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Publication

Tissue-specific regulation of BMP signaling by Drosophila N-glycanase 1.

eLife | 2017

Mutations in the human N-glycanase 1 (NGLY1) cause a rare, multisystem congenital disorder with global developmental delay. However, the mechanisms by which NGLY1 and its homologs regulate embryonic development are not known. Here we show that Drosophila Pngl encodes an N-glycanase and exhibits a high degree of functional conservation with human NGLY1. Loss of Pngl results in developmental midgut defects reminiscent of midgut-specific loss of BMP signaling. Pngl mutant larvae also exhibit a severe midgut clearance defect, which cannot be fully explained by impaired BMP signaling. Genetic experiments indicate that Pngl is primarily required in the mesoderm during Drosophila development. Loss of Pngl results in a severe decrease in the level of Dpp homodimers and abolishes BMP autoregulation in the visceral mesoderm mediated by Dpp and Tkv homodimers. Thus, our studies uncover a novel mechanism for the tissue-specific regulation of an evolutionarily conserved signaling pathway by an N-glycanase enzyme.

Pubmed ID: 28826503 RIS Download

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Associated grants

Agency: NIH HHS, United States
Id: P40 OD018537
Agency: NIDDK NIH HHS, United States
Id: R01 DK109982
Agency: NIGMS NIH HHS, United States
Id: R01 GM084135
Agency: NICHD NIH HHS, United States
Id: U54 HD083092

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