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Caroli disease

MedGen UID:
57924
Concept ID:
C0162510
Congenital Abnormality; Disease or Syndrome
Synonyms: Caroli disease isolated; Congenital polycystic dilatation of intrahepatic bile ducts; Cystic dilatation of the intrahepatic biliary tree
SNOMED CT: Caroli disease (717232005)
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).
Not genetically inherited
MedGen UID:
988794
Concept ID:
CN307044
Finding
Source: Orphanet
clinical entity without genetic inheritance.
 
Monarch Initiative: MONDO:0010913
OMIM®: 600643
Orphanet: ORPHA53035

Definition

A rare congenital disorder characterised by multifocal, segmental dilatation of the large intrahepatic bile ducts. It may present at any age and predominantly affects females. Less than 250 cases have been described worldwide. Caroli disease is characterised by bile ductal ectasia without other apparent hepatic abnormalities. It presents with recurrent bacterial cholangitis, biliary stones causing biliary pain or episodes of pancreatitis. The more common variant of this disease, named Caroli syndrome, is characterised by dilatations of the large bile duct associated with congenital hepatic fibrosis. The aetiology of Caroli disease is unknown and its occurrence is sporadic, whereas Caroli syndrome is generally inherited in an autosomal recessive manner. [from SNOMEDCT_US]

Term Hierarchy

Professional guidelines

PubMed

Brown ZJ, Baghdadi A, Kamel I, Labiner HE, Hewitt DB, Pawlik TM
HPB (Oxford) 2023 Jan;25(1):14-25. Epub 2022 Oct 5 doi: 10.1016/j.hpb.2022.09.010. PMID: 36257874
European Association for the Study of the Liver
J Hepatol 2022 Oct;77(4):1083-1108. Epub 2022 Jun 18 doi: 10.1016/j.jhep.2022.06.002. PMID: 35728731
Soares KC, Goldstein SD, Ghaseb MA, Kamel I, Hackam DJ, Pawlik TM
Pediatr Surg Int 2017 Jun;33(6):637-650. Epub 2017 Mar 31 doi: 10.1007/s00383-017-4083-6. PMID: 28364277

Recent clinical studies

Etiology

Burgmaier K, Broekaert IJ, Liebau MC
Adv Kidney Dis Health 2023 Sep;30(5):468-476. doi: 10.1053/j.akdh.2023.01.005. PMID: 38097335
Brown ZJ, Baghdadi A, Kamel I, Labiner HE, Hewitt DB, Pawlik TM
HPB (Oxford) 2023 Jan;25(1):14-25. Epub 2022 Oct 5 doi: 10.1016/j.hpb.2022.09.010. PMID: 36257874
Soares KC, Goldstein SD, Ghaseb MA, Kamel I, Hackam DJ, Pawlik TM
Pediatr Surg Int 2017 Jun;33(6):637-650. Epub 2017 Mar 31 doi: 10.1007/s00383-017-4083-6. PMID: 28364277
Halbritter J, Porath JD, Diaz KA, Braun DA, Kohl S, Chaki M, Allen SJ, Soliman NA, Hildebrandt F, Otto EA; GPN Study Group
Hum Genet 2013 Aug;132(8):865-84. Epub 2013 Apr 5 doi: 10.1007/s00439-013-1297-0. PMID: 23559409Free PMC Article
Zhang DY, Ji ZF, Shen XZ, Liu HY, Pan BJ, Dong L
J Dig Dis 2012 Sep;13(9):491-5. doi: 10.1111/j.1751-2980.2012.00619.x. PMID: 22908976

Diagnosis

Brown ZJ, Baghdadi A, Kamel I, Labiner HE, Hewitt DB, Pawlik TM
HPB (Oxford) 2023 Jan;25(1):14-25. Epub 2022 Oct 5 doi: 10.1016/j.hpb.2022.09.010. PMID: 36257874
European Association for the Study of the Liver
J Hepatol 2022 Oct;77(4):1083-1108. Epub 2022 Jun 18 doi: 10.1016/j.jhep.2022.06.002. PMID: 35728731
Soares KC, Goldstein SD, Ghaseb MA, Kamel I, Hackam DJ, Pawlik TM
Pediatr Surg Int 2017 Jun;33(6):637-650. Epub 2017 Mar 31 doi: 10.1007/s00383-017-4083-6. PMID: 28364277
Issar P, Issar SK
Indian J Gastroenterol 2014 Sep;33(5):500. Epub 2013 Sep 11 doi: 10.1007/s12664-013-0403-5. PMID: 24022473
Mumoli N, Cei M
Mayo Clin Proc 2007 Feb;82(2):208. doi: 10.4065/82.2.208. PMID: 17290729

