Pediatric brain AVM resection with simultaneous cranioplasty in hybrid operating room

Pediatric brain AVM resection with simultaneous cranioplasty in hybrid operating room | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Research Article Pediatric brain AVM resection with simultaneous cranioplasty in hybrid operating room Peng SUN, Yutong LIU, Mading ZHOU, Jianxin DU, Gao ZENG This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-9016262/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 4 You are reading this latest preprint version Abstract Purpose Brain arteriovenous malformation (AVM) was the most common cause of spontaneous cerebral hemorrhage in children. Many children with ruptured AVM required emergency decompressive craniectomy (DC). Reports focusing on the management of residual AVM after DC were rare. We reviewed children who underwent AVM resection with simultaneous cranioplasty to summarize treatment strategies. Method We reviewed children who underwent brain AVM resection with simultaneous cranioplasty in a hybrid operating room between 2016 and 2025. Results Between 2016 and 2025, 15 cases of AVM resection with simultaneous cranioplasty were performed at this center in a hybrid operating room. The mean interval between DC and AVM resection was 5.3 months (range, 1–25 months). The mean mRS score was 1.8 before AVM resection. Complete AVM resection was confirmed in all patients by intraoperative DSA in the hybrid operating room. Cranioplasty materials included titanium mesh in 8 cases, PEEK in 5 cases, and autologous bone flap in 2 cases. The mean follow-up duration was 50.6 months (range, 8.6–122.1 months) in 14 patients, as 1 patient was lost to follow-up. Ten patients (10/14, 71.4%) achieved good outcomes, and the mean mRS score at the last follow-up was 1.1. AVM recurrence was confirmed by DSA in 1 patient 17 months after surgery. Conclusion Microsurgical resection of AVM, which provided the highest cure rate, combined with simultaneous cranioplasty, might have been an ideal treatment modality for children with residual AVM after DC. brain arteriovenous malformation cranioplasty microsurgery resection hybrid operating room Figures Figure 1 Figure 2 Figure 3 Introduction Brain arteriovenous malformation (AVM) was characterized by a tangle of dysplastic vessels (nidus) fed by arteries and drained by veins without intervening capillaries, forming a high-flow, low-resistance shunt between the arterial and venous systems. AVM was the most common cause of spontaneous cerebral hemorrhage in children. Approximately half (41/83) of children with ruptured AVM experienced severe hemorrhage, defined as a modified Rankin Scale (mRS) score >3 or requiring emergency hematoma evacuation [1]. Additionally, 35.8% (38/106) developed coma after hemorrhage [2]. Therefore, many children with ruptured AVM required emergency decompressive surgery. Some reports showed that hematoma evacuation or decompressive craniectomy (DC) without AVM resection was acceptable, as the incidence of AVM re-rupture before curative treatment was low [3–6]. For children who underwent DC, cranioplasty should have been performed as early as appropriate [7]. However, cranioplasty should have been undertaken only after AVM cure was confirmed. Otherwise, re-rupture of residual AVM after cranioplasty would have made subsequent treatment more complicated. Among available treatment modalities, microsurgical resection offered the highest cure rate, with recent studies reporting rates of 85%–99% [8–12]. Reports focusing on the management of residual AVM after DC were rare. In our opinion, the optimal strategy in this situation might have been AVM resection with simultaneous cranioplasty in a hybrid operating room. We reviewed children who underwent AVM resection with simultaneous cranioplasty at our hospital to summarize treatment strategies and provide recommendations regarding surgical details. Methods We reviewed children who underwent brain AVM resection with simultaneous cranioplasty in a hybrid operating room between 2016 and 2025. Data on clinical presentation, angioarchitectural characteristics, management, and outcomes were collected. All included cases had a preoperative DSA-confirmed intracranial AVM. The Spetzler–Martin grade [13] and supplementary grading scale [14] were applied. All patients underwent surgery in a DSA hybrid operating room. Cranioplasty was performed after complete AVM resection was confirmed by intraoperative DSA. Postoperative neurological deficits were evaluated in all patients, and outcome was assessed using the mRS; a score <2 was defined as a good outcome. Imaging follow-up was conducted with contrast-enhanced MRI or CTA at 6 months, 1 year, and annually thereafter. DSA was performed only if noninvasive imaging suggested AVM recurrence. Results Between 2016 and 2025, 15 cases of AVM resection with simultaneous cranioplasty were performed at this center in a hybrid operating room. There were 8 males and 7 females, with a mean age at onset of 9.7 years (range, 4–15 years). All patients presented with ruptured BAVM and consciousness disturbance. Emergency hematoma evacuation and DC were performed at other hospitals. The mean interval between DC and AVM resection was 5.3 months (range, 1–25 months). No re-rupture occurred during this interval. The mean preoperative mRS score was 1.8. Residual AVM was confirmed by DSA in all patients. AVMs were located in the cerebral cortical hemispheres (frontal, parietal, occipital, or temporal lobes) in 11 cases, in the basal ganglia in 3 cases, and in the cerebellum in 1 case. In 4 cases, the