Shwachman-Diamond syndrome and solid tumors: Three new patients from the French Registry for Severe Chronic Neutropenia and literature review

Pediatr Blood Cancer. 2021 Jul;68(7):e29071. doi: 10.1002/pbc.29071. Epub 2021 Apr 19.

Abstract

Shwachman-Diamond syndrome with Shwachman-Bodian-Diamond syndrome (SBDS) biallelic variants is a rare disorder that predisposes the carrier to malignant hemopathies but solid-cancer predisposition is poorly known. Among 155 cases entered in the French Registry for Severe Chronic Neutropenia, three were identified with malignant solid tumors (ovary, breast, and esophagus). All cancers occurred during the fifth decade and, despite being localized at diagnosis, were rapidly fatal thereafter. No cancer was observed post transplantation in the 14 HSCT survivors. Based on the literature and our patient data, we can merely advance that this complication is predominantly diagnosed in adults.

Keywords: Shwachman-Diamond syndrome; congenital neutropenia; solid cancer.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Female
  • Humans
  • Neoplasms* / complications
  • Neoplasms* / epidemiology
  • Neoplasms* / genetics
  • Neutropenia* / epidemiology
  • Neutropenia* / etiology
  • Neutropenia* / genetics
  • Registries
  • Shwachman-Diamond Syndrome* / complications

Supplementary concepts

  • Neutropenia, severe chronic