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      ANCA-Associated Intrahepatic Duct Injury Associated with Levamisole-Adulterated Cocaine

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          Abstract

          Damage to the liver or kidney can occur through direct toxic effects; however, damage can also be drug-induced immune-mediated. Levamisole-adulterated cocaine (LAC) is known to cause antineutrophil cytoplasmic antibody- (ANCA-) associated vasculitis and glomerulonephritis leading to acute kidney injury and end-stage renal disease. It remains unclear whether LAC is associated with hepatic duct damage. Here, we report a case with biopsy-proven evidence of intrahepatic duct damage months after being diagnosed with ANCA-associated crescentic and sclerosing glomerulonephritis caused by LAC use. This case represents the first report of LAC-induced ANCA-associated hepatic duct cholestasis in the setting of previous LAC-induced ANCA-positive glomerulonephritis.

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          Primary sclerosing cholangitis – a comprehensive review

          Primary sclerosing cholangitis (PSC) is a rare disorder characterised by multi-focal bile duct strictures and progressive liver disease. Inflammatory bowel disease is usually present and there is a high risk of cholangiocarcinoma and colorectal cancer. Most patients ultimately require liver transplantation, after which disease recurrence may occur. With limited therapeutic options and a lack of proven surveillance strategies, patients currently have significant unmet needs. In the present seminar, we provide a comprehensive review of the status of the field. We emphasise developments related to patient stratification and disease behaviour, and provide an overview of management options from a practical, patient-centered perspective. We survey advances made in the understanding of PSC pathogenesis and summarise the ongoing efforts to develop an effective therapy based on these insights.
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            Characterization, outcome, and prognosis in 273 patients with primary sclerosing cholangitis: A single center study.

            Primary sclerosing cholangitis (PSC) is a chronic cholestatic liver disease with varying severity and progression. This study describes the natural history of PSC patients and evaluates the prognostic significance of clinical, biochemical, and cholangiographic findings constructing a novel prognostic model. A population of 273 German PSC patients was studied with a median follow-up time of 76 months (range 1-280 months). Survival curves were analyzed by the Kaplan-Meier method, and prognostic significance of clinical, biochemical, and cholangiographic features recorded at the time of diagnosis was evaluated by multivariate analysis using Cox proportional-hazards regression models. The estimated median survival from the time of diagnosis to death or time of liver transplantation was 9.6 yr. One hundred eight (39.6%) patients underwent liver transplantation. Hepatobiliary malignancies were found in 39 (14.3%) patients of the entire PSC population. Age, low albumin, persistent bilirubin elevation longer than 3 months, hepatomegaly, splenomegaly, dominant bile duct stenosis, and intra- and extrahepatic ductal changes at the time of diagnosis were found to be independent risk factors correlating with poor prognosis and were used to construct a new prognostic model. A persistent bilirubin elevation for longer than 3 months from the time of diagnosis could be identified as a novel marker correlating with a poor outcome. A new prognostic model was developed to predict progression of PSC, which may be useful in timing of liver transplantation.
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              Incidence of primary sclerosing cholangitis: a systematic review and meta-analysis.

              Incidence studies of primary sclerosing cholangitis (PSC) are important for describing the disease's burden and for shedding light on the disease's etiology. The purposes of this study were to conduct a systematic review of the incidence studies of PSC with a meta-analysis and to investigate possible geographic variations and temporal trends in the incidence of the disease. A systematic literature search of MEDLINE (1950-2010) and Embase (1980-2010) was conducted to identify studies investigating the incidence of PSC. The incidence of PSC was summarized with an incidence rate (IR) and 95% confidence intervals. The test of heterogeneity was performed with the Q statistic. Secondary variables extracted from the articles included the following: the method of case ascertainment, the country, the time period, the age, the male/female incidence rate ratio (IRR), and the incidence of PSC subtypes (small-duct or large-duct PSC and inflammatory bowel disease). Stratified and sensitivity analyses were performed to explore heterogeneity between studies and to assess effects of study quality. Time trends were used to explore differences in the incidence across time. The search retrieved 1669 potentially eligible citations; 8 studies met the inclusion criteria. According to a random-effects model, the pooled IR was 0.77 (0.45-1.09) per 100,000 person-years. However, significant heterogeneity was observed between studies (P < 0.001). Sensitivity analyses excluding non-population-based studies increased the overall IR to 1.00 (0.82-1.17) and eliminated the heterogeneity between studies (P = 0.08). The IRR for males versus females was 1.70 (1.34-2.07), and the median age was 41 years (35-47 years). All studies investigating time trends reported an overall increase in the incidence of PSC.
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                Author and article information

                Contributors
                Journal
                Case Reports Hepatol
                Case Reports Hepatol
                CRIHEP
                Case Reports in Hepatology
                Hindawi
                2090-6587
                2090-6595
                2020
                22 December 2020
                : 2020
                : 8867183
                Affiliations
                1University of Central Florida, School of Medicine, 6850 Lake Nona Blvd, Orlando, FL 32827, USA
                2North Florida Regional Medical Center, 6500 W Newberry Rd, Gainesville, FL 32605, USA
                Author notes

                Academic Editor: Ned Snyder

                Author information
                https://orcid.org/0000-0002-6366-0116
                Article
                10.1155/2020/8867183
                7773451
                33425405
                d6abad88-6f23-4742-b1c7-e640072e25b7
                Copyright © 2020 Joshua K. Salabei et al.

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 1 June 2020
                : 8 December 2020
                : 10 December 2020
                Categories
                Case Report

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