In Reply: Integrated Clinical Genetic Analysis of Meningiomas Causing Bony Hyperostosis Shows More Severe Clinical Course and Overexpression of Secreted Pro-Osteogenic Factors

To the Editor: We thank Dr Ferreira and colleagues for their thoughtful comments and interest in our recent article published in Neurosurgery.1,2 We appreciate the additional clinical perspective they offer by emphasizing the importance of dedicated multidisciplinary teams to treat hyperostotic meningiomas.1,3,4 As referenced, we identified a more severe clinical course among patients treated for hyperostotic meningiomas.2 We agree with the respondents that craniectomy is frequently required in such cases, as reflected in our own findings. Because our study included genetic analyses, our patient cohort was relatively recent and had limited follow-up (mean follow-up = 2 years). With this significant limitation, we did not observe a statistically significant difference in recurrence-free survival within hyperostotic subgroup when craniectomy was performed (P = .07). Nevertheless, additional research is needed to identify which patients may benefit from up-front craniectomy in hyperostotic meningiomas. Similar to the respondents' practice, we routinely use prefabricated custom 3D-printed polyether-ether-ketone implants with craniotomy guides in select cases. A recent study demonstrated that prefabricated implants provide better functional outcomes and fewer complications than intraoperative implant construction.5 We fully concur with the respondents that additional, often multidisciplinary, planning should be undertaken in the management of these challenging tumors. We also appreciate that the respondents mention the use of 68Ga-DOTATATE positron emission tomography/computed tomography as a means of detecting intraosseous involvement.6 We have begun to use this modality to identify residual or recurrent disease, particularly when planning postoperative radiotherapy in molecularly aggressive, MenG C tumors.7-10 This approach relies on tumor expression of somatostatin receptors. Although not discussed in our manuscript, somatostatin receptor 1 (SSTR1) transcription was upregulated in the hyperostosis cohort (Log2FC = 1.488, P-adj = 1.39E-02) but expressed at a low level (baseMean = 40.2). Expression of SSTR2 and SSTR4 did not differ significantly between groups. We appreciate the respondents for identifying this interesting radio-genomic correlation and agree that further study is warranted. We thank the respondents for their insightful discussion regarding the complex management of hyperostotic meningiomas and for highlighting the evolving concept of oncoplastic neurosurgery. At our institution, multidisciplinary care extends beyond our collaborations with neuropathology, neuroradiology, neuro-oncology, and radiation oncology to include other surgical specialties.11 We frequently partner with plastic and reconstructive surgeons for complex wound closures after redo or irradiated cases, oculoplastic surgeons for extensive orbital involvement, and otolaryngology (ENT) surgeons for broad skull base disease. These represent only a subset of multidisciplinary relationships that can arise in the care of patients with complex meningioma. Further research is needed to define and optimize the role of such collaboration in primary tumor resections. We appreciate the authors' engagement with our work and share their optimism that continued multidisciplinary efforts will drive technical and clinical advances to improve outcomes for these patients.