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Late stenosis of the small intestine and colon, an atypical feature of chronic graft-versus-host disease. National retrospective study in French pediatric allograft centers

Bone Marrow Transplantation volume 59, pages 1316–1319 (2024)Cite this article

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While esophageal stenosis is pathognomonic of chronic gastro intestinal graft versus host disease (GI cGVHD), little is known about small intestine and colonic strictures following hematopoietic stem cell transplantation (HSCT) [1, 2]. Unlike chronic inflammatory bowel diseases strictures that result from transmural inflammation leading to excessive fibrosis via smooth muscle cells and fibroblasts activation [3], pathophysiology and treatment algorithm of HSCT associated intestine stenosis are unclear. Less than 30 adult and pediatric cases have been reported, most with severe acute or chronic GI GVHD [4,5,6,7,8,9,10]. Severe inflammation as well as fibrotic evolution of cGVHD may play a central role in these stenosis. Other factors (CMV colitis, EBV lymphproliferation, drug toxicity, etc.) could interact during this phenomenon. Most frequent treatment was surgery. Whether GVHD therapy can improve this condition is unclear. To address these issues, we conducted a multicenter retrospective study in French pediatric HSCT units.

Thirteen patients (Supplementary Table 1), aged 2–17 years, transplanted between 2008 and 2020, were identified. All signed consent for data collection. Median interval between transplantation and stenosis diagnosis was 420 days (IQR 299–545). All had acute steroid refractory and chronic moderate to severe GI GVHD. Ten patients had grade IV GI aGVHD (77%) treated with a median of 5 treatment lines [3–7]. Chronic GI GVHD maximum NIH score was 3 for ten patients and 2 for the others. Five patients presented extra-intestinal cGVHD, mainly cutaneous (n = 3) and pulmonary (n = 2). Median cGVHD treatment lines was 3 [2–3]. Eight patients received anti-TNFα monoclonal antibodies (mAbs) at some point during GVHD history (Supplementary Table 2). One patient had CMV colitis at 5 months post HSCT, successfully treated with ganciclovir 30 days before stenosis visualization.

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Data availability

The datasets generated during and/or analyzed during the current study are not publicly available. A study approved by the ethics committee of Rouen university hospital with the No. (E2023-20) in accordance with French legislation. This protocol proposes to report the results of a retrospective study, not covered by the law No. 2012-300 of March 5, 2012 (known as the Jardé law) relating to research involving the person human and with its implementing decrees promulgated in 2016 and 2017 with immediate effect.

Information has been given, no patient or legal representative has expressed opposition. That research involving human subjects, human material, or human data is in accordance with the Declaration of Helsinki.

References

  1. Zeiser R, Blazar BR. Pathophysiology of chronic graft-versus-host disease and therapeutic targets. N. Engl J Med. 2017;377:2565–79.

    Article  CAS  PubMed  Google Scholar 

  2. Patey-Mariaud de Serre N, Reijasse D, Verkarre V, Canioni D, Colomb V, Haddad E, et al. Chronic intestinal graft-versus-host disease: clinical, histological and immunohistochemical analysis of 17 children. Bone Marrow Transpl. 2002;29:223–30.

    Article  CAS  Google Scholar 

  3. Chan WPW, Mourad F, Leong RW. Crohn’s disease associated strictures. J Gastroenterol Hepatol. 2018;33:998–1008.

    Article  PubMed  Google Scholar 

  4. Gutierrez CA, Raval MV, Vester HR, Chaudhury S, von Allmen D, Rothstein DH. Surgical treatment of intestinal complications of graft versus host disease in the pediatric population: case series and review of literature. J Pediatr Surg. 2017;52:1718–17.

    Article  PubMed  Google Scholar 

  5. Herr AL, Latulippe JF, Carignan S, Mitchell A, Bélanger R, Roy J. Is severe intestinal chronic graft-versus-host disease an indication for surgery? A report of two cases. Transplantation. 2004;77:1617–20.

    Article  PubMed  Google Scholar 

  6. Spencer GD, Shulman HM, Myerson D, Thomas ED, McDonald GB. Diffuse intestinal ulceration after marrow transplantation. Hum Pathol. 1986;17:621–33.

