Journal article

Serine and lipid metabolism in macular disease and peripheral neuropathy

ML Gantner, K Eade, M Wallace, MK Handzlik, R Fallon, J Trombley, R Bonelli, S Giles, S Harkins-Perry, TFC Heeren, L Sauer, Y Ideguchi, M Baldini, L Scheppke, MI Dorrell, M Kitano, BJ Hart, C Cai, T Nagasaki, MG Badur Show all

New England Journal of Medicine | MASSACHUSETTS MEDICAL SOC | Published : 2019

Abstract

BACKGROUND Identifying mechanisms of diseases with complex inheritance patterns, such as macular telangiectasia type 2, is challenging. A link between macular telangiectasia type 2 and altered serine metabolism has been established previously. METHODS Through exome sequence analysis of a patient with macular telangiectasia type 2 and his family members, we identified a variant in SPTLC1 encoding a subunit of serine palmitoyltransferase (SPT). Because mutations affecting SPT are known to cause hereditary sensory and autonomic neuropathy type 1 (HSAN1), we examined 10 additional persons with HSAN1 for ophthalmologic disease. We assayed serum amino acid and sphingoid base levels, including leve..

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University of Melbourne Researchers

Grants

Awarded by National Cancer Institute


Funding Acknowledgements

Supported by the Lowy Medical Research Institute, with supplementary funding from the National Health and Medical Research Council of Australia (to Dr. Bahlo), a core grant from the National Eye Institute (EY14800, to Dr. Bernstein), the University of Melbourne Research Scholarship Program (to Mr. Bonelli), a grant from the National Science Foundation (1454425, to Dr. Metallo), and a grant from the National Institutes of Health (NCI R01CA188652, to Dr. Metallo). Heidelberg Engineering provided a Heidelberg Spectralis confocal imaging system to Dr. Bernstein free of charge for use in this study.