AVM was distant from the initial bone window (Fig1, 2), requiring extension of the incision and enlargement of the bone window. Spetzler–Martin grades were 1–2 in 7 cases (46.7%), 3 in 6 cases (40.0%), and 4 in 2 cases (13.3%). Supplementary Spetzler–Martin grades were 2–3 in 4 cases (26.7%) and 4–6 in 11 cases (73.3%). Preoperative embolization was performed in 4 cases. Complete AVM resection was confirmed in all patients by intraoperative DSA in the hybrid operating room. Residual AVM was detected on the first intraoperative DSA in 3 cases (20%). Further exploration and resection were undertaken, and repeat DSA confirmed no residual lesion in these cases. Cranioplasty materials included titanium mesh in 8 cases, PEEK in 5 cases, and autologous bone flap in 2 cases. Complications included suspected intracranial infection in 4 cases, although CSF cultures were negative in all cases. No wound healing complications or subcutaneous effusion occurred. No new neurological deficits were observed. The mean clinical follow-up duration was 50.6 months (range, 8.6–122.1 months) in 14 patients, as 1 patient was lost to follow-up. Ten patients (71.4%) achieved good outcomes, and the mean mRS score at the last follow-up was 1.1. The mean imaging follow-up duration was 25.7 months (range, 6.5–62.6 months), including MRI or CTA in 11 cases and DSA in 3 cases. AVM recurrence was confirmed by DSA in 1 patient 17 months after surgery. She underwent stereotactic radiotherapy (SRS) and experienced no re-rupture during 13 months of follow-up. Illustrative cases Case 1 (Fig.1) A 10-year-old boy suddenly developed headache accompanied by consciousness disturbance and underwent emergency hematoma evacuation and DC. (Supplementary Spetzler–Martin grade 2: S1V0E1/A1B0C1) Case 2 (Fig.2) An 11-year-old girl suddenly developed headache with consciousness disturbance and underwent emergency hematoma evacuation and DC. Surgical resection was performed after embolization. (Supplementary Spetzler–Martin grade 5: S2V1E0/A1B0C1) Case 3 (Fig.3) An 11-year-old boy suddenly developed headache accompanied by right limb weakness. Emergency hematoma evacuation was performed. Subsequently, two embolization procedures were conducted; however, residual BAVM persisted, necessitating surgical resection. (Supplementary Spetzler–Martin grade 5: S2V0E1/A1B0C1) Discussion Reports showed that approximately half (41/83) of children with ruptured AVM experienced severe hemorrhage, defined as an mRS score >3 or requiring emergency hematoma evacuation [1]. Additionally, 35.8% (38/106) developed coma after hemorrhage [2]. The mean hematoma volume was 30.4 cm³ (32.1 cm³ for supratentorial and 17.4 cm³ for infratentorial hemorrhage) [15]. Dofer [3] reported 56 cases of pediatric brain AVM, and evacuation of a space-occupying hematoma was required in the majority (55.5%). Therefore, many children with ruptured AVM required emergency decompressive surgery. For emergency surgery, it was strongly recommended not to address any vascular lesion encountered without prior angiographic evaluation [5]. Current literature indicated that emergency management of ruptured AVM consisting of hematoma evacuation alone or simple DC was safe, and the rate of AVM re-rupture during this interval was relatively low [3–6]. Beecher [6] reported 102 cases of ruptured AVM treated after a 4-week delay, with 6 cases of re-rupture, and proposed that the re-rupture rate was low (<1%) during the 4-week interval. Barone [5] reported that outcomes were better in patients who underwent surgery in the subacute phase, defined as ≥48 hours after rupture, compared with those treated in the acute phase. The author defined the period between 5 and 28 days after hemorrhage as the “subacute” phase, during which the hematoma liquefied and surgical conditions were optimal. At present, most authors preferred delayed AVM treatment, which might have offered several advantages. Hematoma could have obscured the surgical field and radiographic visualization of the AVM, thereby affecting complete resection. Delayed surgery after hematoma absorption might have reduced the risk of residual AVM. Emergency surgery during the acute phase, when edema surrounded the hematoma, might have increased brain injury. Delayed AVM resection after simple hematoma evacuation or DC provided a recovery period and avoided consecutive brain injury. A liquefying hematoma might have offered a surgical corridor to deep AVMs or facilitated deep nidus dissection. In addition, preoperative embolization could have been planned if necessary [4–6]. Regarding cranioplasty, most studies based on traumatic brain injury (TBI) supported early reconstruction [16–17]. Frassanito [16] proposed that early cranioplasty offered several advantages, including easier dissection of tissue planes, prevention of negative post-craniectomy sequelae (trephined syndrome and sinking skin flap syndrome), and, in young children, a greater likelihood of restoring symmetrical skull growth. The author’s policy was to reconstruct the skull 2–3 weeks after DC. Ozoner [17] suggested that early cranioplasty had the potential to enhance neurological recovery after severe TBI. Early improvement in CSF distribution and absorption presumably reduced the risk of hydrocephalus, and increased cerebral perfusion after cranioplasty was another plausible factor contributing to neurocognitive recovery. Based on a literature review, the earliest feasible timing for cranioplasty was approximately 34 days after DC. However, cranioplasty should have been performed only after AVM cure was confirmed. Otherwise, re-rupture