brain sciences Case Report Preoperative Devascularization of Choroid Plexus Tumors: Specific Issues about Anatomy and Embolization Technique Valentina Baro 1,*, Joseph Domenico Gabrieli 2, Giacomo Cester 2 , Ignazio D’Errico 2, Andrea Landi 1 , Luca Denaro 1 and Francesco Causin 2 1 Academic Neurosurgery, Department of Neuroscience, University of Padova, 35100 Padova, Italy; [email protected] (A.L.); [email protected] (L.D.) 2 Neuroradiology Unit, University of Padova, 35100 Padova, Italy; [email protected] (J.D.G.); [email protected] (G.C.); [email protected] (I.D.); [email protected] (F.C.) * Correspondence: [email protected] Abstract: (1) Background: Surgical treatment of choroid plexus tumors is challenging, burdened by a notable risk of bleeding. Neoadjuvant chemotherapy and preoperative embolization have been attempted, with encouraging results; however, the consensus on these procedures is lacking. (2) Methods: We present a case of a 10-month-old girl who underwent preoperative embolization of a hemorrhagic choroid plexus carcinoma of the lateral ventricle via the anterior choroidal artery, followed by total resection. (3) Results: The endovascular procedure was successfully completed, despite the rectification of the anterior choroidal artery associated with the absence of flow proximal to the plexal point. Minimal bleeding was observed during resection and the patient remained neurologically intact. (4) Conclusions: The time from entrance to exit in the anterior choroidal artery Citation: Baro, V.; Gabrieli, J.D.; Cester, G.; D’Errico, I.; Landi, A.; should be monitored and regarded as a potential ‘occlusion time’ in this specific group of patients. Denaro, L.; Causin, F. Preoperative Nevertheless, our case supports the feasibility and effectiveness of preoperative embolization of a Devascularization of Choroid Plexus choroid plexus carcinoma of the lateral ventricle via the anterior choroidal artery, without complica- Tumors: Specific Issues about tions. Furthermore, we suggest the use of a fast-embolic agent, such as N-butyl cyanoacrylate glue, Anatomy and Embolization as the preferred agent for this specific pathology and patient population. Technique. Brain Sci. 2021, 11, 540. https://doi.org/10.3390/ Keywords: tumor embolization; pediatric neurosurgery; pediatric brain tumor brainsci11050540 Academic Editor: Helen L. Fillmore 1. Introduction Received: 15 March 2021 Choroid plexus tumors (CPT) are rare neoplasms of neuro-ectodermal origin, account- Accepted: 23 April 2021 Published: 25 April 2021 ing for less than 1% of all brain tumors and 1%–4% of all pediatric brain tumors. Children younger than 2 years of age are mostly affected, and the incidence of the choroid plexus Publisher’s Note: MDPI stays neutral carcinoma (CPC) is 20%–40% of CPT in patients under 18 months of age [1–3]. Choroid with regard to jurisdictional claims in plexus papilloma (CPP) is the benign form of these tumors—total resection is curative published maps and institutional affil- without adjuvant chemotherapy—confirmed by a 5-year overall survival of 100% [2]. In iations. contrast, the complete resection of CPC is often hindered by the hypervascularity of the lesion and the usual invasion of the contiguous brain parenchyma, leading to a high rate of subtotal resection and recurrences with a 5-year overall survival of 27%, despite adjuvant treatments [1,4,5]. The mortality rate is predominantly due to intraoperative significant blood loss in children with limited circulating volume, and it has been reported to be as Copyright: © 2021 by the authors. high as 13.3% [4,6]. Licensee MDPI, Basel, Switzerland. This article is an open access article The use of neoadjuvant chemotherapy in CPC has been reported as an approach distributed under the terms and that leads to a decrease in blood loss in patients undergoing surgery [7]. Preoperative conditions of the Creative Commons embolization, albeit limited to a small number of patients, has been attempted since the end Attribution (CC BY) license (https:// of the 90s, with encouraging results in terms of reducing blood loss, which, in turn, reduces creativecommons.org/licenses/by/ morbidity and mortality and increases the likelihood of total resection (Supplementary 4.0/). Table S1) [4,8–15]. However, the small and tortuous vasculature of small children, and the Brain Sci. 2021, 11, 540. https://doi.org/10.3390/brainsci11050540 https://www.mdpi.com/journal/brainsci BrainBrain Sci. Sci.2021 2021, 11,, 11 540, x FOR PEER REVIEW 2 of 28 of 8 typicalTable S1) involvement [4,8–15]. However, of eloquent the small arteries, and representtortuous vasculature a significant of small hindrance. children, In thisand the paper, wetypical describe involvement the case ofof aeloquent 10-month-old arteries, patient repres withent a asignificant right lateral hindrance. ventricle In choroid this paper, plexus we describe the case of a 10-month-old patient with a right lateral ventricle choroid plexus carcinoma who underwent successful preoperative embolization. In addition, the relevant carcinoma who underwent successful preoperative embolization. In addition, the relevant literature is reviewed and discussed. literature is reviewed and discussed. 