Case Report
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Feb 16, 2023; 11(5): 1188-1197
Published online Feb 16, 2023. doi: 10.12998/wjcc.v11.i5.1188
Early postsurgical lethal outcome due to splenic littoral cell angioma: A case report
Fan Jia, Han Lin, Yi-Long Li, Jin-Ling Zhang, Liang Tang, Peng-Tian Lu, Yu-Qing Wang, Yi-Feng Cui, Xiu-Hua Yang, Zhao-Yang Lu
Fan Jia, Xiu-Hua Yang, Department of Abdominal Ultrasound, First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
Han Lin, Yi-Feng Cui, Zhao-Yang Lu, Department of Hepatic Surgery, Key Laboratory of Hepatosplenic Surgery, Ministry of Education, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
Yi-Long Li, Department of Pancreatic and Biliary Surgery, Key Laboratory of Hepatosplenic Surgery, Ministry of Education, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
Jin-Ling Zhang, Liang Tang, Department of Radiology and Nuclear Medicine, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
Peng-Tian Lu, Department of Pathology, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
Yu-Qing Wang, Department of Hematopathology, First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
Author contributions: Jia F and Lin H two authors contributed equally to this work, contributed to data extraction, analysis and interpretation, reviewed literature, manuscript writing and editing; Li YL, Zhang JL, Tang L, Lu PT, Wang YQ, and Cui YF provided technical support and professional advice and contributed to manuscript writing and editing; Yang XH and Lu ZY supervised the project until its completion and contributed equally to this work; all authors contributed to the interpretation of the results; all authors have read and approved the final manuscript.
Supported by the National Natural Science Foundation of China, No. 81972230; Hubei Technological Innovation Special Fund (CN), No. CXPJJH11800004-004, No. CXPJJH122002-063, and No. CXPJJH122002-092; University Nursing Program for Young Scholar with Creative Talents in Heilongjiang Province, No. UNPYSCT-2017064; Postdoctoral Foundation of Hei Long Jiang Province, No. LBH-Z20196 and No. LBH-Z20178; China Postdoctoral Science Foundation, No. 2021MD703835; Wu Jie-Ping Medical Foundation, No. 320.6750.2021-23-22; Open Fund of Key Laboratory of Hepatosplenic Surgery, Ministery of Education, Harbin, China, No. GPKF202204; Excellent Youth Science Fund of the First Hopital of Harbin Medical University, No. 2021Y01; and Scientifc Foundation of the First Afliated Hospital of Harbin Medical University, No. 2021B03.
Informed consent statement: Informed written consent was obtained from the patient’s family member for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have 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 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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Xiu-Hua Yang, MD, PhD, Chief Physician, Professor, Department of Abdominal Ultrasound, First Affiliated Hospital of Harbin Medical University, No. 23 Youzheng Road, Nangang District, Harbin 150001, Heilongjiang Province, China. yangxiuhua@hrbmu.edu.cn
Received: November 14, 2022
Peer-review started: November 14, 2022
First decision: November 30, 2022
Revised: December 9, 2022
Accepted: January 16, 2023
Article in press: January 16, 2023
Published online: February 16, 2023
Abstract
BACKGROUND

Littoral cell angioma (LCA) is a rare benign vascular tumor of the spleen. Given its rarity, standard diagnostic and therapeutic recommendations have yet to be developed for reported cases. Splenectomy is the only method of obtaining a pathological diagnosis and providing treatment to obtain a favorable prognosis.

CASE SUMMARY

A 33-year-old female presented with abdominal pain for one month. Computed tomography and ultrasound revealed splenomegaly with multiple lesions and two accessory spleens. The patient underwent laparoscopic total splenectomy and accessory splenectomy, and splenic LCA was confirmed by pathology. Four months after surgery, the patient presented with acute liver failure, was readmitted, rapidly progressed to multiple organ dysfunction syndrome and died.

CONCLUSION

Preoperative diagnosis of LCA is challenging. We systematically reviewed online databases to identify the relevant literature and found a close relationship between malignancy and immunodysregulation. When a patient suffers from both splenic tumors and malignancy or immune-related disease, LCA is possible. Due to potential malignancy, total splenectomy (including accessory spleen) and regular follow-up after surgery are recommended. If LCA is diagnosed after surgery, a comprehensive postoperative examination is needed.

Keywords: Littoral cell angioma, Imaging features, Malignancy, Immunodysregulation, Treatment, Case report

Core Tip: Littoral cell angioma (LCA) is a rare benign vascular tumor of the spleen. No standard diagnostic and therapeutic recommendations are available. We report a patient with LCA and without comorbidities who died of multiple organ dysfunction syndrome 4 mo after surgery, which is extremely rare. Systematic analysis of relevant cases in the PubMed, Embase, Web of Science and the Cochrane Library databases revealed that LCA has a close relationship with malignancy and immunodysregulation. The possibility of LCA should not be overlooked when a patient presents with splenic tumors and malignancy or immune-related disease. Considering its potential malignant behavior, total splenectomy (including accessory spleen) and regular follow-up after surgery are recommended.