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- W2070108401 abstract "There are numerous indications that otosclerosis represents a local manifestation of a generalized disorder of GAG and/or collagen metabolism, a contention supported by the heritable nature of the disease. Zechner and Manolidis reported a pronounced increase of total GAGs in urine of patients with active otosclerosis [Arch. Klin. Exp. Ohren Nasen Kehlkopfheilkd. 202, 634 (1972)]. We were not able to confirm their reported increase in total urinary GAGs. Therefore we searched for more subtle indicators of aberrant GAG metabolism, such as changes in ratios of individual GAGs. For this purpose it was necessary to develop a highly sensitive technique to quantitate individual GAGs; however, again no significant differences were found: dermatan sulfate (0.48 ± 0.06 and 0.32 ± 0.04), heparan sulfate (0.43 ± 0.06 and 0.40 ± 0.04), chondroitin-4-sulfate (0.60 ± 0.07 and 0.71 ± 0.08), and chondroitin-6-sulfate (1.25 ± 0.14 and 1.41 ± 0.16) in controls (N = 22) and active otosclerosis (N = 20), respectively (X̄ ± SEM μg hexuronic acid/mg creatinine). These negative data by no means rule out a possible error in GAG metabolism. More definitive conclusions should emerge from radiotracer (35SO4) studies for the detection of errors in GAG synthesis and/or degradation in skin fibroblast cultures. [Supported by Research Fund of American Otological Society and NIH Grant NS-06575." @default.
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- W2070108401 date "1984-05-01" @default.
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- W2070108401 title "Otosclerosis: A generalized disorder of glycosaminoglycan (CAG) metabolism?" @default.
- W2070108401 doi "https://doi.org/10.1121/1.2021640" @default.
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