TY - JOUR
T1 - Linkage of infantile Bartter syndrome with sensorineural deafness to chromosome 1p
AU - Brennan, Theresa M.H.
AU - Landau, Daniel
AU - Shalev, Hana
AU - Lamb, Fred
AU - Schutte, Brian C.
AU - Walder, Roxanne Y.
AU - Mark, Allyn L.
AU - Carmi, Rivka
AU - Sheffield, Val C.
N1 - Funding Information:
We are very grateful to the family members for their cooperation during this study. We also thank J. Beck, T. Rokhlina and S. Yosefsberg for their technical assistance and helpful discussions. This work was supported by the following National Institutes of Health grants: the Institutional Research Fellowship for Kidney Disease, Hypertension, and Cell Biology; Specialized Center of Research for Molecular Genetics of Hypertension HL-55006; and HG00457. We also thank the Roy J. Carver Charitable Trust for providing support.
PY - 1998/2
Y1 - 1998/2
N2 - Bartter syndrome (BS) is a family of disorders manifested by hypokalemic hypochloremic metabolic alkalosis with normotensive hyperreninemic hyperaldosteronism. We evaluated a unique, inbred Bedouin kindred in which sensorineural deafness (SND) cosegregates with an infantile variant of the BS phenotype. Using a DNA-pooling strategy, we screened the human genome and successfully demonstrated linkage of this unique syndrome to chromosome 1p31. The genes for two kidney-specific chloride channels and a sodium/hydrogen antiporter, located near this region, were excluded as candidate genes. Although the search for the disease-causing gene in this family continues, this linkage further demonstrates the genetic heterogeneity of BS. In addition, the cosegregation of these phenotypes allows us to postulate that a single genetic alteration may be responsible for the SND and the BS phenotype. The identification and characterization of this gene would lead to a better understanding of the normal physiology of the kidney and the inner ear.
AB - Bartter syndrome (BS) is a family of disorders manifested by hypokalemic hypochloremic metabolic alkalosis with normotensive hyperreninemic hyperaldosteronism. We evaluated a unique, inbred Bedouin kindred in which sensorineural deafness (SND) cosegregates with an infantile variant of the BS phenotype. Using a DNA-pooling strategy, we screened the human genome and successfully demonstrated linkage of this unique syndrome to chromosome 1p31. The genes for two kidney-specific chloride channels and a sodium/hydrogen antiporter, located near this region, were excluded as candidate genes. Although the search for the disease-causing gene in this family continues, this linkage further demonstrates the genetic heterogeneity of BS. In addition, the cosegregation of these phenotypes allows us to postulate that a single genetic alteration may be responsible for the SND and the BS phenotype. The identification and characterization of this gene would lead to a better understanding of the normal physiology of the kidney and the inner ear.
UR - http://www.scopus.com/inward/record.url?scp=0031937693&partnerID=8YFLogxK
U2 - 10.1086/301708
DO - 10.1086/301708
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C2 - 9463315
AN - SCOPUS:0031937693
SN - 0002-9297
VL - 62
SP - 355
EP - 361
JO - American Journal of Human Genetics
JF - American Journal of Human Genetics
IS - 2
ER -