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Right isomerism

MedGen UID:
64222
Concept ID:
C0175707
Congenital Abnormality; Disease or Syndrome
Synonyms: Bilateral right-sidedness sequence; Ivemark syndrome
SNOMED CT: Isomerism of right atrial appendage (253336000); Ivemark syndrome (253336000); RAI - right sided atrial isomerism (253336000)
 
HPO: HP:0031855
OMIM®: 208530

Definition

Right atrial isomerism (RAI) is a severe complex congenital heart defect resulting from embryonic disruption of proper left-right axis determination. RAI is usually characterized by complete atrioventricular septal defect with a common atrium and univentricular AV connection, total anomalous pulmonary drainage, and transposition or malposition of the great arteries. Affected individuals present at birth with severe cardiac failure. Other associated abnormalities include bilateral trilobed lungs, midline liver, and asplenia, as well as situs inversus affecting other organs. Left atrial isomerism (LAI) is a related disorder with a somewhat better prognosis. LAI is characterized by bilateral superior vena cava, interruption of the intrahepatic portion of the inferior vena cava, partial anomalous pulmonary venous drainage, and ventricular septal defect. Patients with LAI may have polysplenia and bilateral bilobed lungs, as well as situs inversus affecting other organs. Both RAI and LAI malformation complexes have classically been referred to as Ivemark syndrome (summary by Eronen et al., 2004 and Kaasinen et al., 2010). [from OMIM]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVRight isomerism

Professional guidelines

PubMed

Kahramanoglu O, Demirci O, Uygur L, Erol N, Schiattarella A, Rapisarda AMC
Pediatr Cardiol 2024 Feb;45(2):377-384. Epub 2023 Dec 16 doi: 10.1007/s00246-023-03353-0. PMID: 38103069
Paladini D, Pistorio A, Wu LH, Meccariello G, Lei T, Tuo G, Donarini G, Marasini M, Xie HN
Ultrasound Obstet Gynecol 2018 Jul;52(1):24-34. doi: 10.1002/uog.18907. PMID: 28926132
Loomba RS, Nijhawan K, Anderson R
World J Pediatr Congenit Heart Surg 2016 Jan;7(1):54-62. doi: 10.1177/2150135115601831. PMID: 26714995

Recent clinical studies

Etiology

Ishidou M, Hirose K, Ikai A, Sakamoto K
Asian Cardiovasc Thorac Ann 2022 Jun;30(5):540-548. Epub 2021 Sep 15 doi: 10.1177/02184923211045216. PMID: 34524926
Acherman RJ, Evans WN
Prenat Diagn 2019 Dec;39(13):1220-1224. Epub 2019 Nov 13 doi: 10.1002/pd.5571. PMID: 31600425
Ryerson LM, Pharis S, Pockett C, Soni R, Fruitman D, Guleserian KJ, Nater M, Raynor SC, Mackie AS, Dicken B
Pediatrics 2018 Aug;142(2) doi: 10.1542/peds.2017-4267. PMID: 30049892
Boutet A
Mech Dev 2017 Oct;147:49-60. Epub 2017 Oct 3 doi: 10.1016/j.mod.2017.09.002. PMID: 28986126
Loomba RS, Willes RJ, Kovach JR, Anderson RH
Congenit Heart Dis 2016 Jan-Feb;11(1):7-18. Epub 2015 Jul 29 doi: 10.1111/chd.12288. PMID: 26219620

Diagnosis

Spicer DE, Chowdhury UK, Anderson RH, Pandey NN, Sankhyan LK, George N, Goja S, Malik V
Eur J Cardiothorac Surg 2022 Jun 15;62(1) doi: 10.1093/ejcts/ezac139. PMID: 35234855
Chowdhury UK, Anderson RH, Sankhyan LK, George N, Goja S, Pandey NN, Arvind B, Tharranath I
J Card Surg 2021 Nov;36(11):4280-4291. Epub 2021 Aug 15 doi: 10.1111/jocs.15907. PMID: 34392559
Ryerson LM, Pharis S, Pockett C, Soni R, Fruitman D, Guleserian KJ, Nater M, Raynor SC, Mackie AS, Dicken B
Pediatrics 2018 Aug;142(2) doi: 10.1542/peds.2017-4267. PMID: 30049892
Paladini D, Pistorio A, Wu LH, Meccariello G, Lei T, Tuo G, Donarini G, Marasini M, Xie HN
Ultrasound Obstet Gynecol 2018 Jul;52(1):24-34. doi: 10.1002/uog.18907. PMID: 28926132
Loomba RS, Willes RJ, Kovach JR, Anderson RH
Congenit Heart Dis 2016 Jan-Feb;11(1):7-18. Epub 2015 Jul 29 doi: 10.1111/chd.12288. PMID: 26219620

