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VACTERL association

MedGen UID:
365892
Concept ID:
C1735591
Congenital Abnormality
Synonyms: VACTEL Association; VACTERL Syndrome; Vertebral Anal Tracheoesophageal Esophageal Radial anomalies
SNOMED CT: Vertebral abnormalities, anal atresia, cardiac abnormalities, tracheo-esophageal fistula, renal anomalies, limb defects (VACTERL) syndrome (431395004); Vertebral abnormalities, anal atresia, cardiac abnormalities, tracheo-esophageal fistula, renal anomalies, limb defects syndrome (431395004); VACTERL syndrome (431395004)

Definition

VACTERL association is a disorder that affects many body systems. VACTERL stands for vertebral defects, anal atresia, cardiac defects, tracheo-esophageal fistula, renal anomalies, and limb abnormalities. People diagnosed with VACTERL association typically have at least three of these characteristic features. Affected individuals may have additional abnormalities that are not among the characteristic features of VACTERL association.

Defects in the bones of the spine (vertebrae) are present in 60 to 80 percent of people with VACTERL association. These defects may include misshapen vertebrae, fused vertebrae, and missing or extra vertebrae. In some people, spinal problems require surgery or cause health problems, such as back pain of varying severity, throughout life. Sixty to 90 percent of individuals with VACTERL association have narrowing or blockage of the anus (anal atresia). Anal atresia may be accompanied by abnormalities of the genitalia and urinary tract (genitourinary anomalies). Heart (cardiac) defects occur in 40 to 80 percent of individuals with VACTERL association. Cardiac defects can range in severity from a life-threatening problem to a subtle defect that does not cause health problems. Fifty to 80 percent of people with VACTERL association have a tracheo-esophageal fistula, which is an abnormal connection (fistula) between the esophagus and the windpipe (trachea). Tracheo-esophageal fistula can cause problems with breathing and feeding early in life and typically requires surgical correction in infancy. Kidney (renal) anomalies occur in 50 to 80 percent of individuals with VACTERL association. Affected individuals may be missing one or both kidneys or have abnormally developed or misshapen kidneys, which can affect kidney function. Limb abnormalities are seen in 40 to 50 percent of people with VACTERL association. These abnormalities most commonly include poorly developed or missing thumbs or underdeveloped forearms and hands.

Some of the features of VACTERL association can be subtle and are not identified until late in childhood or adulthood, making diagnosis of this condition difficult. [from MedlinePlus Genetics]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVVACTERL association

Professional guidelines

PubMed

Zhang W, Chen J
Prenat Diagn 2022 Apr;42(4):482-483. Epub 2022 Jan 31 doi: 10.1002/pd.6100. PMID: 35037266
van den Hondel D, Wijers CH, van Bever Y, de Klein A, Marcelis CL, de Blaauw I, Sloots CE, IJsselstijn H
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Debost-Legrand A, Goumy C, Laurichesse-Delmas H, Déchelotte P, Perthus I, Francannet C, Lémery D, Gallot D
Birth Defects Res A Clin Mol Teratol 2015 Oct;103(10):880-6. Epub 2015 Jun 2 doi: 10.1002/bdra.23346. PMID: 26033534

Recent clinical studies

Etiology

Tonni G, Koçak Ç, Grisolia G, Rizzo G, Araujo Júnior E, Werner H, Ruano R, Sepulveda W, Bonasoni MP, Lituania M; “International Perinatology Research Group (IPRG)”
Fetal Pediatr Pathol 2023 Aug;42(4):651-674. Epub 2023 May 17 doi: 10.1080/15513815.2023.2206905. PMID: 37195727
Altintas B, Giri N, McReynolds LJ, Best A, Alter BP
Haematologica 2023 Jan 1;108(1):69-82. doi: 10.3324/haematol.2021.279981. PMID: 35417938Free PMC Article
Chong C, Hamza Y, Tan YW, Paul A, Garriboli M, Wright AJ, Olsburgh J, Taylor C, Sinha MD, Mishra P, Taghizadeh A
J Pediatr Urol 2022 Apr;18(2):150.e1-150.e6. Epub 2022 Feb 7 doi: 10.1016/j.jpurol.2022.01.019. PMID: 35283020
Solomon BD
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Spitz L
Orphanet J Rare Dis 2007 May 11;2:24. doi: 10.1186/1750-1172-2-24. PMID: 17498283Free PMC Article

