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MUL is caused by loss-of-function mutations in TRIM37, which encodes an E3 ubiquitin ligase belonging to the tripartite motif (TRIM) protein family and having both peroxisomal and nuclear localization. We describe a congenic Trim37 knock-out mouse (Trim37\u2212\/\u2212) model for MUL. Trim37\u2212\/\u2212 mice were viable and had normal weight development until approximately 12\u2005months of age, after which they started to manifest increasing problems in wellbeing and weight loss. Assessment of skeletal parameters with computer tomography revealed significantly smaller skull size, but no difference in the lengths of long bones in Trim37\u2212\/\u2212 mice as compared with wild-type. Both male and female Trim37\u2212\/\u2212 mice were infertile, the gonads showing germ cell aplasia, hilus and Leydig cell hyperplasia and accumulation of lipids in and around Leydig cells. Male Trim37\u2212\/\u2212 mice had elevated levels of follicle-stimulating and luteinizing hormones, but maintained normal levels of testosterone. Six-month-old Trim37\u2212\/\u2212 mice had elevated fasting blood glucose and low fasting serum insulin levels. At 1.5\u2005years Trim37\u2212\/\u2212 mice showed non-compaction cardiomyopathy, hepatomegaly, fatty liver and various tumors. The amount and morphology of liver peroxisomes seemed normal in Trim37\u2212\/\u2212 mice. The most consistently seen phenotypes in Trim37\u2212\/\u2212 mice were infertility and the associated hormonal findings, whereas there was more variability in the other phenotypes observed. Trim37\u2212\/\u2212 mice recapitulate several features of the human MUL disease and thus provide a good model to study disease pathogenesis related to TRIM37 deficiency, including infertility, non-alcoholic fatty liver disease, cardiomyopathy and tumorigenesis.<\/jats:p>","DOI":"10.1242\/bio.016246","type":"journal-article","created":{"date-parts":[[2016,4,5]],"date-time":"2016-04-05T00:43:54Z","timestamp":1459817034000},"page":"584-595","source":"Crossref","is-referenced-by-count":26,"title":["<i>Trim37<\/i>-deficient mice recapitulate several features of the multi-organ disorder Mulibrey nanism"],"prefix":"10.1242","volume":"5","author":[{"given":"Kaisa M.","family":"Kettunen","sequence":"first","affiliation":[{"name":"Folkh\u00e4lsan Institute of Genetics, FI-00290 Helsinki, Finland"},{"name":"Research Programs Unit, Molecular Neurology, University of Helsinki, FI-00290 Helsinki, Finland"},{"name":"Neuroscience Center, University of Helsinki, FI-00790 Helsinki, Finland"},{"name":"Institute for Molecular Medicine Finland FIMM, University of Helsinki, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Riitta","family":"Karikoski","sequence":"additional","affiliation":[{"name":"Department of Pathology, Central Hospital of Tavastia, FI-13530 H\u00e4meenlinna, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Riikka H.","family":"H\u00e4m\u00e4l\u00e4inen","sequence":"additional","affiliation":[{"name":"Department of Neurobiology, A. 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Virtanen Institute for Molecular Sciences, University of Eastern Finland, KuopioFI-70211, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teija T.","family":"Toivonen","sequence":"additional","affiliation":[{"name":"Folkh\u00e4lsan Institute of Genetics, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vasily D.","family":"Antonenkov","sequence":"additional","affiliation":[{"name":"Biocenter Oulu and Faculty of Biochemistry and Molecular Medicine, University of Oulu, FI-90220 Oulu, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natalia","family":"Kulesskaya","sequence":"additional","affiliation":[{"name":"Neuroscience Center, University of Helsinki, FI-00790 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vootele","family":"Voikar","sequence":"additional","affiliation":[{"name":"Neuroscience Center, University of Helsinki, FI-00790 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maarit","family":"H\u00f6ltt\u00e4-Vuori","sequence":"additional","affiliation":[{"name":"Department of Anatomy, Faculty of Medicine, University of Helsinki, FI-00290 Helsinki, Finland"},{"name":"Minerva Foundation Institute for Medical Research, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elina","family":"Ikonen","sequence":"additional","affiliation":[{"name":"Department of Anatomy, Faculty of Medicine, University of Helsinki, FI-00290 Helsinki, Finland"},{"name":"Minerva Foundation Institute for Medical Research, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kirsi","family":"Sainio","sequence":"additional","affiliation":[{"name":"Biochemistry and Developmental Biology, Institute of Biomedicine, University of Helsinki, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anu","family":"Jalanko","sequence":"additional","affiliation":[{"name":"Genomics and Biomarkers Unit, National Institute for Health and Welfare, Biomedicum, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Susann","family":"Karlberg","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Children's Hospital, University of Helsinki and Helsinki University Hospital, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Niklas","family":"Karlberg","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Children's Hospital, University of Helsinki and Helsinki University Hospital, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marita","family":"Lipsanen-Nyman","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Children's Hospital, University of Helsinki and Helsinki University Hospital, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jorma","family":"Toppari","sequence":"additional","affiliation":[{"name":"Departments of Physiology and Pediatrics, University of Turku, FI-20520 Turku, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matti","family":"Jauhiainen","sequence":"additional","affiliation":[{"name":"Genomics and Biomarkers Unit, National Institute for Health and Welfare, Biomedicum, FI-00290 Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J. 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