Classical Ehlers-Danlos syndrome with cranio-cervical instability in an infant due to a novel COL5A1 gene mutation

Ehlers-Danlos syndromes (EDSs) are a group of connective tissue disorders with diverse clinical and genetic profiles. Classical Ehlers-Danlos syndrome (cEDS), the second most common type, is characterised by skin hyperextensibility, atrophic scars and joint hypermobility, primarily due to mutations in COL5A1 and COL5A2 genes. We report a case of an infant with severe cEDS presenting with motor developmental delay, joint hyperextensibility and cranio-cervical instability. Genetic analysis identified a novel heterozygous missense mutation in COL5A1 (c.386G>T) and a non-segregating variant in COL1A2. The child's clinical features were more severe compared with his mother and grandmother, highlighting the variable expression of cEDS. Treatment included vitamin D and iron supplementation, physiotherapy and avoidance of excessive stretching. The child later required surgical intervention for cervical dislocation. This case emphasises the importance of clinical examination of family members and targeted genetic testing along with individualised management for severe cEDS presentations.

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Publication Details

Journal
BMJ Case Reports
Published
2024-12-01
DOI
https://doi.org/10.1136/bcr-2024-262894
Citations
1
Primary Topic
Connective tissue disorders research
Type
article
Field-Weighted Citation Impact
0.55
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article

Classical Ehlers-Danlos syndrome with cranio-cervical instability in an infant due to a novel COL5A1 gene mutation

Balasubramaniyan Muthuvel, Rajalakshmi Selvaraj, Haridharanee Thomas Paulraj, Vidhya Chinnasamy
1 citations
BMJ Case Reports
Connective tissue disorders research
0.55
article

Classical Ehlers-Danlos syndrome with cranio-cervical instability in an infant due to a novel COL5A1 gene mutation

Balasubramaniyan Muthuvel, Rajalakshmi Selvaraj, Haridharanee Thomas Paulraj, Vidhya Chinnasamy
article en
1 citations

Abstract

Ehlers-Danlos syndromes (EDSs) are a group of connective tissue disorders with diverse clinical and genetic profiles. Classical Ehlers-Danlos syndrome (cEDS), the second most common type, is characterised by skin hyperextensibility, atrophic scars and joint hypermobility, primarily due to mutations in COL5A1 and COL5A2 genes. We report a case of an infant with severe cEDS presenting with motor developmental delay, joint hyperextensibility and cranio-cervical instability. Genetic analysis identified a novel heterozygous missense mutation in COL5A1 (c.386G>T) and a non-segregating variant in COL1A2. The child's clinical features were more severe compared with his mother and grandmother, highlighting the variable expression of cEDS. Treatment included vitamin D and iron supplementation, physiotherapy and avoidance of excessive stretching. The child later required surgical intervention for cervical dislocation. This case emphasises the importance of clinical examination of family members and targeted genetic testing along with individualised management for severe cEDS presentations.

BMJ Case ReportsVol. 17(12)
Trichy SRM Medical College Hospital and Research Centre (IN)
Openalex Percentile: Top 23%
Connective tissue disorders research
0.55
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Classical Ehlers-Danlos syndrome with cranio-cervical instability in an infant due to a novel COL5A1 gene mutation — Balasubramaniyan Muthuvel, Rajalakshmi Selvaraj, et al. · BMJ Case Reports (2024) | TGRS Research Map | TGRS