Pharmacokinetics of CuGTSM, a Novel Drug Candidate, in a Mouse Model of Menkes Disease

Yamagishi, Yoshiaki; Kudo, Toshiyuki; Oyumi, Masafumi; Sakamoto, Yusuke; Takahashi, Kazuki; Akashi, Taiki; Kobayashi, Shohei; Kawakami, T; Goda, Hitomi; Sato, Yasuhiro; Mimaki, Masakazu; Kodama, Hiroko; Munakata, Mitsutoshi; Makino, Kosho; Takahashi, Hideyo; Fukami, Toshiro; K, Ito

doi:10.1007/s11095-021-03090-0

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“…A combination therapy using copper and lipophilic chelators, such as diethyldithiocarbamate (DEDTC), disulfiram (a dimer of DEDTC), and dimethyldithiocarbamate, has been studied using a macular mouse model. The results suggest that this combination therapy improves copper concentration and cuproenzyme activities in the brain of the macular mouse [ [35] , [36] , [37] , [38] , [39] ]. A combination therapy using copper injections and oral disulfiram was administered to two patients with MD at the age of 8 and 10 years, respectively.…”

Section: Discussionmentioning

confidence: 99%