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https://www.readbyqxmd.com/read/29163677/trim37-promotes-tumor-cell-proliferation-and-drug-resistance-in-pediatric-osteosarcoma
#1
Yanling Tao, Meiyun Xin, Huanchen Cheng, Zongxuan Huang, Tiantian Hu, Teng Zhang, Jianlong Wang
Osteosarcoma (OS) is among the most frequently occurring bone tumors, particularly in children. Clinical treatment of OS is limited due to several factors including resistance to chemotherapy drugs and metastasis, and the underlying molecular mechanisms remain unclear. In the present study, tripartite motif containing 37 (TRIM37) expression levels were upregulated in tumor samples and associated with the development of drug resistance in OS. Furthermore, chemotherapy drug treatment (doxorubicin, cisplatin and methotrexate) induced TRIM37 expression in OS cells in vitro...
December 2017: Oncology Letters
https://www.readbyqxmd.com/read/28815877/new-intragenic-rearrangements-in-non-finnish-mulibrey-nanism
#2
Florence Jobic, Gilles Morin, Catherine Vincent-Delorme, Estelle Cadet, Rosalie Cabry, Michèle Mathieu-Dramard, Henri Copin, Jacques Rochette, Guillaume Jedraszak
Prenatal growth is a complex dynamic process controlled by various genetic and environmental factors. Among genetic syndromes characterized by growth restriction, MULIBREY nanism represents a rare autosomal recessive condition presenting with severe pre- and post-natal growth failure, characteristic dysmorphic features but normal neurological development. The phenotype of MULIBREY nanism is variable and overlaps with others such as the Silver-Russell syndrome. We report here three patients in two distinct non-Finnish families from North France who were first suspected to have Silver-Russell syndrome which failed to be confirmed on molecular analyses...
August 17, 2017: American Journal of Medical Genetics. Part A
https://www.readbyqxmd.com/read/28724525/trim37-a-novel-e3-ligase-for-pex5-mediated-peroxisomal-matrix-protein-import
#3
Wei Wang, Zhi-Jie Xia, Jean-Claude Farré, Suresh Subramani
Most proteins destined for the peroxisomal matrix depend on the peroxisomal targeting signals (PTSs), which require the PTS receptor PEX5, whose deficiency causes fatal human peroxisomal biogenesis disorders (PBDs). TRIM37 gene mutations cause muscle-liver-brain-eye (mulibrey) nanism. We found that TRIM37 localizes in peroxisomal membranes and ubiquitylates PEX5 at K464 by interacting with its C-terminal 51 amino acids (CT51), which is required for PTS protein import. PEX5 mutations (K464A or ΔCT51), or TRIM37 depletion or mutation, reduce PEX5 abundance by promoting its proteasomal degradation, thereby impairing its functions in cargo binding and PTS protein import in human cells...
September 4, 2017: Journal of Cell Biology
https://www.readbyqxmd.com/read/28496510/constrictive-pericarditis-and-primary-amenorrhea-with-syndactyly-in-an-iranian-female-mulibrey-nanism-syndrome
#4
Tahereh Davarpasand, Maryam Sotoudeh Anvari, Mohammad Naderan, Mohammad Ali Boroumand, Hossein Ahmadi
Mulibrey nanism is a rare autosomal recessive syndrome caused by a mutation in the TRIM37 gene with severe growth retardation and multiple organ involvement. Early diagnosis is important because 50% of the patients develop congestive heart failure owing to constrictive pericarditis, and this condition plays a critical role in the final prognosis. A 37-year-old female patient presented with symptoms of dyspnea on exertion and shortness of breath. She had severe growth failure and craniofacial dysmorphic feature...
October 3, 2016: Journal of Tehran Heart Center
https://www.readbyqxmd.com/read/28432469/renal-findings-in-patients-with-mulibrey-nanism
#5
Johanna Sivunen, Susann Karlberg, Jouko Lohi, Niklas Karlberg, Marita Lipsanen-Nyman, Hannu Jalanko
BACKGROUND: Mulibrey nanism (MUL) is a rare inherited disease caused by genetic defects affecting peroxisomal TRIM37 protein. MUL affects multiple organs, leading to growth retardation and early onset type 2 diabetes. We aimed to characterize the structure and function of kidneys and the urinary tract in a large cohort of Finnish MUL patients. METHODS: Ultrasound, magnetic resonance imaging (MRI), and autopsy findings of the kidneys and urinary tract from 101 MUL patients were retrospectively analyzed...
