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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http://dx.doi.org/10.1136/bcr-2024-262894 | DOI Listing |
BMJ Case Rep
December 2024
Department of Pediatrics, Trichy SRM Medical College Hospital and Research Centre, Tiruchirappalli, Tamil Nadu, India.
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.
View Article and Find Full Text PDFJ Clin Med
February 2025
Department of Internal Medicine, Endocrinology and Diabetology, National Medical Institute of the Ministry of the Interior and Administration, 02-507 Warsaw, Poland.
The purpose of this study was to assess bone densitometry parameters in patients with classical and hypermobile Ehlers-Danlos syndrome (EDS) and to determine whether the hypermobile subtype increases the risk of low bone mass, which is particularly important in this patient group, since the genetic mutation responsible for this subtype is still unknown. In order to conduct this study, we collaborated with the EDS society in Poland. A total of 30 females of reproductive age who were included in the study were divided into two groups: Group 1-those with classical EDS ( = 9) and Group 2-those with hypermobile EDS ( = 21).
View Article and Find Full Text PDFClin Transl Gastroenterol
January 2025
Department of Clinical Genetics, Erasmus MC, University Medical Center, Rotterdam, the Netherlands.
Introduction: Classical-like Ehlers-Danlos syndrome type 1 (clEDS1) is a very rare form of Ehlers-Danlos syndrome caused by tenascin-X deficiency, with only 56 individuals reported in medical literature. Tenascin-X is an extracellular matrix protein needed for collagen stability. Previous publications propose that individuals with clEDS1 might be at risk of gastrointestinal (GI) tract perforations and/or tracheal ruptures.
View Article and Find Full Text PDFBMJ Case Rep
January 2025
Medical Department, Lyell McEwin Hospital, Elizabeth Vale, South Australia, Australia.
Ehlers-Danlos syndromes (EDS) are a group of connective tissue disorders associated with skin, ligament, blood vessel and organ abnormalities. Skin hyperextensibility, joint hypermobility and widened atrophic scars are characteristic of classical EDS. Vascular complications, though rare in classical EDS, can be life-threatening, and this necessitates one to look for vascular associations in non-vascular, such as classical, forms of EDS due to the heterogeneity of the syndrome.
View Article and Find Full Text PDFPLoS One
October 2024
Personalised Medicine Centre, School of Medicine, Ulster University, C-TRIC Building, Altnagelvin Hospital, Derry, Londonderry, Northern Ireland.
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