Database : MEDLINE
Search on : Caroli and Disease [Words]
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[PMID]: 25013645
[Au] Autor:Zahmatkeshan M; Bahador A; Geramizade B; Emadmarvasti V; Malekhosseini SA
[Ad] Address:Department of pediatrics, school of medicine, Shiraz University of Medical Sciences, Shiraz, Iran....
[Ti] Title:Liver transplantation for caroli disease.
[So] Source:Int J Organ Transplant Med;3(4):189-91, 2012.
[Is] ISSN:2008-6482
[Cp] Country of publication:Iran
[La] Language:eng
[Ab] Abstract:Caroli disease is a rare congenital disorder characterized by multifocal, segmental dilatation of intrahepatic bile ducts. Patients with Caroli disease who have recurrent bouts of biliary infection, particularly those who also have complications related to portal hypertension may require liver transplantation. In liver transplant ward of Shiraz University of Medical Science we had 4 patients with Caroli disease who were transplanted. Herein, we describe the demographic characteristics and post-transplant course of the patients. These patients presented with liver failure, recurrent cholangitis and portal hypertension sequelae unresponsive to medical treatment. The mean age of patients was 24.5 (range: 18-36) years, the mean MELD score was 17.5 (range: 11-23), three patients were female; one was male. All of the patients had good post-transplantation course except for one patient who developed post-operative biliary stricture for whom biliary reconstruction was done.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1407
[Da] Date of entry for processing:140711
[St] Status:PubMed-not-MEDLINE

  2 / 1193 MEDLINE  
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[PMID]: 24958584
[Au] Autor:Grieb D; Feldkamp A; Lang T; Melter M; Stroszczynski C; Brassel F; Meila D
[Ad] Address:Departments of Radiology, and Neuroradiology and....
[Ti] Title:Caroli disease associated with vein of galen malformation in a male child.
[So] Source:Pediatrics;134(1):e284-8, 2014 Jul.
[Is] ISSN:1098-4275
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:We report the first case of a male child with both Caroli disease and vein of Galen malformation. The neonate presented to our department with congestive heart failure as a result of the intracranial arteriovenous high-flow shunt. Over time, several endovascular embolizations led to a complete angiographic occlusion of the shunt. Additionally, the diagnosis of Caroli disease was made at the age of 2 months. He developed choledocholithiasis necessitating endoscopic sphincterotomy and stone extraction. As a prolonged medical treatment he received ursodeoxycholic acid and antibiotics. A coincidence of Caroli disease and vein of Galen malformation has not yet been described. Both diseases are very rare, leading to the question of whether there is a link in the pathogenesis. Based on the few previously described underlying mechanisms, we develop hypotheses about the relationship between both rare diseases. We consider overexpression of vascular endothelial growth factor and its receptors as a possible common molecular mechanism in their pathogenesis. We also highlight the critical role of increased expression of the Notch ligand Jagged 1 both in the development of cerebral arteriovenous malformations in general and in the formation of dilated intrahepatic bile ducts (eg, in Caroli disease).
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1407
[Js] Journal subset:AIM; IM
[St] Status:In-Data-Review
[do] DOI:10.1542/peds.2013-0747

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[PMID]: 24969198
[Au] Autor:Shenoy P; Zaki SA; Shanbag P; Bhongade S
[Ad] Address:Division of Pediatric Nephrology, Department of Pediatrics, Lokmanya Tilak Municipal General Hospital and Medical College, Sion, Mumbai, India.
[Ti] Title:Caroli's syndrome with autosomal recessive polycystic kidney disease.
[So] Source:Saudi J Kidney Dis Transpl;25(4):840-3, 2014 Jul-Aug.
[Is] ISSN:1319-2442
[Cp] Country of publication:Saudi Arabia
[La] Language:eng
[Ab] Abstract:Caroli's syndrome (CS) is a rare congenital disorder characterized by multiple segmental cystic or saccular dilatations of the intrahepatic bile ducts and congenital hepatic fibrosis. We report a 9-year-old boy who was diagnosed with CS and autosomal recessive poly-cystic kidney disease. On screening, his 5-month-old asymptomatic sister had multiple dilated biliary radicals with multiple bilateral renal cystic lesions. Both the patient and the affected sibling have been advised regular follow-up for monitoring the progression of the disease. In conclusion, patients with CS should be screened for renal cystic lesions and vice versa even if they are asymptomatic. Also, as the disease is inherited in an autosomal recessive manner, it is important to screen family members for early diagnosis and management.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1406
[Js] Journal subset:IM
[St] Status:In-Data-Review

