CC BY-NC-ND 4.0 · J Neurol Surg Rep 2024; 85(01): e25-e28
DOI: 10.1055/a-2277-4296
Case Report

Necrotizing Parasagittal Meningioma in Patient with Systemic Lupus Erythematosus after Treatments with Methotrexate and Hydroxychloroquine

Thitikan Wangapakul
1   Neurosurgical Unit, Yala Regional Hospital, Yala, Thailand
,
Ambar Elizabeth Riley Moguel
2   Neurosurgical Department, ISSSTE “1ro de Octubre,” Mexico City, Mexico
,
Abdel Raouf Kayssi
3   Arkansas Neuroscience Institute, Arkansas, United States
› Author Affiliations

Abstract

Objective Meningiomas are the most common extra-axial tumors of the central nervous system. Meningiomas are particularly problematic when they invade deep or vital structures, causing the tumors to be inoperable. Nonsurgical adjunctive or salvage treatments to shrink a meningioma with multiple recurrences, located in deep-seated area or surgically unfit area, remain underexplored. The authors report a rare case of a spontaneously necrotic meningioma (World Health Organization [WHO] grade I) in a patient with systemic lupus erythematosus on chronic methotrexate and hydroxychloroquine.

Case Study A 29-year-old female with systemic lupus erythematosus had been treated with methotrexate and hydroxychloroquine for 7 years. She presented with episodes of seizures and hemiparesis. Neuroimaging revealed a possible necrotic meningioma in the left parietal parasagittal area. Subsequent intraoperative findings showed lytic tissue of the tumor, and by histopathology results the tumor was classified as WHO grade I with massive necrosis. After craniotomy with tumor removal, the patient's motor function fully recovered without recurrent seizures.

Discussions Necrotizing of small and benign meningioma is rarely found but otherwise interesting. The cause of this phenomenon is not yet understood thoroughly. In this case, we suspected various possible causes such as vasculitis interrupting blood supply, use of immunosuppressive drugs such as hydroxychloroquine or methotrexate, or, less likely, latent infections in the immunocompromised patient. Despite the lack of more evidence supports, this finding encourages further study of nonsurgical or salvage treatment of inoperable meningioma, so sequalae after refractory recurrences of meningioma can be prevented, and patient treatment outcomes can be improved.



Publication History

Received: 21 November 2023

Accepted: 09 February 2024

Accepted Manuscript online:
27 February 2024

Article published online:
28 March 2024

© 2024. The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/)

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 
  • References

  • 1 Wiemels J, Wrensch M, Claus EB. Epidemiology and etiology of meningioma. J Neurooncol 2010; 99 (03) 307-314
  • 2 Goldbrunner R, Stavrinou P, Jenkinson MD. et al. EANO guideline on the diagnosis and management of meningiomas. Neuro-oncol 2021; 23 (11) 1821-1834
  • 3 Apra C, Peyre M, Kalamarides M. Current treatment options for meningioma. Expert Rev Neurother 2018; 18 (03) 241-249
  • 4 D'Ambrosio AL, Bruce JN. Treatment of meningioma: an update. Curr Neurol Neurosci Rep 2003; 3 (03) 206-214
  • 5 Chen WC, Perlow HK, Choudhury A. et al. Radiotherapy for meningiomas. J Neurooncol 2022; 160 (02) 505-515
  • 6 Mooney MA, Essayed W, Patel V, Devlin PM, Al-Mefty O. Brachytherapy as salvage treatment for meningioma with malignant progression after exhausting other treatment options: 2-dimensional operative video. Oper Neurosurg (Hagerstown) 2022; 22 (05) e215-e215
  • 7 Sherman WJ, Raizer JJ. Chemotherapy: what is its role in meningioma?. Expert Rev Neurother 2012; 12 (10) 1189-1195 , quiz 1196
  • 8 Chamberlain MC. The role of chemotherapy and targeted therapy in the treatment of intracranial meningioma. Curr Opin Oncol 2012; 24 (06) 666-671
  • 9 Kano T, Kobayashi M, Yoshida K, Kawase T. Central tumor necrosis of a large meningioma following acute anemia caused by hysterectomy. Neurol Med Chir (Tokyo) 2009; 49 (09) 424-426
  • 10 Matyja E, Grajkowska W, Nauman P, Bonicki W, Bojarski P, Marchel A. Necrotic rhabdoid meningiomas with aggressive clinical behavior. Clin Neuropathol 2010; 29 (05) 307-316
  • 11 Góes P, Santos BFO, Suzuki FS. et al. Necrosis is a consistent factor to recurrence of meningiomas: should it be a stand-alone grading criterion for grade II meningioma?. J Neurooncol 2018; 137 (02) 331-336
  • 12 Garcia-Segura ME, Erickson AW, Jairath R, Munoz DG, Das S. Necrosis and brain invasion predict radio-resistance and tumor recurrence in atypical meningioma: a retrospective cohort study. Neurosurgery 2020; 88 (01) E42-E48
  • 13 Bernstein M, Villamil A, Davidson G, Erlichman C. Necrosis in a meningioma following systemic chemotherapy. Case report. J Neurosurg 1994; 81 (02) 284-287
  • 14 Ackland SP, Schilsky RL. High-dose methotrexate: a critical reappraisal. J Clin Oncol 1987; 5 (12) 2017-2031
  • 15 Onorati AV, Dyczynski M, Ojha R, Amaravadi RK. Targeting autophagy in cancer. Cancer 2018; 124 (16) 3307-3318
  • 16 Golden EB, Cho HY, Hofman FM, Louie SG, Schönthal AH, Chen TC. Quinoline-based antimalarial drugs: a novel class of autophagy inhibitors. Neurosurg Focus 2015; 38 (03) E12
  • 17 Compter I, Eekers DBP, Hoeben A. et al. Chloroquine combined with concurrent radiotherapy and temozolomide for newly diagnosed glioblastoma: a phase IB trial. Autophagy 2021; 17 (09) 2604-2612
  • 18 Wear D, Bhagirath E, Balachandar A, Vegh C, Pandey S. Autophagy inhibition via hydroxychloroquine or 3-methyladenine enhances chemotherapy-induced apoptosis in neuro-blastoma and glioblastoma. Int J Mol Sci 2023; 24 (15) 12052
  • 19 Zhang LN, Shi TY, Yang YJ, Zhang FC. An SLE patient with prolactinoma and recurrent granulomatous mastitis successfully treated with hydroxychloroquine and bromocriptine. Lupus 2014; 23 (04) 417-420
  • 20 Canavero S, Pagni CA. Meningioma and Takayasu disease: Case report. Ital J Neuro Scim 1990; 11 (04) 391-394
  • 21 Solyman OM, Vizcaino MA, Fu R, Henderson AD. Neurosarcoidosis Masquerading as Giant Cell Arteritis With Incidental Meningioma. Journal of Neuro-Ophthalmology 2021; 41 (01) e122-e124