of residual AVM after cranioplasty would have made subsequent treatment more complicated. After AVM rupture, the risk of re-rupture during the first year was approximately 6%–15.8%, decreasing to 2%–7.9% annually thereafter [5–6]. Untreated AVM diagnosed in childhood carried a lifelong rupture risk. Therefore, pediatric AVM should have been managed proactively, with cure as the ultimate goal. Among the three treatment modalities, the cure rate of endovascular embolization alone remained relatively low, with recent studies reporting rates of 46%–91% [18–19], and recurrence was frequent. Lauzer [20] reported a recurrence rate of 36.4% in limited embolization cases. Complete obliteration rates after SRS were 36% at 2 years, 46%–84% at 3 years, and 51%–95% at 5 years post-irradiation [21–24]. Although the cure rate was acceptable, obliteration required a prolonged period, during which rupture risk persisted. Waiting for obliteration after SRS would have delayed the optimal timing for cranioplasty. Microsurgical AVM resection provided the highest cure rate, with recent studies reporting rates of 85%–99% [8–12]. Therefore, the preferred strategy for children with residual AVM after DC might have been AVM resection with simultaneous cranioplasty. The optimal interval between DC and AVM resection remained uncertain. A prolonged interval might have increased the risk of re-rupture, whereas delayed cranioplasty could have resulted in extensive dural calcification and angulation of the bone window edge, thereby affecting reconstruction. Conversely, an excessively short interval might have caused consecutive brain injury and hindered functional recovery. In our series, the interval ranged from 1 to 25 months, and in most cases (11/15, 73.3%), it was 3–6 months. Based on the literature and our findings, an interval of 1–6 months might have been reasonable. A particular consideration in this surgery was the relationship between AVM location and the DC bone window. In our series, 4 cases had AVMs located distant from the initial bone window. The initial DC procedures were performed at other hospitals, where DSA might have been unavailable in the emergency setting. This resulted in unclear AVM characteristics and an inappropriate bone window range in these cases. Elongation of the incision and enlargement of the bone window were therefore required. Three-dimensional DSA with fusion imaging of the skull and AVM (Fig 3, image D), or fusion imaging of CT and MRI (Fig 1, image D), assisted by an electrode patch placed on the skin (Fig 1, image E), allowed more accurate localization of the lesion and its projection. This approach reduced the extent of dural opening and minimized injury during separation of adhesions between the dura and underlying brain tissue. In addition, careful attention was required when opening the dura to avoid injury to the superficial draining vein (Fig 3, image G). A hybrid operating room allowed simultaneous open surgery and endovascular procedures, offering significant advantages in AVM management. Its primary benefit was the ability to detect residual AVM using intraoperative DSA, thereby ensuring complete resection and preventing residual lesions. AVMs with higher Spetzler–Martin grades, eloquent location, complex angioarchitecture, diffuse morphology, or prior surgery were more challenging to resect and more prone to residual disease. Such cases were particularly suitable for management in a hybrid operating room [25,26]. In our series, 11 cases (73.3%) had diffuse AVMs and 9 cases (60%) were located in eloquent areas, which increased surgical difficulty. Residual AVM was detected on the first intraoperative DSA in 3 cases (20%). Without a hybrid operating room, the recurrence rate might have been higher. Therefore, for resection of residual AVM after DC, a hybrid operating room might have been preferable. This study had several limitations. It was retrospective in design, and the initial surgeries were performed at other hospitals. The clinical status at onset was not fully documented, which might have affected outcomes. In addition, the follow-up duration was relatively short. Previous reports indicated that pediatric AVM could recur more than 10 years after surgery [27]. Therefore, long-term follow-up was necessary. Conclusion Brain AVM was the most common cause of spontaneous cerebral hemorrhage in children. Many children with ruptured AVM required emergency decompressive surgery. After stabilization, cranioplasty should have been performed as early as appropriate to facilitate functional recovery. However, cranioplasty should have been undertaken only after confirmation of AVM cure. Microsurgical AVM resection, which provided the highest cure rate, combined with simultaneous cranioplasty, might have been the optimal treatment strategy for children with residual AVM after DC. Declarations Funding This research is funded by “The first batch of tasks (specifically assigned) for the scientific and technological innovation special program of XiongAn New Area in 2023 (2023XAGG0072)” Conflicts of interest The authors declare that they have no competing interests. Availability of data and material The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request. Code availability Not applicable Authors' contributions PS: Conceptualization, Methodology, Investigation, Writing Original Draft Preparation, Writing Review and Editing. MZ: Data Curation. YTL: Software. JD: Supervision. GZ: Conceptualization, Writing Review and Editing, Supervision. All authors have read and agreed to the published version of the manuscript. Ethics approval The present study was approved by the Ethics Committee of Xuanwu Hospital, Capital Medical University. All procedures were performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments Consent to participate Written informed consent was obtained from the patients and their parents. Consent for publication Not applicable References Ma L, Chen XL, Chen Y, Wu CX, Ma J, Zhao YL (2017) Subsequent haemorrhage in children with untreated brain arteriovenous malformation: Higher risk with unbalanced inflow and outflow angioarchitecture. Eur Radiol 27(7):2868–2876. 