  4 / 1193 MEDLINE  
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[PMID]: 23375562
[Au] Autor:Prestia A; Caroli A; Herholz K; Reiman E; Chen K; Jagust WJ; Frisoni GB; Translational Outpatient Memory Clinic Working Group; Alzheimer's Disease Neuroimaging Initiative
[Ad] Address:Laboratory of Epidemiology Neuroimaging and Telemedicine, and Unit for the Clinical Translation of Research, IRCCS Centro San Giovanni di Dio FBF, Brescia, Italy.
[Ti] Title:Diagnostic accuracy of markers for prodromal Alzheimer's disease in independent clinical series.
[So] Source:Alzheimers Dement;9(6):677-86, 2013 Nov.
[Is] ISSN:1552-5279
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:OBJECTIVE: To capitalize on data from different clinical series to compare sensitivity and specificity of individual biomarkers for predicting mild cognitive impairment (MCI) progression to Alzheimer's disease (AD). METHODS: Medial temporal atrophy, cortical hypometabolism, and cerebrospinal fluid biomarkers were assessed in 18 patients with mild cognitive impairment (MCI) with prodromal AD (pAD; conversion time, 26 ± 12 months) and 18 stable MCI (sMCI) patients from the Translational Outpatient Memory Clinic cohort, as well as in 24 pAD patients (conversion time, 36 ± 12 months) and 33 sMCI patients from the Alzheimer's Disease Neuroimaging Initiative cohort. Medial temporal atrophy was measured by manual, semi-automated, and automated hippocampal volumetry; cortical hypometabolism was measured using several indices of AD-related hypometabolism pattern; and cerebrospinal fluid markers were amyloid ß (Aß)42 and total tau protein concentrations. For each biomarker, sensitivity for pAD, specificity for sMCI, and diagnostic accuracy were computed. RESULTS: Sensitivity to predict MCI conversion to AD in the Alzheimer's Disease Neuroimaging Initiative and Translational Outpatient Memory Clinic cohorts was 79% and 94% based on Aß42, 46% and 28% based on hippocampal volumes, 33% to 66% and 56% to 78% based on different hypometabolism indices, and 46% and 61% based on total tau levels, respectively. Specificity to exclude sMCI was 27% and 50% based on Aß42, 76% and 94% based on hippocampal volumes, 58% to 67% and 55% to 83% based on different hypometabolism indices, and 61% and 83% based on total tau levels, respectively. CONCLUSIONS: Current findings suggest that Aß42 concentrations and hippocampal volumes may be used in combination to best identify pAD.
[Mh] MeSH terms primary: Alzheimer Disease/diagnosis
Amyloid beta-Peptides/cerebrospinal fluid
Biological Markers/analysis
Cognition Disorders/cerebrospinal fluid
Cognition Disorders/diagnosis
Hippocampus/pathology
Peptide Fragments/cerebrospinal fluid
[Mh] MeSH terms secundary: Aged
Aged, 80 and over
Cognition Disorders/complications
Databases, Factual/statistics & numerical data
Disease Progression
Female
Fluorodeoxyglucose F18/diagnostic use
Hippocampus/radionuclide imaging
Humans
Magnetic Resonance Imaging
Male
Middle Aged
Positron-Emission Tomography
Sensitivity and Specificity
Statistics, Nonparametric
Time Factors
[Pt] Publication type:JOURNAL ARTICLE; RESEARCH SUPPORT, N.I.H., EXTRAMURAL; RESEARCH SUPPORT, NON-U.S. GOV'T
[Nm] Name of substance:0 (Amyloid beta-Peptides); 0 (Biological Markers); 0 (Peptide Fragments); 0 (amyloid beta-protein (1-42)); 0Z5B2CJX4D (Fluorodeoxyglucose F18)
[Em] Entry month:1406
[Js] Journal subset:IM
[Da] Date of entry for processing:131108
[St] Status:MEDLINE

