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Endoscopic Assessment and Serial Balloon Dilatation in a Toddler With Dyskeratosis Congenita-Hoyeraal-Hreidarsson Syndrome Following Bone Marrow Transplant: A Case Report.
Rodriguez, Kurt; Shargo, Ryan; Ekblad, Morgan; Sunkersett, Gauri; Karjoo, Sara; Betensky, Marisol; Wilsey, Michael J.
Afiliación
  • Rodriguez K; From the Johns Hopkins All Children's Hospital, Saint Petersburg, FL.
  • Shargo R; University of South Florida, Reisterstown, MD.
  • Ekblad M; University of South Florida, Reisterstown, MD.
  • Sunkersett G; AbbVie, South San Francisco, CA.
  • Karjoo S; From the Johns Hopkins All Children's Hospital, Saint Petersburg, FL.
  • Betensky M; From the Johns Hopkins All Children's Hospital, Saint Petersburg, FL.
  • Wilsey MJ; From the Johns Hopkins All Children's Hospital, Saint Petersburg, FL.
JPGN Rep ; 4(2): e291, 2023 May.
Article en En | MEDLINE | ID: mdl-37200733
We report a 3-year-old patient with suspected oropharyngeal graft-versus-host disease (GVHD) who developed progressive dysphagia to solids and liquids. The patient has a history of Dyskeratosis Congenita-Hoyeraal-Hreidarsson Syndrome with associated bone marrow failure requiring a nonmyeloablative matched sibling hematopoietic stem cell transplant. Esophagram revealed significant narrowing in the cricopharyngeal region. Subsequent esophagoscopy showed a proximal, high-grade pinhole esophageal stricture that was very difficult to visualize and cannulate. High-grade esophageal strictures are uncommon in very young children with GVHD. We believe the patient's underlying Dyskeratosis Congenita-Hoyeraal-Hreidarsson Syndrome in the setting of inflammatory changes seen in GVHD following hematopoietic stem cell transplant set the stage for a high-grade esophageal obstruction. The patient's symptoms improved with serial endoscopic balloon dilation.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: JPGN Rep Año: 2023 Tipo del documento: Article Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: JPGN Rep Año: 2023 Tipo del documento: Article Pais de publicación: Estados Unidos