Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Dec 6, 2022; 10(34): 12637-12647
Published online Dec 6, 2022. doi: 10.12998/wjcc.v10.i34.12637
Inflammatory myofibroblastic tumor of the central nervous system: A case report
Zhen-Jin Su, Ze-Shang Guo, Heng-Tong Wan, Xin-Yu Hong
Zhen-Jin Su, Ze-Shang Guo, Heng-Tong Wan, Xin-Yu Hong, Department of Neurosurgical Oncology, The First Hospital of Jilin University, Changchun 130000, Jilin Province, China
Author contributions: Su ZJ, Guo ZS, and Wan HT designed the study, collected and analyzed the data, explained the data, drafted the manuscript, and performed the literature review; Hong XY performed the surgery and contributed to preoperative and postoperative imaging and analysis and interpretation of pathological and immunohistochemical results; Su ZJ revised the language of the manuscript and made critical revisions to finalize the version submitted; Hong XY provided critical expertise, approved the submitted article, and agreed to be responsible for all aspects of the work; All authors made read and approved the submitted version.
Informed consent statement: Informed written consent was obtained from the patient and her family for publication of this report and any accompanying images.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
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: Xin-Yu Hong, PhD, Neurosurgeon, Professor, Department of Neurosurgical Oncology, The First Hospital of Jilin University, No. 71, Xinmin Street, Changchun City, Changchun 130000, Jilin Province, China. hongxy@jlu.edu.cn
Received: June 30, 2022
Peer-review started: June 30, 2022
First decision: August 4, 2022
Revised: September 16, 2022
Accepted: November 7, 2022
Article in press: November 7, 2022
Published online: December 6, 2022
Processing time: 154 Days and 22 Hours
Abstract
BACKGROUND

An inflammatory myofibroblastic tumor (IMT) occurring in the central nervous system is very rare, and thus its pathogenesis is unknown. This case report and literature review aimed to explore the pathogenesis, clinical features, imaging findings, pathological characteristics, immunohistochemical characteristics, diagnoses, treatments, and risks of postoperative recurrence of IMT in the central nervous system.

CASE SUMMARY

A 67-year-old woman was admitted to the hospital with an exophthalmic protrusion and double vision in the left eye that had persisted for 3 mo. Magnetic resonance imaging (MRI) showed a 2.4 cm × 1.3 cm heterogeneous large mass in the bottom of the left anterior cranial fossa, which was closely related to the dura mater. Before surgery, we suspected the mass to be meningioma. The entire mass was successfully removed under neuronavigation and electrophysiological monitoring, and postoperative pathology indicated an IMT with extensive infiltration of chronic inflammatory cells and scattered multinucleated giant cells. Head MRI at the 3-mo follow-up showed that the tumor at the bottom of left anterior cranial fossa had been completely resected without recurrence.

CONCLUSION

From the histological, immunohistochemical, and genetic analyses, the present case suggests that the pathogenesis of IMT-CNS is related to autoimmunity.

Keywords: Inflammatory myofibroblastic tumor; Central nervous system; Pathogeny; Diagnosis; Treatment; Risk of postoperative recurrence; Case report

Core Tip: Inflammatory myofibroblastic tumors (IMTs) rarely occur in the central nervous system (CNS), and the pathogenesis of IMT-CNS is unknown. We present a 67-year-old woman with IMT-CNS who presented with an exophthalmic protrusion and double vision in the left eye lasting for 3 mo. A 2.4 cm × 1.3 cm mass was found by magnetic resonance imaging at the left anterior cranial fossa base, and postoperative pathology indicated an IMT. Histological, immunohistochemical, and genetic analyses indicated that the IMT-CNS pathogenesis may be autoimmunity related. The main treatments for INT-CNS are gross tumor resection or rituximab infusion combined with high-dose prednisone.