Unique CD18 mutations involving a deletion in the extracellular stalk region and a major truncation of the cytoplasmic domain in a patient with leukocyte adhesion deficiency type 1

Blood. 2004 Feb 1;103(3):1105-13. doi: 10.1182/blood-2003-08-2780. Epub 2003 Sep 25.

Abstract

Two novel CD18 mutations were identified in a patient who was a compound heterozygote with type 1 leukocyte adhesion deficiency and whose phenotype was typical except that he exhibited hypertrophic scarring. A deletion of 36 nucleotides in exon 12 (1622del36) predicted the net loss of 12 amino acid (aa) residues in the third cysteine-rich repeat of the extracellular stalk region (mut-1). A nonsense mutation in exon 15 (2200G>T), predicted a 36-aa truncation of the cytoplasmic domain (mut-2). Lymphocyte function-associated antigen 1 (LFA-1) and macrophage antigen-1 (Mac-1) containing the mut-1 beta(2) subunit were expressed at very low levels compared with wild-type (wt) beta(2). Mac-1 and LFA-1 expression with the mut-2 beta(2) subunit were equivalent to results with wt beta(2). Binding function of Mac-1 with mut-2 beta(2) was equivalent to that with wt beta(2). However, binding function of LFA-1 with the mut-2 beta(2) subunit was reduced by 50% versus wt beta(2). It was concluded that (1) the portion of the CD18 stalk region deleted in mut-1 is critical for beta(2) integrin heterodimer expression but the portion of the cytoplasmic domain truncated in mut-2 is not; and (2) the mut-2 cytoplasmic domain truncation impairs binding function of LFA-1 but not of Mac-1. Studies with the patient's neutrophils (PMNs) were consistent with functional impairment of LFA-1 but not of Mac-1.

Publication types

  • Case Reports
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Adult
  • Amino Acid Sequence
  • Animals
  • Base Sequence
  • CD18 Antigens / chemistry*
  • CD18 Antigens / genetics*
  • CD18 Antigens / metabolism
  • COS Cells
  • Case-Control Studies
  • Cell Adhesion
  • Codon, Nonsense*
  • DNA, Complementary / genetics
  • Heterozygote
  • Humans
  • In Vitro Techniques
  • Intercellular Adhesion Molecule-1 / metabolism
  • Leukocyte-Adhesion Deficiency Syndrome / classification
  • Leukocyte-Adhesion Deficiency Syndrome / genetics*
  • Leukocyte-Adhesion Deficiency Syndrome / immunology*
  • Lymphocyte Function-Associated Antigen-1 / metabolism
  • Macrophage-1 Antigen / chemistry
  • Macrophage-1 Antigen / metabolism
  • Male
  • Molecular Sequence Data
  • Phenotype
  • Protein Structure, Tertiary
  • Recombinant Proteins / chemistry
  • Recombinant Proteins / genetics
  • Recombinant Proteins / metabolism
  • Rosette Formation
  • Sequence Deletion*
  • Transfection

Substances

  • CD18 Antigens
  • Codon, Nonsense
  • DNA, Complementary
  • Lymphocyte Function-Associated Antigen-1
  • Macrophage-1 Antigen
  • Recombinant Proteins
  • Intercellular Adhesion Molecule-1