Back to Search Start Over

Angiokeratoma corporis diffusum (Anderson-Fabry's disease): a case report.

Authors :
Massi D
Martinelli F
Battini ML
Comin CE
Franchi A
Gioia O
Santucci M
Source :
Journal of the European Academy of Dermatology and Venereology : JEADV [J Eur Acad Dermatol Venereol] 2000 Mar; Vol. 14 (2), pp. 127-30.
Publication Year :
2000

Abstract

We report on a 14-year-old boy who presented with a 4-year history of acral pains and febrile episodes. On physical examination, numerous small reddish papules were present on his abdomen, located predominantly on the periumbelical region. Renal function was within normal limits. Ophthalmological examination revealed whorled opacities of the cornea (cornea verticillata) and dilated tortuous conjunctival vessels. Histopathological examination of one of the cutaneous papules showed several dilated blood vessels in the superficial dermis surrounded by collarettes of thickened rete ridges, consistent with a diagnosis of angiokeratoma. The electron-microscopic study of a skin specimen demonstrated the presence of dilated lysosomes with deposition of electron-dense bodies, some of which with laminated structure, in endothelial cells and fibroblasts. These findings were regarded as indicative of Fabry's disease. Subsequent biochemical analysis confirmed the presence of a alpha-galactosidase A deficiency in leukocytes. In conclusion, we described the clinical, histopathological and submicroscopic findings of a case of Fabry's disease, in which the combination of electron microscopic and biochemical approaches allowed the correct diagnosis.

Details

Language :
English
ISSN :
0926-9959
Volume :
14
Issue :
2
Database :
MEDLINE
Journal :
Journal of the European Academy of Dermatology and Venereology : JEADV
Publication Type :
Academic Journal
Accession number :
10972099
Full Text :
https://doi.org/10.1046/j.1468-3083.2000.00030.x