Abstract:
:The Chediak-Higashi syndrome (CHS) is an inherited disorder of humans and of several animal species, characterized by partial albinism, pseudohemophilia, increased susceptibility to disease, and large inclusions in all granule-forming cells. In this study, various parameters of adenine nucleotide biochemistry were examined in beige mouse kidney tissue and in peripheral blood leukocytes from CHS mink. There were no differences in the total protein content, total ATPase activity or the magnesium (Mg2+) ATPase or the sodium-potassium (Na(+)-K+) ATPase activities, the concentrations of ATP, ADP, and AMP, or the adenylate energy charge (AEC) in kidney extracts from beige and normal mice. In studies of leukocytes, there were no differences in the concentrations of ATP, ADP, AMP, and cAMP or the AECs in total leukocyte preparations and in extracts from granulocytes or nongranulocytes. These results can be explained by any one of several hypotheses: no storage pool of adenine nucleotides exists in the tissues examined; or the alleged storage pool is not affected by CHS; or the quantity of nucleotides in the alleged storage pool is too minute to be evaluated by current techniques; or the CHS defect might cause a shift from the storage pool to the metabolic pool.
journal_name
Exp Mol Patholjournal_title
Experimental and molecular pathologyauthors
Jamison KC,Padgett GA,Giesy JP,Bell TGdoi
10.1006/exmp.1993.1004subject
Has Abstractpub_date
1993-02-01 00:00:00pages
40-52issue
1eissn
0014-4800issn
1096-0945pii
S0014-4800(83)71004-Xjournal_volume
58pub_type
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