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A multicenter study of women with nonclassical congenital adrenal hyperplasia: relationship between genotype and phenotype

Mol Genet Metab. 2000 Nov;71(3):527-34. doi: 10.1006/mgme.2000.3036.

Abstract

Characteristic presentation of nonclassical adrenal hyperplasia (NCAH) due to 21-hydroxylase deficiency was compared between women carrying a severe and a mild CYP21 mutation (Group 1, N = 26) versus homozygotes for mild mutations (Group 2, N = 8). The diagnosis was based on elevated ACTH-stimulated 17OH-progesterone (17OHP). Genotyping for 10 mutations was performed by PCR-based techniques. Jewish patients predominated among Group 2 (25% vs 11.5% in Group 1); however, 85% of all patients were non-Jewish Caucasians. Average age of presentation was 23-25 years, and did not differ between groups. Hirsutism, and to a lesser extent oligomenorrhea and acne, were more prevalent among Group 1 women. There was a trend to higher basal 17OHP among Group 1 patients (mean +/- SEM; 1354+/-323 vs 714+/-129 ng/dl, P< or =0.25). The lack of significant difference was perhaps due to the relatively few homozygotes for 2 mild mutations (24%). V281L was carried on approximately 48% of all alleles, and about 16% carried either P30L or P453S. Approximately 38% of alleles and 77% of patients carried a classic mutation. These data have important implications for genetic counseling. In summary, we describe differences in clinical, hormonal, and genetic characteristics among a multiethnic group of females with NCAH.

Publication types

  • Multicenter Study

MeSH terms

  • 17-alpha-Hydroxyprogesterone / blood
  • Acne Vulgaris / complications
  • Adolescent
  • Adrenal Hyperplasia, Congenital / complications
  • Adrenal Hyperplasia, Congenital / enzymology
  • Adrenal Hyperplasia, Congenital / genetics*
  • Adult
  • Alleles
  • Amino Acid Substitution
  • Body Mass Index
  • Child
  • Female
  • Gene Frequency
  • Genotype
  • Hirsutism / complications
  • Humans
  • Middle Aged
  • Mutation
  • Oligomenorrhea / complications
  • Phenotype
  • Steroid 21-Hydroxylase / genetics*

Substances

  • 17-alpha-Hydroxyprogesterone
  • Steroid 21-Hydroxylase