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Unsurprisingly, mutations in dynein are causative of various developmental neuropathies and motor neuron diseases. These \u2018dyneinopathies\u2019 define a broad spectrum of diseases with no known correlation between mutation identity and disease state. To circumvent complications associated with dynein studies in human cells, we employed budding yeast as a screening platform to characterize the motility properties of seventeen disease-correlated dynein mutants. Using this system, we determined the molecular basis for several classes of etiologically related diseases. Moreover, by engineering compensatory mutations, we alleviated the mutant phenotypes in two of these cases, one of which we confirmed with recombinant human dynein. In addition to revealing molecular insight into dynein regulation, our data provide additional evidence that the type of disease may in fact be dictated by the degree of dynein dysfunction.<\/jats:p>","DOI":"10.7554\/elife.47246","type":"journal-article","created":{"date-parts":[[2019,7,31]],"date-time":"2019-07-31T08:00:22Z","timestamp":1564560022000},"source":"Crossref","is-referenced-by-count":55,"title":["Molecular basis for dyneinopathies reveals insight into dynein regulation and dysfunction"],"prefix":"10.7554","volume":"8","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2571-6377","authenticated-orcid":true,"given":"Matthew G","family":"Marzo","sequence":"first","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, Colorado State University, Fort Collins, United States"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jacqueline M","family":"Griswold","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Molecular 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