Home LiteratureArticle Details
PMID: 2988520 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S. Review

Adrenal dysfunction in glycerol kinase deficiency.

Biochemical medicine ·Vol. 33 ·No. 2 ·1985-04-00 ·Pages 189-99

Seltzer WK, Firminger H, Klein J, Pike A, Fennessey P, McCabe ER

Abstract

The infantile form of glycerol kinase deficiency appears to be an X-linked disorder which is consistently characterized by developmental delay and adrenal cortical insufficiency and hypoplasia. We propose that the inherited deficiency of outer mitochondrial membrane-bound glycerol kinase restricts glycerophospholipid synthesis, and, hence, the activation of steroidogenesis. This would limit the conversion of cholesterol to pregnenolone, the precursor for glucocorticoids in the adrenal cortex. The deficiency in cortisol production, with a lack of feedback to the pituitary, would result in increased ACTH production and hypertrophy of the fascicular zone at the same time that replication of the cells within this zone would be inhibited. Similarly, the decreased mineralocorticoid production by the sparse glomerulosal zone would limit the ability of the individual to respond to stress, and would result in development of potentially fatal hyponatremia and hyperkalemia. Organization of the pathway for glycerophospholipid synthesis at the outer mitochondrial membrane would make this pathway particularly vulnerable to mutations disrupting the compartmented production of the parent compound, glycerol 3-phosphate, by mitochondrial-bound glycerol kinase.

MeSH Terms
Adrenal Cortex/pathology Adrenal Glands/enzymology Adrenal Insufficiency/enzymology Animals Cattle Cell Compartmentation Child Child, Preschool Gas Chromatography-Mass Spectrometry Glycerol Kinase/deficiency Humans Hydrocortisone/therapeutic use Male Metabolism, Inborn Errors/enzymology Models, Biological Oxidative Phosphorylation Phospholipids/biosynthesis Phosphotransferases/deficiency Steroids/urine Tissue Distribution
Chemicals
Phospholipids Steroids Phosphotransferases Glycerol Kinase Hydrocortisone
Authors & Affiliations
6 authors, click to expand affiliations / ORCID
Seltzer W K
Firminger H
Klein J
Pike A
Fennessey P
McCabe E R
Article Info
Journal
Biochemical medicine
Abbr.
Biochem Med
ISSN
0006-2944
Published
1985-04-00
Pages
189-99
Language
English
Region
United States
NLM ID
0151424
Subset
IM
Grants
NICHD NIH HHS · 5P30 HD0424 · United States
NICHD NIH HHS · 5PO1 HD08315 · United States
NIADDK NIH HHS · R01 AM26265 · United States
Analysis Services
Analysis Services

Contact

No. 2 Wenbo Road, Zhangqiu District, Jinan, Shandong

Qilu Normal University · Genelibs Bioinformatics Lab

750 Shunhua Rd, Jinan

2F, Bldg F, University Science Park

Tel: 0531-88819269

WeChat Official Account

Follow our WeChat subscription account for real-time updates and the latest in medical and biological research.


Business Email

E-mail: product@genelibs.com