April 22, 2017: Pediatric Nephrology: Journal of the International Pediatric Nephrology Association
https://www.readbyqxmd.com/read/28098873/trim37-promotes-epithelial%C3%A2-mesenchymal-transition-in-colorectal-cancer
#6
Cheng-En Hu, Jun Gan
There is substantial research on the oncogenic role of tripartite motif containing 37 (TRIM37); however, its importance in colorectal cancer (CRC) remains to be elucidated. The present study used reverse transcription‑quantitative polymerase chain reaction, immunohistochemistry and western blotting to detect the expression level of TRIM37 in CRC. The importance of TRIM37 in cell proliferation, invasion and metastasis of CRC were investigated through overexpressing or knocking‑down of TRIM37 in CRC cell lines, to observe its function...
March 2017: Molecular Medicine Reports
https://www.readbyqxmd.com/read/27832544/in-vitro-assay-to-study-histone-ubiquitination-during-transcriptional-regulation
#7
Jogender Tushir-Singh, Sanchita Bhatnagar
In mammals, gene expression is largely controlled at the transcriptional level. In response to environmental or intrinsic signaling, gene expression is often fine-tuned by epigenetic modifications, including DNA methylation and histone modifications. One such histone modification is ubiquitination that predominately occurs in mono-ubiquitinated forms on histone H2A and H2B. We recently identified and characterized a novel E3 ligase called TRIM37 that ubiquitinates H2A. This study highlights the consequence of aberrant histone ubiquitination at the promoters of tumor suppressor genes in breast cancer...
2017: Methods in Molecular Biology
https://www.readbyqxmd.com/read/27432897/53bp1-and-usp28-mediate-p53-activation-and-g1-arrest-after-centrosome-loss-or-extended-mitotic-duration
#8
Franz Meitinger, John V Anzola, Manuel Kaulich, Amelia Richardson, Joshua D Stender, Christopher Benner, Christopher K Glass, Steven F Dowdy, Arshad Desai, Andrew K Shiau, Karen Oegema
In normal human cells, centrosome loss induced by centrinone-a specific centrosome duplication inhibitor-leads to irreversible, p53-dependent G1 arrest by an unknown mechanism. A genome-wide CRISPR/Cas9 screen for centrinone resistance identified genes encoding the p53-binding protein 53BP1, the deubiquitinase USP28, and the ubiquitin ligase TRIM37. Deletion of TP53BP1, USP28, or TRIM37 prevented p53 elevation in response to centrosome loss but did not affect cytokinesis failure-induced arrest or p53 elevation after doxorubicin-induced DNA damage...
July 18, 2016: Journal of Cell Biology
https://www.readbyqxmd.com/read/27044324/trim37-deficient-mice-recapitulate-several-features-of-the-multi-organ-disorder-mulibrey-nanism
#9
Kaisa M Kettunen, Riitta Karikoski, Riikka H Hämäläinen, Teija T Toivonen, Vasily D Antonenkov, Natalia Kulesskaya, Vootele Voikar, Maarit Hölttä-Vuori, Elina Ikonen, Kirsi Sainio, Anu Jalanko, Susann Karlberg, Niklas Karlberg, Marita Lipsanen-Nyman, Jorma Toppari, Matti Jauhiainen, J Kalervo Hiltunen, Hannu Jalanko, Anna-Elina Lehesjoki
Mulibrey nanism (MUL) is a rare autosomal recessive multi-organ disorder characterized by severe prenatal-onset growth failure, infertility, cardiopathy, risk for tumors, fatty liver, and type 2 diabetes. 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(-/-)) model for MUL. Trim37(-/-) mice were viable and had normal weight development until approximately 12 months of age, after which they started to manifest increasing problems in wellbeing and weight loss...
2016: Biology Open
https://www.readbyqxmd.com/read/26395261/trim37-promoted-the-growth-and-migration-of-the-pancreatic-cancer-cells
#10
Jianxin Jiang, She Tian, Chao Yu, Meiyuan Chen, Chengyi Sun
Increasing evidence indicated that tripartite motif containing 37 (TRIM37) was involved in the tumorigenesis of several cancer types. However, its expression pattern and biological functions in pancreatic ductal adenocarcinoma (PDAC) remained unknown. In this study, real-time PCR, Western blot and immunohistochemistry was performed to examine the expression of TRIM37 in the pancreatic cancerous tissues. Colony formation assay and cell migration assay were performed to study the functions of TRIM37 in pancreatic cancer cells...