10.1007/s00330-016-4645-3 Blauwblomme T, Bourgeois M, Meyer P et al (2014) Long-term outcome of 106 consecutive pediatric ruptured brain arteriovenous malformations after combined treatment. Stroke 45(6):1664–1671. 10.1161/STROKEAHA.113.004292 Dorfer C, Czech T, Bavinzski G et al (2010) Multimodality treatment of cerebral AVMs in children: a single-centre 20 years experience. 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Acta Neurochir (Wien) 165(6):1565–1573. 10.1007/s00701-023-05612-8 Additional Declarations No competing interests reported. Cite Share Download PDF Status: Under Review Version 1 posted Reviewers invited by journal 09 Apr, 2026 Editor assigned by journal 05 Mar, 2026 Submission checks completed at journal 05 Mar, 2026 First submitted to journal 03 Mar, 2026 You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-9016262","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":620341091,"identity":"ce7fab43-9c64-4fb8-9a5a-27a9442e360a","order_by":0,"name":"Peng SUN","email":"","orcid":"","institution":"Capital Medical University","correspondingAuthor":false,"prefix":"","firstName":"Peng","middleName":"","lastName":"SUN","suffix":""},{"id":620341099,"identity":"4d6af43e-efc4-4e7e-8205-83db361cfd6b","order_by":1,"name":"Yutong LIU","email":"","orcid":"","institution":"Capital Medical University","correspondingAuthor":false,"prefix":"","firstName":"Yutong","middleName":"","lastName":"LIU","suffix":""},{"id":620341101,"identity":"f37697bf-919a-4654-a046-1bda144bf694","order_by":2,"name":"Mading ZHOU","email":"","orcid":"","institution":"Capital Medical University","correspondingAuthor":false,"prefix":"","firstName":"Mading","middleName":"","lastName":"ZHOU","suffix":""},{"id":620341102,"identity":"111f36f9-b060-4a90-bd63-895ab4e8dffa","order_by":3,"name":"Jianxin DU","email":"","orcid":"","institution":"Capital Medical University","correspondingAuthor":false,"prefix":"","firstName":"Jianxin","middleName":"","lastName":"DU","suffix":""},{"id":620341108,"identity":"2605e732-349f-47ef-a380-bfb565f8365b","order_by":4,"name":"Gao ZENG","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA0klEQVRIiWNgGAWjYHAD5gMHPvwgTQtb4sGZPaRp4TE+zMFGhDr59t7DrysqauXk+898OMzAwyDPL3YAvxbGnnNplmfOHDc2uJG74XCBBYPhzNkJ+LUwS+SYGTa2HUvcIMG74fAMHoYEg9sEtLDJv4Fomd9/5sFhHjYitPBI8Bg/bGyrSWw4kMNAnBYJnhwzxoYzB4B+STMABrIEYb/It58x/thQUQcMscOPP3z4YSPPL01AC8g7EgwMh+G2ElQOAswfGBjqiFI5CkbBKBgFIxQAAJAGRsa7T/MMAAAAAElFTkSuQmCC","orcid":"","institution":"Capital Medical University","correspondingAuthor":true,"prefix":"","firstName":"Gao","middleName":"","lastName":"ZENG","suffix":""}],"badges":[],"createdAt":"2026-03-03 05:53:13","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-9016262/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-9016262/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":107254723,"identity":"b700c79a-7f93-4ae6-81b9-664f107e9882","added_by":"auto","created_at":"2026-04-19 12:05:14","extension":"jpg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":1121658,"visible":true,"origin":"","legend":"\u003cp\u003eA. CT indicated right occipital lobe hemorrhage. B. Drainage vein (arrow) visible on the contrast MRI. C. DSA indicated right occipital lobe AVM (arrow indicated drainage vein). D. Fusion image of CT and MRI showed the AVM located away from the bone window. E. The projection position of the AVM was confirmed by DSA in the hybrid operating room with the help of an electrode patch on the skin (red arrow). F. Surgical incision (arrow) refers to the projection position of the AVM. G. DSA performed immediately after surgery in the hybrid operating room indicated the AVM was completely resected. H. Cranioplasty with PEEK and new bone flap. I. CT reconstruction 1 year post surgery. J. Contrast MRI 2 years post surgery showed no recurrence.\u003c/p\u003e","description":"","filename":"case1.jpg","url":"https://assets-eu.researchsquare.com/files/rs-9016262/v1/c9ec5627e14017ba92aea6f9.jpg"},{"id":107254725,"identity":"d0546aa0-7d3e-4717-82c6-5e2f5fb099ef","added_by":"auto","created_at":"2026-04-19 12:05:14","extension":"jpg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":900498,"visible":true,"origin":"","legend":"\u003cp\u003eA. CT indicated right frontal lobe hemorrhage. B, C. Anteroposterior and lateral view of DSA indicated right frontal AVM with transit feeding arteries (arrow) from the right middle cerebral artery (MCA). D, E. DSA after embolization of AVM. F. Cast of embolization agent (arrow) indicated the boundary of the AVM and the AVM located beneath the bone window. G. Contrast MRI showed AVM (red arrow) and small amount of hematoma (white arrow). H. The feeding artery was occluded by an aneurysm clip (arrow). I, J. DSA performed immediately after surgery in the hybrid operating room indicated the AVM was completely resected. K. CT reconstruction 3 months post surgery showed little absorption of autologous bone flap. L. Contrast MRI 4 years post surgery showed no recurrence.