Immunology of Retinitis Pigmentosa and Gene Therapy-Associated Uveitis

被引:4
|
作者
Yang, Paul [1 ]
Mustafi, Debarshi [2 ,3 ,4 ]
Pepple, Kathryn L. [2 ]
机构
[1] Oregon Hlth & Sci Univ, Casey Eye Inst, Portland, OR 97239 USA
[2] Univ Washington, Roger & Karalis Johnson Retina Ctr, Dept Ophthalmol, Seattle, WA 98109 USA
[3] Brotman Baty Inst Precis Med, Seattle, WA 98109 USA
[4] Seattle Childrens Hosp, Dept Ophthalmol, Seattle, WA 98109 USA
来源
关键词
PHOTORECEPTOR CELL-DEATH; CHRONIC INFLAMMATORY REACTION; ADAPTIVE IMMUNE-RESPONSES; S-ANTIGEN REACTIVITY; RETINAL DEGENERATION; MOUSE MODEL; MYCOPHENOLATE-MOFETIL; MICROGLIAL ACTIVATION; ANIMAL-MODELS; MACULAR EDEMA;
D O I
10.1101/cshperspect.a041305
中图分类号
R-3 [医学研究方法]; R3 [基础医学];
学科分类号
1001 ;
摘要
The underlying immune state of inherited retinal degenerations (IRDs) and retinitis pigmentosa (RP) has been an emerging area of interest, wherein the consequences have never been greater given the widespread recognition of gene therapy-associated uveitis (GTU) in gene therapy clinical trials. Whereas some evidence suggests that the adaptive immune system may play a role, the majority of studies indicate that the innate immune system is likely the primary driver of neuroinflammation in RP. During retinal degeneration, discrete mechanisms activate resident microglia and promote infiltrating macrophages that can either be protective or detrimental to photoreceptor cell death. This persistent stimulation of innate immunity, overlaid by the introduction of viral antigens as part of gene therapy, has the potential to trigger a complex microglia/macrophage-driven proinflammatory state. A better understanding of the immune pathophysiology in IRD and GTU will be necessary to improve the success of developing novel treatments for IRDs.
引用
收藏
页数:21
相关论文
共 50 条
  • [41] Optogenetic therapy for retinitis pigmentosa
    V Busskamp
    S Picaud
    J A Sahel
    B Roska
    Gene Therapy, 2012, 19 : 169 - 175
  • [43] RETINITIS PIGMENTOSA ASSOCIATED WITH GLAUCOMA
    GARTNER, S
    SCHLOSSMAN, A
    AMERICAN JOURNAL OF OPHTHALMOLOGY, 1949, 32 (10) : 1337 - 1553
  • [44] Retinitis pigmentosa associated with vitiligo
    Mansouri, Parvin
    Mortazavi, Mohammadreza
    Madani, Mohammadhossein Malek
    Mazaheri, Maria
    EUROPEAN JOURNAL OF DERMATOLOGY, 2006, 16 (03) : 319 - 320
  • [45] Retinitis pigmentosa associated with hypomagnesaemia
    Liang, Steve Y-W
    Lee, Lawrence R.
    CLINICAL AND EXPERIMENTAL OPHTHALMOLOGY, 2010, 38 (06): : 645 - 647
  • [46] Retinal pigmentary changes in chronic uveitis mimicking retinitis pigmentosa
    D. Damla Sevgi
    Samaneh Davoudi
    Jason Comander
    Lucia Sobrin
    Graefe's Archive for Clinical and Experimental Ophthalmology, 2017, 255 : 1801 - 1810
  • [47] Towards a clinical trial of gene therapy to target retinitis pigmentosa associated with Usher syndrome type IB
    Ferla, R.
    Dell'Aquila, F.
    Doria, M.
    Ferraiuolo, M.
    Noto, A.
    Grazioli, F.
    Ammendola, V.
    Colloca, S.
    Auricchio, A.
    HUMAN GENE THERAPY, 2021, 32 (19-20) : A75 - A75
  • [48] A promising non-viral gene therapy approach for retinitis pigmentosa
    Bigot, Karine
    Gondouin, Pauline
    Montagne, Pierrick
    Benard, Romain
    Picard, Emilie
    Courtois, Yves G.
    Buggage, Ronald
    Bordet, Thierry
    Behar-Cohen, Francine F.
    INVESTIGATIVE OPHTHALMOLOGY & VISUAL SCIENCE, 2019, 60 (09)
  • [50] Perspectives on Evolving Gene Therapy for X-Linked Retinitis Pigmentosa
    Sengupta, Sabyasachi
    JAMA OPHTHALMOLOGY, 2023, 141 (03) : 283 - 284