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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...
Autores principales: | , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
John Wiley & Sons, Inc.
2021
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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. |
format | Online Article Text |
id | pubmed-8100402 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | John Wiley & Sons, Inc. |
record_format | MEDLINE/PubMed |
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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