Vedolizumab use for severe chronic enterocolitis in a child with Hoyeraal–Hreidarsson syndrome: A case report

Abstract

We report a patient with Hoyeraal-Hreidarsson syndrome (HHS), a telomere biology disorder, who developed severe chronic enterocolitis with inflammatory bowel disease-like features. Vedolizumab led to improvement in stool frequency, reduced fecal calprotectin, and clinical remission without infectious complications. Remission was maintained with vedolizumab monotherapy for 2 years after discontinuation of budesonide and parenteral nutrition. This report highlights vedolizumab as a potentially safe therapeutic option for enterocolitis in HHS, emphasizing the value of gut-selective therapy when systemic immunosuppression is risky. Further studies are needed to establish the long-term efficacy and safety of vedolizumab in patients with telomere biology disorders.

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BibTeX

@article{chiba2026vedolizumab,
  title = {{Vedolizumab use for severe chronic enterocolitis in a child with Hoyeraal–Hreidarsson syndrome: A case report}},
  author = {Yasutaka Chiba and Manabu Takahashi and Taro Yoshida and Seiya Tagane and Takeshi Shiohata and Shoko Miura and Manami Akasaka},
  journal = {JPGN reports},
  year = {2026},
  month = oct,
  doi = {10.1002/jpr3.70291},
  url = {https://europepmc.org/article/PMC/PMC13717532},
  note = {CC BY},
}

RIS

TY  - JOUR
TI  - Vedolizumab use for severe chronic enterocolitis in a child with Hoyeraal–Hreidarsson syndrome: A case report
AU  - Yasutaka Chiba
AU  - Manabu Takahashi
AU  - Taro Yoshida
AU  - Seiya Tagane
AU  - Takeshi Shiohata
AU  - Shoko Miura
AU  - Manami Akasaka
PY  - 2026
DA  - 2026/10/29
JO  - JPGN reports
DO  - 10.1002/jpr3.70291
UR  - https://europepmc.org/article/PMC/PMC13717532
AB  - We report a patient with Hoyeraal-Hreidarsson syndrome (HHS), a telomere biology disorder, who developed severe chronic enterocolitis with inflammatory bowel disease-like features. Vedolizumab led to improvement in stool frequency, reduced fecal calprotectin, and clinical remission without infectious complications. Remission was maintained with vedolizumab monotherapy for 2 years after discontinuation of budesonide and parenteral nutrition. This report highlights vedolizumab as a potentially safe therapeutic option for enterocolitis in HHS, emphasizing the value of gut-selective therapy when systemic immunosuppression is risky. Further studies are needed to establish the long-term efficacy and safety of vedolizumab in patients with telomere biology disorders.
SN  - 2691-171X
ER  -

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