Publication Details

Zimmermann-Laband syndrome in monozygotic twins with a mild neurobehavioral phenotype lacking gingival overgrowth-A case report of a novel KCNN3 gene variant

SCHWARZ, M.; RYBA, L.; KŘEPELOVÁ, A.; MOSLEROVÁ, V.; ZELINOVÁ, M.; TURNOVEC, M.; MARTINKOVÁ, J.; KRATOCHVÍLOVÁ, L.; DRAHANSKÝ, M.; MACEK, M.; HAVLOVICOVÁ, M. Zimmermann-Laband syndrome in monozygotic twins with a mild neurobehavioral phenotype lacking gingival overgrowth-A case report of a novel KCNN3 gene variant. American Journal of Medical Genetics, Part A, 2021, vol. 2021, no. 12, p. 1-5. ISSN: 1552-4833.
Czech title
Zimmermannův-Labandův syndrom u monozygotních dvojčat s mírným neurobehaviorálním fenotypem postrádajícím přerůstání dásní - Kazuistika nové varianty genu KCNN3
Type
journal article
Language
English
Authors
SCHWARZ, M.
RYBA, L.
KŘEPELOVÁ, A.
MOSLEROVÁ, V.
ZELINOVÁ, M.
TURNOVEC, M.
MARTINKOVÁ, J.
KRATOCHVÍLOVÁ, L.
Drahanský Martin, prof. Ing., Ph.D.
MACEK, M.
HAVLOVICOVÁ, M.
URL
Keywords

channelopathy, gingival fibromatosis, KCNN3, monozygotic twins, ZimmermannLaband
syndrome

Abstract

Zimmermann-Laband syndrome is a rare, heterogeneous disorder characterized by
gingival hypertrophy or fibromatosis, aplastic/hypoplastic nails, hypoplasia of
the distal phalanges, hypertrichosis, various degrees of intellectual disability,
and distinctive facial features. Three genes are considered causative for ZLS:
KCNH1, KCNN3, and ATP6V1B2. We report on a pair of female concordant monozygotic
twins, both carrying a novel pathogenic variant in the KCNN3 gene, identified
using exome sequencing. Only six ZLS patients with the KCNN3 pathogenic variant
have been reported so far. The twins show facial dysmorphism, hypoplastic distal
phalanges, aplasia or hypoplasia of nails, and hypertrichosis. During infancy,
they showed mild developmental delays, mainly speech. They successfully completed
secondary school education and are socio economically independent. Gingival
overgrowth is absent in both individuals. Our patients exhibited an unusually
mild phenotype compared to published cases, which is an important diagnostic
finding for proper genetic counseling for Zimmermann-Laband syndrome patients and
their families.

Published
2021
Pages
1–5
Journal
American Journal of Medical Genetics, Part A, vol. 2021, no. 12, ISSN 1552-4833
DOI
UT WoS
000730061000001
EID Scopus
BibTeX
@article{BUT176372,
  author="SCHWARZ, M. and RYBA, L. and KŘEPELOVÁ, A. and MOSLEROVÁ, V. and ZELINOVÁ, M. and TURNOVEC, M. and MARTINKOVÁ, J. and KRATOCHVÍLOVÁ, L. and DRAHANSKÝ, M. and MACEK, M. and HAVLOVICOVÁ, M.",
  title="Zimmermann-Laband syndrome in monozygotic twins with a mild neurobehavioral phenotype lacking gingival overgrowth-A case report of a novel KCNN3 gene variant",
  journal="American Journal of Medical Genetics, Part A",
  year="2021",
  volume="2021",
  number="12",
  pages="1--5",
  doi="10.1002/ajmg.a.62616",
  issn="1552-4833",
  url="https://onlinelibrary.wiley.com/doi/10.1002/ajmg.a.62616"
}
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