000119959 001__ 119959 000119959 005__ 20240228135003.0 000119959 0247_ $$2doi$$a10.1523/JNEUROSCI.2560-14.2014 000119959 0247_ $$2pmid$$apmid:25274825 000119959 0247_ $$2altmetric$$aaltmetric:2732877 000119959 037__ $$aDKFZ-2017-00550 000119959 041__ $$aeng 000119959 082__ $$a610 000119959 1001_ $$aMoreno, Natalia$$b0 000119959 245__ $$aLoss of Smarc proteins impairs cerebellar development. 000119959 260__ $$aWashington, DC$$bSoc.44027$$c2014 000119959 264_1 $$2Crossref$$3online$$bSociety for Neuroscience$$c2014-10-01 000119959 264_1 $$2Crossref$$3print$$bSociety for Neuroscience$$c2014-10-01 000119959 264_1 $$2Crossref$$3print$$bSociety for Neuroscience$$c2014-10-01 000119959 3367_ $$2DRIVER$$aarticle 000119959 3367_ $$2DataCite$$aOutput Types/Journal article 000119959 3367_ $$0PUB:(DE-HGF)16$$2PUB:(DE-HGF)$$aJournal Article$$bjournal$$mjournal$$s1490786115_9393 000119959 3367_ $$2BibTeX$$aARTICLE 000119959 3367_ $$2ORCID$$aJOURNAL_ARTICLE 000119959 3367_ $$00$$2EndNote$$aJournal Article 000119959 520__ $$aSMARCA4 (BRG1) and SMARCB1 (INI1) are tumor suppressor genes that are crucially involved in the formation of malignant rhabdoid tumors, such as atypical teratoid/rhabdoid tumor (AT/RT). AT/RTs typically affect infants and occur at various sites of the CNS with a particular frequency in the cerebellum. Here, granule neurons and their progenitors represent the most abundant cell type and are known to give rise to a subset of medulloblastoma, a histologically similar embryonal brain tumor. To test how Smarc proteins influence the development of granule neurons and whether this population may serve as cellular origin for AT/RTs, we specifically deleted Smarca4 and Smarcb1 in cerebellar granule cell precursors. Respective mutant mice displayed severe ataxia and motor coordination deficits, but did not develop any tumors. In fact, they suffered from a severely hypoplastic cerebellum due to a significant inhibition of granule neuron precursor proliferation. Molecularly, this was accompanied by an enhanced activity of Wnt/β-catenin signaling that, by itself, is known to cause a nearly identical phenotype. We further used an hGFAP-cre allele, which deleted Smarcb1 much earlier and in a wider neural precursor population, but we still did not detect any tumor formation in the CNS. In summary, our results emphasize cell-type-dependent roles of Smarc proteins and argue against cerebellar granule cells and other progeny of hGFAP-positive neural precursors as the cellular origin for AT/RTs. 000119959 536__ $$0G:(DE-HGF)POF3-312$$a312 - Functional and structural genomics (POF3-312)$$cPOF3-312$$fPOF III$$x0 000119959 588__ $$aDataset connected to CrossRef, PubMed, 000119959 650_7 $$2NLM Chemicals$$aAtoh1 protein, mouse 000119959 650_7 $$2NLM Chemicals$$aBasic Helix-Loop-Helix Transcription Factors 000119959 650_7 $$2NLM Chemicals$$aChromosomal Proteins, Non-Histone 000119959 650_7 $$2NLM Chemicals$$aGlial Fibrillary Acidic Protein 000119959 650_7 $$2NLM Chemicals$$aNuclear Proteins 000119959 650_7 $$2NLM Chemicals$$aSMARCB1 Protein 000119959 650_7 $$2NLM Chemicals$$aSmarcb1 protein, mouse 000119959 650_7 $$2NLM Chemicals$$aTranscription Factors 000119959 650_7 $$2NLM Chemicals$$aWnt Proteins 000119959 650_7 $$0147336-22-9$$2NLM Chemicals$$aGreen Fluorescent Proteins 000119959 650_7 $$0EC 3.6.1.-$$2NLM Chemicals$$aSmarca4 protein, mouse 000119959 650_7 $$0EC 3.6.4.-$$2NLM Chemicals$$aDNA Helicases 000119959 650_7 $$0EC 4.2.1.11$$2NLM Chemicals$$aPhosphopyruvate Hydratase 000119959 7001_ $$0P:(DE-He78)fe53b38061056c5d390f37ce8d63f505$$aSchmidt, Christin$$b1$$udkfz 000119959 7001_ $$aAhlfeld, Julia$$b2 000119959 7001_ $$aPöschl, Julia$$b3 000119959 7001_ $$aDittmar, Stefanie$$b4 000119959 7001_ $$0P:(DE-He78)f746aa965c4e1af518b016de3aaff5d9$$aPfister, Stefan$$b5 000119959 7001_ $$0P:(DE-He78)4c28e2aade5f44d8eca9dd8e97638ec8$$aKool, Marcel$$b6 000119959 7001_ $$aKerl, Kornelius$$b7 000119959 7001_ $$aSchüller, Ulrich$$b8 000119959 77318 $$2Crossref$$3journal-article$$a10.1523/jneurosci.2560-14.2014$$bSociety for Neuroscience$$d2014-10-01$$n40$$p13486-13491$$tThe Journal of Neuroscience$$v34$$x0270-6474$$y2014 000119959 773__ $$0PERI:(DE-600)1475274-8$$a10.1523/JNEUROSCI.2560-14.2014$$gVol. 34, no. 40, p. 13486 - 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