February 2016: Tumour Biology: the Journal of the International Society for Oncodevelopmental Biology and Medicine
https://www.readbyqxmd.com/read/26208456/over-expression-of-trim37-promotes-cell-migration-and-metastasis-in-hepatocellular-carcinoma-by-activating-wnt-%C3%AE-catenin-signaling
#11
Jianxin Jiang, Chao Yu, Meiyuan Chen, She Tian, Chengyi Sun
Hepatocellular carcinoma (HCC) is the most common cancer in the world especially in East Asia and Africa. Advanced stage, metastasis and frequent relapse are responsible for the poor prognosis of HCC. However, the precise mechanisms underlying HCC remained unclear. So it is urgent to identify the pathological processes and relevant molecules of HCC. TRIM37 is an E3 ligase and has been observed deregulated expression in various tumors. Recent studies of TRIM37 have implicated that TRIM37 played critical roles in cell proliferation and other processes...
September 4, 2015: Biochemical and Biophysical Research Communications
https://www.readbyqxmd.com/read/25470042/trim37-is-a-new-histone-h2a-ubiquitin-ligase-and-breast-cancer-oncoprotein
#12
Sanchita Bhatnagar, Claude Gazin, Lynn Chamberlain, Jianhong Ou, Xiaochun Zhu, Jogender S Tushir, Ching-Man Virbasius, Ling Lin, Lihua J Zhu, Narendra Wajapeyee, Michael R Green
The TRIM37 (also known as MUL) gene is located in the 17q23 chromosomal region, which is amplified in up to ∼ 40% of breast cancers. TRIM37 contains a RING finger domain, a hallmark of E3 ubiquitin ligases, but its protein substrate(s) is unknown. Here we report that TRIM37 mono-ubiquitinates histone H2A, a chromatin modification associated with transcriptional repression. We find that in human breast cancer cell lines containing amplified 17q23, TRIM37 is upregulated and, reciprocally, the major H2A ubiquitin ligase RNF2 (also known as RING1B) is downregulated...
December 4, 2014: Nature
https://www.readbyqxmd.com/read/24357726/whole-genome-integrative-analysis-reveals-expression-signatures-predicting-transformation-in-follicular-lymphoma
#13
Marianne Brodtkorb, Ole Christian Lingjærde, Kanutte Huse, Gunhild Trøen, Marit Hystad, Vera I Hilden, June H Myklebust, Ellen Leich, Andreas Rosenwald, Jan Delabie, Harald Holte, Erlend B Smeland
Transformation of follicular lymphoma (FL) to a more aggressive disease is associated with rapid progression and death. Existing molecular markers for transformation are few and their clinical impact is limited. Here, we report on a whole-genome study of DNA copy numbers and gene expression profiles in serial FL biopsies. We identified 698 genes with high correlation between gene expression and copy number, and the molecular network most enriched for these cis-associated genes. This network includes 14 cis-associated genes directly related to the nuclear factor κB (NF-κB) pathway...
February 13, 2014: Blood
https://www.readbyqxmd.com/read/23769972/discovering-regulators-of-centriole-biogenesis-through-sirna-based-functional-genomics-in-human-cells
#14
Fernando R Balestra, Petr Strnad, Isabelle Flückiger, Pierre Gönczy
Centrioles are essential for forming cilia, flagella, and centrosomes and are thus critical for a range of fundamental cellular processes. Despite their importance, the mechanisms governing centriole biogenesis remain incompletely understood. We performed a high-content genome-wide small-interfering-RNA-based screen to identify genes regulating centriole formation in human cells. We designed an algorithm to automatically detect GFP-Centrin foci that, combined with subsequent manual analysis, allowed us to identify 44 genes required for centriole formation and 32 genes needed for restricting centriole number...
June 24, 2013: Developmental Cell
https://www.readbyqxmd.com/read/23385855/refractory-congestive-heart-failure-following-delayed-pericardectomy-in-a-12-year-old-child-with-mulibrey-nanism-due-to-a-novel-mutation-in-trim37
#15
Matthias Kumpf, Riikka H Hämäläinen, Michael Hofbeck, Winfried Baden
UNLABELLED: Mulibrey nanism (MUL) is a rare autosomal recessive disorder with severe primordial growth retardation and multiorgan involvement, caused by mutations in TRIM37. Early clinical detection is important since more than 50 % of the patients develop congestive heart failure. We report a 12-year-old patient who presented in infancy with severe growth retardation, dysmorphic features, and cleft palate. Clinical diagnosis of MUL was established at the age of 5 years. Postmortem, molecular diagnostic confirmed MUL as a novel 1-bp deletion (c...
