TY - JOUR
T1 - Cystic canal mutants in Caenorhabditis elegans are defective in the apical membrane domain of the renal (excretory) cell
AU - Buechner, Matthew
AU - Hall, David H.
AU - Bhatt, Harshida
AU - Hedgecock, Edward M.
N1 - Funding Information:
This paper was dedicated to Julius Adler on the occasion of his 65th birthday. We thank Andrew Chisholm and Robert Horvitz for sharing their exc alleles; John Plenefisch for initial mapping of exc-6; Steven Jones, David Baillie, and Judith Austin for sharing unpublished results on let-653 and sma-1; Sarah Wurzelman, Christine Roy, and Michael Sepanski for expert assistance in electron microscopy; Michael Edidin, Andrew Fire, and Patricia Wilson for helpful discussions; and Adam Antebi, Harald Hutter, and Carolyn Norris for comments on the manuscript. Several mapping strains were obtained from the Caenorhabditis Genetic Center. M.B. received an NIH postdoctoral fellowship; this work was supported by grants from the NIH (NS26295) (RR12596) and the National Kidney Foundation of Maryland.
PY - 1999/10/1
Y1 - 1999/10/1
N2 - The excretory cell extends a tubular process, or canal, along the basolateral surface of the epidermis to form the nematode renal epithelium. This cell can undergo normal tubulogenesis in isolated cell culture. Mutations in 12 genes cause excretory canal cysts in Caenorhabditis elegans. Genetic interactions, and their similar phenotypes, suggest these genes may encode functionally related proteins. Depending upon genotype and individual canal, defects range from focal cysts, flanked by normal width segments, to regional cysts involving the entire tubule. Oftentimes the enlarged regions are convoluted or partially septated. In mutants with very large cysts, renal function is measurably impaired. Based on histology and ultrastructure, canal cysts likely result from defects of the apical membrane domain. These mutants provide a model of tubulocystic disease without hyperplasia or basement membrane abnormalities. Similar apical mechanisms could regulate tubular morphology of vertebrate nephrons.
AB - The excretory cell extends a tubular process, or canal, along the basolateral surface of the epidermis to form the nematode renal epithelium. This cell can undergo normal tubulogenesis in isolated cell culture. Mutations in 12 genes cause excretory canal cysts in Caenorhabditis elegans. Genetic interactions, and their similar phenotypes, suggest these genes may encode functionally related proteins. Depending upon genotype and individual canal, defects range from focal cysts, flanked by normal width segments, to regional cysts involving the entire tubule. Oftentimes the enlarged regions are convoluted or partially septated. In mutants with very large cysts, renal function is measurably impaired. Based on histology and ultrastructure, canal cysts likely result from defects of the apical membrane domain. These mutants provide a model of tubulocystic disease without hyperplasia or basement membrane abnormalities. Similar apical mechanisms could regulate tubular morphology of vertebrate nephrons.
KW - Excretory canals
KW - Nematode
KW - Polycystic kidney disease
KW - Renal epithelium
KW - Tubulogenesis
UR - http://www.scopus.com/inward/record.url?scp=0033214947&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=0033214947&partnerID=8YFLogxK
U2 - 10.1006/dbio.1999.9398
DO - 10.1006/dbio.1999.9398
M3 - Article
C2 - 10491271
AN - SCOPUS:0033214947
SN - 0012-1606
VL - 214
SP - 227
EP - 241
JO - Developmental Biology
JF - Developmental Biology
IS - 1
ER -