Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Dec 26, 2021; 9(36): 11457-11466
Published online Dec 26, 2021. doi: 10.12998/wjcc.v9.i36.11457
Severe digital ischemia coexists with thrombocytopenia in malignancy-associated antiphospholipid syndrome: A case report and review of literature
Jia-Lei Chen, Xi Yu, Rong Luo, Ming Liu
Jia-Lei Chen, Rong Luo, Ming Liu, Department of Orthopedics, West China Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
Xi Yu, Department of Rehabilitation, West China Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
Author contributions: Chen JL was the patient’s physicians and contributed to manuscript drafting; Yu X was the patient’s physical therapist and helped with acquisition of the data. Chen JL and Luo R reviewed the literature and interpreted the imaging findings; Liu M and Chen JL were responsible for the revision of the manuscript for important intellectual content; all authors have read and approved the final manuscript.
Informed consent statement: Informed written consent was obtained from the patient and the patient's daughter as a legally authorized representative for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
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 that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (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: Jia-Lei Chen, MD, PhD, Attending Doctor, Department of Orthopedics, West China Hospital, Sichuan University, No. 37 Guoxue Alley, Wuhou District, Chengdu 610041, Sichuan Province, China. chenjialei2016@wchscu.cn
Received: July 22, 2021
Peer-review started: July 22, 2021
First decision: September 2, 2021
Revised: September 8, 2021
Accepted: November 18, 2021
Article in press: November 18, 2021
Published online: December 26, 2021
Processing time: 154 Days and 8.3 Hours
Abstract
BACKGROUND

Paraneoplastic syndromes are characterized by atypical clinical manifestations. Several reports of hepatocellular carcinoma (HCC) paraneoplastic phenomena have been reported. They usually manifest as one type in an individual, but it is not common for the two clinical manifestations to occur simultaneously.

CASE SUMMARY

A 52-year-old female patient was admitted to hospital with pale skin and numbness of the second to fifth fingers in the left hand, which rapidly developed into severe digital ischemia. Computed tomography angiography revealed uneven thickness of the left ulnar artery with severe local luminal stenosis. Blood analysis during hospitalization showed persistent mild to medium thrombocytopenia and insensitive to hormonal therapy. Antiphospholipid antibody testing showed high titer of IgG anticardiolipin antibodies (aCLs), IgA aCLs, IgG anti-β2-glycoprotein-I (anti-β2 GPI), and IgA anti-β2 GPI. The exact diagnosis was HCC when the high a-fetoprotein levels, computed tomography findings, and the history of chronic hepatitis B came together. This was a rare case of coexisting manifestations as presenting symptoms of malignancy-associated antiphospholipid syndrome. The patient underwent several operations, antithrombotic treatments and hormonal therapy. However, the patient refused chemotherapy and died 8 wk after diagnosis.

CONCLUSION

This report highlights the importance of atypical clinical changes that could alert the physicians to vigilance for a concomitant underlying malignancy.

Keywords: Antiphospholipid syndrome; Digital ischemia; Thrombocytopenia; Malignancy; Case report

Core Tip: Paraneoplastic syndromes, compared with cancer, are characterized by atypical clinical manifestations. This report described a case of hepatocellular carcinoma in which the presenting symptom was severe digital ischemia, mimicking Raynaud’s phenomenon, accompanied with thrombocytopenia, associated with antiphospholipid antibodies. This report highlights the importance of atypical clinical changes that could alert the physicians to be vigilant for concomitant underlying malignancy.