Proton Radiotherapy for Vestibular Schwannomas in Patients with NF2-Related Schwannomatosis: A Case Series

Jules P.J. Douwes; Kimberley S. Koetsier; Victor S. van Dam; Scott R. Plotkin; Frederick G. Barker; D. Bradley Welling; Jeroen C. Jansen; Erik F. Hensen; Helen A. Shih

doi:10.3390/curroncol30030263

Proton Radiotherapy for Vestibular Schwannomas in Patients with NF2-Related Schwannomatosis: A Case Series

Jules P.J. Douwes^*, Kimberley S. Koetsier, Victor S. van Dam, Scott R. Plotkin, Frederick G. Barker, D. Bradley Welling, Jeroen C. Jansen, Erik F. Hensen, Helen A. Shih

^*Corresponding author for this work

Otorhinolaryngology and Head and Neck Surgery

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Abstract

(1) Background: This study aimed to evaluate the efficacy and treatment-related toxicity of proton radiotherapy (PRT) for vestibular schwannoma (VS) in patients with neurofibromatosis type 2-related schwannomatosis (NF2). (2) Methods: Consecutive NF2 patients treated with PRT for VS between 2004 and 2016 were retrospectively evaluated, focusing on tumor volume, facial and trigeminal nerve function, hearing, tinnitus, vestibular symptoms, and the need for salvage therapy after PRT. (3) Results: Eight patients were included (median age 36 years, 50% female). Median follow-up was 71 months. Five (63%) patients received fractionated PRT and three (38%) received PRT radiosurgery for VS. Six patients (75%) received prior VS surgery; three also received bevacizumab. Six patients (75%) did not require salvage therapy after PRT. Two patients (25%) with residual hearing lost it after PRT, and six had already lost ipsilateral hearing prior to PRT. Tumor and treatment-related morbidity could be evaluated in six patients. Following PRT, conditions that occurred or worsened were: facial paresis in five (83%), trigeminal hypoesthesia in two (33%), tinnitus in two (33%), and vestibular symptoms in four patients (67%). (4) Conclusion: After PRT for VS, the majority of the NF2 patients in the cohort did not require additional therapy. Tumor and/or treatment-related cranial nerve deficits were common. This is at least partly explained by the use of PRT as a salvage treatment after surgery or bevacizumab, in the majority of cases. There remains the further opportunity to elucidate the efficacy and toxicity of proton radiotherapy as a primary treatment.

Original language	English
Pages (from-to)	3473-3483
Number of pages	11
Journal	Current Oncology
Volume	30
Issue number	3
DOIs	https://doi.org/10.3390/curroncol30030263
Publication status	Published - 20 Mar 2023

Bibliographical note

Funding Information:
K.S.K. receives salary partly funded by (1) the Surcharge for Top Consortia for Knowledge and Innovation (TKIs) from the Ministry of Economic Affairs and Climate and the HollandPTC consortium—Erasmus MC, Rotterdam; Holland Proton Therapy Center, Delft; Leiden University Medical Center, Leiden; Delft University of Technology, Delft, the Netherlands, and (2) Varian Medical System (the principal investigator of this research grant is E.F.H.).

Publisher Copyright: © 2023 by the authors.

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@article{7af06fcbaccc4b9088c532da9b0b8ff3,

title = "Proton Radiotherapy for Vestibular Schwannomas in Patients with NF2-Related Schwannomatosis: A Case Series",

abstract = "(1) Background: This study aimed to evaluate the efficacy and treatment-related toxicity of proton radiotherapy (PRT) for vestibular schwannoma (VS) in patients with neurofibromatosis type 2-related schwannomatosis (NF2). (2) Methods: Consecutive NF2 patients treated with PRT for VS between 2004 and 2016 were retrospectively evaluated, focusing on tumor volume, facial and trigeminal nerve function, hearing, tinnitus, vestibular symptoms, and the need for salvage therapy after PRT. (3) Results: Eight patients were included (median age 36 years, 50% female). Median follow-up was 71 months. Five (63%) patients received fractionated PRT and three (38%) received PRT radiosurgery for VS. Six patients (75%) received prior VS surgery; three also received bevacizumab. Six patients (75%) did not require salvage therapy after PRT. Two patients (25%) with residual hearing lost it after PRT, and six had already lost ipsilateral hearing prior to PRT. Tumor and treatment-related morbidity could be evaluated in six patients. Following PRT, conditions that occurred or worsened were: facial paresis in five (83%), trigeminal hypoesthesia in two (33%), tinnitus in two (33%), and vestibular symptoms in four patients (67%). (4) Conclusion: After PRT for VS, the majority of the NF2 patients in the cohort did not require additional therapy. Tumor and/or treatment-related cranial nerve deficits were common. This is at least partly explained by the use of PRT as a salvage treatment after surgery or bevacizumab, in the majority of cases. There remains the further opportunity to elucidate the efficacy and toxicity of proton radiotherapy as a primary treatment.",

author = "Douwes, {Jules P.J.} and Koetsier, {Kimberley S.} and {van Dam}, {Victor S.} and Plotkin, {Scott R.} and Barker, {Frederick G.} and Welling, {D. Bradley} and Jansen, {Jeroen C.} and Hensen, {Erik F.} and Shih, {Helen A.}",

