunoglobulin because of severe hypogammaglobulinemia as a result of dramatic enteric protein loss, therapy with infliximab was initiated, leading to marked clinical and endoscopic improvement, with eventual histological remission. After 3 years therapy was discontinued at the patient’s request, resulting in rapid clinical relapse characterized by recurrent diarrhoea and need for inpatient parenteral nutritional therapy. Subsequent endoscopic and histopathological assessment revealed disease recurrence, with subepithelial collagen band thickening up to 30.2 µm and complete villous atrophy (Marsh IIIc). Re-initiation of infliximab achieved rapid and sustained clinical and histological remission.Summary: Collagenous sprue should be considered in patients with refractory celiac disease unresponsive to a gluten-free diet. Anti–TNF-α immunomodulatory therapy with infliximab can induce rapid and sustained remission, though relapse may occur after treatment cessation, highlighting the importance of ongoing surveillance and individualized long-term immunomodulatory therapy. Rejeski J Conway J Zhou Y Collagenous Sprue 2020 Am J Med Sci 310-11 Rejeski J, Conway J, Zhou Y. Collagenous Sprue. Am J Med Sci. 2020 May;359(5):310-11. DOI: 10.1016/j.amjms.2019.12.008 https://doi.org/10.1016/j.amjms.2019.12.008 0 0 0 0