Dilute Suppressor Dsu Acts Semidominantly to Suppress the Coat Color Phenotype of a Deletion Mutation, Dl20J, of the Murine Dilute Locus

Overview

Journal Proc Natl Acad Sci U S A

Specialty Science

Date 1988 Nov 1

PMID 3141922

Citations 15

Authors

K J Moore

P K Seperack

M C Strobel

D A Swing

N G Copeland

N A Jenkins

Affiliations

Soon will be listed here.

Abstract

The murine dilute suppressor (dsu) gene is the only unlinked trans-acting suppressor identified in mammals. dsu, which was originally reported to be recessive, was recognized by its ability to suppress the coat color phenotype of a retroviral insertion mutation, dv, of the murine dilute (d) locus. This insertion mutation resulted from the integration of an ecotropic murine leukemia virus into noncoding sequences of the dilute gene. Therefore, dsu may act like other allele-specific recessive suppressors identified in Drosophila melanogaster and yeast that suppress mutations induced by retrotransposon insertions. To investigate this possibility, we have examined whether dsu could suppress a spontaneously arising allele of d, dl20J, which is shown here to result from a 3.5-kilobase deletion. These studies indicate that dsu does not function like other eukaryotic suppressor genes that suppress retrotransposon-induced mutations. We also show that dsu is not, as originally reported, a recessive gene but is semidominantly inherited. Collectively, these results allow us to propose a mechanism for the suppressor activity of dsu.

Citing Articles

The Structure of Melanoregulin Reveals a Role for Cholesterol Recognition in the Protein's Ability to Promote Dynein Function.

Rout A, Wu X, Starich M, Strub M, Hammer J, Tjandra N Structure. 2018; 26(10):1373-1383.e4.

PMID: 30174147 PMC: 6170685. DOI: 10.1016/j.str.2018.07.009.

Melanoregulin, product of the dsu locus, links the BLOC-pathway and OA1 in organelle biogenesis.

Rachel R, Nagashima K, OSullivan T, Frost L, Stefano F, Marigo V PLoS One. 2012; 7(9):e42446.

PMID: 22984402 PMC: 3439427. DOI: 10.1371/journal.pone.0042446.

Melanoregulin regulates a shedding mechanism that drives melanosome transfer from melanocytes to keratinocytes.

Wu X, Masedunskas A, Weigert R, Copeland N, Jenkins N, Hammer J Proc Natl Acad Sci U S A. 2012; 109(31):E2101-9.

PMID: 22753477 PMC: 3412008. DOI: 10.1073/pnas.1209397109.

The myosin Va head domain binds to the neurofilament-L rod and modulates endoplasmic reticulum (ER) content and distribution within axons.

Rao M, Mohan P, Kumar A, Yuan A, Montagna L, Campbell J PLoS One. 2011; 6(2):e17087.

PMID: 21359212 PMC: 3040190. DOI: 10.1371/journal.pone.0017087.

Melanoregulin (MREG) modulates lysosome function in pigment epithelial cells.

Damek-Poprawa M, Diemer T, Lopes V, Lillo C, Harper D, Marks M J Biol Chem. 2009; 284(16):10877-89.

PMID: 19240024 PMC: 2667774. DOI: 10.1074/jbc.M808857200.

References

MARKERT C, Silvers W . The Effects of Genotype and Cell Environment on Melanoblast Differentiation in the House Mouse. Genetics. 1956; 41(3):429-50. PMC: 1209793. DOI: 10.1093/genetics/41.3.429. View

Rinchik E, Russell L, Copeland N, Jenkins N . Molecular genetic analysis of the dilute-short ear (d-se) region of the mouse. Genetics. 1986; 112(2):321-42. PMC: 1202704. DOI: 10.1093/genetics/112.2.321. View

Denhardt D . A membrane-filter technique for the detection of complementary DNA. Biochem Biophys Res Commun. 1966; 23(5):641-6. DOI: 10.1016/0006-291x(66)90447-5. View

RUSSELL E . A Quantitative Histological Study of the Pigment Found in the Coat Color Mutants of the House Mouse. II. Estimates of the Total Volume of Pigment. Genetics. 1948; 33(3):228-36. PMC: 1209407. DOI: 10.1093/genetics/33.3.228. View

