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Renal-hepatic-pancreatic dysplasia(RHPD)

MedGen UID:
382215
Concept ID:
C2673883
Disease or Syndrome
Synonym: RHPD
SNOMED CT: Renal hepatic pancreatic dysplasia (763891005); Ivemark II syndrome (763891005); Renohepaticopancreatic dysplasia (763891005)
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
 
Monarch Initiative: MONDO:0017417
OMIM® Phenotypic series: PS208540
Orphanet: ORPHA294415

Definition

A rare genetic developmental defect during embryogenesis syndrome with the triad of pancreatic fibrosis (and cysts, with a reduction of parenchymal tissue), renal dysplasia (with peripheral cortical cysts, primitive collecting ducts, glomerular cysts and metaplastic cartilage) and hepatic dysgenesis (enlarged portal areas containing numerous elongated binary profiles with a tendency to perilobular fibrosis). Situs abnormalities, skeletal anomalies and anencephaly have also been associated. Patients that survive the neonatal period present renal insufficiency, chronic jaundice and insulin-dependant diabetes. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVRenal-hepatic-pancreatic dysplasia

Recent clinical studies

Etiology

Kapur RP, Cole B, Zhang M, Lin J, Fligner CL
Pediatr Dev Pathol 2013 May-Jun;16(3):191-200. Epub 2013 Feb 25 doi: 10.2350/12-12-1281-OA.1. PMID: 23438674
Frank V, Habbig S, Bartram MP, Eisenberger T, Veenstra-Knol HE, Decker C, Boorsma RA, Göbel H, Nürnberg G, Griessmann A, Franke M, Borgal L, Kohli P, Völker LA, Dötsch J, Nürnberg P, Benzing T, Bolz HJ, Johnson C, Gerkes EH, Schermer B, Bergmann C
Hum Mol Genet 2013 Jun 1;22(11):2177-85. Epub 2013 Feb 14 doi: 10.1093/hmg/ddt070. PMID: 23418306
Fiskerstrand T, Houge G, Sund S, Scheie D, Leh S, Boman H, Knappskog PM
J Mol Diagn 2010 Jan;12(1):125-31. Epub 2009 Dec 10 doi: 10.2353/jmoldx.2010.090033. PMID: 20007846Free PMC Article
Carles D, Serville F, Dubecq JP, Gonnet JM
Eur J Pediatr 1988 May;147(4):431-2. doi: 10.1007/BF00496429. PMID: 3396599
Bernstein J, Chandra M, Creswell J, Kahn E, Malouf NN, McVicar M, Weinberg AG, Wybel RE
Am J Med Genet 1987 Feb;26(2):391-403. doi: 10.1002/ajmg.1320260218. PMID: 3812591

Diagnosis

Zhu H, Zhao ZH, Zhu SY, Xiong F, He LH, Zhang Y, Wang J
BMC Pediatr 2022 Oct 18;22(1):603. doi: 10.1186/s12887-022-03659-7. PMID: 36253741Free PMC Article
Inaguma Y, Kaito H, Morisada N, Iijima K, Tanaka R
Pediatr Int 2019 Feb;61(2):210-212. Epub 2019 Feb 7 doi: 10.1111/ped.13758. PMID: 30734414
Rajagopalan R, Grochowski CM, Gilbert MA, Falsey AM, Coleman K, Romero R, Loomes KM, Piccoli DA, Devoto M, Spinner NB
Am J Med Genet A 2016 Mar;170(3):750-3. Epub 2015 Dec 24 doi: 10.1002/ajmg.a.37512. PMID: 26697755
Hussain Z, Masroor I, Haider QU, Alam T
J Coll Physicians Surg Pak 2011 Apr;21(4):242-4. PMID: 21453625
Bernstein J, Chandra M, Creswell J, Kahn E, Malouf NN, McVicar M, Weinberg AG, Wybel RE
Am J Med Genet 1987 Feb;26(2):391-403. doi: 10.1002/ajmg.1320260218. PMID: 3812591

Prognosis

Gunther K, Imseis EM, Samuel JP, Hillman EA, Ojala TH, Jahnukainen T, Hillman PR
Mol Genet Genomic Med 2023 Apr;11(4):e2135. Epub 2023 Feb 8 doi: 10.1002/mgg3.2135. PMID: 36756677Free PMC Article
Peña-Padilla C, Marshall CR, Walker S, Scherer SW, Tavares-Macías G, Razo-Jiménez G, Bobadilla-Morales L, Acosta-Fernández E, Corona-Rivera A, Mendoza-Londono R, Corona-Rivera JR
Clin Genet 2017 Apr;91(4):640-646. Epub 2017 Feb 16 doi: 10.1111/cge.12924. PMID: 27874174
Witters I, Devriendt K, Spinnewijn D, Moerman P, Van Assche FA, Fryns JP
Am J Med Genet 2002 Jan 22;107(3):233-6. doi: 10.1002/ajmg.10131. PMID: 11807905
Torra R, Alós L, Ramos J, Estivill X
J Med Genet 1996 May;33(5):409-12. doi: 10.1136/jmg.33.5.409. PMID: 8733053Free PMC Article

Clinical prediction guides

Thuy PX, Jang TK, Moon EY
Int J Mol Sci 2024 Sep 26;25(19) doi: 10.3390/ijms251910369. PMID: 39408701Free PMC Article
Peña-Padilla C, Marshall CR, Walker S, Scherer SW, Tavares-Macías G, Razo-Jiménez G, Bobadilla-Morales L, Acosta-Fernández E, Corona-Rivera A, Mendoza-Londono R, Corona-Rivera JR
Clin Genet 2017 Apr;91(4):640-646. Epub 2017 Feb 16 doi: 10.1111/cge.12924. PMID: 27874174
Rajagopalan R, Grochowski CM, Gilbert MA, Falsey AM, Coleman K, Romero R, Loomes KM, Piccoli DA, Devoto M, Spinner NB
Am J Med Genet A 2016 Mar;170(3):750-3. Epub 2015 Dec 24 doi: 10.1002/ajmg.a.37512. PMID: 26697755
Frank V, Habbig S, Bartram MP, Eisenberger T, Veenstra-Knol HE, Decker C, Boorsma RA, Göbel H, Nürnberg G, Griessmann A, Franke M, Borgal L, Kohli P, Völker LA, Dötsch J, Nürnberg P, Benzing T, Bolz HJ, Johnson C, Gerkes EH, Schermer B, Bergmann C
Hum Mol Genet 2013 Jun 1;22(11):2177-85. Epub 2013 Feb 14 doi: 10.1093/hmg/ddt070. PMID: 23418306
Fiskerstrand T, Houge G, Sund S, Scheie D, Leh S, Boman H, Knappskog PM
J Mol Diagn 2010 Jan;12(1):125-31. Epub 2009 Dec 10 doi: 10.2353/jmoldx.2010.090033. PMID: 20007846Free PMC Article

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