\u003c/p\u003e","description":"","filename":"case2.jpg","url":"https://assets-eu.researchsquare.com/files/rs-9016262/v1/0857a27020ebc3d63c1551f2.jpg"},{"id":107254724,"identity":"0eb8c20f-0f03-4221-8af9-e51b48e0530f","added_by":"auto","created_at":"2026-04-19 12:05:14","extension":"jpg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":956238,"visible":true,"origin":"","legend":"\u003cp\u003eA. CT indicated left parietal lobe hemorrhage. B. DSA indicated left parietal lobe AVM. C. DSA after embolization indicated residual AVM. D. Contrast MRI showed AVM (arrow). E. DTI showed the pyramid tract (pink) located anterior-medially to the AVM (arrow). F. 3D DSA with fusion of skull images showed the residual AVM and the drainage vein (red arrow) located behind the embolization agent. G. Image during surgery showed the drainage vein (black arrow) and the embolization agent (white arrow). H. DSA performed immediately after surgery in the hybrid operating room indicated the AVM was completely resected. I. Non-subtractive image showed residual embolization agent (arrow). J. Contrast MRI 1 year post surgery showed no recurrence.\u003c/p\u003e","description":"","filename":"case3.jpg","url":"https://assets-eu.researchsquare.com/files/rs-9016262/v1/7b7b4ec57fadbb51cf471675.jpg"},{"id":107483070,"identity":"712905fd-6297-446b-a0a2-a5a09c99832b","added_by":"auto","created_at":"2026-04-22 02:26:09","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":3125296,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-9016262/v1/b7a58da4-2bc2-4257-951f-6448fbd7d267.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Pediatric brain AVM resection with simultaneous cranioplasty in hybrid operating room","fulltext":[{"header":"Introduction","content":"\u003cp\u003eBrain arteriovenous malformation (AVM) was characterized by a tangle of dysplastic vessels (nidus) fed by arteries and drained by veins without intervening capillaries, forming a high-flow, low-resistance shunt between the arterial and venous systems. AVM was the most common cause of spontaneous cerebral hemorrhage in children. Approximately half (41/83) of children with ruptured AVM experienced severe hemorrhage, defined as a modified Rankin Scale (mRS) score \u0026gt;3 or requiring emergency hematoma evacuation [1]. Additionally, 35.8% (38/106) developed coma after hemorrhage [2]. Therefore, many children with ruptured AVM required emergency decompressive surgery.\u003c/p\u003e\n\u003cp\u003eSome reports showed that hematoma evacuation or decompressive craniectomy (DC) without AVM resection was acceptable, as the incidence of AVM re-rupture before curative treatment was low [3–6]. For children who underwent DC, cranioplasty should have been performed as early as appropriate [7]. However, cranioplasty should have been undertaken only after AVM cure was confirmed. Otherwise, re-rupture of residual AVM after cranioplasty would have made subsequent treatment more complicated. Among available treatment modalities, microsurgical resection offered the highest cure rate, with recent studies reporting rates of 85%–99% [8–12].\u003c/p\u003e\n\u003cp\u003eReports focusing on the management of residual AVM after DC were rare. In our opinion, the optimal strategy in this situation might have been AVM resection with simultaneous cranioplasty in a hybrid operating room.\u003c/p\u003e\n\u003cp\u003eWe reviewed children who underwent AVM resection with simultaneous cranioplasty at our hospital to summarize treatment strategies and provide recommendations regarding surgical details.\u003c/p\u003e"},{"header":"Methods","content":"\u003cp\u003eWe reviewed children who underwent brain AVM resection with simultaneous cranioplasty in a hybrid operating room between 2016 and 2025. Data on clinical presentation, angioarchitectural characteristics, management, and outcomes were collected. All included cases had a preoperative DSA-confirmed intracranial AVM. The Spetzler–Martin grade [13] and supplementary grading scale [14] were applied.\u003c/p\u003e\n\u003cp\u003eAll patients underwent surgery in a DSA hybrid operating room. Cranioplasty was performed after complete AVM resection was confirmed by intraoperative DSA.\u003c/p\u003e\n\u003cp\u003ePostoperative neurological deficits were evaluated in all patients, and outcome was assessed using the mRS; a score \u0026lt;2 was defined as a good outcome.\u003c/p\u003e\n\u003cp\u003eImaging follow-up was conducted with contrast-enhanced MRI or CTA at 6 months, 1 year, and annually thereafter. DSA was performed only if noninvasive imaging suggested AVM recurrence.\u003c/p\u003e"},{"header":"Results","content":"\u003cp\u003eBetween 2016 and 2025, 15 cases of AVM resection with simultaneous cranioplasty were performed at this center in a hybrid operating room. There were 8 males and 7 females, with a mean age at onset of 9.7 years (range, 4–15 years).\u003c/p\u003e\n\u003cp\u003eAll patients presented with ruptured BAVM and consciousness disturbance. Emergency hematoma evacuation and DC were performed at other hospitals. The mean interval between DC and AVM resection was 5.3 months (range, 1–25 months). No re-rupture occurred during this interval. The mean preoperative mRS score was 1.8.\u003c/p\u003e\n\u003cp\u003eResidual AVM was confirmed by DSA in all patients. AVMs were located in the cerebral cortical hemispheres (frontal, parietal, occipital, or temporal lobes) in 11 cases, in the basal ganglia in 3 cases, and in the cerebellum in 1 case. In 4 cases, the AVM was distant from the initial bone window (Fig1, 2), requiring extension of the incision and enlargement of the bone window.