  5 / 1193 MEDLINE  
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[PMID]: 24710345
[Au] Autor:Hao X; Liu S; Dong Q; Zhang H; Zhao J; Su L
[Ad] Address:Pediatric Surgery, The Affiliated Hospital of Medical College, Qingdao University, Qingdao, China....
[Ti] Title:Whole exome sequencing identifies recessive PKHD1 mutations in a Chinese twin family with Caroli disease.
[So] Source:PLoS One;9(4):e92661, 2014.
[Is] ISSN:1932-6203
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:BACKGROUND: Mutations in PKHD1 cause autosomal recessive Caroli disease, which is a rare congenital disorder involving cystic dilatation of the intrahepatic bile ducts. However, the mutational spectrum of PKHD1 and the phenotype-genotype correlations have not yet been fully established. METHODS: Whole exome sequencing (WES) was performed on one twin sample with Caroli disease from a Chinese family from Shandong province. Routine Sanger sequencing was used to validate the WES and to carry out segregation studies. We also described the PKHD1 mutation associated with the genotype-phenotype of this twin. RESULTS: A combination of WES and Sanger sequencing revealed the genetic defect to be a novel compound heterozygous genotype in PKHD1, including the missense mutation c.2507 T>C, predicted to cause a valine to alanine substitution at codon 836 (c.2507T>C, p.Val836Ala), and the nonsense mutation c.2341C>T, which is predicted to result in an arginine to stop codon at codon 781 (c.2341C>T, p.Arg781*). This compound heterozygous genotype co-segregates with the Caroli disease-affected pedigree members, but is absent in 200 normal chromosomes. CONCLUSIONS: Our findings indicate exome sequencing can be useful in the diagnosis of Caroli disease patients and associate a compound heterozygous genotype in PKHD1 with Caroli disease, which further increases our understanding of the mutation spectrum of PKHD1 in association with Caroli disease.
[Pt] Publication type:JOURNAL ARTICLE; RESEARCH SUPPORT, NON-U.S. GOV'T
[Em] Entry month:1404
[Js] Journal subset:IM
[St] Status:In-Process
[do] DOI:10.1371/journal.pone.0092661

  6 / 1193 MEDLINE  
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[PMID]: 24899970
[Au] Autor:Shinto AS; Selvakumar J
[Ad] Address:Narayana Hrudyalaya, 258 A Bommasandra Industrial Area, Anekal Taluk, Bangalore, 99 India ; Nuclear Medicine Department, Amala Institute of Medical Sciences, Amalanagar, Thrissur, 555 Kerala India.
[Ti] Title:Unexpectedly Diagnosed Caroli's Disease on HIDA Scintigraphy in a Patient with Calculous Cholecystitis.
[So] Source:Nucl Med Mol Imaging;44(4):304-6, 2010 Dec.
[Is] ISSN:1869-3474
[Cp] Country of publication:Germany
[La] Language:eng
[Ab] Abstract:Caroli's disease, which is a rare condition with congenital dilatation of the intrahepatic bile ducts, is usually diagnosed postoperatively. The clinical suspicion in a patient with gallstones and choledocholithiasis presenting with dilated intrahepatic biliary radicles and jaundice is usually an obstructive etiology. However, scintigraphic evaluation of this entity, as in this case, gives additional information on liver function, biliary drainage and predisposing conditions like Caroli's disease, which could be missed otherwise.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1406
[Cu] Class update date: 140611
[Lr] Last revision date:140611
[Da] Date of entry for processing:140606
[St] Status:PubMed-not-MEDLINE
[do] DOI:10.1007/s13139-010-0054-8

  7 / 1193 MEDLINE  
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[PMID]: 24413206
[Au] Autor:Badura J; Król R; Kurek A; Hartleb M; Cierpka L
[Ti] Title:Left-hemihepatectomy as a method of treatment of locally limited Caroli disease.
[So] Source:Pol Przegl Chir;85(11):663-5, 2013 Nov.
[Is] ISSN:0032-373X
[Cp] Country of publication:Poland
[La] Language:eng
[Ab] Abstract:The study introduces a case of a 51-year old patient with Caroli's disease of left liver lobe. In 2011 the patient was admitted to Clinic of General, Vascular and Transplantation Surgery. She was after first in her life incident of an acute pancreatitis and subsequent ERCP procedure with left hepatic biliary tract drainage. The lady was qualified to left-hemihepatoctomy, which was successfully conducted in our clinic. The only complication of the procedure was surgical site infection which was properly treated with typical antibiotics. One-year observation occurred no other complications and liver function was fine.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1401
[Js] Journal subset:IM
[St] Status:In-Process

