Targeted Treatment of Individuals With Psychosis Carrying a Copy Number Variant Containing a Genomic Triplication of the Glycine Decarboxylase Gene.

TitleTargeted Treatment of Individuals With Psychosis Carrying a Copy Number Variant Containing a Genomic Triplication of the Glycine Decarboxylase Gene.
Publication TypeJournal Article
Year of Publication2019
AuthorsJ Bodkin, A, Coleman, MJ, Godfrey, LJ, Carvalho, CMB, Morgan, CJ, Suckow, RF, Anderson, T, Öngür, D, Kaufman, MJ, Lewandowski, KE, Siegel, AJ, Waldstreicher, E, Grochowski, CM, Javitt, DC, Rujescu, D, Hebbring, S, Weinshilboum, R, Rodriguez, SBurgos, Kirchhoff, C, Visscher, T, Vuckovic, A, Fialkowski, A, McCarthy, S, Malhotra, D, Sebat, J, Goff, DC, Hudson, JI, Lupski, JR, Coyle, JT, Rudolph, U, Levy, DL
JournalBiol Psychiatry
Volume86
Issue7
Pagination523-535
Date Published2019 Oct 01
ISSN1873-2402
Abstract

BACKGROUND: The increased mutational burden for rare structural genomic variants in schizophrenia and other neurodevelopmental disorders has so far not yielded therapies targeting the biological effects of specific mutations. We identified two carriers (mother and son) of a triplication of the gene encoding glycine decarboxylase, GLDC, presumably resulting in reduced availability of the N-methyl-D-aspartate receptor coagonists glycine and D-serine and N-methyl-D-aspartate receptor hypofunction. Both carriers had a diagnosis of a psychotic disorder.

METHODS: We carried out two double-blind, placebo-controlled clinical trials of N-methyl-D-aspartate receptor augmentation of psychotropic drug treatment in these two individuals. Glycine was used in the first clinical trial, and D-cycloserine was used in the second one.

RESULTS: Glycine or D-cycloserine augmentation of psychotropic drug treatment each improved psychotic and mood symptoms in placebo-controlled trials.

CONCLUSIONS: These results provide two independent proof-of-principle demonstrations of symptom relief by targeting a specific genotype and explicitly link an individual mutation to the pathophysiology of psychosis and treatment response.

DOI10.1016/j.biopsych.2019.04.031
Alternate JournalBiol. Psychiatry
PubMed ID31279534
PubMed Central IDPMC6745274
Grant ListUM1 HG006542 / HG / NHGRI NIH HHS / United States
R35 NS105078 / NS / NINDS NIH HHS / United States
R01 GM106373 / GM / NIGMS NIH HHS / United States
R21 MH105732 / MH / NIMH NIH HHS / United States
U24 MH081810 / MH / NIMH NIH HHS / United States
R01 NS058529 / NS / NINDS NIH HHS / United States
R21 MH097470 / MH / NIMH NIH HHS / United States
T32 HL079888 / HL / NHLBI NIH HHS / United States
R21 MH104505 / MH / NIMH NIH HHS / United States