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A novel homozygous variant in C1QBP causes severe IUGR, edema, and cardiomyopathy in two fetuses

The C1QBP protein (complement component 1 Q subcomponent‐binding protein), encoded by the C1QBP gene, is a multifunctional protein predominantly localized in the mitochondrial matrix. Biallelic variants have previously been shown to give rise to combined respiratory‐chain deficiencies with variable...

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Autores principales: Alstrup, Morten, Vogel, Ida, Sandager, Puk, Blechingberg, Jenny, Becher, Naja, Østergaard, Elsebet
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley & Sons, Inc. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8100402/
https://www.ncbi.nlm.nih.gov/pubmed/33977026
http://dx.doi.org/10.1002/jmd2.12209
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author Alstrup, Morten
Vogel, Ida
Sandager, Puk
Blechingberg, Jenny
Becher, Naja
Østergaard, Elsebet
author_facet Alstrup, Morten
Vogel, Ida
Sandager, Puk
Blechingberg, Jenny
Becher, Naja
Østergaard, Elsebet
author_sort Alstrup, Morten
collection PubMed
description The C1QBP protein (complement component 1 Q subcomponent‐binding protein), encoded by the C1QBP gene, is a multifunctional protein predominantly localized in the mitochondrial matrix. Biallelic variants have previously been shown to give rise to combined respiratory‐chain deficiencies with variable phenotypic presentation, severity, and age at onset, from intrauterine with a mostly lethal course, to a late‐onset mild myopathy. We present two fetuses, one male and one female, of first‐cousin parents, with severe intrauterine growth retardation, oligo/anhydramnios, edema, and cardiomyopathy as the most prominent prenatal symptoms. Both fetuses showed no copy number variants by chromosome microarray analysis. Analysis of a fibroblast culture from one of the fetuses showed deficiency of respiratory chain complex IV, and using exome sequencing, we identified homozygosity for a novel variant in C1QBP in both fetuses. To our knowledge, only six patients with pathogenic variants in C1QBP have been reported previously and with this report, we add a novel pathogenic variant in C1QBP found in two related fetuses.
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spelling pubmed-81004022021-05-10 A novel homozygous variant in C1QBP causes severe IUGR, edema, and cardiomyopathy in two fetuses Alstrup, Morten Vogel, Ida Sandager, Puk Blechingberg, Jenny Becher, Naja Østergaard, Elsebet JIMD Rep Case Reports The C1QBP protein (complement component 1 Q subcomponent‐binding protein), encoded by the C1QBP gene, is a multifunctional protein predominantly localized in the mitochondrial matrix. Biallelic variants have previously been shown to give rise to combined respiratory‐chain deficiencies with variable phenotypic presentation, severity, and age at onset, from intrauterine with a mostly lethal course, to a late‐onset mild myopathy. We present two fetuses, one male and one female, of first‐cousin parents, with severe intrauterine growth retardation, oligo/anhydramnios, edema, and cardiomyopathy as the most prominent prenatal symptoms. Both fetuses showed no copy number variants by chromosome microarray analysis. Analysis of a fibroblast culture from one of the fetuses showed deficiency of respiratory chain complex IV, and using exome sequencing, we identified homozygosity for a novel variant in C1QBP in both fetuses. To our knowledge, only six patients with pathogenic variants in C1QBP have been reported previously and with this report, we add a novel pathogenic variant in C1QBP found in two related fetuses. John Wiley & Sons, Inc. 2021-03-05 /pmc/articles/PMC8100402/ /pubmed/33977026 http://dx.doi.org/10.1002/jmd2.12209 Text en © 2021 The Authors. JIMD Reports published by John Wiley & Sons Ltd on behalf of SSIEM. https://creativecommons.org/licenses/by/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Reports
Alstrup, Morten
Vogel, Ida
Sandager, Puk
Blechingberg, Jenny
Becher, Naja
Østergaard, Elsebet
A novel homozygous variant in C1QBP causes severe IUGR, edema, and cardiomyopathy in two fetuses
title A novel homozygous variant in C1QBP causes severe IUGR, edema, and cardiomyopathy in two fetuses
title_full A novel homozygous variant in C1QBP causes severe IUGR, edema, and cardiomyopathy in two fetuses
title_fullStr A novel homozygous variant in C1QBP causes severe IUGR, edema, and cardiomyopathy in two fetuses
title_full_unstemmed A novel homozygous variant in C1QBP causes severe IUGR, edema, and cardiomyopathy in two fetuses
title_short A novel homozygous variant in C1QBP causes severe IUGR, edema, and cardiomyopathy in two fetuses
title_sort novel homozygous variant in c1qbp causes severe iugr, edema, and cardiomyopathy in two fetuses
topic Case Reports
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8100402/
https://www.ncbi.nlm.nih.gov/pubmed/33977026
http://dx.doi.org/10.1002/jmd2.12209
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