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@ARTICLE{Priglinger:141595,
author = {Priglinger, Claudia and Klopstock, Thomas and Rudolph,
Günter and Priglinger, Siegfried Georg},
title = {[{L}eber's {H}ereditary {O}ptic {N}europathy].},
journal = {Klinische Monatsblätter für Augenheilkunde},
volume = {236},
number = {11},
issn = {0023-2165},
address = {Stuttgart},
publisher = {Thieme78968},
reportid = {DZNE-2020-07919},
pages = {1271-1282},
year = {2019},
abstract = {Leber's hereditary optic neuropathy (LHON) typically
affects young adults with a higher prevalence in men, but
can ultimately occur at any age and also in women. LHON is
caused by point mutations in the mitochondrial DNA, which
lead to a defect in complex I of the mitochondrial
respiratory chain. This in turn causes dysfunction and later
degeneration of retinal ganglion cells, followed by
ascending optic atrophy. Classically, LHON presents as a
subacute unilateral loss of visual acuity, dyschromatopsia
in the red-green axis and a central or centrocecal scotoma.
The partner eye usually develops similar symptoms within
3 - 6 months of onset of the disease. In $25\%$ of
cases, however, the disease begins bilaterally. In the
natural course of the disease, the majority of patients
remain with a visual acuity less than 0.1, even though a
small proportion may experience a spontaneous improvement in
visual acuity. In 2015, the ubiquinone analogue Idebenone
was approved by the European Medicines Agency for the
treatment of LHON. The decisive factors for therapeutic
success are an early start and an appropriate treatment
duration. It should also be noted that a proportion of
patients may experience a delayed response to therapy.
However, a complete recovery of visual acuity is rare even
under therapy. Since patients affected by LHON are mostly
young adults of working age, who go largely blind more or
less acutely, immediate support with magnifying vision aids
and advice on social and vocational rehabilitation is
essential. Alternative therapeutic approaches such as gene
therapy, neuroprotection or stem cell-based aspects are
currently the subject of clinical studies and offer hope for
further perspectives for those affected. Although with
Idebenone a causal therapy has already been approved for
LHON, many questions regarding the pathogenesis of the
disease have not yet been completely clarified. This
particularly concerns gender prevalence and possible
additional triggers or protective factors. In this overview,
the clinical course of LHON, diagnostics and current therapy
recommendations as well as the special features and current
explanatory approaches to incomplete penetrance and symptoms
of LHON are explained.},
keywords = {DNA, Mitochondrial / Female / Genetic Therapy / Humans /
Male / Optic Atrophy, Hereditary, Leber: genetics / Optic
Atrophy, Hereditary, Leber: therapy / Retinal Ganglion Cells
/ Visual Acuity / Young Adult / DNA, Mitochondrial (NLM
Chemicals)},
cin = {Clinical Dementia Research München},
cid = {I:(DE-2719)1111016},
pnm = {344 - Clinical and Health Care Research (POF3-344)},
pid = {G:(DE-HGF)POF3-344},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:31639883},
doi = {10.1055/a-0972-1552},
url = {https://pub.dzne.de/record/141595},
}