\u003c/p\u003e\n\u003cp\u003eSpetzler–Martin grades were 1–2 in 7 cases (46.7%), 3 in 6 cases (40.0%), and 4 in 2 cases (13.3%). Supplementary Spetzler–Martin grades were 2–3 in 4 cases (26.7%) and 4–6 in 11 cases (73.3%).\u003c/p\u003e\n\u003cp\u003ePreoperative embolization was performed in 4 cases. Complete AVM resection was confirmed in all patients by intraoperative DSA in the hybrid operating room. Residual AVM was detected on the first intraoperative DSA in 3 cases (20%). Further exploration and resection were undertaken, and repeat DSA confirmed no residual lesion in these cases. Cranioplasty materials included titanium mesh in 8 cases, PEEK in 5 cases, and autologous bone flap in 2 cases.\u003c/p\u003e\n\u003cp\u003eComplications included suspected intracranial infection in 4 cases, although CSF cultures were negative in all cases. No wound healing complications or subcutaneous effusion occurred. No new neurological deficits were observed.\u003c/p\u003e\n\u003cp\u003eThe mean clinical follow-up duration was 50.6 months (range, 8.6–122.1 months) in 14 patients, as 1 patient was lost to follow-up. Ten patients (71.4%) achieved good outcomes, and the mean mRS score at the last follow-up was 1.1. The mean imaging follow-up duration was 25.7 months (range, 6.5–62.6 months), including MRI or CTA in 11 cases and DSA in 3 cases. AVM recurrence was confirmed by DSA in 1 patient 17 months after surgery. She underwent stereotactic radiotherapy (SRS) and experienced no re-rupture during 13 months of follow-up.\u003c/p\u003e\n\u003cp\u003eIllustrative cases\u003c/p\u003e\n\u003cp\u003eCase 1 (Fig.1)\u003c/p\u003e\n\u003cp\u003eA 10-year-old boy suddenly developed headache accompanied by consciousness disturbance and underwent emergency hematoma evacuation and DC. (Supplementary Spetzler–Martin grade 2: S1V0E1/A1B0C1)\u003c/p\u003e\n\u003cp\u003eCase 2 (Fig.2)\u003c/p\u003e\n\u003cp\u003eAn 11-year-old girl suddenly developed headache with consciousness disturbance and underwent emergency hematoma evacuation and DC. Surgical resection was performed after embolization. (Supplementary Spetzler–Martin grade 5: S2V1E0/A1B0C1)\u003c/p\u003e\n\u003cp\u003eCase 3 (Fig.3)\u003c/p\u003e\n\u003cp\u003eAn 11-year-old boy suddenly developed headache accompanied by right limb weakness. Emergency hematoma evacuation was performed. Subsequently, two embolization procedures were conducted; however, residual BAVM persisted, necessitating surgical resection. (Supplementary Spetzler–Martin grade 5: S2V0E1/A1B0C1)\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eReports showed that approximately half (41/83) of children with ruptured AVM experienced severe hemorrhage, defined as an mRS score \u0026gt;3 or requiring emergency hematoma evacuation [1]. Additionally, 35.8% (38/106) developed coma after hemorrhage [2]. The mean hematoma volume was 30.4 cm³ (32.1 cm³ for supratentorial and 17.4 cm³ for infratentorial hemorrhage) [15]. Dofer [3] reported 56 cases of pediatric brain AVM, and evacuation of a space-occupying hematoma was required in the majority (55.5%). Therefore, many children with ruptured AVM required emergency decompressive surgery.\u003c/p\u003e\n\u003cp\u003eFor emergency surgery, it was strongly recommended not to address any vascular lesion encountered without prior angiographic evaluation [5]. Current literature indicated that emergency management of ruptured AVM consisting of hematoma evacuation alone or simple DC was safe, and the rate of AVM re-rupture during this interval was relatively low [3–6]. Beecher [6] reported 102 cases of ruptured AVM treated after a 4-week delay, with 6 cases of re-rupture, and proposed that the re-rupture rate was low (\u0026lt;1%) during the 4-week interval. Barone [5] reported that outcomes were better in patients who underwent surgery in the subacute phase, defined as ≥48 hours after rupture, compared with those treated in the acute phase. The author defined the period between 5 and 28 days after hemorrhage as the “subacute” phase, during which the hematoma liquefied and surgical conditions were optimal.\u003c/p\u003e\n\u003cp\u003eAt present, most authors preferred delayed AVM treatment, which might have offered several advantages. Hematoma could have obscured the surgical field and radiographic visualization of the AVM, thereby affecting complete resection. Delayed surgery after hematoma absorption might have reduced the risk of residual AVM. Emergency surgery during the acute phase, when edema surrounded the hematoma, might have increased brain injury. Delayed AVM resection after simple hematoma evacuation or DC provided a recovery period and avoided consecutive brain injury. A liquefying hematoma might have offered a surgical corridor to deep AVMs or facilitated deep nidus dissection. In addition, preoperative embolization could have been planned if necessary [4–6].\u003c/p\u003e\n\u003cp\u003eRegarding cranioplasty, most studies based on traumatic brain injury (TBI) supported early reconstruction [16–17]. Frassanito [16] proposed that early cranioplasty offered several advantages, including easier dissection of tissue planes, prevention of negative post-craniectomy sequelae (trephined syndrome and sinking skin flap syndrome), and, in young children, a greater likelihood of restoring symmetrical skull growth. The author’s policy was to reconstruct the skull 2–3 weeks after DC. Ozoner [17] suggested that early cranioplasty had the potential to enhance neurological recovery after severe TBI. Early improvement in CSF distribution and absorption presumably reduced the risk of hydrocephalus, and increased cerebral perfusion after cranioplasty was another plausible factor contributing to neurocognitive recovery. Based on a literature review, the earliest feasible timing for cranioplasty was approximately 34 days after DC.