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Renal hypodysplasia/aplasia 2(RHDA2)

MedGen UID:
816689
Concept ID:
C3810359
Disease or Syndrome
Synonym: RHDA2
 
Gene (location): FGF20 (8p22)
 
Monarch Initiative: MONDO:0014319
OMIM®: 615721

Definition

Renal hypodysplasia/aplasia belongs to a group of perinatally lethal renal diseases, including bilateral renal aplasia, unilateral renal agenesis with contralateral dysplasia (URA/RD), and severe obstructive uropathy. Renal aplasia falls at the most severe end of the spectrum of congenital anomalies of the kidney and urinary tract (CAKUT; 610805), and usually results in death in utero or in the perinatal period. Families have been documented in which bilateral renal agenesis or aplasia coexists with unilateral renal aplasia, renal dysplasia, or renal aplasia with renal dysplasia, suggesting that these conditions may belong to a pathogenic continuum or phenotypic spectrum (summary by Joss et al., 2003; Humbert et al., 2014). For a discussion of genetic heterogeneity of renal hypodysplasia/aplasia, see RHDA1 (191830). [from OMIM]

Clinical features

From HPO
Pulmonary hypoplasia
MedGen UID:
78574
Concept ID:
C0265783
Congenital Abnormality
A congenital abnormality in which the lung parenchyma is not fully developed. It may be associated with other congenital abnormalities.
Potter facies
MedGen UID:
78614
Concept ID:
C0266619
Congenital Abnormality
A facial appearance characteristic of a fetus or neonate due to oligohydramnios experienced in the womb, comprising ocular hypertelorism, low-set ears, receding chin, and flattening of the nose.
Redundant skin
MedGen UID:
154379
Concept ID:
C0581342
Pathologic Function
Loose and sagging skin often associated with loss of skin elasticity.

Professional guidelines

PubMed

Faguer S, Decramer S, Chassaing N, Bellanné-Chantelot C, Calvas P, Beaufils S, Bessenay L, Lengelé JP, Dahan K, Ronco P, Devuyst O, Chauveau D
Kidney Int 2011 Oct;80(7):768-76. Epub 2011 Jul 20 doi: 10.1038/ki.2011.225. PMID: 21775974

Recent clinical studies

Etiology

Yang SS, Tsai JD, Kanematsu A, Han CH
J Infect Chemother 2021 Nov;27(11):1543-1554. Epub 2021 Aug 11 doi: 10.1016/j.jiac.2021.07.014. PMID: 34391623
Seeman T, Weigel F, Blahova K, Fencl F, Pruhova S, Hermes K, Klaus R, Lange-Sperandio B, Grote V, John-Kroegel U
Eur J Pediatr 2021 Dec;180(12):3599-3603. Epub 2021 Jun 26 doi: 10.1007/s00431-021-04165-1. PMID: 34176013
Ricci P, Magalhães P, Krochmal M, Pejchinovski M, Daina E, Caruso MR, Goea L, Belczacka I, Remuzzi G, Umbhauer M, Drube J, Pape L, Mischak H, Decramer S, Schaefer F, Schanstra JP, Cereghini S, Zürbig P
Sci Rep 2019 Feb 18;9(1):2225. doi: 10.1038/s41598-019-38713-5. PMID: 30778115Free PMC Article
Schönfelder EM, Knüppel T, Tasic V, Miljkovic P, Konrad M, Wühl E, Antignac C, Bakkaloglu A, Schaefer F, Weber S; ESCAPE Trial Group
Am J Kidney Dis 2006 Jun;47(6):1004-12. doi: 10.1053/j.ajkd.2006.02.177. PMID: 16731295
Glassberg KI, Stephens FD, Lebowitz RL, Braren V, Duckett JW, Jacobs EC, King LR, Perlmutter AD
J Urol 1987 Oct;138(4 Pt 2):1085-92. doi: 10.1016/s0022-5347(17)43510-5. PMID: 3309374

Diagnosis

Yang SS, Tsai JD, Kanematsu A, Han CH
J Infect Chemother 2021 Nov;27(11):1543-1554. Epub 2021 Aug 11 doi: 10.1016/j.jiac.2021.07.014. PMID: 34391623
Seeman T, Weigel F, Blahova K, Fencl F, Pruhova S, Hermes K, Klaus R, Lange-Sperandio B, Grote V, John-Kroegel U
Eur J Pediatr 2021 Dec;180(12):3599-3603. Epub 2021 Jun 26 doi: 10.1007/s00431-021-04165-1. PMID: 34176013
Beaudoux O, Lebre AS, Doco Fenzy M, Spodenkiewicz M, Canivet E, Colosio C, Poirsier C
Am J Med Genet A 2021 Mar;185(3):937-944. Epub 2021 Jan 13 doi: 10.1002/ajmg.a.62050. PMID: 33438842
Rossanti R, Morisada N, Nozu K, Kamei K, Horinouchi T, Yamamura T, Minamikawa S, Fujimura J, Nagano C, Sakakibara N, Ninchoji T, Kaito H, Ito S, Tanaka R, Iijima K
J Hum Genet 2020 Jun;65(6):541-549. Epub 2020 Mar 16 doi: 10.1038/s10038-020-0741-y. PMID: 32203253
Faguer S, Decramer S, Chassaing N, Bellanné-Chantelot C, Calvas P, Beaufils S, Bessenay L, Lengelé JP, Dahan K, Ronco P, Devuyst O, Chauveau D
Kidney Int 2011 Oct;80(7):768-76. Epub 2011 Jul 20 doi: 10.1038/ki.2011.225. PMID: 21775974

