{
  "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.",
  "abstract_shown": true,
  "affiliations": [
    "Department of Pediatrics Iwate Medical University Yahaba Japan."
  ],
  "article_type": "case study",
  "arxiv": null,
  "author_total": 7,
  "authors": [
    {
      "affs": [
        0
      ],
      "name": "Yasutaka Chiba"
    },
    {
      "affs": [
        0
      ],
      "name": "Manabu Takahashi",
      "orcid": "0000-0003-0908-9227"
    },
    {
      "affs": [
        0
      ],
      "name": "Taro Yoshida"
    },
    {
      "affs": [
        0
      ],
      "name": "Seiya Tagane"
    },
    {
      "affs": [
        0
      ],
      "name": "Takeshi Shiohata"
    },
    {
      "affs": [
        0
      ],
      "name": "Shoko Miura"
    },
    {
      "affs": [
        0
      ],
      "name": "Manami Akasaka"
    }
  ],
  "bibtex": "https://research.draconitic.com/paper/7h1v051wm04yg.bib",
  "cited_by": 0,
  "doi": "10.1002/jpr3.70291",
  "fulltext_url": "https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13717532/?tool=EBI",
  "id": "7h1v051wm04yg",
  "issns": [
    "2691-171X"
  ],
  "landing_url": "https://europepmc.org/article/PMC/PMC13717532",
  "language": "eng",
  "licence": "CC-BY",
  "licence_label": "CC BY",
  "licence_source": "article",
  "licence_url": "https://creativecommons.org/licenses/by/4.0/",
  "metadata_licence": null,
  "native_subjects": [
    [
      "heading",
      "CASE REPORT"
    ],
    [
      "heading",
      "Case Report"
    ],
    [
      "heading",
      "Gastroenterology: Inflammatory Bowel Disease (Europe)"
    ],
    [
      "openalex",
      "1311|Inflammatory Bowel Disease"
    ]
  ],
  "openalex": "W7224361956",
  "pdf_url": "https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13717532/pdf/?tool=EBI",
  "pmcid": "PMC13717532",
  "pmid": "42959554",
  "published": "2026-10-29",
  "publisher": "Wiley",
  "ris": "https://research.draconitic.com/paper/7h1v051wm04yg.ris",
  "sources": [
    {
      "first_seen": "2026-10-31T00:20:37Z",
      "native_id": "PMC:PMC13717532",
      "source": "europepmc",
      "url": "https://europepmc.org/article/PMC/PMC13717532"
    },
    {
      "first_seen": "2026-11-01T00:20:50Z",
      "native_id": "MED:42959554",
      "source": "europepmc",
      "url": "https://europepmc.org/article/MED/42959554"
    },
    {
      "first_seen": "2026-11-02T12:21:50Z",
      "native_id": "PMC13717532",
      "source": "pmc",
      "url": "https://doi.org/10.1002/jpr3.70291"
    }
  ],
  "subjects": [
    {
      "field": "biology",
      "origin": "openalex:1311|Inflammatory Bowel Disease",
      "subfield": "genetics-genomics"
    }
  ],
  "title": "Vedolizumab use for severe chronic enterocolitis in a child with Hoyeraal–Hreidarsson syndrome: A case report",
  "title_text": "Vedolizumab use for severe chronic enterocolitis in a child with Hoyeraal–Hreidarsson syndrome: A case report",
  "type": "peer-reviewed",
  "updated_at": "2026-11-02T12:21:50Z",
  "url": "https://research.draconitic.com/paper/7h1v051wm04yg",
  "venue": "JPGN reports",
  "versions": []
}