Moore K, Swing D, Rinchik E, Mucenski M, Buchberg A, Copeland N . The murine dilute suppressor gene dsu suppresses the coat-color phenotype of three pigment mutations that alter melanocyte morphology, d, ash and ln. Genetics. 1988; 119(4):933-41. PMC: 1203476. DOI: 10.1093/genetics/119.4.933. View

Zachar Z, Davison D, Garza D, Bingham P . A detailed developmental and structural study of the transcriptional effects of insertion of the Copia transposon into the white locus of Drosophila melanogaster. Genetics. 1985; 111(3):495-515. PMC: 1202656. DOI: 10.1093/genetics/111.3.495. View

Jenkins N, Copeland N, Taylor B, Lee B . Dilute (d) coat colour mutation of DBA/2J mice is associated with the site of integration of an ecotropic MuLV genome. Nature. 1981; 293(5831):370-4. DOI: 10.1038/293370a0. View

Engstrom J, Wong A, Maurer R . Interaction of DNA polymerase III gamma and beta subunits in vivo in Salmonella typhimurium. Genetics. 1986; 113(3):499-515. PMC: 1202852. DOI: 10.1093/genetics/113.3.499. View

Thomas P . Hybridization of denatured RNA and small DNA fragments transferred to nitrocellulose. Proc Natl Acad Sci U S A. 1980; 77(9):5201-5. PMC: 350025. DOI: 10.1073/pnas.77.9.5201. View

10.

Parkhurst S, Corces V . Interactions among the gypsy transposable element and the yellow and the suppressor of hairy-wing loci in Drosophila melanogaster. Mol Cell Biol. 1986; 6(1):47-53. PMC: 367482. DOI: 10.1128/mcb.6.1.47-53.1986. View

11.

Parkhurst S, Corces V . Forked, gypsys, and suppressors in Drosophila. Cell. 1985; 41(2):429-37. DOI: 10.1016/s0092-8674(85)80016-7. View

12.

Siracusa L, Russell L, Jenkins N, Copeland N . Allelic variation within the Emv-15 locus defines genomic sequences closely linked to the agouti locus on mouse chromosome 2. Genetics. 1987; 117(1):85-92. PMC: 1203190. DOI: 10.1093/genetics/117.1.85. View

13.

Copeland N, Hutchison K, Jenkins N . Excision of the DBA ecotropic provirus in dilute coat-color revertants of mice occurs by homologous recombination involving the viral LTRs. Cell. 1983; 33(2):379-87. DOI: 10.1016/0092-8674(83)90419-1. View

14.

Weil C, Oakley C, Oakley B . Isolation of mip (microtubule-interacting protein) mutations of Aspergillus nidulans. Mol Cell Biol. 1986; 6(8):2963-8. PMC: 367866. DOI: 10.1128/mcb.6.8.2963-2968.1986. View

15.

Goldberg D . Isolation and partial characterization of the Drosophila alcohol dehydrogenase gene. Proc Natl Acad Sci U S A. 1980; 77(10):5794-8. PMC: 350157. DOI: 10.1073/pnas.77.10.5794. View

16.

Kato N, Kato M, Larsson E, Rydnert J, Ohlsson R, Cohen M . Tissue-specific expression of human provirus ERV3 mRNA in human placenta: two of the three ERV3 mRNAs contain human cellular sequences. J Virol. 1987; 61(7):2182-91. PMC: 254242. DOI: 10.1128/JVI.61.7.2182-2191.1987. View

17.

Sweet H . Dilute suppressor, a new suppressor gene in the house mouse. J Hered. 1983; 74(4):305-6. DOI: 10.1093/oxfordjournals.jhered.a109794. View

18.

Kost T, Theodorakis N, Hughes S . The nucleotide sequence of the chick cytoplasmic beta-actin gene. Nucleic Acids Res. 1983; 11(23):8287-301. PMC: 326582. DOI: 10.1093/nar/11.23.8287. View

19.

Wahl G, Stern M, Stark G . Efficient transfer of large DNA fragments from agarose gels to diazobenzyloxymethyl-paper and rapid hybridization by using dextran sulfate. Proc Natl Acad Sci U S A. 1979; 76(8):3683-7. PMC: 383897. DOI: 10.1073/pnas.76.8.3683. View

20.

Hu F, LESNEY P . THE ISOLATION AND CYTOLOGY OF TWO PIGMENT CELL STRAINS FROM B-16 MOUSE MELANOMAS. Cancer Res. 1964; 24:1634-43. View