note = "Funding Information: K.S.K. receives salary partly funded by (1) the Surcharge for Top Consortia for Knowledge and Innovation (TKIs) from the Ministry of Economic Affairs and Climate and the HollandPTC consortium—Erasmus MC, Rotterdam; Holland Proton Therapy Center, Delft; Leiden University Medical Center, Leiden; Delft University of Technology, Delft, the Netherlands, and (2) Varian Medical System (the principal investigator of this research grant is E.F.H.). Publisher Copyright: {\textcopyright} 2023 by the authors.",

year = "2023",

month = mar,

day = "20",

doi = "10.3390/curroncol30030263",

language = "English",

volume = "30",

pages = "3473--3483",

journal = "Current Oncology",

issn = "1198-0052",

publisher = "Multidisciplinary Digital Publishing Institute (MDPI)",

number = "3",

}

TY - JOUR

T1 - Proton Radiotherapy for Vestibular Schwannomas in Patients with NF2-Related Schwannomatosis

T2 - A Case Series

AU - Douwes, Jules P.J.

AU - Koetsier, Kimberley S.

AU - van Dam, Victor S.

AU - Plotkin, Scott R.

AU - Barker, Frederick G.

AU - Welling, D. Bradley

AU - Jansen, Jeroen C.

AU - Hensen, Erik F.

AU - Shih, Helen A.

N1 - Funding Information: K.S.K. receives salary partly funded by (1) the Surcharge for Top Consortia for Knowledge and Innovation (TKIs) from the Ministry of Economic Affairs and Climate and the HollandPTC consortium—Erasmus MC, Rotterdam; Holland Proton Therapy Center, Delft; Leiden University Medical Center, Leiden; Delft University of Technology, Delft, the Netherlands, and (2) Varian Medical System (the principal investigator of this research grant is E.F.H.). Publisher Copyright: © 2023 by the authors.

PY - 2023/3/20

Y1 - 2023/3/20

N2 - (1) Background: This study aimed to evaluate the efficacy and treatment-related toxicity of proton radiotherapy (PRT) for vestibular schwannoma (VS) in patients with neurofibromatosis type 2-related schwannomatosis (NF2). (2) Methods: Consecutive NF2 patients treated with PRT for VS between 2004 and 2016 were retrospectively evaluated, focusing on tumor volume, facial and trigeminal nerve function, hearing, tinnitus, vestibular symptoms, and the need for salvage therapy after PRT. (3) Results: Eight patients were included (median age 36 years, 50% female). Median follow-up was 71 months. Five (63%) patients received fractionated PRT and three (38%) received PRT radiosurgery for VS. Six patients (75%) received prior VS surgery; three also received bevacizumab. Six patients (75%) did not require salvage therapy after PRT. Two patients (25%) with residual hearing lost it after PRT, and six had already lost ipsilateral hearing prior to PRT. Tumor and treatment-related morbidity could be evaluated in six patients. Following PRT, conditions that occurred or worsened were: facial paresis in five (83%), trigeminal hypoesthesia in two (33%), tinnitus in two (33%), and vestibular symptoms in four patients (67%). (4) Conclusion: After PRT for VS, the majority of the NF2 patients in the cohort did not require additional therapy. Tumor and/or treatment-related cranial nerve deficits were common. This is at least partly explained by the use of PRT as a salvage treatment after surgery or bevacizumab, in the majority of cases. There remains the further opportunity to elucidate the efficacy and toxicity of proton radiotherapy as a primary treatment.

AB - (1) Background: This study aimed to evaluate the efficacy and treatment-related toxicity of proton radiotherapy (PRT) for vestibular schwannoma (VS) in patients with neurofibromatosis type 2-related schwannomatosis (NF2). (2) Methods: Consecutive NF2 patients treated with PRT for VS between 2004 and 2016 were retrospectively evaluated, focusing on tumor volume, facial and trigeminal nerve function, hearing, tinnitus, vestibular symptoms, and the need for salvage therapy after PRT. (3) Results: Eight patients were included (median age 36 years, 50% female). Median follow-up was 71 months. Five (63%) patients received fractionated PRT and three (38%) received PRT radiosurgery for VS. Six patients (75%) received prior VS surgery; three also received bevacizumab. Six patients (75%) did not require salvage therapy after PRT. Two patients (25%) with residual hearing lost it after PRT, and six had already lost ipsilateral hearing prior to PRT. Tumor and treatment-related morbidity could be evaluated in six patients. Following PRT, conditions that occurred or worsened were: facial paresis in five (83%), trigeminal hypoesthesia in two (33%), tinnitus in two (33%), and vestibular symptoms in four patients (67%). (4) Conclusion: After PRT for VS, the majority of the NF2 patients in the cohort did not require additional therapy. Tumor and/or treatment-related cranial nerve deficits were common. This is at least partly explained by the use of PRT as a salvage treatment after surgery or bevacizumab, in the majority of cases. There remains the further opportunity to elucidate the efficacy and toxicity of proton radiotherapy as a primary treatment.

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U2 - 10.3390/curroncol30030263

DO - 10.3390/curroncol30030263

M3 - Article

C2 - 36975476

AN - SCOPUS:85151165476

SN - 1198-0052

VL - 30

SP - 3473

EP - 3483

JO - Current Oncology

JF - Current Oncology

IS - 3

ER -

Proton Radiotherapy for Vestibular Schwannomas in Patients with NF2-Related Schwannomatosis: A Case Series

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