Therapy

Karagyozov P, Dobreva I, Tishkov I, Draganov K, Velikova K
Gastrointest Endosc 2022 Aug;96(2):377-378. Epub 2022 Apr 19 doi: 10.1016/j.gie.2022.04.013. PMID: 35452632
Petters LM, Vogel TP, Munoz FM, Hernandez JA, Koohmaraie S, Nowicki MJ, Zumbro CE, Mysore KR
Am J Transplant 2021 Jul;21(7):2596-2599. Epub 2021 Apr 2 doi: 10.1111/ajt.16572. PMID: 33754452Free PMC Article
Hogan MC, Torres VE
Nephrol News Issues 2008 Aug;22(9):29-31. PMID: 18778002
Ananthakrishnan AN, Saeian K
Curr Gastroenterol Rep 2007 Apr;9(2):151-5. doi: 10.1007/s11894-007-0010-7. PMID: 17418061
Burt MJ, Chambers ST, Chapman BA, Strack MF, Troughton WD
J Gastroenterol Hepatol 1994 Mar-Apr;9(2):194-7. doi: 10.1111/j.1440-1746.1994.tb01242.x. PMID: 8003654

Prognosis

Burgmaier K, Broekaert IJ, Liebau MC
Adv Kidney Dis Health 2023 Sep;30(5):468-476. doi: 10.1053/j.akdh.2023.01.005. PMID: 38097335
Fahrner R, Dennler SG, Inderbitzin D
World J Gastroenterol 2020 Aug 21;26(31):4718-4728. doi: 10.3748/wjg.v26.i31.4718. PMID: 32884228Free PMC Article
Soares KC, Goldstein SD, Ghaseb MA, Kamel I, Hackam DJ, Pawlik TM
Pediatr Surg Int 2017 Jun;33(6):637-650. Epub 2017 Mar 31 doi: 10.1007/s00383-017-4083-6. PMID: 28364277
Zhang DY, Ji ZF, Shen XZ, Liu HY, Pan BJ, Dong L
J Dig Dis 2012 Sep;13(9):491-5. doi: 10.1111/j.1751-2980.2012.00619.x. PMID: 22908976
Harjai MM, Bal RK
Pediatr Surg Int 2000;16(5-6):431-2. doi: 10.1007/s003839900323. PMID: 10955583

Clinical prediction guides

Fahrner R, Dennler SG, Inderbitzin D
World J Gastroenterol 2020 Aug 21;26(31):4718-4728. doi: 10.3748/wjg.v26.i31.4718. PMID: 32884228Free PMC Article
Fahrner R, Dennler SGC, Dondorf F, Ardelt M, Rauchfuss F, Settmacher U
J Visc Surg 2019 Apr;156(2):91-95. Epub 2018 Jun 19 doi: 10.1016/j.jviscsurg.2018.06.001. PMID: 29929811
Huang Z, Zhang F, Luo Z
Gastroenterology 2017 Mar;152(4):697-698. Epub 2017 Feb 1 doi: 10.1053/j.gastro.2016.09.052. PMID: 28157514
Wang ZX, Li YG, Wang RL, Li YW, Li ZY, Wang LF, Yang HY, Zhu Y, Wang Y, Bai YF, He TT, Zhang XF, Xiao XH
HPB (Oxford) 2015 Mar;17(3):278-83. Epub 2014 Oct 19 doi: 10.1111/hpb.12330. PMID: 25327281Free PMC Article
Bakoyiannis A, Delis S, Triantopoulou C, Dervenis C
World J Gastroenterol 2013 Nov 21;19(43):7603-19. doi: 10.3748/wjg.v19.i43.7603. PMID: 24282350Free PMC Article

Recent systematic reviews

Fahrner R, Dennler SG, Inderbitzin D
World J Gastroenterol 2020 Aug 21;26(31):4718-4728. doi: 10.3748/wjg.v26.i31.4718. PMID: 32884228Free PMC Article
Ercolani G, Grazi GL, Pinna AD
Dig Surg 2010;27(1):68-75. Epub 2010 Apr 1 doi: 10.1159/000268628. PMID: 20357454

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