October 2013: European Journal of Pediatrics
https://www.readbyqxmd.com/read/21966607/asthma-predictive-genetic-markers-in-gene-expression-profiling-of-peripheral-blood-mononuclear-cells
#16
Seung Woo Shin, Tae Jeong Oh, Se-Min Park, Jong Sook Park, An Soo Jang, Sung Woo Park, Soo Taek Uh, Sungwhan An, Choon-Sik Park
PURPOSE: We sought to identify asthma-related genes and to examine the potential of these genes to predict asthma, based on expression levels. METHODS: The subjects were 42 asthmatics and 10 normal healthy controls. PBMC RNA was subjected to microarray analysis using a 35K array; t-tests were used to identify genes that were expressed differentially between the two groups. A multiple logistic regression analysis was applied to the differentially expressed genes, and area under the curve (AUC) values from receiver operating characteristic (ROC) curves were obtained...
October 2011: Allergy, Asthma & Immunology Research
https://www.readbyqxmd.com/read/21865362/testicular-failure-and-male-infertility-in-the-monogenic-mulibrey-nanism-disorder
#17
Susann Karlberg, Jorma Toppari, Niklas Karlberg, Mirja Nurmio, Riitta Karikoski, Hannu Jalanko, Marita Lipsanen-Nyman
CONTEXT: Few monogenic mutations causing human male infertility have been identified to date. OBJECTIVE: We studied pubertal development and fecundity in males with Mulibrey nanism (MUL) caused by mutations in the TRIM37 gene. DESIGN, SETTING, AND PATIENTS: Twenty-eight male MUL patients of the Finnish national cohort aged 8.7 to 50.0 yr (median age, 28.8) at the end of observation were followed for 10 yr beginning from 2000-2001. MAIN OUTCOME MEASURES: Clinical characteristics, reproductive hormone levels, semen quality, and testicular histology were assessed...
November 2011: Journal of Clinical Endocrinology and Metabolism
https://www.readbyqxmd.com/read/21604015/prognostic-value-of-acquired-uniparental-disomy-aupd-in-primary-breast-cancer
#18
Musaffe Tuna, Marcel Smid, John W M Martens, John A Foekens
Many studies have examined DNA copy number changes or gene expression profiling and their association with clinical outcomes in breast cancer. However, until now no study has investigated whether acquired uniparental disomy (aUPD), in which both chromosomes in a pair are derived from the same parent, may have an association with clinical outcome including initiation and recurrence of breast cancer. In this study, we used high-density SNP and expression microarrays data from primary tumors of 313 lymph node-negative breast cancer patients who had not received adjuvant systemic therapy to evaluate the association of aUPD with metastasis-free survival (MFS) and overall survival (OS)...
February 2012: Breast Cancer Research and Treatment
https://www.readbyqxmd.com/read/19334051/high-frequency-of-tumours-in-mulibrey-nanism
#19
Niklas Karlberg, Susann Karlberg, Riitta Karikoski, Sakari Mikkola, Marita Lipsanen-Nyman, Hannu Jalanko
Mulibrey nanism (MUL) is a monogenic disorder with prenatal-onset growth failure, typical clinical characteristics, cardiopathy and tendency for a metabolic syndrome. It is caused by recessive mutations in the TRIM37 gene encoding for the peroxisomal TRIM37 protein with ubiquitin-ligase activity. In this work, the frequency and pathology of malignant and benign tumours were analysed in a national cohort of 89 Finnish MUL patients aged 0.7-76 years. The subjects had a clinical and radiological evaluation, and histological and immunohistocemical analyses on specimens obtained from biopsy, surgery or autopsy, were performed...
June 2009: Journal of Pathology
https://www.readbyqxmd.com/read/19329943/gynecological-tumors-in-mulibrey-nanism-and-role-for-ring-finger-protein-trim37-in-the-pathogenesis-of-ovarian-fibrothecomas
#20
Susann Karlberg, Marita Lipsanen-Nyman, Heini Lassus, Jukka Kallijärvi, Anna-Elina Lehesjoki, Ralf Butzow
Mulibrey nanism is an autosomal recessive growth disorder caused by mutations in the TRIM37 gene encoding a protein of unknown function. More than half of female patients with Mulibrey nanism develop benign mesenchymal tumors of ovarian sex cord-stromal origin. In this work, we characterize the gynecological tumors of female patients with Mulibrey nanism in detail. In addition to tumors of the fibrothecoma group, 18% (4/22) of the patients were observed with epithelial neoplasias, including 2 ovarian adenofibromas, 1 ovarian poorly differentiated adenocarcinoma and 1 endometrial adenocarcinoma...
April 2009: Modern Pathology: An Official Journal of the United States and Canadian Academy of Pathology, Inc
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