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Knockout of Murine Mamld1 Impairs Testicular Growth and Daily Sperm Production but Permits Normal Postnatal Androgen Production and Fertility

MAMLD1 has been implicated in testicular function in both human and mouse fetuses. Although three patients with MAMLD1 mutations were reported to have hypergonadotropic hypogonadism in their teens, the functional significance of MAMLD1 in the postnatal testis remains unclear. Here, we analyzed the p...

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Autores principales: Miyado, Mami, Yoshida, Kaoru, Miyado, Kenji, Katsumi, Momori, Saito, Kazuki, Nakamura, Shigeru, Ogata, Tsutomu, Fukami, Maki
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5486121/
https://www.ncbi.nlm.nih.gov/pubmed/28629181
http://dx.doi.org/10.3390/ijms18061300
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author Miyado, Mami
Yoshida, Kaoru
Miyado, Kenji
Katsumi, Momori
Saito, Kazuki
Nakamura, Shigeru
Ogata, Tsutomu
Fukami, Maki
author_facet Miyado, Mami
Yoshida, Kaoru
Miyado, Kenji
Katsumi, Momori
Saito, Kazuki
Nakamura, Shigeru
Ogata, Tsutomu
Fukami, Maki
author_sort Miyado, Mami
collection PubMed
description MAMLD1 has been implicated in testicular function in both human and mouse fetuses. Although three patients with MAMLD1 mutations were reported to have hypergonadotropic hypogonadism in their teens, the functional significance of MAMLD1 in the postnatal testis remains unclear. Here, we analyzed the phenotype of Mamld1 knockout (KO) male mice at reproductive ages. The reproductive organs of KO male mice were morphologically unremarkable, except for relatively small testes. Seminiferous tubule size and number of proliferating spermatogonia/spermatocytes were reduced in the KO testis. Daily sperm production of KO mice was mildly attenuated, whereas total sperm counts in epididymal semen remained normal. Sperm motility and morphology, as well as androgen levels in serum and testicular tissues and the number of pups born from cross-mated wildtype (WT) female mice, were comparable between WT and KO male mice. These results indicate that MAMLD1 contributes to the maintenance of postnatal testicular growth and daily sperm production but is dispensable for androgen biosynthesis and fertility. MAMLD1 likely plays supporting roles in multiple and continuous steps of male reproduction.
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spelling pubmed-54861212017-06-29 Knockout of Murine Mamld1 Impairs Testicular Growth and Daily Sperm Production but Permits Normal Postnatal Androgen Production and Fertility Miyado, Mami Yoshida, Kaoru Miyado, Kenji Katsumi, Momori Saito, Kazuki Nakamura, Shigeru Ogata, Tsutomu Fukami, Maki Int J Mol Sci Article MAMLD1 has been implicated in testicular function in both human and mouse fetuses. Although three patients with MAMLD1 mutations were reported to have hypergonadotropic hypogonadism in their teens, the functional significance of MAMLD1 in the postnatal testis remains unclear. Here, we analyzed the phenotype of Mamld1 knockout (KO) male mice at reproductive ages. The reproductive organs of KO male mice were morphologically unremarkable, except for relatively small testes. Seminiferous tubule size and number of proliferating spermatogonia/spermatocytes were reduced in the KO testis. Daily sperm production of KO mice was mildly attenuated, whereas total sperm counts in epididymal semen remained normal. Sperm motility and morphology, as well as androgen levels in serum and testicular tissues and the number of pups born from cross-mated wildtype (WT) female mice, were comparable between WT and KO male mice. These results indicate that MAMLD1 contributes to the maintenance of postnatal testicular growth and daily sperm production but is dispensable for androgen biosynthesis and fertility. MAMLD1 likely plays supporting roles in multiple and continuous steps of male reproduction. MDPI 2017-06-19 /pmc/articles/PMC5486121/ /pubmed/28629181 http://dx.doi.org/10.3390/ijms18061300 Text en © 2017 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Miyado, Mami
Yoshida, Kaoru
Miyado, Kenji
Katsumi, Momori
Saito, Kazuki
Nakamura, Shigeru
Ogata, Tsutomu
Fukami, Maki
Knockout of Murine Mamld1 Impairs Testicular Growth and Daily Sperm Production but Permits Normal Postnatal Androgen Production and Fertility
title Knockout of Murine Mamld1 Impairs Testicular Growth and Daily Sperm Production but Permits Normal Postnatal Androgen Production and Fertility
title_full Knockout of Murine Mamld1 Impairs Testicular Growth and Daily Sperm Production but Permits Normal Postnatal Androgen Production and Fertility
title_fullStr Knockout of Murine Mamld1 Impairs Testicular Growth and Daily Sperm Production but Permits Normal Postnatal Androgen Production and Fertility
title_full_unstemmed Knockout of Murine Mamld1 Impairs Testicular Growth and Daily Sperm Production but Permits Normal Postnatal Androgen Production and Fertility
title_short Knockout of Murine Mamld1 Impairs Testicular Growth and Daily Sperm Production but Permits Normal Postnatal Androgen Production and Fertility
title_sort knockout of murine mamld1 impairs testicular growth and daily sperm production but permits normal postnatal androgen production and fertility
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5486121/
https://www.ncbi.nlm.nih.gov/pubmed/28629181
http://dx.doi.org/10.3390/ijms18061300
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