\u003c/p\u003e\n\u003cp\u003eHowever, cranioplasty should have been performed only after AVM cure was confirmed. Otherwise, re-rupture of residual AVM after cranioplasty would have made subsequent treatment more complicated. After AVM rupture, the risk of re-rupture during the first year was approximately 6%–15.8%, decreasing to 2%–7.9% annually thereafter [5–6]. Untreated AVM diagnosed in childhood carried a lifelong rupture risk. Therefore, pediatric AVM should have been managed proactively, with cure as the ultimate goal. Among the three treatment modalities, the cure rate of endovascular embolization alone remained relatively low, with recent studies reporting rates of 46%–91% [18–19], and recurrence was frequent. Lauzer [20] reported a recurrence rate of 36.4% in limited embolization cases. Complete obliteration rates after SRS were 36% at 2 years, 46%–84% at 3 years, and 51%–95% at 5 years post-irradiation [21–24]. Although the cure rate was acceptable, obliteration required a prolonged period, during which rupture risk persisted. Waiting for obliteration after SRS would have delayed the optimal timing for cranioplasty. Microsurgical AVM resection provided the highest cure rate, with recent studies reporting rates of 85%–99% [8–12]. Therefore, the preferred strategy for children with residual AVM after DC might have been AVM resection with simultaneous cranioplasty.\u003c/p\u003e\n\u003cp\u003eThe optimal interval between DC and AVM resection remained uncertain. A prolonged interval might have increased the risk of re-rupture, whereas delayed cranioplasty could have resulted in extensive dural calcification and angulation of the bone window edge, thereby affecting reconstruction. Conversely, an excessively short interval might have caused consecutive brain injury and hindered functional recovery. In our series, the interval ranged from 1 to 25 months, and in most cases (11/15, 73.3%), it was 3–6 months. Based on the literature and our findings, an interval of 1–6 months might have been reasonable.\u003c/p\u003e\n\u003cp\u003eA particular consideration in this surgery was the relationship between AVM location and the DC bone window. In our series, 4 cases had AVMs located distant from the initial bone window. The initial DC procedures were performed at other hospitals, where DSA might have been unavailable in the emergency setting. This resulted in unclear AVM characteristics and an inappropriate bone window range in these cases. Elongation of the incision and enlargement of the bone window were therefore required. Three-dimensional DSA with fusion imaging of the skull and AVM (Fig 3, image D), or fusion imaging of CT and MRI (Fig 1, image D), assisted by an electrode patch placed on the skin (Fig 1, image E), allowed more accurate localization of the lesion and its projection. This approach reduced the extent of dural opening and minimized injury during separation of adhesions between the dura and underlying brain tissue. In addition, careful attention was required when opening the dura to avoid injury to the superficial draining vein (Fig 3, image G).\u003c/p\u003e\n\u003cp\u003eA hybrid operating room allowed simultaneous open surgery and endovascular procedures, offering significant advantages in AVM management. Its primary benefit was the ability to detect residual AVM using intraoperative DSA, thereby ensuring complete resection and preventing residual lesions. AVMs with higher Spetzler–Martin grades, eloquent location, complex angioarchitecture, diffuse morphology, or prior surgery were more challenging to resect and more prone to residual disease. Such cases were particularly suitable for management in a hybrid operating room [25,26]. In our series, 11 cases (73.3%) had diffuse AVMs and 9 cases (60%) were located in eloquent areas, which increased surgical difficulty. Residual AVM was detected on the first intraoperative DSA in 3 cases (20%). Without a hybrid operating room, the recurrence rate might have been higher. Therefore, for resection of residual AVM after DC, a hybrid operating room might have been preferable.\u003c/p\u003e\n\u003cp\u003eThis study had several limitations. It was retrospective in design, and the initial surgeries were performed at other hospitals. The clinical status at onset was not fully documented, which might have affected outcomes. In addition, the follow-up duration was relatively short. Previous reports indicated that pediatric AVM could recur more than 10 years after surgery [27]. Therefore, long-term follow-up was necessary.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eBrain AVM was the most common cause of spontaneous cerebral hemorrhage in children. Many children with ruptured AVM required emergency decompressive surgery. After stabilization, cranioplasty should have been performed as early as appropriate to facilitate functional recovery. However, cranioplasty should have been undertaken only after confirmation of AVM cure. Microsurgical AVM resection, which provided the highest cure rate, combined with simultaneous cranioplasty, might have been the optimal treatment strategy for children with residual AVM after DC.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003eFunding\u003c/p\u003e\n\u003cp\u003eThis research is funded by “The first batch of tasks (specifically assigned) for the scientific and technological innovation special program of XiongAn New Area in 2023 (2023XAGG0072)”\u003c/p\u003e\n\u003cp\u003eConflicts of interest\u003c/p\u003e\n\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e\n\u003cp\u003eAvailability of data and material\u003c/p\u003e\n\u003cp\u003eThe datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.