  8 / 1193 MEDLINE  
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[PMID]: 23593929
[Au] Autor:Telega G; Cronin D; Avner ED
[Ad] Address:Department of Pediatrics, Children's Hospital Health System of Wisconsin and Medical College of Wisconsin, Milwaukee, WI 53226, USA. gtelega@mcw.edu
[Ti] Title:New approaches to the autosomal recessive polycystic kidney disease patient with dual kidney-liver complications.
[So] Source:Pediatr Transplant;17(4):328-35, 2013 Jun.
[Is] ISSN:1399-3046
[Cp] Country of publication:Denmark
[La] Language:eng
[Ab] Abstract:Improved neonatal medical care and renal replacement technology have improved the long-term survival of patients with ARPKD. Ten-yr survival of those surviving the first year of life is reported to be 82% and is continuing to improve further. However, despite increases in overall survival and improved treatment of systemic hypertension and other complications of their renal disease, nearly 50% of survivors will develop ESRD within the first decade of life. In addition to renal pathology, patients with ARPKD develop ductal plate malformations with cystic dilation of intra- and extrahepatic bile ducts resulting in CHF and Caroli syndrome. Many patients with CHF will develop portal hypertension with resulting esophageal varices, splenomegaly, hypersplenism, protein losing enteropathy, and gastrointestinal bleeding. Management of portal hypertension may require EBL of esophageal varices or porto-systemic shunting. Complications of hepatic involvement can include ascending cholangitis, cholestasis with malabsorption of fat-soluble vitamins, and rarely benign or malignant liver tumors. Patients with ARPKD who eventually reach ESRD, and ultimately require kidney transplantation, present a unique set of complications related to their underlying hepato-biliary disease. In this review, we focus on new approaches to these challenging patients, including the indications for liver transplantation in ARPKD patients with severe chronic kidney disease awaiting kidney transplant. While survival in patients with ARPKD and isolated kidney transplant is comparable to that of age-matched pediatric patients who have received kidney transplants due to other primary renal diseases, 64-80% of the mortality occurring in ARPKD kidney transplant patients is attributed to cholangitis/sepsis, which is related to their hepato-biliary disease. Recent data demonstrate that surgical mortality among pediatric liver transplant recipients is decreased to <10% at one yr. The immunosuppressive regimen used for kidney transplant recipients is adequate for most liver transplant recipients. We therefore suggest that in a select group of ARPKD patients with recurrent cholangitis or complications of portal hypertension, combined liver-kidney transplant is a viable option. Although further study is necessary to confirm our approach, we believe that combined liver-kidney transplantation can potentially decrease overall mortality and morbidity in carefully selected ARPKD patients with ESRD and clinically significant CHF.
[Mh] MeSH terms primary: Kidney Transplantation/methods
Liver Transplantation/methods
Polycystic Kidney, Autosomal Recessive/therapy
[Mh] MeSH terms secundary: Caroli Disease/complications
Child
Cholangitis/complications
Cholestasis/complications
Humans
Hypertension, Portal/complications
Immunosuppressive Agents/therapeutic use
Kidney Failure, Chronic/complications
Kidney Failure, Chronic/therapy
Liver Diseases/complications
Liver Failure/complications
Liver Failure/therapy
Liver Neoplasms/complications
Patient Selection
Polycystic Kidney, Autosomal Recessive/complications
Risk
[Pt] Publication type:JOURNAL ARTICLE; REVIEW
[Nm] Name of substance:0 (Immunosuppressive Agents)
[Em] Entry month:1401
[Cu] Class update date: 140603
[Lr] Last revision date:140603
[Js] Journal subset:IM
[Da] Date of entry for processing:130522
[St] Status:MEDLINE
[do] DOI:10.1111/petr.12076

  9 / 1193 MEDLINE  
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[PMID]: 24834275
[Au] Autor:Baghbanian M; Salmanroghani H; Baghbanian A
[Ad] Address:Department of Gastroenterology, Shaheed Sadoughi University of Medical Sciences, Yazd, Iran.
[Ti] Title:Cholangiocarcinoma or Caroli disease: a case presentation.
[So] Source:Gastroenterol Hepatol Bed Bench;6(4):214-6, 2013.
[Is] ISSN:2008-2258
[Cp] Country of publication:Iran
[La] Language:eng
[Ab] Abstract:Early diagnosis and appropriate treatment of cholangiocarcinoma is problematic. Cross sectional imaging and tumor marker CA 19-9 are not absolutely reliable and tissue sampling is difficult. We present a patient with cholangitis and cystic dilation of intra-hepatic bile ducts that primarily diagnosed as Caroli's disease in imaging and needle biopsy but laparotomy and surgical biopsy revealed cholangiocarcinoma.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1405
[Cu] Class update date: 140519
[Lr] Last revision date:140519
[Da] Date of entry for processing:140516
[St] Status:PubMed-not-MEDLINE

  10 / 1193 MEDLINE  
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[PMID]: 24730630
[Au] Autor:Viswanathan S; Kumar D
[Ad] Address:Department of Pediatrics, Rainbow Babies and Children's Hospital, University Hospitals, Cleveland, Ohio, USA.
[Ti] Title:Diagnostic challenge of large congenital liver cyst in the newborn.
[So] Source:Pediatr Int;56(2):267-70, 2014 Apr.
[Is] ISSN:1442-200X
[Cp] Country of publication:Australia
[La] Language:eng
[Ab] Abstract:Liver cysts in the newborn often pose significant diagnostic challenges. Described herein is a case of large congenital liver cyst that was difficult to diagnose both antenatally and postnatally and which was later diagnosed as Caroli disease.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1404
[Js] Journal subset:IM
[St] Status:In-Process
[do] DOI:10.1111/ped.12260


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