2. Case Description 2. Case Description A 10-month-old female with regular neurological development and no previous A 10-month-old female with regular neurological development and no previous medical history was admitted to our hospital for an acute onset of vomiting, followed medical history was admitted to our hospital for an acute onset of vomiting, followed by by a progressive deterioration of her neurological status, requiring tracheal intubation. a progressive deterioration of her neurological status, requiring tracheal intubation. The The computed tomography scan showed a large right intraventricular mass with recent computed tomography scan showed a large right intraventricular mass with recent bleed- bleeding associated with moderate midline shift. Magnetic resonance imaging (MRI) with ing associated with moderate midline shift. Magnetic resonance imaging (MRI) with con- contrast media and time-of-flight magnetic resonance angiography (ToF MRA) confirmed trast media and time-of-flight magnetic resonance angiography (ToF MRA) confirmed the the presence of a large, hemorrhagic, intensely enhancing intraventricular mass, centered in presence of a large, hemorrhagic, intensely enhancing intraventricular mass, centered in thethe atrium atrium of of the the right right lateral lateral ventricle, ventricle, without without invasion invasion of of the the adjacent adjacent brain brain parenchyma, paren- fedchyma, by a fed moderately by a moderately enlarged enlarged anterior anterior choroidal choroidal artery artery (Figure (Figure1). Whole1). Whole spine spine MRI excludedMRI excluded signs signs of cerebrospinal of cerebrospinal fluid fluid seeding. seeding. The patientThe patient was was scheduled scheduled for for the the following fol- daylowing to perform day to perform endovascular endovascular embolization embolization and surgical and surgical excision. excision. FigureFigure 1. (1.A ()A Gradient-echo) Gradient-echo image image shows shows intralesional, intralesional, perilesional perilesional and and intraventricular intraventricular hemorrhagichemorrhagic components.components. (B,C)) T2 axialT2 (axialB) and (B) Volume and Volume Rendering Rendering of ToF of MRAToF MRA (C) show(C) show the coursethe course of the of the anterior anterior choroidal choroidal artery. artery. The The shifted shifted white white line line represents, schematically, the course of the artery; dashed arrows indicate the exact location of the highlighted vessel. represents, schematically, the course of the artery; dashed arrows indicate the exact location of the highlighted vessel. Segment 1 is the cisternal portion of the artery which courses across the carotid, crural and ambient cysterns, up to the Segmentchoroidal 1 is fissure, the cisternal where it portion enters the of thevent arteryricle; the which asterisk courses * corresponds across the to carotid,the plexal crural point and that ambientmarks the cysterns, entrance upin the to the choroidalventricle, fissure, and segment where it2 is enters the intraventricular the ventricle; the segment. asterisk Segme * correspondsnt 3 is the tumoral to the plexal segment point of the that artery marks running the entrance along the in the ventricle,surface andof the segment tumor (see 2 is theFigure intraventricular 4 for the complete segment. anatom Segmentical description). 3 is the tumoral segment of the artery running along the surface of the tumor (see Figure 4 for the complete anatomical description). A preoperative three vessel angiography (bilateral internal carotid artery and domi- nantA vertebral preoperative artery) three confirmed vessel angiographythe presence of (bilateral a slightly internal enlarged carotid right artery anterior and cho- domi- nantroidal vertebral artery feeding artery) the confirmed tumoral the blush. presence No feeders of a slightly were observed enlarged from right the anterior posterior choroidal cir- arteryculation; feeding embolization the tumoral was blush. obtained No feederswith n-butyl were cyanoacrylate observed from (NBCA) the posterior superselective circulation; embolizationinjection (0.4 mL) was via obtained a microcatheter with n-butyl (Magic cyanoacrylate 1.2F-Balt, Montmorency, (NBCA) superselective France; aided injection