Repeat stereotactic radiosurgery for persistent cerebral arteriovenous malformations in pediatric patients

Author:

Garcia Gracie1,Mantziaris Georgios1,Pikis Stylianos1,Dumot Chloe1,Lunsford L. Dade2,Niranjan Ajay2,Wei Zhishuo2,Srinivasan Priyanka2,Tang Lilly W.2,Liscak Roman3,May Jaromir3,Lee Cheng-Chia45,Yang Huai-Che45,Peker Selcuk6,Samanci Yavuz6,Nabeel Ahmed M.78,Reda Wael A.78,Tawadros Sameh R.79,Abdel Karim Khaled79,El-Shehaby Amr M. N.79,Emad Eldin Reem710,Elazzazi Ahmed Hesham9,Martínez Moreno Nuria11,Martínez Álvarez Roberto11,Padmanaban Varun12,Jareczek Francis J.12,McInerney James12,Cockroft Kevin M.12,Alzate Juan Diego13,Kondziolka Douglas13,Tripathi Manjul14,Sheehan Jason P.1

Affiliation:

1. Department of Neurological Surgery, University of Virginia, Charlottesville, Virginia;

2. Department of Neurological Surgery, University of Pittsburgh Medical Center, Pennsylvania;

3. Department of Stereotactic and Radiation Neurosurgery, Nemocnice Na Homolce Hospital, Prague, Czech Republic;

4. Department of Neurosurgery, Neurological Institute, Taipei Veterans General Hospital, Taipei, Taiwan;

5. School of Medicine, National Yang Ming Chiao Tung University, Taipei, Taiwan;

6. Department of Neurosurgery, Koç University School of Medicine, Istanbul, Turkey;

7. Gamma Knife Center Cairo, Nasser Institute Hospital, Cairo, Egypt;

8. Neurosurgery Department, Benha University, Qalubiya, Egypt;

9. Neurosurgery and Clinical Oncology Department, Ain Shams University, Cairo, Egypt;

10. Radiation Oncology Department, National Cancer Institute, Cairo University, Cairo, Egypt;

11. Radiosurgery Unit, Hospital Ruber Internacional, Madrid, Spain;

12. Department of Neurosurgery, Pennsylvania State Milton S. Hershey Medical Center, Hershey, Pennsylvania;

13. Department of Neurosurgery, NYU Langone, New York, New York; and

14. Department of Neurosurgery, Postgraduate Institute of Medical Education and Research, Chandigarh, India

Abstract

OBJECTIVE The purpose of this study was to describe the long-term outcomes and associated risks related to repeat stereotactic radiosurgery (SRS) for persistent arteriovenous malformations (AVMs) in pediatric patients. METHODS Under the auspices of the International Radiosurgery Research Foundation, this retrospective multicenter study analyzed pediatric patients who underwent repeat, single-session SRS between 1987 and 2022. The primary outcome variable was a favorable outcome, defined as nidus obliteration without hemorrhage or neurological deterioration. Secondary outcomes included rates and probabilities of hemorrhage, radiation-induced changes (RICs), and cyst or tumor formation. RESULTS The cohort included 83 pediatric patients. The median patient age was 11 years at initial SRS and 15 years at repeat SRS. Fifty-seven children (68.7%) were managed exclusively using SRS, and 42 (50.6%) experienced hemorrhage prior to SRS. Median AVM diameter and volume were substantially different between the first (25 mm and 4.5 cm3, respectively) and second (16.5 mm and 1.6 cm3, respectively) SRS, while prescription dose and isodose line remained similar. At the 5-year follow-up evaluation from the second SRS, nidus obliteration was achieved in 42 patients (50.6%), with favorable outcome in 37 (44.6%). The median time to nidus obliteration and hemorrhage was 35.5 and 38.5 months, respectively. The yearly cumulative probability of favorable outcome increased from 2.5% (95% CI 0.5%–7.8%) at 1 year to 44% (95% CI 32%–55%) at 5 years. The probability of achieving obliteration followed a similar pattern and reached 51% (95% CI 38%–62%) at 5 years. The 5-year risk of hemorrhage during the latency period after the second SRS reached 8% (95% CI 3.2%–16%). Radiographically, 25 children (30.1%) had RICs, but only 5 (6%) were symptomatic. Delayed cyst formation occurred in 7.2% of patients, with a median onset of 47 months. No radiation-induced neoplasia was observed. CONCLUSIONS The study results showed nidus obliteration in most pediatric patients who underwent repeat SRS for persistent AVMs. The risks of symptomatic RICs and latency period hemorrhage were quite low. These findings suggest that repeat radiosurgery should be considered when treating pediatric patients with residual AVM after prior SRS. Further study is needed to define the role of repeat SRS more fully in this population.

Publisher

Journal of Neurosurgery Publishing Group (JNSPG)

Reference35 articles.

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2. Stereotactic radiosurgery for pediatric brain arteriovenous malformations: long-term outcomes;Chen CJ,2020

3. A population-based study of brain arteriovenous malformation: long-term treatment outcomes;ApSimon HT,2002

4. Acute fatal hemorrhage from previously undiagnosed cerebral arteriovenous malformations in children: a single-center experience;Riordan CP,2018

5. Untreated brain arteriovenous malformation: patient-level meta-analysis of hemorrhage predictors;Kim H,2014

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