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Article

The role of SPAG1 in the assembly of axonemal dyneins in human airway epithelia.

Journal of cell science - 15 Mar 2022

Smith Amanda J, Bustamante-Marin Ximena M, Yin Weining, Sears Patrick R, Herring Laura E, Dicheva Nedyalka N, López-Giráldez Francesc, Mane Shrikant, Tarran Robert, Leigh Margaret W, Knowles Michael R, Zariwala Maimoona A, Ostrowski Lawrence E

Abstract excerpt

Mutations in SPAG1, a dynein axonemal assembly factor (DNAAF) that facilitates the assembly of dynein arms in the cytoplasm before their transport into the cilium, result in primary ciliary dyskinesia (PCD), a genetically heterogenous disorder characterized by chronic oto-sino-pulmonary disease, infertility and laterality defects. To further elucidate the role of SPAG1 in dynein assembly, we examined its...

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Identifiers

PMID: 35178554
DOI: 10.1242/jcs.259512
PMCID: PMC8995097

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