Literature DB >> 7049883

Clinical variability of congenital adrenal hyperplasia due to 11 beta-hydroxylase deficiency.

A Rösler, E Leiberman, J Sack, H Landau, A Benderly, S W Moses, T Cohen.   

Abstract

Studies in 18 Jewish families from Morocco, Tunis, Turkey and Iran revealed 26 patients with congenital adrenal hyperplasia due to 11 beta-hydroxylase deficiency. The clinical expression of androgen excess varied widely in affected females, and range from solely enlarged clitoris in the mildest forms to severely hypertrophied clitoris with penile urethra and fused labial-scrotal folds in the most extreme forms of masculinization. Intermediate degrees of severity were manifested by ambiguous genitalia. There was no correlation between the degree of virilization and the signs of mineralocorticoid excess. Severe volume-induced hypertension leading to vascular accidents and death were also observed in severe as well as in mildly virilized patients, while completely masculinized females were sometime normotensive. Overt hypokalemia was present in 6 patients but was not a constant feature of hypertensives. However, all affected individuals, except for 2 infants, had very low levels of plasma renin activity suggesting that a state of volume expansion was indeed present in the majority of cases, even though changes in blood pressure did not always occur. The clinical expression of this disorder is characterized by a wide range of variability in the signs of both androgen and mineralocorticoid excess, which do not necessarily correlate with the quantity of hormones secreted.

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Year:  1982        PMID: 7049883     DOI: 10.1159/000179494

Source DB:  PubMed          Journal:  Horm Res        ISSN: 0301-0163


  6 in total

Review 1.  Clinical perspectives in congenital adrenal hyperplasia due to 11β-hydroxylase deficiency.

Authors:  Krupali Bulsari; Henrik Falhammar
Journal:  Endocrine       Date:  2016-12-07       Impact factor: 3.633

2.  Left ventricular failure due to a rare variant of congenital adrenal hyperplasia.

Authors:  Sonal Bhatia; Mamta N Muranjan; Keya R Lahiri
Journal:  Indian J Pediatr       Date:  2012-01-10       Impact factor: 1.967

3.  A mutation in CYP11B1 (Arg-448----His) associated with steroid 11 beta-hydroxylase deficiency in Jews of Moroccan origin.

Authors:  P C White; J Dupont; M I New; E Leiberman; Z Hochberg; A Rösler
Journal:  J Clin Invest       Date:  1991-05       Impact factor: 14.808

Review 4.  Bilateral testicular enlargement due to adrenal remnant in a patient with C11 hydroxylase deficiency congenital adrenal hyperplasia.

Authors:  A A M Ghazi; F Hadayegh; G Khakpour; F Azizi; J C Melby
Journal:  J Endocrinol Invest       Date:  2003-01       Impact factor: 4.256

5.  Hypertension in a neonate with 11 beta-hydroxylase deficiency.

Authors:  M Mimouni; H Kaufman; A Roitman; C Morag; N Sadan
Journal:  Eur J Pediatr       Date:  1985-01       Impact factor: 3.183

6.  Congenital adrenal hyperplasia due to 11-beta-hydroxylase deficiency with skeletal abnormalities.

Authors:  K M Ajlouni; M A Arnaout; Y Qoussous
Journal:  J Endocrinol Invest       Date:  1996-05       Impact factor: 4.256

  6 in total

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