    Article  CAS  PubMed  Google Scholar 

  7. Evans J, Percy J, Eckstein R, Ma D, Schnitzler M. Surgery for intestinal graft-versus-host disease: report of two cases. Dis Colon Rectum. 1998;41:1573–6.

    Article  CAS  PubMed  Google Scholar 

  8. Tordjman M, Ouachee M, Bonnard A, Tilea B, Yakouben K, Viala J, et al. Small bowel stenosis: a manifestation of chronic graft-versus-host disease in children? Hum Pathol. 2018;72:174–9.

    Article  PubMed  Google Scholar 

  9. Yoon JY, Kim H, Kang HJ, Park KD, Shin HY, Ahn HS. Chronic graft versus host disease with small bowel obstruction after unrelated hematopoietic stem cell transplantation in a patient with acute myeloid leukemia. Korean J Hematol. 2012;47:142–5.

    Article  PubMed  PubMed Central  Google Scholar 

  10. Gavel G, Marven S, Evans MJ, Walker J, Vora AJ. Obliterative enteritis complicating graft versus host disease. Bone Marrow Transpl. 2003;32:1097–8.

    Article  CAS  Google Scholar 

  11. Derlin T, Laqmani A, Veldhoen S, Apostolova I, Ayuk F, Adam G, et al. Magnetic resonance enterography for assessment of intestinal graft-versus-host disease after allogeneic stem cell transplantation. Eur Radio. 2015;25:1229–37.

    Article  Google Scholar 

  12. T Cuvelier GDE, Schoettler M, Buxbaum NP, Pinal-Fernandez I, Schmalzing M, Distler JHW, et al. toward a better understanding of the atypical features of chronic graft-versus-host disease: a report from the 2020 National Institutes of Health Consensus Project Task Force. Transpl Cell Ther. 2022;28:426–45.

    Article  Google Scholar 

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Authors and Affiliations

  1. Pediatrics, Hospital Charles Nicolle CHU Rouen, Rouen, France

    Hana Boulkroun

  2. Pediatric Gastro-Enterology CHU Caen Normandie, Caen, France

    Edouard Lacotte

  3. Pediatric Hematology Oncology, Center Pellingrin, CHU Bordeaux, Bordeaux, France

    Marie Angoso

  4. Pediatric Hematology Oncology and Stem Cells Transplantation, Robert Debré Hospital Paris,GH APHP-Nord, Paris University, Paris, France

    Jean Hugues Dalle

  5. Anesthesia, Reanimation, CHU Rouen, Rouen, France

    Julien Kallout

  6. Pediatric Hematology Oncology, CHU Toulouse, Toulouse, France

    Marie Nolla

  7. Pediatric Hematology, Oncology, Rennes South Hospital CHU, Rennes, France

    Agathe Picard

  8. Pediatric Hematology, Oncology, Hospital Hautepierre CHU Strasbourg, Strasbourg, France

    Catherine Paillard

  9. Pediatric Hematology Oncology, CHU de Grenoble, Grenoble, France

    Dominique Plantaz

  10. Pediatric Hematology, Oncology, Center Leon Berard Lyon, Lyon, France

    Cécile Renard

  11. Pediatric Hematology, Immunology, Oncology, CHU Nantes, Nantes, France

    Fanny Rialland

  12. Pediatric Hematology, Immunology, Oncology and Stem Cells Transplantation, Rouen University Hospital Charles Nicolle CHU Rouen, Rouen, France

    Pascale Schneider & Nimrod Buchbinder

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  1. Hana Boulkroun
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Contributions

HB, NB designed the study, and were primarily responsible for writing of the manuscript; HB and NB contributed equally. They had access to full trial data; EL contributed to study design; NB, MA, JHD, MN, AP, CP, DP, CR, FR, EL, PS contributed patients and edited the manuscript; JK provided all bio-statistical support and contributed to data analysis.

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Correspondence to Hana Boulkroun.

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Boulkroun, H., Lacotte, E., Angoso, M. et al. Late stenosis of the small intestine and colon, an atypical feature of chronic graft-versus-host disease. National retrospective study in French pediatric allograft centers. Bone Marrow Transplant 59, 1316–1319 (2024). https://doi.org/10.1038/s41409-024-02330-2

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  • DOI: https://doi.org/10.1038/s41409-024-02330-2

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