\u003c/p\u003e\n\u003cp\u003eCode availability\u003c/p\u003e\n\u003cp\u003eNot applicable\u003c/p\u003e\n\u003cp\u003eAuthors' contributions\u003c/p\u003e\n\u003cp\u003ePS: Conceptualization, Methodology, Investigation, Writing Original Draft Preparation, Writing Review and Editing. MZ: Data Curation. YTL: Software. JD: Supervision. GZ: Conceptualization, Writing Review and Editing, Supervision. All authors have read and agreed to the published version of the manuscript.\u003c/p\u003e\n\u003cp\u003eEthics approval\u003c/p\u003e\n\u003cp\u003eThe present study was approved by the Ethics Committee of Xuanwu Hospital, Capital Medical University. All procedures were performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments\u003c/p\u003e\n\u003cp\u003eConsent to participate\u003c/p\u003e\n\u003cp\u003eWritten informed consent was obtained from the patients and their parents.\u003c/p\u003e\n\u003cp\u003eConsent for publication\u003c/p\u003e\n\u003cp\u003eNot applicable\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eMa L, Chen XL, Chen Y, Wu CX, Ma J, Zhao YL (2017) Subsequent haemorrhage in children with untreated brain arteriovenous malformation: Higher risk with unbalanced inflow and outflow angioarchitecture. 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Acta Neurochir (Wien) 165(6):1565\u0026ndash;1573. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.1007/s00701-023-05612-8\u003c/span\u003e\u003cspan address=\"10.1007/s00701-023-05612-8\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"childs-nervous-system","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"cnsy","sideBox":"Learn more about [Child's Nervous System](http://link.springer.com/journal/381)","snPcode":"381","submissionUrl":"https://submission.nature.com/new-submission/381/3","title":"Child's Nervous System","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false},"keywords":"brain arteriovenous malformation, cranioplasty, microsurgery resection, hybrid operating room","lastPublishedDoi":"10.21203/rs.3.rs-9016262/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-9016262/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003ePurpose\u003c/h2\u003e \u003cp\u003eBrain arteriovenous malformation (AVM) was the most common cause of spontaneous cerebral hemorrhage in children. Many children with ruptured AVM required emergency decompressive craniectomy (DC). Reports focusing on the management of residual AVM after DC were rare. We reviewed children who underwent AVM resection with simultaneous cranioplasty to summarize treatment strategies.\u003c/p\u003e\u003ch2\u003eMethod\u003c/h2\u003e \u003cp\u003eWe reviewed children who underwent brain AVM resection with simultaneous cranioplasty in a hybrid operating room between 2016 and 2025.\u003c/p\u003e\u003ch2\u003eResults\u003c/h2\u003e \u003cp\u003eBetween 2016 and 2025, 15 cases of AVM resection with simultaneous cranioplasty were performed at this center in a hybrid operating room. The mean interval between DC and AVM resection was 5.3 months (range, 1\u0026ndash;25 months). The mean mRS score was 1.8 before AVM resection. Complete AVM resection was confirmed in all patients by intraoperative DSA in the hybrid operating room. Cranioplasty materials included titanium mesh in 8 cases, PEEK in 5 cases, and autologous bone flap in 2 cases. The mean follow-up duration was 50.6 months (range, 8.6\u0026ndash;122.1 months) in 14 patients, as 1 patient was lost to follow-up. Ten patients (10/14, 71.4%) achieved good outcomes, and the mean mRS score at the last follow-up was 1.1. AVM recurrence was confirmed by DSA in 1 patient 17 months after surgery.\u003c/p\u003e\u003ch2\u003eConclusion\u003c/h2\u003e \u003cp\u003eMicrosurgical resection of AVM, which provided the highest cure rate, combined with simultaneous cranioplasty, might have been an ideal treatment modality for children with residual AVM after DC.\u003c/p\u003e","manuscriptTitle":"Pediatric brain AVM resection with simultaneous cranioplasty in hybrid operating room","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2026-04-19 12:05:10","doi":"10.21203/rs.3.rs-9016262/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"reviewersInvited","content":"","date":"2026-04-09T10:29:09+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2026-03-06T04:33:01+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2026-03-06T04:29:02+00:00","index":"","fulltext":""},{"type":"submitted","content":"Child's Nervous System","date":"2026-03-03T05:36:53+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"childs-nervous-system","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"cnsy","sideBox":"Learn more about [Child's Nervous System](http://link.springer.com/journal/381)","snPcode":"381","submissionUrl":"https://submission.nature.com/new-submission/381/3","title":"Child's Nervous System","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false}}],"origin":"","ownerIdentity":"b163e054-a474-46f0-ab39-b939ab88d61d","owner":[],"postedDate":"April 19th, 2026","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2026-04-19T12:05:10+00:00","versionOfRecord":[],"versionCreatedAt":"2026-04-19 12:05:10","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-9016262","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-9016262","identity":"rs-9016262","version":["v1"]},"buildId":"XKTyCvWXoU3ODBz1xrDgd","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}