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Management of Multiple Meningiomas Georgios Tsermoulas, Mudr, Msc, FRCS(SN),1 Mazda K

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Management of Multiple Meningiomas Georgios Tsermoulas, Mudr, Msc, FRCS(SN),1 Mazda K CLINICAL ARTICLE J Neurosurg 128:1403–1409, 2018 Management of multiple meningiomas Georgios Tsermoulas, MUDr, MSc, FRCS(SN),1 Mazda K. Turel, MCh,1 Jared T. Wilcox, MD, MSc,1 David Shultz, MD, PhD,2 Richard Farb, MD, FRCPC,3 Gelareh Zadeh, MD, PhD, FRCSC,1 and Mark Bernstein, MD, MHSc, FRCS(C)1 Divisions of 1Neurosurgery and 3Neuroradiology, Toronto Western Hospital; and 2Department of Radiation Oncology, Princess Margaret Cancer Centre, Toronto, Ontario, Canada OBJECTIVE Multiple meningiomas account for 1%–10% of meningiomas. This study describes epidemiological aspects of the disease and its management, which is more challenging than for single tumors. METHODS A consecutive series of adult patients with ≥ 2 spatially separated meningiomas was reviewed. Patients with neurofibromatosis Type 2 were excluded. The authors collected clinical, imaging, histological, and treatment data to ob- tain information on epidemiology, management options, and outcomes of active treatment and surveillance. RESULTS A total of 133 consecutive patients were included over 25 years, with a total of 395 synchronous and 53 metachronous meningiomas, and a median of 2 tumors per patient. One hundred six patients had sporadic disease, 26 had radiation-induced disease, and 1 had familial meningiomatosis. At presentation, half of the patients were asympto- matic. In terms of their maximum cross-sectional diameter, the tumors were small (≤ 2 cm) in 67% and large (> 4 cm) in 11% of the meningiomas. Fifty-four patients had upfront treatment, and 31 had delayed treatment after an observation period (mean 4 years). One in 4 patients had ≥ 2 meningiomas treated. Overall, 64% of patients had treatment for 142 tumors—67 with surgery and 18 with radiotherapy alone. The mean follow-up was 7 years, with 13% of treated patients receiving salvage therapy. Approximately 1 in 4 patients who underwent surgery had ≥ 1 WHO Grade II or III menin- gioma. Meningiomas of different histological subtypes and grades in the same patient were not uncommon. CONCLUSIONS Multiple meningiomas are often asymptomatic, probably because the majority are small and a signifi- cant proportion are induced by radiation. Approximately two-thirds of patients with multiple meningiomas require therapy, but only one-third of all meningiomas need active treatment. The authors recommend surveillance for stable and asymp- tomatic meningiomas and therapy for those that are symptomatic or growing. https://thejns.org/doi/abs/10.3171/2017.2.JNS162608 KEY WORDS multiple meningiomas; radiation-induced meningioma; asymptomatic meningioma; craniotomy; surveillance; oncology ENINGIOMAS are common brain tumors, account- the clinician has to decide whether the disease is symptom- ing for approximately one-third of all primary atic, which lesion is responsible, if treatment is warranted, brain tumors in adults. The majority are single. which treatment option is best, when to initiate treatment, MMultiple meningiomas are defined as the presence of ≥ 2 and how to manage incidental multiple meningiomas. spatially separated synchronous or metachronous lesions, The literature on this subject is relatively scarce. Most and they occur in up to 10% of meningiomas.15,18 They can published papers are case reports or small case series, and be sporadic, radiation induced, or familial. The 2 familial there is a gap in the knowledge on the best management syndromes commonly associated with the disorder are neu- of this disease. The objective of this study was to review rofibromatosis Type 2 (NF2) and familial meningiomatosis. our practice to answer the aforementioned questions and The pathogenesis, natural history, and management of to provide guidance for the management of patients with single meningiomas have been extensively studied; how- multiple meningiomas. ever, similar knowledge on multiple meningiomas remains limited. These lesions represent a heterogeneous group with different etiologies, probably constituting distinct en- Methods tities from single meningiomas. In addition, multiple me- This is a retrospective observational study of a con- ningiomas pose special management challenges because secutive cohort of patients with multiple meningiomas ABBREVIATIONS NF2 = neurofibromatosis Type 2; SRS = stereotactic radiosurgery. SUBMITTED October 18, 2016. ACCEPTED February 2, 2017. INCLUDE WHEN CITING Published online July 21, 2017; DOI: 10.3171/2017.2.JNS162608. ©AANS, 2018 J Neurosurg Volume 128 • May 2018 1403 Unauthenticated | Downloaded 10/05/21 02:00 PM UTC G. Tsermoulas et al. managed at Toronto Western Hospital between 1991 and Results 2016. The study received approval from the local Research