Therapy

Ishidou M, Hirose K, Ikai A, Sakamoto K
Asian Cardiovasc Thorac Ann 2022 Jun;30(5):540-548. Epub 2021 Sep 15 doi: 10.1177/02184923211045216. PMID: 34524926
Ryerson LM, Pharis S, Pockett C, Soni R, Fruitman D, Guleserian KJ, Nater M, Raynor SC, Mackie AS, Dicken B
Pediatrics 2018 Aug;142(2) doi: 10.1542/peds.2017-4267. PMID: 30049892
Paladini D, Pistorio A, Wu LH, Meccariello G, Lei T, Tuo G, Donarini G, Marasini M, Xie HN
Ultrasound Obstet Gynecol 2018 Jul;52(1):24-34. doi: 10.1002/uog.18907. PMID: 28926132
Boutet A
Mech Dev 2017 Oct;147:49-60. Epub 2017 Oct 3 doi: 10.1016/j.mod.2017.09.002. PMID: 28986126
Changlani DK, Kotecha M, Changlani TD, Varghese R, Kumar RS
Heart Lung Circ 2012 Sep;21(9):598-605. Epub 2012 Jun 20 doi: 10.1016/j.hlc.2012.05.739. PMID: 22726404

Prognosis

Hirose K, Ikai A, Ito H, Ishidou M, Nakatani E, Sakamoto K
Eur J Cardiothorac Surg 2022 Sep 2;62(4) doi: 10.1093/ejcts/ezac080. PMID: 35147681
Ryerson LM, Pharis S, Pockett C, Soni R, Fruitman D, Guleserian KJ, Nater M, Raynor SC, Mackie AS, Dicken B
Pediatrics 2018 Aug;142(2) doi: 10.1542/peds.2017-4267. PMID: 30049892
Loomba RS, Nijhawan K, Anderson R
World J Pediatr Congenit Heart Surg 2016 Jan;7(1):54-62. doi: 10.1177/2150135115601831. PMID: 26714995
Loomba RS, Willes RJ, Kovach JR, Anderson RH
Congenit Heart Dis 2016 Jan-Feb;11(1):7-18. Epub 2015 Jul 29 doi: 10.1111/chd.12288. PMID: 26219620
Weichert J, Hartge D, Germer U, Axt-Fliedner R, Gembruch U
Ultrasound Obstet Gynecol 2011 May;37(5):543-8. Epub 2011 Mar 25 doi: 10.1002/uog.7764. PMID: 20922781

Clinical prediction guides

Spicer DE, Chowdhury UK, Anderson RH, Pandey NN, Sankhyan LK, George N, Goja S, Malik V
Eur J Cardiothorac Surg 2022 Jun 15;62(1) doi: 10.1093/ejcts/ezac139. PMID: 35234855
Acherman RJ, Evans WN
Prenat Diagn 2019 Dec;39(13):1220-1224. Epub 2019 Nov 13 doi: 10.1002/pd.5571. PMID: 31600425
Ryerson LM, Pharis S, Pockett C, Soni R, Fruitman D, Guleserian KJ, Nater M, Raynor SC, Mackie AS, Dicken B
Pediatrics 2018 Aug;142(2) doi: 10.1542/peds.2017-4267. PMID: 30049892
Boutet A
Mech Dev 2017 Oct;147:49-60. Epub 2017 Oct 3 doi: 10.1016/j.mod.2017.09.002. PMID: 28986126
Loomba RS, Willes RJ, Kovach JR, Anderson RH
Congenit Heart Dis 2016 Jan-Feb;11(1):7-18. Epub 2015 Jul 29 doi: 10.1111/chd.12288. PMID: 26219620

Recent systematic reviews

Paladini D, Pistorio A, Wu LH, Meccariello G, Lei T, Tuo G, Donarini G, Marasini M, Xie HN
Ultrasound Obstet Gynecol 2018 Jul;52(1):24-34. doi: 10.1002/uog.18907. PMID: 28926132
Loomba RS, Nijhawan K, Anderson R
World J Pediatr Congenit Heart Surg 2016 Jan;7(1):54-62. doi: 10.1177/2150135115601831. PMID: 26714995
Loomba RS, Willes RJ, Kovach JR, Anderson RH
Congenit Heart Dis 2016 Jan-Feb;11(1):7-18. Epub 2015 Jul 29 doi: 10.1111/chd.12288. PMID: 26219620

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