Literature DB >> 26293594

Exome Sequencing of SLC30A2 Identifies Novel Loss- and Gain-of-Function Variants Associated with Breast Cell Dysfunction.

Samina Alam1,2, Stephen R Hennigar3, Carla Gallagher4, David I Soybel1,2, Shannon L Kelleher5,6,7,8.   

Abstract

The zinc (Zn) transporter ZnT2 (SLC30A2) is expressed in specialized secretory cells including breast, pancreas and prostate, and imports Zn into mitochondria and vesicles. Mutations in SLC30A2 substantially reduce milk Zn concentration ([Zn]) and cause severe Zn deficiency in exclusively breastfed infants. Recent studies show that ZnT2-null mice have low milk [Zn], in addition to profound defects in mammary gland function during lactation. Here, we used breast milk [Zn] to identify novel non-synonymous ZnT2 variants in a population of lactating women. We also asked whether specific variants induce disturbances in intracellular Zn management or cause cellular dysfunction in mammary epithelial cells. Healthy, breastfeeding women were stratified into quartiles by milk [Zn] and exonic sequencing of SLC30A2 was performed. We found that 36% of women tested carried non-synonymous ZnT2 variants, all of whom had milk Zn levels that were distinctly above or below those in women without variants. We identified 12 novel heterozygous variants. Two variants (D(103)E and T(288)S) were identified with high frequency (9 and 16%, respectively) and expression of T(288)S was associated with a known hallmark of breast dysfunction (elevated milk sodium/potassium ratio). Select variants (A(28)D, K(66)N, Q(71)H, D(103)E, A(105)P, Q(137)H, T(288)S and T(312)K) were characterized in vitro. Compared with wild-type ZnT2, these variants were inappropriately localized, and most resulted in either 'loss-of-function' or 'gain-of-function', and altered sub-cellular Zn pools, Zn secretion, and cell cycle check-points. Our study indicates that SLC30A2 variants are common in this population, dysregulate Zn management and can lead to breast cell dysfunction. This suggests that genetic variation in ZnT2 could be an important modifier of infant growth/development and reproductive health/disease. Importantly, milk [Zn] level may serve as a bio-reporter of breast function during lactation.

Entities:  

Keywords:  Breast dysfunction; Breast milk zinc; Lactation; SLC30A2; Zinc secretion; ZnT2

Mesh:

Substances:

Year:  2015        PMID: 26293594     DOI: 10.1007/s10911-015-9338-z

Source DB:  PubMed          Journal:  J Mammary Gland Biol Neoplasia        ISSN: 1083-3021            Impact factor:   2.673


  52 in total

1.  Crystal structure of the cytosolic domain of the cation diffusion facilitator family protein.

Authors:  Takashi Higuchi; Motoyuki Hattori; Yoshiki Tanaka; Ryuichiro Ishitani; Osamu Nureki
Journal:  Proteins       Date:  2009-08-15

2.  Identification of the Zn2+ binding site and mode of operation of a mammalian Zn2+ transporter.

Authors:  Ehud Ohana; Eitan Hoch; Chen Keasar; Taiho Kambe; Ofer Yifrach; Michal Hershfinkel; Israel Sekler
Journal:  J Biol Chem       Date:  2009-04-14       Impact factor: 5.157

3.  In situ dimerization of multiple wild type and mutant zinc transporters in live cells using bimolecular fluorescence complementation.

Authors:  Inbal Lasry; Yarden Golan; Bluma Berman; Noy Amram; Fabian Glaser; Yehuda G Assaraf
Journal:  J Biol Chem       Date:  2014-01-22       Impact factor: 5.157

4.  ZnT4 provides zinc to zinc-dependent proteins in the trans-Golgi network critical for cell function and Zn export in mammary epithelial cells.

Authors:  Nicholas H McCormick; Shannon L Kelleher
Journal:  Am J Physiol Cell Physiol       Date:  2012-05-23       Impact factor: 4.249

5.  Mutant prolactin receptor and familial hyperprolactinemia.

Authors:  Paul J Newey; Caroline M Gorvin; Rajesh V Thakker
Journal:  N Engl J Med       Date:  2014-03-06       Impact factor: 91.245

6.  Lethal milk mutation results in dietary zinc deficiency in nursing mice.

Authors:  J A Piletz; R E Ganschow
Journal:  Am J Clin Nutr       Date:  1978-04       Impact factor: 7.045

7.  X-ray fluorescence microscopy reveals accumulation and secretion of discrete intracellular zinc pools in the lactating mouse mammary gland.

