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An Infant With Primrose Syndrome: A Case Report

Primrose syndrome is a rare autosomal dominant disorder that is characterized by recognizable facial phenotype, sensorineural hearing loss, hypotonia, and developmental delay. All reported probands show de novo ZBTB20 pathogenic variant. Since its discovery in 1982, Primrose syndrome has remained an...

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Detalles Bibliográficos
Autores principales: Long, Calista, DeRose, Barry, Lal, Anthony B, Imboden, Elizabeth
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Cureus 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10625483/
https://www.ncbi.nlm.nih.gov/pubmed/37927765
http://dx.doi.org/10.7759/cureus.46546
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author Long, Calista
DeRose, Barry
Lal, Anthony B
Imboden, Elizabeth
author_facet Long, Calista
DeRose, Barry
Lal, Anthony B
Imboden, Elizabeth
author_sort Long, Calista
collection PubMed
description Primrose syndrome is a rare autosomal dominant disorder that is characterized by recognizable facial phenotype, sensorineural hearing loss, hypotonia, and developmental delay. All reported probands show de novo ZBTB20 pathogenic variant. Since its discovery in 1982, Primrose syndrome has remained an underdiagnosed condition. Awareness of presentation and prompt diagnostic workup are crucial for early identification and proper management. In this case report, we discuss a case of Primrose syndrome diagnosed in an infant born at Wellspan Hospital in York, PA. The patient exhibited classic phenotypic features, including a high hairline, high-arched palate, and brachycephaly at birth, as well as an absent corpus callosum observed on postnatal MRI and genotypic findings of a pathogenic variant in ZBTB20.
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spelling pubmed-106254832023-11-05 An Infant With Primrose Syndrome: A Case Report Long, Calista DeRose, Barry Lal, Anthony B Imboden, Elizabeth Cureus Pediatrics Primrose syndrome is a rare autosomal dominant disorder that is characterized by recognizable facial phenotype, sensorineural hearing loss, hypotonia, and developmental delay. All reported probands show de novo ZBTB20 pathogenic variant. Since its discovery in 1982, Primrose syndrome has remained an underdiagnosed condition. Awareness of presentation and prompt diagnostic workup are crucial for early identification and proper management. In this case report, we discuss a case of Primrose syndrome diagnosed in an infant born at Wellspan Hospital in York, PA. The patient exhibited classic phenotypic features, including a high hairline, high-arched palate, and brachycephaly at birth, as well as an absent corpus callosum observed on postnatal MRI and genotypic findings of a pathogenic variant in ZBTB20. Cureus 2023-10-05 /pmc/articles/PMC10625483/ /pubmed/37927765 http://dx.doi.org/10.7759/cureus.46546 Text en Copyright © 2023, Long et al. https://creativecommons.org/licenses/by/3.0/This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Pediatrics
Long, Calista
DeRose, Barry
Lal, Anthony B
Imboden, Elizabeth
An Infant With Primrose Syndrome: A Case Report
title An Infant With Primrose Syndrome: A Case Report
title_full An Infant With Primrose Syndrome: A Case Report
title_fullStr An Infant With Primrose Syndrome: A Case Report
title_full_unstemmed An Infant With Primrose Syndrome: A Case Report
title_short An Infant With Primrose Syndrome: A Case Report
title_sort infant with primrose syndrome: a case report
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10625483/
https://www.ncbi.nlm.nih.gov/pubmed/37927765
http://dx.doi.org/10.7759/cureus.46546
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