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This dysfunction leads to the accumulation of undegraded substrates, such as heparan sulfate, ultimately leading to progressive neuroinflammation and neurodegeneration. Despite well\u2010defined genetic causes, no disease\u2010modifying therapies exist for Sanfilippo syndrome. While microglia, the brain's resident immune cells, can play both protective and pathogenic roles, the contribution of neuroinflammation to LSD pathology remains underexplored. This review examines the contribution of neuroinflammation to Sanfilippo syndrome, emphasizing emerging mechanisms involving TLR4 signaling, inflammasome activation, the cGAS\u2010STING pathway, and lysosomal biogenesis regulators such as TFE family transcription factors. We also discuss the potential of cellular therapies to modulate neuroimmune responses and offer new therapeutic avenues. By integrating insights from neuroimmunology and lysosomal biology, we aim to identify shared mechanisms and therapeutic targets across Sanfilippo syndrome and related LSDs.<\/jats:p>","DOI":"10.1002\/bies.70083","type":"journal-article","created":{"date-parts":[[2025,10,18]],"date-time":"2025-10-18T12:38:21Z","timestamp":1760791101000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["The Neuroimmune Landscape of the Lysosomal Storage Disorder Sanfilippo Syndrome"],"prefix":"10.1002","volume":"47","author":[{"given":"Rafael A.","family":"Badell\u2010Grau","sequence":"first","affiliation":[{"name":"Department of Pediatrics University of California  San Diego California USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Johannes C. 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