Pediatric brain arteriovenous malformation recurrence: a cohort study, systematic review and meta-analysis

Hak, Jean-François; Boulouis, Grégoire; Kerleroux, Basile; Benichi, Sandro; Stricker, Sarah; Gariel, Florent; Garzelli, Lorenzo; Meyer, Philippe; Kossorotoff, Manoëlle; Boddaert, Nathalie; Vidal, Vincent; Girard, Nadine; Dangouloff-Ros, Volodia; Brunelle, Françis; Fullerton, Heather J.; Hetts, Steven W.; Blauwblomme, Thomas; Naggara, Olivier

doi:10.1136/neurintsurg-2021-017777

Cited by 16 publications

(14 citation statements)

References 40 publications

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“…Once an AVM appears to be obliterated on a post-treatment DSA, it is unclear what the optimal imaging follow-up strategy should be. Pediatric patients, in particular, have been reported to have recurrence of apparently cured brain AVMs; such recurrences may be more common in younger patients who have presented with ruptured AVMs as opposed to unruptured AVMs 146–150…”

Section: Recurrence After Treatment and Follow-up Strategiesmentioning

confidence: 99%

Endovascular treatment in the multimodality management of brain arteriovenous malformations: report of the Society of NeuroInterventional Surgery Standards and Guidelines Committee

Leacy

Ansari

Schirmer

et al. 2022

J NeuroIntervent Surg

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BackgroundThe purpose of this review is to summarize the data available for the role of angiography and embolization in the comprehensive multidisciplinary management of brain arteriovenous malformations (AVMsMethodsWe performed a structured literature review for studies examining the indications, efficacy, and outcomes for patients undergoing endovascular therapy in the context of brain AVM management. We graded the quality of the evidence. Recommendations were arrived at through a consensus conference of the authors, then with additional input from the full Society of NeuroInterventional Surgery (SNIS) Standards and Guidelines Committee and the SNIS Board of Directors.ResultsThe multidisciplinary evaluation and treatment of brain AVMs continues to evolve. Recommendations include: (1) Digital subtraction catheter cerebral angiography (DSA)—including 2D, 3D, and reformatted cross-sectional views when appropriate—is recommended in the pre-treatment assessment of cerebral AVMs. (I, B-NR). (2) It is recommended that endovascular embolization of cerebral arteriovenous malformations be performed in the context of a complete multidisciplinary treatment plan aiming for obliteration of the AVM and cure. (I, B-NR). (3) Embolization of brain AVMs before surgical resection can be useful to reduce intraoperative blood loss, morbidity, and surgical complexity. (IIa, B-NR). (4) The role of primary curative embolization of cerebral arteriovenous malformations is uncertain, particularly as compared with microsurgery and radiosurgery with or without adjunctive embolization. Further research is needed, particularly with regard to risk for AVM recurrence. (III equivocal, C-LD). (5) Targeted embolization of high-risk features of ruptured brain AVMs may be considered to reduce the risk for recurrent hemorrhage. (IIb, C-LD). (6) Palliative embolization may be useful to treat symptomatic AVMs in which curative therapy is otherwise not possible. (IIb, B-NR). (7) The role of AVM embolization as an adjunct to radiosurgery is not well-established. Further research is needed. (III equivocal, C-LD). (8) Imaging follow-up after apparent cure of brain AVMs is recommended to assess for recurrence. Although non-invasive imaging may be used for longitudinal follow-up, DSA remains the gold standard for residual or recurrent AVM detection in patients with concerning imaging and/or clinical findings. (I, C-LD). (9) Improved national and international reporting of patients of all ages with brain AVMs, their treatments, side effects from treatment, and their long-term outcomes would enhance the ability to perform clinical trials and improve the rigor of research into this rare condition. (I, C-EO).ConclusionsAlthough the quality of evidence is lower than for more common conditions subjected to multiple randomized controlled trials, endovascular therapy has an important role in the management of brain AVMs. Prospective studies are needed to strengthen the data supporting these recommendations.

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Section: Recurrence After Treatment and Follow-up Strategiesmentioning

confidence: 99%

Endovascular treatment in the multimodality management of brain arteriovenous malformations: report of the Society of NeuroInterventional Surgery Standards and Guidelines Committee

Leacy

Ansari

Schirmer

et al. 2022

J NeuroIntervent Surg

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“…However, there is a lack of consensus regarding specific medical treatments to cure the disease. The rate of complications in patients undergoing microsurgery, embolization, and radiosurgery is high, ranging from 5.1% to 7.4%, leading to death or persistent neurological dysfunction,4 especially in pediatric patients 5. Therefore, there is a need to identify risk factors associated with the disease and unfavorable clinical outcomes.…”

Section: Introductionmentioning

confidence: 99%

Associated genetic variants and potential pathogenic mechanisms of brain arteriovenous malformation

