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GalSph and GluSph are useful biomarkers for both diagnosis and monitoring of treatment effects. We have developed and validated a sensitive, accurate, high\u2010throughput assay for simultaneous determination of the concentration of GalSph and GluSph in mouse serum. GalSph and GluSph and their deuterated internal standards were extracted by protein precipitation in quantitative recoveries, baseline separated by hydrophilic interaction chromatography and detected by positive\u2010ion electrospray mass spectrometry in multiple reaction monitoring mode. Total run time was 7\u00a0min. The lower limit of quantification was 0.2\u00a0ng\/mL for both GalSph and GluSph. Sample stability, assay precision and accuracy, and method robustness were demonstrated. This method has been successfully applied to measurement of these lipid biomarkers in a natural history study in twitcher (Krabbe) mice.<\/jats:p>","DOI":"10.1002\/bmc.4235","type":"journal-article","created":{"date-parts":[[2018,3,8]],"date-time":"2018-03-08T01:50:38Z","timestamp":1520473838000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":24,"title":["A HILIC\u2010MS\/MS method for simultaneous quantification of the lysosomal disease markers galactosylsphingosine and glucosylsphingosine in mouse serum"],"prefix":"10.1002","volume":"32","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1703-2167","authenticated-orcid":false,"given":"Rohini","family":"Sidhu","sequence":"first","affiliation":[{"name":"Department of Medicine Washington University School of Medicine  St Louis MO 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