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Article
March 1966

Hepatic Phosphorylase Defect: Studies on Peripheral Blood

Author Affiliations

NEW YORK; DURHAM, NC; NEW YORK
From the Babies Hospital and Department of Pediatrics, College of Physicians and Surgeons, Columbia University, New York (Drs. Wallis and Harris), and Department of Pediatrics, Duke University Medical School, Durham (Dr. Sidbury). Dr. Wallis is on leave from the Queen Elizabeth Hospital for Sick Children, London.

Am J Dis Child. 1966;111(3):278-282. doi:10.1001/archpedi.1966.02090060088008
Abstract

STUDIES OF THE genetics of glycogen storage disease have been hampered by the obvious difficulty of obtaining tissue from apparently healthy relatives for enzyme assays.

In 1961, Williams and Field1 from the United States and Hülsmann et al2 from Belgium reported very low leukocyte phosphorylase activity in five patients with proven hepatic phosphorylase defect. In two of the families there was also low leukocyte phosphorylase activity in the mother, but normal values in the father and male siblings. The mothers were quite healthy in both instances and a sex-linked, dominant form of inheritance was postulated by both sets of authors.

Sidbury et al3,4 have studied the erythrocyte glycogen concentration and its β-amylase degradation in many cases of glycogen storage disease. High concentrations were found in limit dextrinosis and in hepatic phosphorylase defect. Abnormally short and long chain structure of glycogen could be demonstrated.

These peripheral blood studies

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