PCR detection of Y-specific sequences in patients with Ullrich-Turner syndrome: clinical implications and limitations

Am J Med Genet. 1998 Apr 1;76(4):283-7.

Abstract

Cytogenetic analysis of patients with Ullrich-Turner syndrome (UTS) may fail to detect low levels of Y chromosome mosaicism or Y-derived marker chromosomes. More sensitive polymerase chain reaction (PCR)-based tests have been developed; however, applicability of these data to prognosis of virilization and gonadoblastoma development has not been investigated adequately. We used a multiplex PCR-based method to detect two Y-specific sequences, SRY and AMGLY. Thirteen patients with UTS without cytogenetically detected Y chromosomes were studied. Y-specific sequences were detected in 5 patients by multiplex PCR. A cryptic translocation involving the Y chromosome was found in one patient with severe virilization of external genitalia and a male phenotype. Y chromosomal mosaicism was detected in peripheral blood and in both gonads of one patient, and only in the left gonad of another patient. Existence of a Y-derived marker was demonstrated in 2 patients, one of whom had no testicular tissue or virilization. Consistent with previous reports, we conclude that PCR is more sensitive than classical cytogenetic analysis and detects patients with Y-specific sequences in blood cells. However, the absence of Y-specific material in blood is not a sufficient reason to reject surgical treatment in case of virilization.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Amelogenin
  • Child
  • DNA / blood
  • DNA-Binding Proteins / chemistry*
  • DNA-Binding Proteins / genetics
  • Dental Enamel Proteins / genetics
  • Female
  • Gonadoblastoma / etiology
  • Gonadoblastoma / genetics
  • Gonads
  • Humans
  • Karyotyping
  • Male
  • Mosaicism
  • Nuclear Proteins*
  • Phenotype
  • Polymerase Chain Reaction / methods*
  • Sex-Determining Region Y Protein
  • Transcription Factors*
  • Translocation, Genetic
  • Turner Syndrome / blood
  • Turner Syndrome / complications
  • Turner Syndrome / genetics*
  • Virilism / etiology
  • Virilism / genetics
  • X Chromosome
  • Y Chromosome*

Substances

  • Amelogenin
  • DNA-Binding Proteins
  • Dental Enamel Proteins
  • Nuclear Proteins
  • SRY protein, human
  • Sex-Determining Region Y Protein
  • Transcription Factors
  • DNA