# Classical radiolucent lesion of the mandible in a child, uncommon diagnosis

Amaury Derquenne, Marine Anquetil, Hubert Desal, Hélios Bertin

► To cite this version:

Amaury Derquenne, Marine Anquetil, Hubert Desal, Hélios Bertin. Classical radiolucent lesion of the mandible in a child, uncommon diagnosis. Journal of Stomatology, Oral and Maxillofacial Surgery, 2024, 125 (4), pp.101932. 10.1016/j.jormas.2024.101932. hal-05460273

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Submitted on 15 Jan 2026

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Short Communication

# Classical radiolucent lesion of the mandible in a child, uncommon diagnosis

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CrossMark

Amaury Derquenne<sup>a,*</sup>, Marine Anquetil<sup>a</sup>, Hubert Desal<sup>b,c</sup>, Hélios Bertin<sup>a,d</sup>

ARTICLE INFO

Article History:

Received 15 May 2024

Accepted 29 May 2024

Available online 30 May 2024

A 11-year-old boy with no previous history was referred to our department of oral and maxillofacial surgery after two bleeding episodes from intraoral origin. The bleeding was nocturnal, unsolicited, of small volume, and with spontaneous resolution. Clinical examination revealed only mobility of the right mandibular second molar, with no mass syndrome and no mandibular or labiomental hypoesthesia. Hemodynamic parameters were stable, and the patient was pain-free. The initial cone-beam computed tomography (CBCT) showed a unique radiolucent blowing lesion of the right mandibular body and ramus extended over 52-mm long axis (Fig. 1A). Given the initial clinical situation, an injected cervico-facial CT-scan was carried out, confirming the voluminous mandibular lesion associated with contrast in the osteolytic cavity after iodine injection (Fig. 1B). What is your diagnosis?

Given the hypervascularization on the CT-scan, an arteriovenous malformation (AVM) with intra-osseous aneurysm of the right

inferior alveolar artery was suspected and confirmed by arteriography (Fig. 2). Embolization of the vascular malformation was performed by radial artery approach, with intralesional injection of ethylene vinyl alcohol copolymer (Onyx® 18) (MicroTherapeutics, Irvine CA) distal to the lower right alveolar artery (Fig. 3A). A control arteriogram was performed at 3 months showing no significant arteriovenous shunt or residual vascular anomaly (Fig. 3B). The postoperative period was marked by recurrent cellulitis in mandibular sector IV. Curettage of the embolization material was performed after a reassuring arteriogram at 3 months, showing no significant arteriovenous shunt or residual vascular anomaly. Given recurrent infectious episodes, curettage of the embolization material was performed at 6 months, allowing for ossification of the mandibular lesion and stabilization of the mandibular molars.

Mandibular AVM is a rare pathology, with just over a hundred cases described in the literature to date [1]. Due to an intra-osseous

<figure><img src="image_5.png" /><figcaption>**Fig. 1.** Initial radiographic presentation. Panoramic reconstruction from CBCT showing the voluminous blowing lesion of the right mandible extended from the lingula to the first lower molar (A). Injected cervicofacial CT scan showing pathological contrast enhancement within the bone lesion (B).</figcaption></figure>

<footnote><sup>a</sup> Nantes Université, CHU Nantes, Service de chirurgie maxillo-faciale et stomatologie, F-44000 Nantes, France</footnote>

<footnote><sup>b</sup> Nantes Université, CHU Nantes, Neuroradiologie diagnostique et interventionnelle, F-44000 Nantes, France</footnote>

<footnote><sup>c</sup> Nantes Université, INSERM UMR 1087, Institut du thorax, F-44000 Nantes, France</footnote>

<footnote><sup>d</sup> Nantes Université, Univ Angers, CHU Nantes, INSERM, CNRS, CRCI2NA, F-44000 Nantes, France</footnote>

<footnote>* Corresponding author.</footnote>

<footnote>E-mail address: amaury.derquenne@chu-nantes.fr (A. Derquenne).</footnote>
<figure><img src="image_6.png" /><figcaption>**Fig. 2.** Arteriography of the right external carotid artery (*) showing the internal maxillary artery (single white arrow) giving rise to the inferior alveolar artery (double white arrow). Voluminous arteriovenous aneurysm in the distal part of the inferior alveolar artery in its intramandibular portion.</figcaption></figure>

<figure><img src="image_7.png" /><figcaption>**Fig. 3.** Procedure for exclusion of intraosseous arteriovenous malformation by placement of Onyx® 18 (A). Arteriographic control at three months showing no recurrence of the vascular malformation (B).</figcaption></figure>

development, diagnosis is often delayed, with symptoms onset at the time of cortical bone lysis or tooth eruption. It is a dynamic lesion, based on the principles of angiogenesis (ischemia-repermeabilization-neovascular recruitment) which may explain its expansion and recurrence after embolization. Treatment is based essentially on embolization; in the absence of soft-tissue extension and recurrence on control arteriography, bone curettage may allow mandibular bone regeneration [2]. Ciccarese et al. reported 3 cases of osteitis and abscess post-embolization in 6 treated patients [1]. Furthermore, embolization exposes young patients to ionizing radiation [3]. Nevertheless, sclerotherapy which requires less radiation has no place for such AVM developed in bone. Total bone resection including hemimandibulectomy is the most radical technique, but significantly reduces the risk of subsequent recurrence [4]. In this case, mandibular reconstruction is performed, usually using a fibula flap. This technique remains aggressive in children, with the risk of altering mandibular growth. More recently, the use of rapamycin (Sirolimus), by inducting a VEGF inhibition, showed interesting results in the reduction of the activity of the AVM, but with no effect on the volume

of the malformation [5]. However, the low number of cases of mandibular intraosseous AVMs described makes it impossible to conduct clinical trials or consensus management recommendations.

**Patient consent**

The patient reported in this manuscript provided written informed consent for the publication of the case details.

**Declaration of competing interest**

All authors have no conflicts of interest to disclose.

**CRediT authorship contribution statement**

**Amaury Derquenne:** Writing – original draft, Investigation.
**Marine Anquetil:** Investigation. **Hubert Desal:** Supervision. **Hélios Bertin:** Writing – review & editing, Supervision.
References

[1] Ciccarese F, Chauvel-Picard J, Guibaud L, Viremouneix L, Perrot J-L, Breton P, et al. Management of intra-osseous arteriovenous malformations of the mandible. J Stomatol Oral Maxillofac Surg 2022;123:e544-8. doi: 10.1016/j.jormas.2022.03.004.

[2] Brusati R, Galioto S, Biglioli F, Goisis M. Conservative treatment of arteriovenous malformations of the mandible. Int J Oral Maxillofac Surg 2001;30:397-401. doi: 10.1054/ijom.2001.0120.

[3] L G, N E, A P, M K, J T, G H, et al. Patient radiation exposure from embolo-sclerotherapy of peripheral vascular malformations. J Vasc Surg 2021;73. doi: 10.1016/j.jvs.2020.08.152.

[4] Shum JW, Clayman L. Resection and immediate reconstruction of a pediatric vascular malformation in the mandible: case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontology 2010;109:517-24. doi: 10.1016/j.tripleo.2009.10.020.

[5] Freixo C, Ferreira V, Martins J, Almeida R, Caldeira D, Rosa M, et al. Efficacy and safety of sirolimus in the treatment of vascular anomalies: a systematic review. J Vasc Surg 2020;71:318-27. doi: 10.1016/j.jvs.2019.06.217.