Diagnosis

Tonni G, Koçak Ç, Grisolia G, Rizzo G, Araujo Júnior E, Werner H, Ruano R, Sepulveda W, Bonasoni MP, Lituania M; “International Perinatology Research Group (IPRG)”
Fetal Pediatr Pathol 2023 Aug;42(4):651-674. Epub 2023 May 17 doi: 10.1080/15513815.2023.2206905. PMID: 37195727
van Lennep M, Singendonk MMJ, Dall'Oglio L, Gottrand F, Krishnan U, Terheggen-Lagro SWJ, Omari TI, Benninga MA, van Wijk MP
Nat Rev Dis Primers 2019 Apr 18;5(1):26. doi: 10.1038/s41572-019-0077-0. PMID: 31000707
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Solomon BD
Orphanet J Rare Dis 2011 Aug 16;6:56. doi: 10.1186/1750-1172-6-56. PMID: 21846383Free PMC Article

Therapy

Gill KG
Pediatr Radiol 2022 Apr;52(4):777-785. Epub 2021 Nov 3 doi: 10.1007/s00247-021-05200-x. PMID: 34731287
Society for Maternal-Fetal Medicine (SMFM), Gandhi M, Rac MWF, McKinney J
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Hilger A, Schramm C, Draaken M, Mughal SS, Dworschak G, Bartels E, Hoffmann P, Nöthen MM, Reutter H, Ludwig M
Pediatr Surg Int 2012 Jul;28(7):725-9. Epub 2012 Mar 16 doi: 10.1007/s00383-012-3073-y. PMID: 22422375
Kim J, Kim P, Hui CC
Clin Genet 2001 May;59(5):306-15. doi: 10.1034/j.1399-0004.2001.590503.x. PMID: 11359461

Prognosis

Chong C, Hamza Y, Tan YW, Paul A, Garriboli M, Wright AJ, Olsburgh J, Taylor C, Sinha MD, Mishra P, Taghizadeh A
J Pediatr Urol 2022 Apr;18(2):150.e1-150.e6. Epub 2022 Feb 7 doi: 10.1016/j.jpurol.2022.01.019. PMID: 35283020
Brosens E, Ploeg M, van Bever Y, Koopmans AE, IJsselstijn H, Rottier RJ, Wijnen R, Tibboel D, de Klein A
Eur J Med Genet 2014 Aug;57(8):440-52. Epub 2014 Jun 13 doi: 10.1016/j.ejmg.2014.05.009. PMID: 24931924
Solomon BD
Orphanet J Rare Dis 2011 Aug 16;6:56. doi: 10.1186/1750-1172-6-56. PMID: 21846383Free PMC Article
Spitz L
Orphanet J Rare Dis 2007 May 11;2:24. doi: 10.1186/1750-1172-2-24. PMID: 17498283Free PMC Article
Kuo MF, Tsai Y, Hsu WM, Chen RS, Tu YK, Wang HS
J Neurosurg 2007 Mar;106(3 Suppl):201-4. doi: 10.3171/ped.2007.106.3.201. PMID: 17465385

Clinical prediction guides

Chong C, Hamza Y, Tan YW, Paul A, Garriboli M, Wright AJ, Olsburgh J, Taylor C, Sinha MD, Mishra P, Taghizadeh A
J Pediatr Urol 2022 Apr;18(2):150.e1-150.e6. Epub 2022 Feb 7 doi: 10.1016/j.jpurol.2022.01.019. PMID: 35283020
Boer LL, Morava E, Klein WM, Schepens-Franke AN, Oostra RJ
Birth Defects Res 2017 Jun 1;109(10):791-804. Epub 2017 May 16 doi: 10.1002/bdr2.1049. PMID: 28509418
Lubinsky M
Am J Med Genet A 2015 Nov;167A(11):2594-8. Epub 2015 Jul 21 doi: 10.1002/ajmg.a.37257. PMID: 26198446
Brosens E, Ploeg M, van Bever Y, Koopmans AE, IJsselstijn H, Rottier RJ, Wijnen R, Tibboel D, de Klein A
Eur J Med Genet 2014 Aug;57(8):440-52. Epub 2014 Jun 13 doi: 10.1016/j.ejmg.2014.05.009. PMID: 24931924
Kim J, Kim P, Hui CC
Clin Genet 2001 May;59(5):306-15. doi: 10.1034/j.1399-0004.2001.590503.x. PMID: 11359461

Recent systematic reviews

Amirhassankhani S, Lloyd MS
J Craniofac Surg 2018 Mar;29(2):372-375. doi: 10.1097/SCS.0000000000004201. PMID: 29239919
Bartels E, Schulz AC, Mora NW, Pineda-Alvarez DE, Wijers CHW, Marcelis CM, Stressig R, Ritgen J, Schmiedeke E, Mattheisen M, Draaken M, Hoffmann P, Hilger AC, Dworschak GC, Baudisch F, Ludwig M, Bagci S, Müller A, Gembruch U, Geipel A, Berg C, Bartmann P, Nöthen MM, van Rooij IALM, Solomon BD, Reutter HM
Clin Dysmorphol 2012 Oct;21(4):191-195. doi: 10.1097/MCD.0b013e328358243c. PMID: 22895008Free PMC Article

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