Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Hepatol. Jan 27, 2019; 11(1): 127-132
Published online Jan 27, 2019. doi: 10.4254/wjh.v11.i1.127
Non-uremic calciphylaxis associated with alcoholic hepatitis: A case report
Yasser M Sammour, Haitham M Saleh, Mohamed M Gad, Brayden Healey, Melissa Piliang
Yasser M Sammour, Mohamed M Gad, Heart and Vascular Institute, Cleveland Clinic, Cleveland, OH 44195, United States
Haitham M Saleh, Department of Dermatology, Ain Shams University, Cairo 11566, Egypt
Brayden Healey, College of Osteopathic Medicine of the Pacific-Northwest, Western University of Health Sciences, Lebanon, OR 97355, United States
Melissa Piliang, Department of Dermatology, Cleveland Clinic, Cleveland 44195, OH, United States
Author contributions: Sammour YM designed the research and collected the patient’s clinical data; Piliang M provided the patient’s histopathological information; Sammour YM, Saleh HM, Gad MM, Healey B, Piliang M analyzed the data and wrote the paper.
Informed consent statement: A phone consent was obtained from the patient’s husband after the patient passed away. The phone conversation was documented in the patient’s electronic medical record.
Conflict-of-interest statement: No conflict of interest to disclose.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Yasser M Sammour, MD, Research Fellow, Heart and Vascular Institute, Cleveland Clinic, 9500 Euclid Ave, J2-606, Cleveland, OH 44195, United States. sammouy@ccf.org
Telephone: +1-216-3346144
Received: September 4, 2018
Peer-review started: September 4, 2018
First decision: October 15, 2018
Revised: December 2, 2018
Accepted: January 3, 2019
Article in press: January 4, 2019
Published online: January 27, 2019
Abstract
BACKGROUND

Calciphylaxis is a form of vascular calcification more commonly associated with renal disease. While the exact mechanism of calciphylaxis is poorly understood, most cases are due to end stage kidney disease. However, it can also be found in patients without kidney disease and in such cases is termed non-uremic calciphylaxis for which have multiple proposed etiologies.

CASE SUMMARY

We describe a case of a thirty-year-old morbidly obese Caucasian female who had a positive history of alcoholic hepatitis and presented with painful calciphylaxis wounds of the abdomen, hips, and thighs. The hypercoagulability panel showed low levels of Protein C and normal Protein S, low Antithrombin III and positive lupus anticoagulant and negative anticardiolipin. Wound biopsy confirmed the diagnosis of non-uremic calciphylaxis in the setting of alcoholic liver disease. The calciphylaxis wounds did not improve when Sodium Thiosulfate was used alone. The patient underwent a series of bedside and surgical debridement. Broad spectrum antibiotics were also used for secondary wound bacterial infections. The patient passed away shortly after due to sepsis and multiorgan failure.

CONCLUSION

Non-uremic Calciphylaxis can occur in the setting of alcoholic liver disease. The treatment of choice is still unknown.

Keywords: Calciphylaxis, Alcoholic hepatitis, Vascular calcification, Sodium thiosulfate, Debridement, Case report

Core tip: In this case report, we present a patient with alcoholic liver disease and low levels of Protein C who developed calciphylaxis and died shortly after due to complications. The pathogenesis is not completely understood but the disruption of calcium-phosphate-byproduct has been implicated to play a role in the disease process. Liver dysfunction can lead to low levels of coagulation inhibitors specifically Protein C and Protein S. The aim of the medical treatment is to lower the calcium-phosphate-byproduct and decrease the vascular calcification. The use of surgical wound debridement is less established.