Cellular radiosensitivity in human severe-combined-immunodeficiency (SCID) syndromes

Radiother Oncol. 1997 Jan;42(1):53-7. doi: 10.1016/s0167-8140(96)01865-8.

Abstract

Purpose: The aim of the work was to establish to what extent a variety of human severe-combined-immunodeficiency (SCID) disorders are associated with in vitro cellular hypersensitivity to ionizing radiation.

Materials and methods: A study was made of fibroblast strains established from individuals with adenosine deaminase deficiency, T(-)B(-) SCID, Omenn's syndrome and a SCID heterozygote. For comparison, an assessment was also made of the radiosensitivity of a series of fibroblast strains derived from: normal donors, a patients with ataxia-telangiectasia (A-T) and an A-T heterozygote. Radiosensitivity was determined using a clonogenic assay following both high (HDR) and low (LDR) dose-rate irradiation.

Results: Following HDR irradiation, the fibroblast strains derived from the different human SCID disorders displayed a wide range of radiosensitivity: the adenosine deaminase deficiency cells were similar in radiosensitivity to normal fibroblasts, T(-)B(-) cells were as hypersensitive to radiation as A-T cells and the Omenn's syndrome cells showed intermediate radiosensitivity. However, whereas all four normal cell strains studied showed significant LDR sparing, none of the SCID fibroblasts did.

Conclusions: These data indicate that human SCID is variable in terms of radiosensitivity depending on the particular defect. In addition, the lack of LDR sparing of radiation-induced damage suggests the involvement of some form(s) of DNA repair defect in all the human SCID syndromes.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adenosine Deaminase / deficiency
  • Cell Survival
  • Cells, Cultured / radiation effects
  • Colony-Forming Units Assay
  • Dose-Response Relationship, Radiation
  • Fibroblasts / cytology
  • Fibroblasts / radiation effects*
  • Humans
  • Radiation Dosage
  • Radiation Tolerance / immunology*
  • Reference Values
  • Severe Combined Immunodeficiency / genetics
  • Severe Combined Immunodeficiency / immunology*

Substances

  • Adenosine Deaminase