During a 25-year period, there were 147 potentially eli- Ethics Board. Our Neurosurgery Division has a dedicated gible patients. Patients were excluded for missing data (n neuro-oncology service and a multidisciplinary team of = 8), discharge at presentation (n = 5), and mortality in the clinicians with expertise in the management of meningio- postoperative period (n = 1, pulmonary embolism). We in- mas that serves a population area of approximately 5 mil- cluded 133 patients in the final analysis, with a total of 448 lion. meningiomas. The baseline characteristics of the cohort are Inclusion criteria were adult patients (≥ 18 years) with ≥ summarized in Table 1. There were 29 men and 104 women diagnosis of 2 separate meningiomas at presentation with a mean age at presentation of 58 years (range 18–91 or diagnosis of a single meningioma at presentation who years). One hundred six patients had sporadic disease, 26 developed additional meningiomas during follow-up; and patients had radiation-induced meningiomas, and only 1 multiple meningiomas of sporadic, radiation-induced, and case was associated with familial meningiomatosis. All pa- familial (non-NF2) types. Exclusion criteria were patients tients with radiation-induced meningiomas underwent cra- with NF2 types (multiple meningiomas and bilateral or nial radiation therapy many years before presentation, most unilateral vestibular schwannoma), because their manage- in childhood for a different brain tumor. The vast majority ment is more complex and usually focuses on the schwan- of meningiomas were synchronous tumors (n = 395, 88%), nomas; patients without follow-up data; or those who and 34 patients developed 53 metachronous meningiomas. developed metachronous meningiomas deemed to be me- Patients with multiple meningiomas in our series present- tastases or recurrence of a previous meningioma. The lat- ed at similar ages compared with those with single meningi- ter cases included patients with high-grade meningiomas omas, but the female/male ratio was higher (3.5:1) compared who developed meningiomas at follow-up or patients who with the 2:1 ratio reported for single lesions (p = 0.002).40 developed tumors in the vicinity of a previously resected The female/male ratio was 4.6:1 for sporadic and 1.9:1 for meningioma, irrespective of grade. radiation-induced multiple meningioma cases (p = 0.11). The meningiomas were deemed radiation induced when the Cahan criteria were fulfilled as follows: the patient Presentation and Imaging had received radiation therapy; the neoplasm occurred in the previously irradiated field; a latency period of several The modes of presentation were as follows. Sixty-six years had elapsed (5 years in the original description); the cases were discovered incidentally and were asymptomat- tumor was histologically and/or radiographically different from the primary; and syndromes associated with neo- TABLE 1. Epidemiological characteristics of 133 patients with plasms had been excluded.9 448 multiple meningiomas The Radiology Department database was searched us- Characteristic No. (%) ing the words “multiple” or “second” or “additional” or “another” and “meningioma” or “meningiomas” for the Sex study period, and all of the search results (n = 2735) were Male 29 (22) reviewed. The following retrospective data were collect- Female 104 (78) ed for the list of eligible patients: demographic data (age Mean age, yrs 58 and sex), etiology, presentation (symptomatic or asymp- Etiology, patients/tumors tomatic), imaging findings, histological characteristics, and treatment method and outcome. We categorized the Sporadic 106 (80)/333 (74) tumors based on size, according to their maximum cross- Radiation induced 26 (20)/110 (25) sectional diameter (small [≤ 2 cm], medium [> 2 and ≤ 4 Familial meningiomatosis 1 (1)/5 (1) cm], and large [> 4 cm]), and location (convexity, midline No. of meningiomas [parasagittal, falcine/tentorial, intraventricular], or skull Mean per patient 3 base). Management options included upfront treatment or Synchronous 395 a wait-and-see approach. Treatment included resection or radiation therapy, in the form of stereotactic radiosurgery Metachronous 53 (SRS) or fractionated radiotherapy. Adjuvant radiothera- Size at presentation, synchronous py was administered in selected cases. The outcome for Small 264 (67) treated patients was local control measured by the need for Medium 87 (22) further intervention for the recurrent tumors. The outcome Large 44 (11) for patients managed expectantly was tumor growth and Location at presentation, synchronous development of symptoms measured by the need for treat- ment during the surveillance period. Convexity 156 (39) The statistical analysis was performed with XLSTAT Midline 138 (35) version 2016.04.32523 (copyright Addinsoft 1995–2016). Skull base 101 (26) The descriptive statistics of the clinical,
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