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000120463 1001_ $$0P:(DE-He78)a9542bb104fe3f4d562e1d275e03f5ba$$aFeng, Weijun$$b0$$eFirst author
000120463 245__ $$aChd7 is indispensable for mammalian brain development through activation of a neuronal differentiation programme.
000120463 260__ $$aLondon$$bNature Publishing Group$$c2017
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000120463 520__ $$aMutations in chromatin modifier genes are frequently associated with neurodevelopmental diseases. We herein demonstrate that the chromodomain helicase DNA-binding protein 7 (Chd7), frequently associated with CHARGE syndrome, is indispensable for normal cerebellar development. Genetic inactivation of Chd7 in cerebellar granule neuron progenitors leads to cerebellar hypoplasia in mice, due to the impairment of granule neuron differentiation, induction of apoptosis and abnormal localization of Purkinje cells, which closely recapitulates known clinical features in the cerebella of CHARGE patients. Combinatory molecular analyses reveal that Chd7 is required for the maintenance of open chromatin and thus activation of genes essential for granule neuron differentiation. We further demonstrate that both Chd7 and Top2b are necessary for the transcription of a set of long neuronal genes in cerebellar granule neurons. Altogether, our comprehensive analyses reveal a mechanism with chromatin remodellers governing brain development via controlling a core transcriptional programme for cell-specific differentiation.
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000120463 7001_ $$0P:(DE-He78)0ac2bd1a9fb1823a351ee4434d80808b$$aKawauchi, Daisuke$$b1$$eFirst author
000120463 7001_ $$0P:(DE-He78)0e2afbe79bacc08410665f049d5208fa$$aKörkel-Qu, Huiqin$$b2
000120463 7001_ $$0P:(DE-He78)29aa3379308b1c2bde2b1876186aae49$$aDeng, Huan$$b3
000120463 7001_ $$0P:(DE-He78)a5f297f34b5deb64061d3a915d1bafab$$aSerger, Elisabeth$$b4
000120463 7001_ $$0P:(DE-He78)a4101d4d75f0b7d1f24f67ccbe63164b$$aSieber, Laura$$b5
000120463 7001_ $$aLieberman, Jenna Ariel$$b6
000120463 7001_ $$aJimeno-González, Silvia$$b7
000120463 7001_ $$0P:(DE-He78)b84967c4f073b71405404f3719c788cd$$aLambo, Sander$$b8
000120463 7001_ $$0P:(DE-He78)1c2351213e0199e6bd4efaceac511917$$aHanna, Bola$$b9
000120463 7001_ $$0P:(DE-He78)044b8e7cc52bce80fe9afd70f8f4d44e$$aHarim, Yassin$$b10
000120463 7001_ $$0P:(DE-He78)e40e066e33767cfd54fa29f1464e7ca5$$aJansen, Malin$$b11
000120463 7001_ $$0P:(DE-He78)654da7a5feabd5cf7a7dbef6ec324053$$aNeuerburg, Anna$$b12
000120463 7001_ $$0P:(DE-He78)806c4988c4c63fae4c598bc7975ca277$$aFriesen, Olga$$b13
000120463 7001_ $$0P:(DE-He78)2a8fbc2efe7e5e468472d57f724fe39b$$aZuckermann, Marc$$b14
000120463 7001_ $$0P:(DE-HGF)0$$aRajendran, Vijayanad$$b15
000120463 7001_ $$00000-0003-0557-679X$$aGronych, Jan$$b16
000120463 7001_ $$aAyrault, Olivier$$b17
000120463 7001_ $$0P:(DE-He78)8d9c904a6cea14d4c99c78ba46e41f93$$aKorshunov, Andrey$$b18
000120463 7001_ $$0P:(DE-He78)551bb92841f634070997aa168d818492$$aJones, David$$b19
000120463 7001_ $$0P:(DE-He78)4c28e2aade5f44d8eca9dd8e97638ec8$$aKool, Marcel$$b20
000120463 7001_ $$aNorthcott, Paul A$$b21
000120463 7001_ $$0P:(DE-He78)e13b4363c5fe858044ef8a39c02c870c$$aLichter, Peter$$b22
000120463 7001_ $$aCortés-Ledesma, Felipe$$b23
000120463 7001_ $$0P:(DE-He78)f746aa965c4e1af518b016de3aaff5d9$$aPfister, Stefan$$b24
000120463 7001_ $$0P:(DE-He78)76aeb2431f7458c9261e69c5420390c6$$aLiu, Haikun$$b25$$eLast author
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