Therapy

Tudorache E, Sellier-Leclerc AL, Lenoir M, Tubiana-Rufi N, Bensman A, Bellanne-Chantelot C, Ulinski T
Pediatr Diabetes 2012 Sep;13(6):e35-9. Epub 2012 Jan 20 doi: 10.1111/j.1399-5448.2011.00842.x. PMID: 22260488
Fischbach M, Terzic J, Menouer S, Provot E, Laugel V
Clin Nephrol 2001 Dec;56(6):S17-20. PMID: 11770806
Van Dyck M, Proesmans W
Clin Nephrol 2001 Dec;56(6):S13-6. PMID: 11770805
Hohenfellner K, Wingen AM, Nauroth O, Wühl E, Mehls O, Schaefer F
Pediatr Nephrol 2001 Apr;16(4):356-61. doi: 10.1007/s004670100567. PMID: 11354781

Prognosis

Yang SS, Tsai JD, Kanematsu A, Han CH
J Infect Chemother 2021 Nov;27(11):1543-1554. Epub 2021 Aug 11 doi: 10.1016/j.jiac.2021.07.014. PMID: 34391623
Beaudoux O, Lebre AS, Doco Fenzy M, Spodenkiewicz M, Canivet E, Colosio C, Poirsier C
Am J Med Genet A 2021 Mar;185(3):937-944. Epub 2021 Jan 13 doi: 10.1002/ajmg.a.62050. PMID: 33438842
Tasic V, Ristoska-Bojkovska N, Gucev Z, Lozanovski VJ
Ren Fail 2015;37(9):1440-3. Epub 2015 Sep 3 doi: 10.3109/0886022X.2015.1074488. PMID: 26335537
Faguer S, Decramer S, Chassaing N, Bellanné-Chantelot C, Calvas P, Beaufils S, Bessenay L, Lengelé JP, Dahan K, Ronco P, Devuyst O, Chauveau D
Kidney Int 2011 Oct;80(7):768-76. Epub 2011 Jul 20 doi: 10.1038/ki.2011.225. PMID: 21775974
Sanna-Cherchi S, Ravani P, Corbani V, Parodi S, Haupt R, Piaggio G, Innocenti ML, Somenzi D, Trivelli A, Caridi G, Izzi C, Scolari F, Mattioli G, Allegri L, Ghiggeri GM
Kidney Int 2009 Sep;76(5):528-33. Epub 2009 Jun 17 doi: 10.1038/ki.2009.220. PMID: 19536081

Clinical prediction guides

Negrisolo S, Benetti E
Int J Mol Sci 2023 Feb 19;24(4) doi: 10.3390/ijms24044165. PMID: 36835576Free PMC Article
Martens H, Hennies I, Getwan M, Christians A, Weiss AC, Brand F, Gjerstad AC, Christians A, Gucev Z, Geffers R, Seeman T, Kispert A, Tasic V, Bjerre A, Lienkamp SS, Haffner D, Weber RG
Eur J Hum Genet 2020 Dec;28(12):1681-1693. Epub 2020 Jul 31 doi: 10.1038/s41431-020-0678-9. PMID: 32737436Free PMC Article
Rossanti R, Morisada N, Nozu K, Kamei K, Horinouchi T, Yamamura T, Minamikawa S, Fujimura J, Nagano C, Sakakibara N, Ninchoji T, Kaito H, Ito S, Tanaka R, Iijima K
J Hum Genet 2020 Jun;65(6):541-549. Epub 2020 Mar 16 doi: 10.1038/s10038-020-0741-y. PMID: 32203253
Schönfelder EM, Knüppel T, Tasic V, Miljkovic P, Konrad M, Wühl E, Antignac C, Bakkaloglu A, Schaefer F, Weber S; ESCAPE Trial Group
Am J Kidney Dis 2006 Jun;47(6):1004-12. doi: 10.1053/j.ajkd.2006.02.177. PMID: 16731295
Peruzzi L, Lombardo F, Amore A, Merlini E, Restagno G, Silvestro L, Papalia T, Coppo R
J Urol 2005 Aug;174(2):713-7. doi: 10.1097/01.ju.0000164739.13408.e2. PMID: 16006956

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