Discovery of Metabolic Biomarkers for Duchenne Muscular Dystrophy within a Natural History Study

被引:29
作者
Boca, Simina M. [1 ,2 ,3 ]
Nishida, Maki [1 ]
Harris, Michael [1 ]
Rao, Shruti [1 ]
Cheema, Amrita K. [2 ,4 ]
Gill, Kirandeep [2 ]
Seol, Haeri [5 ,6 ]
Morgenroth, Lauren P. [5 ,6 ]
Henricson, Erik [7 ]
McDonald, Craig [7 ]
Mah, Jean K. [8 ]
Clemens, Paula R. [9 ,10 ]
Hoffman, Eric P. [5 ,6 ]
Hathout, Yetrib [5 ,6 ]
Madhavan, Subha [1 ,2 ]
机构
[1] Georgetown Univ, Med Ctr, Innovat Ctr Biomed Informat, Washington, DC 20007 USA
[2] Georgetown Univ, Med Ctr, Dept Oncol, Washington, DC 20007 USA
[3] Georgetown Univ, Med Ctr, Dept Biostat Bioinformat & Biomath, Washington, DC 20007 USA
[4] Georgetown Univ, Med Ctr, Dept Biochem & Mol & Cellular Biol, Washington, DC 20007 USA
[5] Childrens Natl Med Ctr, Washington, DC 20010 USA
[6] George Washington Univ, Washington, DC USA
[7] Univ Calif Davis, Sch Med, Dept Phys Med & Rehabil, Davis, CA 95616 USA
[8] Univ Calgary, Dept Pediat, Alberta Childrens Hosp, Calgary, AB T2N 1N4, Canada
[9] Dept Veteran Affairs Med Ctr, Neurol Serv, Pittsburgh, PA USA
[10] Univ Pittsburgh, Dept Neurol, Pittsburgh, PA 15260 USA
基金
美国国家卫生研究院;
关键词
DISEASE PROGRESSION; PROTEOMICS;
D O I
10.1371/journal.pone.0153461
中图分类号
O [数理科学和化学]; P [天文学、地球科学]; Q [生物科学]; N [自然科学总论];
学科分类号
07 ; 0710 ; 09 ;
摘要
Serum metabolite profiling in Duchenne muscular dystrophy (DMD) may enable discovery of valuable molecular markers for disease progression and treatment response. Serum samples from 51 DMD patients from a natural history study and 22 age-matched healthy volunteers were profiled using liquid chromatography coupled to mass spectrometry (LC-MS) for discovery of novel circulating serum metabolites associated with DMD. Fourteen metabolites were found significantly altered (1% false discovery rate) in their levels between DMD patients and healthy controls while adjusting for age and study site and allowing for an interaction between disease status and age. Increased metabolites included arginine, creatine and unknown compounds at m/z of 357 and 312 while decreased metabolites included creatinine, androgen derivatives and other unknown yet to be identified compounds. Furthermore, the creatine to creatinine ratio is significantly associated with disease progression in DMD patients. This ratio sharply increased with age in DMD patients while it decreased with age in healthy controls. Overall, this study yielded promising metabolic signatures that could prove useful to monitor DMD disease progression and response to therapies in the future.
引用
收藏
页数:18
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