Authors:  Nicholas McCormick; Vanessa Velasquez; Lydia Finney; Stefan Vogt; Shannon L Kelleher
Journal:  PLoS One       Date:  2010-06-11       Impact factor: 3.240

Review 8.  Roles of zinc and metallothionein-3 in oxidative stress-induced lysosomal dysfunction, cell death, and autophagy in neurons and astrocytes.

Authors:  Sook-Jeong Lee; Jae-Young Koh
Journal:  Mol Brain       Date:  2010-10-26       Impact factor: 4.041

9.  Analysis of zinc transporter, hZnT4 ( Slc30A4), gene expression in a mammary gland disorder leading to reduced zinc secretion into milk.

Authors:  Agnes Michalczyk; George Varigos; Anthony Catto-Smith; Rachael C Blomeley; M Leigh Ackland
Journal:  Hum Genet       Date:  2003-05-13       Impact factor: 4.132

10.  Copper, iron, and zinc contents of human milk at early stages of lactation.

Authors:  R M Feeley; R R Eitenmiller; J B Jones; H Barnhart
Journal:  Am J Clin Nutr       Date:  1983-03       Impact factor: 7.045

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  14 in total

Review 1.  Biological underpinnings of breastfeeding challenges: the role of genetics, diet, and environment on lactation physiology.

Authors:  Sooyeon Lee; Shannon L Kelleher
Journal:  Am J Physiol Endocrinol Metab       Date:  2016-06-28       Impact factor: 4.310

2.  In silico mapping of quantitative trait loci (QTL) regulating the milk ionome in mice identifies a milk iron locus on chromosome 1.

Authors:  Darryl L Hadsell; Louise A Hadsell; Monique Rijnkels; Yareli Carcamo-Bahena; Jerry Wei; Peter Williamson; Michael A Grusak
Journal:  Mamm Genome       Date:  2018-08-02       Impact factor: 2.957

3.  Zinc transporter 2 interacts with vacuolar ATPase and is required for polarization, vesicle acidification, and secretion in mammary epithelial cells.

Authors:  Sooyeon Lee; Olivia C Rivera; Shannon L Kelleher
Journal:  J Biol Chem       Date:  2017-11-07       Impact factor: 5.157

4.  Molecular Basis of Transient Neonatal Zinc Deficiency: NOVEL ZnT2 MUTATIONS DISRUPTING ZINC BINDING AND PERMEATION.

Authors:  Yarden Golan; Naoya Itsumura; Fabian Glaser; Bluma Berman; Taiho Kambe; Yehuda G Assaraf
Journal:  J Biol Chem       Date:  2016-05-02       Impact factor: 5.157

5.  Whole-Genome Resequencing of Holstein Bulls for Indel Discovery and Identification of Genes Associated with Milk Composition Traits in Dairy Cattle.

Authors:  Jianping Jiang; Yahui Gao; Yali Hou; Wenhui Li; Shengli Zhang; Qin Zhang; Dongxiao Sun
Journal:  PLoS One       Date:  2016-12-28       Impact factor: 3.240

Review 6.  Genetic Variations as Modifying Factors to Dietary Zinc Requirements-A Systematic Review.

Authors:  Kaitlin J Day; Melissa M Adamski; Aimee L Dordevic; Chiara Murgia
Journal:  Nutrients       Date:  2017-02-17       Impact factor: 5.717

7.  ZnT2-Mediated Zinc Import Into Paneth Cell Granules Is Necessary for Coordinated Secretion and Paneth Cell Function in Mice.

Authors:  Abigail B Podany; Justin Wright; Regina Lamendella; David I Soybel; Shannon L Kelleher
Journal:  Cell Mol Gastroenterol Hepatol       Date:  2016-01-08

8.  Evaluation of the roles of the cytosolic N-terminus and His-rich loop of ZNT proteins using ZNT2 and ZNT3 chimeric mutants.

Authors:  Kazuhisa Fukue; Naoya Itsumura; Natsuko Tsuji; Katsutoshi Nishino; Masaya Nagao; Hiroshi Narita; Taiho Kambe
Journal:  Sci Rep       Date:  2018-09-20       Impact factor: 4.379

9.  A genetic variant in SLC30A2 causes breast dysfunction during lactation by inducing ER stress, oxidative stress and epithelial barrier defects.

Authors:  Sooyeon Lee; Yandong Zhou; Donald L Gill; Shannon L Kelleher
Journal:  Sci Rep       Date:  2018-02-23       Impact factor: 4.379

10.  Zinc Content in Breast Milk and Its Association with Maternal Diet.

Authors:  Līva Aumeistere; Inga Ciproviča; Dace Zavadska; Konstantīns Bavrins; Anastasija Borisova
Journal:  Nutrients       Date:  2018-10-05       Impact factor: 5.717

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