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@ARTICLE{Oder:131599,
author = {D. Oder and D. Liu and K. Hu and N. Üçeyler and T.
Salinger and J. Müntze and K. Lorenz and R. Kandolf and
H.-J. Gröne$^*$ and C. Sommer and G. Ertl and C. Wanner and
P. Nordbeck},
title = {α-{G}alactosidase {A} {G}enotype {N}215{S} {I}nduces a
{S}pecific {C}ardiac {V}ariant of {F}abry {D}isease.},
journal = {Circulation / Cardiovascular genetics},
volume = {10},
number = {5},
issn = {1942-3268},
address = {Philadelphia, Pa.},
publisher = {Lippincott, Williams $\&$ Wilkins},
reportid = {DKFZ-2017-06231},
pages = {e001691 -},
year = {2017},
note = {Herz-Kreislauf},
abstract = {Hypertrophic cardiomyopathy is the most common type of
cardiomyopathy, but many patients lack
sarcomeric/myofilament mutations. We studied whether
cardio-specific α-galactosidase A gene variants are
misinterpreted as hypertrophic cardiomyopathy because of the
lack of extracardiac organ involvement.All subjects who
tested positive for the N215S genotype (n=26, 13 females,
mean age 49±17 [range, 14-74] years) were characterized in
this prospective monocentric longitudinal cohort study to
determine genotype-specific clinical characteristics of the
N215S (c.644A>G [p.Asn215Ser]) α-galactosidase A gene
variant. All subjects were initially referred with suspicion
of genetically determined hypertrophic cardiomyopathy.
Cardiac hypertrophy (interventricular septum, 12±4 [7-23]
mm; left ventricular posterior wall, 11±4 [7-21] mm; left
ventricular mass, 86±41 [46-195] g/m2) was progressive,
systolic function mainly preserved (cardiac index 2.8±0.6
[1.9-3.9] L/min per m2), and diastolic function mildly
abnormal. Cardiac magnetic resonance imaging revealed
replacement fibrosis in loco typico (18/26, $69\%),$
particularly in subjects >50 years. Elderly subjects had
advanced heart failure, and 6 $(23\%)$ were suggested for
implantable cardioverter-defibrillator therapy. Leukocyte
α-galactosidase A enzyme activity was mildly reduced in 19
subjects and lyso-globotriaosylceramide slightly elevated
(median, 4.9; interquartile range, 1.3-9.1 ng/mL).
Neurological and renal impairments (serum creatinine,
0.87±0.20; median, 0.80; interquartile range, 0.70-1.01
mg/dL; glomerular filtration rate, 102±23; median, 106;
interquartile range, 84-113 mL/min) were discreet. Only 2
subjects developed clinically relevant
proteinuria.α-Galactosidase A genotype N215S does not lead
to the development of a classical Fabry phenotype but
induces a specific cardiac variant of Fabry disease
mimicking nonobstructive hypertrophic cardiomyopathy. The
lack of prominent noncardiac impairment leads to a
significant delay in diagnosis and Fabry-specific therapy.},
cin = {G130},
ddc = {610},
cid = {I:(DE-He78)G130-20160331},
pnm = {322 - Genetics and Pathophysiology (POF3-322)},
pid = {G:(DE-HGF)POF3-322},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:29018006},
doi = {10.1161/CIRCGENETICS.116.001691},
url = {https://inrepo02.dkfz.de/record/131599},
}