Radiosensitivity in a newborn with microcephalia: A case report of Nijmegen breakage syndrome

Birth Defects Res. 2024 May;116(5):e2346. doi: 10.1002/bdr2.2346.

Abstract

Aim: Nijmegen breakage syndrome (NBS) is an autosomal recessive DNA repair disorder which is characterized by immunodeficiency and increased risk of lymphoproliferative malignancy.

Case: We observed an increase in the rate of chromosomal rearrangements in the cultured cells following an incidental radiograph for craniosynostosis in a newborn who was followed up due to microcephaly. We identified a homozygous deletion of c.657_661delACAAA/p.Lys219fs (rs587776650) in the NBN gene through whole exome sequencing.

Conclusion: It is crucial to thoroughly examine the clinical features of newborns with microcephaly and consider chromosomal instability syndromes just like Nijmegen breakage syndrome. Not overlooking radiosensitivity, which is a characteristic feature of this syndrome, is a vital condition to the patient's survival time.

Keywords: NBN gene; Nijmegen breakage syndrome (NBS); chromosomal rearrangements; microcephaly; radiosensitivity.

Publication types

  • Case Reports

MeSH terms

  • Cell Cycle Proteins / genetics
  • Exome Sequencing / methods
  • Female
  • Homozygote
  • Humans
  • Infant, Newborn
  • Male
  • Microcephaly* / genetics
  • Nijmegen Breakage Syndrome* / genetics
  • Nuclear Proteins / genetics
  • Radiation Tolerance* / genetics

Substances

  • NBN protein, human