Minimal Efficacy of Nitisinone Treatment in a Novel Mouse Model of Oculocutaneous Albinism, Type 3

Onojafe Ighovie F.; Megan Lucyanne H.; Melch Madeline G.; Aderemi Joseph O.; Alur Ramakrishna P.; Abu-Asab Mones S.; Chan Chi-Chao; Bernardini Isa M.; Albert Jessica S.; Cogliati Tiziana; Adams David R.; Brooks Brian P.

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Minimal Efficacy of Nitisinone Treatment in a Novel Mouse Model of Oculocutaneous Albinism, Type 3

机译：核酮治疗在血管外形仿生新型小鼠模型中的最小疗效，3型

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PURPOSE. Oral nitisinone has been shown to increase fur and ocular pigmentation in a mouse model of oculocutaneous albinism (OCA) due to hypomorphic mutations in tyrosinase (TYR), OCA1B. This study determines if nitisinone can improve ocular and/or fur pigmentation in a mouse model of OCA type 3 (OCA3), caused by mutation of the tyrosinase-related protein 1 (Tyrp1) gene.

机译：目的。由于酪氨酸酶（Tyr），OCA1b中的酪氨酸酶（Tyr），OCA1B中的警长突变，已经显示口腔核苷酸在血管外囊症（OCA）的小鼠模型中增加皮草和眼色素沉着。该研究确定核酮可以在由酪氨酸酶相关蛋白1（TyrP1）基因的突变引起的OCA型3（OCA3）的小鼠模型中改善眼部和/或毛皮色素沉着。

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来源
《Investigative ophthalmology & visual science》 |2018年第12期|共8页
作者
Onojafe Ighovie F.; Megan Lucyanne H.; Melch Madeline G.; Aderemi Joseph O.; Alur Ramakrishna P.; Abu-Asab Mones S.; Chan Chi-Chao; Bernardini Isa M.; Albert Jessica S.; Cogliati Tiziana; Adams David R.; Brooks Brian P.;
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作者单位

NEI Ophthalm Genet &

Visual Funct Branch NIH Bethesda MD 20892 USA;

NEI Ophthalm Genet &

Visual Funct Branch NIH Bethesda MD 20892 USA;

NEI Ophthalm Genet &

Visual Funct Branch NIH Bethesda MD 20892 USA;

NEI Ophthalm Genet &

Visual Funct Branch NIH Bethesda MD 20892 USA;

NEI Ophthalm Genet &

Visual Funct Branch NIH Bethesda MD 20892 USA;

NEI Immunol Lab NIH Bldg 10 Bethesda MD 20892 USA;

NEI Immunol Lab NIH Bldg 10 Bethesda MD 20892 USA;

NHGRI Med Genet Branch NIH Bethesda MD 20892 USA;

NHGRI Med Genet Branch NIH Bethesda MD 20892 USA;

NEI Ophthalm Genet &

Visual Funct Branch NIH Bethesda MD 20892 USA;

NHGRI Med Genet Branch NIH Bethesda MD 20892 USA;

NEI Ophthalm Genet &

Visual Funct Branch NIH Bethesda MD 20892 USA;

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原文格式 PDF
正文语种 eng
中图分类眼科学;
关键词
albinism; visual acuity; retinal pigment epithelium;

机译：白化病;视力;视网膜颜料上皮;

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专利

1. Minimal Efficacy of Nitisinone Treatment in a Novel Mouse Model of Oculocutaneous Albinism, Type 3 [J] . Onojafe Ighovie F., Megan Lucyanne H., Melch Madeline G., Investigative ophthalmology & visual science . 2018,第12期

机译：核酮治疗在血管外形仿生新型小鼠模型中的最小疗效，3型
2. Tyrosinase processing and intracellular trafficking is disrupted in mouse primary melanocytes carrying the underwhite (uw) mutation. A model for oculocutaneous albinism (OCA) type 4 [J] . Costin GE., Valencia JC., Vieira WD., Journal of Cell Science . 2003,第15期

机译：酪氨酸酶加工和细胞内运输被破坏的小鼠原代黑素细胞中的白色（uw）突变。眼皮肤白化病（OCA）4型的模型
3. Tyrosinase processing and intracellular trafficking is disrupted in mouse primary melanocytes carrying the underwhite (uw) mutation. A model for oculocutaneous albinism (OCA) type 4. [J] . Costin GE, Valencia JC, Vieira WD, Journal of Cell Science . 2003,第Pta15期

机译：酪氨酸酶加工和细胞内运输被破坏的小鼠原代黑素细胞中的白色（uw）突变。眼皮肤白化病（OCA）4型的模型。
4. Topical calcitriol prior to photodynamic therapy enhances treatment efficacy in non-melanoma skin cancer mouse models [C] . Kishore Rollakanti, Sanjay Anand, Edward V. Maytin Optical methods for tumor treatment and detection: mechanisms and techniques in photodynamic therapy XXIV . 2015

机译：光动力疗法前的局部骨化三醇可增强非黑色素瘤皮肤癌小鼠模型的治疗功效
5. Investigations of the biodistribution and phototherapeutic utility of two palladium(II)-containing macrocyclic drugs in a tumour-bearing mouse model: A study of the pharmacokinetics and tissue distribution of the potential photodynamic drug palladium-bacteriopheophorbide as well as the in vivo treatment efficacy and tissue distribution of the potential photothermal drug palladium(II) octabutoxynaphthalocyanine. [D] . DeGroot, Jennifer Leanne. 2002

机译：两种含钯（II）的大环药物在荷瘤小鼠模型中的生物分布和光疗实用性的研究：潜在的光动力药物钯-细菌脱镁叶绿酸的药代动力学和组织分布以及体内治疗效果和研究光热药物钯（II）八丁氧基萘酞菁的组织分布。
6. Minimal Efficacy of Nitisinone Treatment in a Novel Mouse Model of Oculocutaneous Albinism Type 3 [O] . Ighovie F. Onojafe, Lucyanne H. Megan, Madeline G. Melch, -1

机译：在一种新型的眼皮肤白化病3型小鼠模型中尼替尼酮治疗的最低功效
7. Nitisinone improves eye and skin pigmentation defects in a mouse model of oculocutaneous albinism [O] . Onojafe, Ighovie F., Adams, David R., Simeonov, Dimitre R., 2011

机译：尼替尼酮可改善眼皮肤白化病小鼠模型中的眼睛和皮肤色素沉着缺陷

Minimal Efficacy of Nitisinone Treatment in a Novel Mouse Model of Oculocutaneous Albinism, Type 3

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