1971•New England Journal of MedicineRequires access

Ceramide Trihexosidosis (Fabry's Disease) without Skin Lesions

Joe T.R. Clarke, Juergen Knaack, John C. Crawhall, Leonhard S. Wolfe

Open publisher page 82 citations

Abstract

Histologic, histochemical and electron microscopical studies of kidney tissue taken by open biopsy from two unrelated men with unexplained proteinuria revealed changes typical of Fabry's disease although neither patient had any other manifestations of the disease. Angiokeratoma and corneal opacities were absent. The diagnoses were confirmed by the demonstration of excessive excretion of trihexosylceramide and digalactosylceramide in the urine. Decreased activities of the leukocyte lysosomal hydrolase, α-galactosidase, were also found in both patients.

About this research paper

What this paper is about

Histologic, histochemical and electron microscopical studies of kidney tissue taken by open biopsy from two unrelated men with unexplained proteinuria revealed changes typical of Fabry's disease although neither patient had any other manifestations of the disease. Angiokeratoma and corneal opacities were absent. The diagnoses were confirmed by the demonstration of excessive excretion of trihexosylceramide and digalactosylceramide in the urine. Decreased activities of the leukocyte lysosomal hydrolase, α-galactosidase, were also found in both patients.

Why it matters

OpenAlex reports 82 citations for this work. Citation counts describe recorded attention and do not establish research quality.

Key contribution

A contribution statement is not available in the OpenAlex record.

Method / approach

Method details are not available in the OpenAlex metadata.

Main findings

Findings are not separately available in the OpenAlex metadata.

Limitations

Limitations are not available in the OpenAlex metadata.

Applications

Application details are not available in the OpenAlex metadata.

Available abstract

Histologic, histochemical and electron microscopical studies of kidney tissue taken by open biopsy from two unrelated men with unexplained proteinuria revealed changes typical of Fabry's disease although neither patient had any other manifestations of the disease. Angiokeratoma and corneal opacities were absent. The diagnoses were confirmed by the demonstration of excessive excretion of trihexosylceramide and digalactosylceramide in the urine. Decreased activities of the leukocyte lysosomal hydrolase, α-galactosidase, were also found in both patients.

Key concepts: Fabry disease, Fabry's disease, Globotriaosylceramide, Angiokeratoma, Medicine, Proteinuria, Pathology, Disease

Related papers

Back to paper searchBrowse research topicsOriginal source
Ceramide Trihexosidosis (Fabry's Disease) without Skin Lesions — Research Paper | ScholarLens