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Kyphoscoliotic Ehlers-Danlos syndrome

MedGen UID:
1639430
Concept ID:
C4551497
Disease or Syndrome
Synonym: EDS VI
SNOMED CT: Ehlers-Danlos syndrome kyphoscoliotic type (718211004); Ehlers-Danlos syndrome type 6 (718211004); Ehlers-Danlos syndrome type VI (718211004)
 
Related genes: FKBP14, PLOD1
 
Orphanet: ORPHA536545

Definition

The Ehlers-Danlos syndromes (EDS) are a group of heritable connective tissue disorders that share the common features of skin hyperextensibility, articular hypermobility, and tissue fragility. The major characteristics of kyphoscoliotic-type EDS are severe muscle hypotonia at birth, generalized joint laxity, scoliosis at birth, and scleral fragility and rupture of the ocular globe (Beighton et al., 1998). Nevo syndrome, previously thought to be a distinct entity, is identical to EDS type VI (Voermans et al., 2009). Genetic Heterogeneity of Ehlers-Danlos Syndrome, Kyphoscoliotic Type See EDSSKCL2 (614557), caused by mutation in the FKBP14 gene (614505). Classification of Ehlers-Danlos Syndromes The current classification of Ehlers-Danlos syndromes is based on a 2017 international classification described by Malfait et al. (2017), which recognizes 13 EDS subtypes. This classification revised the 'Villefranche classification' reported by Beighton et al. (1998). Beighton et al. (1998) reported on a revised nosology of the Ehlers-Danlos syndromes, designated the Villefranche classification. Major and minor diagnostic criteria were defined for each type and complemented whenever possible with laboratory findings. Six main descriptive types were substituted for earlier types numbered with Roman numerals: classic type (EDS I and II), hypermobility type (EDS III), vascular type (EDS IV), kyphoscoliosis type (EDS VI), arthrochalasia type (EDS VIIA and VIIB), and dermatosparaxis type (EDS VIIC). Six other forms were listed, including a category of 'unspecified forms.' [from OMIM]

Professional guidelines

PubMed

Liang B, Yu D, Zhao W, Wang Y, Wang X, Wu X, Chen L, Chen M, Zhang M, Chen X, Lin N, Huang H, Xu L
J Hum Genet 2022 Nov;67(11):629-638. Epub 2022 Jul 27 doi: 10.1038/s10038-022-01062-9. PMID: 35896820

Recent clinical studies

Etiology

Colman M, Vroman R, Dhooge T, Malfait Z, Symoens S, Burnyté B, Nampoothiri S, Kariminejad A, Malfait F, Syx D
Hum Mutat 2022 Dec;43(12):1994-2009. Epub 2022 Sep 12 doi: 10.1002/humu.24456. PMID: 36054293
Ruiz-Botero F, Ramírez-Montaño D, Pachajoa H
Arch Argent Pediatr 2019 Jun 1;117(3):e274-e278. doi: 10.5546/aap.2019.eng.e274. PMID: 31063316
Giunta C, Baumann M, Fauth C, Lindert U, Abdalla EM, Brady AF, Collins J, Dastgir J, Donkervoort S, Ghali N, Johnson DS, Kariminejad A, Koch J, Kraenzlin M, Lahiri N, Lozic B, Manzur AY, Morton JEV, Pilch J, Pollitt RC, Schreiber G, Shannon NL, Sobey G, Vandersteen A, van Dijk FS, Witsch-Baumgartner M, Zschocke J, Pope FM, Bönnemann CG, Rohrbach M
Genet Med 2018 Jan;20(1):42-54. Epub 2017 Jun 15 doi: 10.1038/gim.2017.70. PMID: 28617417Free PMC Article

Diagnosis

Wiegand A, Kastury R, Neogi A, Mani A, Bale A, Cox A
Cold Spring Harb Mol Case Stud 2023 Jun;9(3) Epub 2023 Jul 11 doi: 10.1101/mcs.a006281. PMID: 37433679Free PMC Article
Ni X, Jin C, Jiang Y, Wang O, Li M, Xing X, Xia W
BMC Med Genet 2020 Oct 31;21(1):214. doi: 10.1186/s12881-020-01154-3. PMID: 33129265Free PMC Article
Shin YL, Park YN, Jang MA
J Korean Med Sci 2020 Mar 16;35(10):e96. doi: 10.3346/jkms.2020.35.e96. PMID: 32174067Free PMC Article
Ruiz-Botero F, Ramírez-Montaño D, Pachajoa H
Arch Argent Pediatr 2019 Jun 1;117(3):e274-e278. doi: 10.5546/aap.2019.eng.e274. PMID: 31063316
Giunta C, Baumann M, Fauth C, Lindert U, Abdalla EM, Brady AF, Collins J, Dastgir J, Donkervoort S, Ghali N, Johnson DS, Kariminejad A, Koch J, Kraenzlin M, Lahiri N, Lozic B, Manzur AY, Morton JEV, Pilch J, Pollitt RC, Schreiber G, Shannon NL, Sobey G, Vandersteen A, van Dijk FS, Witsch-Baumgartner M, Zschocke J, Pope FM, Bönnemann CG, Rohrbach M
Genet Med 2018 Jan;20(1):42-54. Epub 2017 Jun 15 doi: 10.1038/gim.2017.70. PMID: 28617417Free PMC Article

Therapy

Ni X, Jin C, Jiang Y, Wang O, Li M, Xing X, Xia W
BMC Med Genet 2020 Oct 31;21(1):214. doi: 10.1186/s12881-020-01154-3. PMID: 33129265Free PMC Article

Clinical prediction guides

Ishikawa Y, Bonna A, Gould DB, Farndale RW
Int J Mol Sci 2023 Oct 13;24(20) doi: 10.3390/ijms242015156. PMID: 37894834Free PMC Article
Colman M, Vroman R, Dhooge T, Malfait Z, Symoens S, Burnyté B, Nampoothiri S, Kariminejad A, Malfait F, Syx D
Hum Mutat 2022 Dec;43(12):1994-2009. Epub 2022 Sep 12 doi: 10.1002/humu.24456. PMID: 36054293
Shin YL, Park YN, Jang MA
J Korean Med Sci 2020 Mar 16;35(10):e96. doi: 10.3346/jkms.2020.35.e96. PMID: 32174067Free PMC Article

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