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A growing number of studies indicate that SMA is a multi\u2010system disease. The cerebellum has received little attention even though it plays an important role in motor function and widespread pathology has been reported in the cerebella of SMA patients. In this study, we assessed SMA pathology in the cerebellum using structural and diffusion magnetic resonance imaging, immunohistochemistry, and electrophysiology with the SMN\u03947 mouse model. We found a significant disproportionate loss in cerebellar volume, decrease in afferent cerebellar tracts, selective lobule\u2010specific degeneration of Purkinje cells, abnormal lobule foliation and astrocyte integrity, and a decrease in spontaneous firing of cerebellar output neurons in the SMA mice compared to controls. Our data suggest that defects in cerebellar structure and function due to decreased survival motor neuron (SMN) levels impair the functional cerebellar output affecting motor control, and that cerebellar pathology should be addressed to achieve comprehensive treatment and therapy for SMA patients.<\/jats:p>","DOI":"10.1111\/bpa.13162","type":"journal-article","created":{"date-parts":[[2023,5,23]],"date-time":"2023-05-23T00:46:04Z","timestamp":1684802764000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":15,"title":["Cerebellar structural, astrocytic, and neuronal abnormalities in the\n                    <scp>SMN\u03947<\/scp>\n                    mouse model of spinal muscular atrophy"],"prefix":"10.1111","volume":"33","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6300-8999","authenticated-orcid":false,"given":"Nicholas C.","family":"Cottam","sequence":"first","affiliation":[{"name":"Department of Biological Sciences 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