Ichthyosiform dermatosis with superficial blister formation and peeling: evidence for a desmosomal anomaly and altered epidermal vitamin A metabolism

Details

Serval ID
serval:BIB_93A60255E56B
Type
Article: article from journal or magazin.
Publication sub-type
Case report (case report): feedback on an observation with a short commentary.
Collection
Publications
Institution
Title
Ichthyosiform dermatosis with superficial blister formation and peeling: evidence for a desmosomal anomaly and altered epidermal vitamin A metabolism
Journal
Journal of the American Academy of Dermatology
Author(s)
Mevorah  B., Salomon  D., Siegenthaler  G., Hohl  D., Meier  M. L., Saurat  J. H., Frenk  E.
ISSN
0190-9622 (Print)
Publication state
Published
Issued date
02/1996
Volume
34
Number
2 Pt 2
Pages
379-85
Notes
Case Reports
Journal Article
Research Support, Non-U.S. Gov't --- Old month value: Feb
Abstract
We describe a man with generalized congenital ichthyosiform dermatosis, severe cheilitis, and palmar and plantar hyperkeratosis with superficial blistering. Low-dose acitretin therapy induced areas of peeling skin, similar to that seen in the peeling skin syndrome. Histologically, the skin was moderately hyperkeratotic and the palmar blisters were subcorneal. Electron microscopy revealed that the splitting occurred within the desmosomal plaque. Ultrastructural and biochemical investigations indicated epidermal hypervitaminosis A, probably related to alteration of epidermal retinoic acid metabolism. This disease is proposed as a hitherto unreported variant of the peeling skin syndrome.
Keywords
Adolescent Cheilitis/etiology Desmosomes/ultrastructure Epidermis/metabolism Humans Ichthyosis/classification/metabolism/*pathology Keratoderma, Palmoplantar/etiology Male Receptors, Retinoic Acid/metabolism Skin/*pathology Vitamin A/*metabolism
Pubmed
Web of science
Create date
25/01/2008 17:36
Last modification date
20/08/2019 15:56
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