Liu

Zhang

et al. 2022

J NeuroIntervent Surg

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BackgroundThe pathogenic mechanism of brain arteriovenous malformation (bAVM) is poorly understood. A growing body of evidence indicates that genetic factors play crucial roles in bAVM. This study examined genetic variants associated with bAVM through quantitative synthesis and qualitative description of literature.MethodsFive databases were searched to gather potentially relevant articles published up to January 2022. STATA 14.0 software was used for statistical analyses. Pooled odds ratios and 95% confidence intervals were calculated with random effect models, and heterogeneity was assessed using the Cochran Q test and quantified with the I2 test. Sensitivity and publication bias were analyzed to test the robustness of the associations. Variants identified in only one study or with great heterogeneity were not suitable for pooling association analysis, and therefore a qualitative systematic review was performed.ResultsIn total, 30 papers were included in a systematic review involving 4709 cases and 7832 controls, where 17 papers were in a meta-analysis. A suggested association of bAVM was observed with ACVRL1 rs2071219 in the additive model and CDKN2B-AS1 rs1333040 in the recessive and additive models. Other variants of genes that could not be analyzed were summarized by qualitative description. These genes were mostly involved in bone morphogenic protein/transforming growth factor beta (BMP/TGF-β), vascular endothelial growth factor/vascular endothelial growth factor receptor (VEGF/VEGFR), and RAS-mitogen activated protein kinase (MAPK) signaling and inflammation.ConclusionsAccording to our meta-analysis, ACVRL1 rs2071219 and CDKN2B-AS1 rs1333040 were potentially associated with bAVM. Multiple pathological signaling pathways could affect disease development. Future studies should aim to determine the interaction of candidate genes with environmental risk factors and to elucidate detailed mechanisms of action of variants and genes.1

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“…2,3 Although each AVM intervention shares the common goal of nidal obliteration, there is intertreatment variability with regard to hemorrhage and recurrence rates after angiographic cure. 2 Due to the rarity of these 2 events, this longitudinal multicenter retrospective cohort study aims to define the brain hemorrhage and nidal recurrence risks of obliterated AVMs in patients treated with stereotactic radiosurgery (SRS).…”

mentioning

confidence: 99%

Hemorrhage and Recurrence of Obliterated Brain Arteriovenous Malformations Treated With Stereotactic Radiosurgery

Chen

Ding

Kumar

et al. 2022

Stroke

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Background: Although complete nidal obliteration of brain arteriovenous malformations (AVM) is generally presumed to represent durable cure, postobliteration hemorrhage, and AVM recurrence have become increasingly recognized phenomena. The goal of the study was to define hemorrhage and nidal recurrence risks of obliterated AVMs treated with stereotactic radiosurgery (SRS). Methods: This is a retrospective cohort study from the International Radiosurgery Research Foundation comprising AVM patients treated between 1987 and 2020. Patients with AVM obliteration on digital subtraction angiography (DSA) were included. Outcomes were (1) hemorrhage and (2) AVM recurrence. Follow-up duration began at the time of AVM obliteration and was censored at subsequent hemorrhage, AVM recurrence, additional AVM treatment, or loss to follow-up. Annualized risk and survival analyses were performed. A sensitivity analysis comprising patients with AVM obliteration on magnetic resonance imaging or DSA was also performed for postobliteration hemorrhage. Results: The study cohort comprised 1632 SRS-treated patients with AVM obliteration on DSA. Pediatric patients comprised 15% of the cohort, and 42% of AVMs were previously ruptured. The mean imaging follow-up after AVM obliteration was 22 months. Among 1607 patients with DSA-confirmed AVM obliteration, 16 hemorrhages (1.0%) occurred over 2223 patient-years of follow-up (0.72%/y). Of the 1543 patients with DSA-confirmed AVM obliteration, 5 AVM recurrences (0.32%) occurred over 2071 patient-years of follow-up (0.24%/y). Of the 16 patients with postobliteration hemorrhage, AVM recurrence was identified in 2 (12.5%). In the sensitivity analysis comprising 1939 patients with post-SRS AVM obliteration on magnetic resonance imaging or DSA, 16 hemorrhages (0.83%) occurred over 2560 patient-years of follow-up (0.63%/y). Conclusions: Intracranial hemorrhage and recurrent arteriovenous shunting after complete nidal obliteration are rare in AVM patients treated with SRS, and each phenomenon harbors an annual risk of <1%. Although routine postobliteration DSA cannot be recommended to SRS-treated AVM patients, long-term neuroimaging may be advisable in these patients.

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Pediatric brain arteriovenous malformation recurrence: a cohort study, systematic review and meta-analysis

Cited by 16 publications

References 40 publications

Endovascular treatment in the multimodality management of brain arteriovenous malformations: report of the Society of NeuroInterventional Surgery Standards and Guidelines Committee

Endovascular treatment in the multimodality management of brain arteriovenous malformations: report of the Society of NeuroInterventional Surgery Standards and Guidelines Committee

Associated genetic variants and potential pathogenic mechanisms of brain arteriovenous malformation

Hemorrhage and Recurrence of Obliterated Brain Arteriovenous Malformations Treated With Stereotactic Radiosurgery

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