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      Bile cast nephropathy in a patient with cholangiocarcinoma – a case report

      case-report

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          Key Clinical Message

          Bile cast nephropathy is characterized by the presence of bile casts associated with renal failure and/or proximal tubulopathy in cases of severe hyperbilirubinemia. The clinician should carefully examine the urine samples for characteristic bile‐stained granular casts in suspected case.

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          Most cited references26

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          Bile cast nephropathy is a common pathologic finding for kidney injury associated with severe liver dysfunction.

          Cholemic nephrosis represents a spectrum of renal injury from proximal tubulopathy to intrarenal bile cast formation found in patients with severe liver dysfunction. However, the contribution of this diagnosis has been largely forgotten in the modern literature. To more precisely define this, we conducted a clinicopathologic study of 44 subjects (41 autopsies and 3 renal biopsies) from jaundiced patients at the University of Chicago. Of these, 24 patients had bile casts with involvement of distal nephron segments in 18 mild cases and extension to proximal tubules for 6 severe cases. Eleven of 13 patients with hepatorenal syndrome and all 10 with cirrhosis (due to alcoholism) had tubular bile casts. These casts significantly correlated with higher serum total and direct bilirubin levels, and a trend toward higher serum creatinine, AST, and ALT levels. Bile casts may contribute to the kidney injury of severely jaundiced patients by direct bile and bilirubin toxicity, and tubular obstruction. Both mechanisms are analogous to the injury by myeloma or myoglobin casts. Accounting for the presence of renal bile casts provides a more complete representation of the renal injury that can occur in this unique clinical setting. Thus, bile cast nephropathy is an appropriate term for the severe form of injury observed in the spectrum of cholemic nephrosis. Additional studies are needed to establish the significance of this parameter for patient management in different clinical settings.
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            Acute kidney injury: a guide to diagnosis and management.

            Acute kidney injury is characterized by abrupt deterioration in kidney function, manifested by an increase in serum creatinine level with or without reduced urine output. The spectrum of injury ranges from mild to advanced, sometimes requiring renal replacement therapy. The diagnostic evaluation can be used to classify acute kidney injury as prerenal, intrinsic renal, or postrenal. The initial workup includes a patient history to identify the use of nephrotoxic medications or systemic illnesses that might cause poor renal perfusion or directly impair renal function. Physical examination should assess intravascular volume status and identify skin rashes indicative of systemic illness. The initial laboratory evaluation should include measurement of serum creatinine level, complete blood count, urinalysis, and fractional excretion of sodium. Ultrasonography of the kidneys should be performed in most patients, particularly in older men, to rule out obstruction. Management of acute kidney injury involves fluid resuscitation, avoidance of nephrotoxic medications and contrast media exposure, and correction of electrolyte imbalances. Renal replacement therapy (dialysis) is indicated for refractory hyperkalemia; volume overload; intractable acidosis; uremic encephalopathy, pericarditis, or pleuritis; and removal of certain toxins. Recognition of risk factors (e.g., older age, sepsis, hypovolemia/shock, cardiac surgery, infusion of contrast agents, diabetes mellitus, preexisting chronic kidney disease, cardiac failure, liver failure) is important. Team-based approaches for prevention, early diagnosis, and aggressive management are critical for improving outcomes.
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              Bile acids trigger cholemic nephropathy in common bile-duct-ligated mice.

              Tubular epithelial injury represents an underestimated but important cause of renal dysfunction in patients with cholestasis and advanced liver disease, but the underlying mechanisms are unclear. To address the hypothesis that accumulation and excessive alternative urinary elimination of potentially toxic bile acids (BAs) may contribute to kidney injury in cholestasis, we established a mouse model for detailed in vivo time course as well as treatment studies. Three-day common bile duct ligation (CBDL) induced renal tubular epithelial injury predominantly at the level of aquaporin 2-positive collecting ducts with tubular epithelial and basement membrane defects. This was followed by progressive interstitial nephritis and tubulointerstitial renal fibrosis in 3-, 6-, and 8-week CBDL mice. Farnesoid X receptor knockout mice (with a hydrophilic BA pool) were completely protected from CBDL-induced renal fibrosis. Prefeeding of hydrophilic norursodeoxycholic acid inhibited renal tubular epithelial injury in CBDL mice. In addition, we provide evidence for renal tubular injury in cholestatic patients with cholemic nephropathy.
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                Author and article information

                Contributors
                kittrawee@gmail.com
                Journal
                Clin Case Rep
                Clin Case Rep
                10.1002/(ISSN)2050-0904
                CCR3
                Clinical Case Reports
                John Wiley and Sons Inc. (Hoboken )
                2050-0904
                05 March 2018
                May 2018
                : 6
                : 5 ( doiID: 10.1111/ccr3.2018.6.issue-5 )
                : 779-783
                Affiliations
                [ 1 ] Division of Nephrology Faculty of Medicine Khon Kaen University Khon Kaen Thailand
                [ 2 ] Department of Medicine Faculty of Medicine Khon Kaen University Khon Kaen Thailand
                Author notes
                [*] [* ] Correspondence

                Kittrawee Kritmetapak, Division of Nephrology, Faculty of Medicine, Khon Kaen University, Khon Kaen 40002, Thailand. Tel: +6643‐363‐746; E‐mail: kittrawee@ 123456gmail.com

                Author information
                http://orcid.org/0000-0002-4506-5874
                Article
                CCR31465
                10.1002/ccr3.1465
                5930206
                9a1766cc-dc58-410f-80ea-22b8d91dad02
                © 2018 The Authors. Clinical Case Reports published by John Wiley & Sons Ltd.

                This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.

                History
                : 17 October 2017
                : 28 January 2018
                : 09 February 2018
                Page count
                Figures: 3, Tables: 0, Pages: 5, Words: 2908
                Categories
                Case Report
                Case Reports
                Custom metadata
                2.0
                ccr31465
                May 2018
                Converter:WILEY_ML3GV2_TO_NLMPMC version:version=5.3.7.2 mode:remove_FC converted:02.05.2018

                bile cast nephropathy,renal failure,proximal tubulopathy,hyperbilirubinemia

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