دورية أكاديمية
Expression characterization and functional implication of the collagen-modifying Leprecan proteins in mouse gonadal tissue and mature sperm.
| العنوان: | Expression characterization and functional implication of the collagen-modifying Leprecan proteins in mouse gonadal tissue and mature sperm. |
|---|---|
| المؤلفون: | Zimmerman SM; Department of Physiology & Biophysics, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Besio R; Department of Physiology & Biophysics, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Heard-Lipsmeyer ME; Department of Physiology & Biophysics, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Dimori M; Department of Physiology & Biophysics, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Castagnola P; IRCSS AOU-San Martino-IST, Genoa, Italy., Swain FL; Department of Orthopaedic Surgery, Center for Orthopaedic Research, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Gaddy D; Department of Physiology & Biophysics, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Diekman AB; Department of Biochemistry, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Morello R; Department of Physiology & Biophysics, University of Arkansas for Medical Sciences, Little Rock, AR, USA.; Department of Orthopaedic Surgery, Center for Orthopaedic Research, University of Arkansas for Medical Sciences, Little Rock, AR, USA.; Division of Genetics, University of Arkansas for Medical Sciences, Little Rock, AR, USA. |
| المصدر: | AIMS genetics [AIMS Genet] 2018; Vol. 5 (1), pp. 24-40. Date of Electronic Publication: 2018 Feb 07. |
| نوع المنشور: | Journal Article |
| اللغة: | English |
| بيانات الدورية: | Publisher: AIMS Press Country of Publication: United States NLM ID: 101634348 Publication Model: Print-Electronic Cited Medium: Print ISSN: 2377-1143 (Print) Linking ISSN: 23771143 NLM ISO Abbreviation: AIMS Genet Subsets: PubMed not MEDLINE |
| أسماء مطبوعة: | Original Publication: Springfield, MO : AIMS Press, [2014]-[2019] |
| مستخلص: | The Leprecan protein family which includes the prolyl 3-hydroxylase enzymes (P3H1, P3H2, and P3H3), the closely related cartilage-associated protein (CRTAP), and SC65 (Synaptonemal complex 65, aka P3H4, LEPREL4), is involved in the post-translational modification of fibrillar collagens. Mutations in CRTAP , P3H1 and P3H2 cause human genetic diseases. We recently showed that SC65 forms a stable complex in the endoplasmic reticulum with P3H3 and lysyl hydroxylase 1 and that loss of this complex leads to defective collagen lysyl hydroxylation and causes low bone mass and skin fragility. Interestingly, SC65 was initially described as a synaptonemal complex-associated protein, suggesting a potential additional role in germline cells. In the present study, we describe the expression of SC65, CRTAP and other Leprecan proteins in postnatal mouse reproductive organs. We detect SC65 expression in peritubular cells of testis up to 4 weeks of age but not in cells within seminiferous tubules, while its expression is maintained in ovarian follicles until adulthood. Similar to bone and skin, SC65 and P3H3 are also tightly co-expressed in testis and ovary. Moreover, we show that CRTAP, a protein normally involved in collagen prolyl 3-hydroxylation, is highly expressed in follicles and stroma of the ovary and in testes interstitial cells at 4 weeks of age, germline cells and mature sperm. Importantly, CrtapKO mice have a mild but significant increase in morphologically abnormal mature sperm (17% increase compared to WT). These data suggest a role for the Leprecans in the post-translational modification of collagens expressed in the stroma of the reproductive organs. While we could not confirm that SC65 is part of the synaptonemal complex, the expression of CRTAP in the seminiferous tubules and in mature sperm suggest a role in the testis germ cell lineage and sperm morphogenesis. Competing Interests: Conflict of interest All authors declare no conflicts of interest in this paper. |
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| معلومات مُعتمدة: | R01 AR060823 United States AR NIAMS NIH HHS; U54 TR001629 United States TR NCATS NIH HHS |
| فهرسة مساهمة: | Keywords: CRTAP; Leprecans; SC65; ovary; sperm morphology; testis |
| تواريخ الأحداث: | Date Created: 20181113 Latest Revision: 20200930 |
| رمز التحديث: | 20231215 |
| مُعرف محوري في PubMed: | PMC6221197 |
| DOI: | 10.3934/genet.2018.1.24 |
| PMID: | 30417103 |
| قاعدة البيانات: | MEDLINE |
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