Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Feb 6, 2019; 7(3): 373-381
Published online Feb 6, 2019. doi: 10.12998/wjcc.v7.i3.373
Castleman disease presenting with jaundice: A case report and review of literature
Bo Zhai, Hai-Yang Ren, Wei-Dong Li, Shiva Reddy, Shu-Jun Zhang, Xue-Ying Sun
Bo Zhai, Hai-Yang Ren, Wei-Dong Li, Department of General Surgery, The Fourth Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
Bo Zhai, Wei-Dong Li, Xue-Ying Sun, Hepatosplenic Surgery Center, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
Shiva Reddy, Xue-Ying Sun, Department of Molecular Medicine and Pathology, Faculty of Medical and Health Sciences, The University of Auckland, Auckland 1142, New Zealand
Shu-Jun Zhang, Department of Pathology, The Fourth Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
Author contributions: Sun XY and Zhang SJ designed the report; Zhai B and Ren HY collected the patient’s clinical data; Li WD and Zhang SJ performed histological and immunohistochemical analyses; Reddy S participated in study design and revised the manuscript; Zhai B and Sun XY analyzed the data and wrote the paper; all the authors have finally approved the version of the article to be published.
Supported by the National Key Research and Development Program of China, No. 2017YFC1308602; and the National Natural Scientific Foundation of China, No. 81472321 and No. 81401975.
Informed consent statement: Consent was obtained from the patient for publication of this report and accompanying images.
Conflict-of-interest statement: The authors have no conflict of interests to declare.
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: Xue-Ying Sun, MD, PhD, Professor, The Hepatosplenic Surgery Center, The First Affiliated Hospital of Harbin Medical University, No. 23, Youzheng Street, Harbin 150001, Heilongjiang Province, China. sunxueying@hrbmu.edu.cn
Telephone: +86-451-53643628 Fax: +86-451-53643628
Received: October 30, 2018
Peer-review started: October 30, 2018
First decision: November 14, 2018
Revised: November 26, 2018
Accepted: December 12, 2018
Article in press: December 12, 2018
Published online: February 6, 2019
Abstract
BACKGROUND

Castleman disease (CD) is a rare lymphoproliferative disorder that presents with various symptoms. CD accompanied with jaundice is uncommon since there are only 11 cases reported in the literature.

CASE SUMMARY

Here we report a 62-year-old woman who was admitted to the hospital with signs and symptoms of intermittent jaundice. Biochemical tests showed higher serum levels of total and direct bilirubin, and normal serum levels of tumor markers and interleukin-6. Contrast-enhanced computed tomography detected a 6 cm × 4 cm × 2.5 cm mass between the hepatoduodenal ligament and the inferior vena cava. The mass was successfully excised and the patient had a complete resolution of symptoms. A diagnosis of idiopathic unicentric CD was made based upon histological examination, which demonstrated the pathological features of CD of mixed type, including hyperplasia of follicular lymphoids with abundant plasma cells, degenerative germinal centers, interfollicular vascularity and hyaline degeneration. The diagnosis was corroborated by immunohistochemical analysis which detected multiple biomarkers.

CONCLUSION

This is the first study that describes the clinicopathological features of CD presenting with jaundice, which may deepen and extend our understanding of this disease.

Keywords: Castleman disease, Jaundice, Case report, Clinicopathology, Immunohistochemistry

Core tip: Castleman disease (CD) is a rare lymphoproliferative disorder that presents with various symptoms. Here we report an uncommon case of CD accompanied with intermittent jaundice caused by extrinsic compression of a mass located between the hepatoduodenal ligament and the inferior vena cava. Histological examination demonstrated the pathological features of idiopathic unicentric CD of mixed type, supported by the immunohistochemical detection of multiple markers. The clinicopathological and immunohistochemical study on the case of CD provides new insights and diagnostic criteria of this orphan disease.