healthcare Review Diode Laser Management of Primary Extranasopharyngeal Angiofibroma Presenting as Maxillary Epulis: Report of a Case and Literature Review Saverio Capodiferro 1,† , Luisa Limongelli 1,†, Silvia D’Agostino 2,* , Angela Tempesta 1 , Marco Dolci 2, Eugenio Maiorano 3 and Gianfranco Favia 1 1 Department of Interdisciplinary Medicine, University of Bari Aldo Moro, 70121 Bari, Italy; [email protected] (S.C.); [email protected] (L.L.); [email protected] (A.T.); [email protected] (G.F.) 2 Department of Medical, Oral and Biotechnological Sciences, University of Chieti Pescara, 66100 Chieti, Italy; [email protected] 3 Department of Emergency and Organ Transplantation, University of Bari Aldo Moro, 70121 Bari, Italy; [email protected] * Correspondence: [email protected]; Tel.: +39-3930246351 † These Authors equally contributed to the article. Abstract: Juvenile nasopharyngeal angiofibroma is a rare vascular neoplasm, mostly occurring in adolescent males, and representing 0.05% of all head and neck tumors. Nevertheless, it is usually recognized as the most common benign mesenchymal neoplasm of the nasopharynx. Usually, it originates from the posterolateral wall of the nasopharynx and, although histologically benign, classi- cally shows a locally aggressive behavior with bone destruction as well as spreading through natural foramina and/or fissures to the nasopharynx, nasal and paranasal cavities, spheno-palatine foramen, Citation: Capodiferro, S.; Limongelli, infratemporal fossa and, very rarely, to the cranial cavity. Extranasopharyngeal angiofibroma is L.; D’Agostino, S.; Tempesta, A.; considered a distinct entity due to older age at presentation, different localizations (outside the Dolci, M.; Maiorano, E.; Favia, G. nasopharyngeal pterygopalatine fossa) and attenuated clinical course. Extranasopharyngeal angiofi- Diode Laser Management of Primary broma has been sporadically described in the oral cavity. We report a case of extranasopharyngeal Extranasopharyngeal Angiofibroma angiofibroma with primary and exclusive involvement of the adherent gingiva of the anterior maxilla, Presenting as Maxillary Epulis: managed by preoperative diode laser trans-mucosal photocoagulation and subsequent surgical Report of a Case and Literature removal. The current literature on primary extranasopharyngeal angiofibroma is also reviewed. Review. Healthcare 2021, 9, 33. https://doi.org/10.3390/ Keywords: nasopharyngeal angiofibroma; extranasopharyngeal angiofibroma; diode laser; trans- healthcare9010033 mucosal photocoagulation; oral cavity Received: 13 November 2020 Accepted: 29 December 2020 Published: 1 January 2021 1. Introduction Publisher’s Note: MDPI stays neu- Juvenile nasopharyngeal angiofibroma (JNA) is an uncommon vascular tumor, mainly tral with regard to jurisdictional clai- occurring in the nasal and paranasal cavities of adolescent males and representing approx- ms in published maps and institutio- imately 0.05% of all tumors of the head and neck [1–4]. Morphologically, JNA would fit nal affiliations. the criteria for benign mesenchymal neoplasms but may show a locally aggressive clinical course in view of local extension into the adjacent tissues, which often precludes complete surgical removal, with possible tumor persistence and recurrences in a relatively high num- ber of patients (20%) [4,5]. This represents a true challenge for JNA management, as such Copyright: © 2021 by the authors. Li- tumors frequently arise in the posterior-lateral wall of the nasal cavity close to the superior censee MDPI, Basel, Switzerland. margin of the sphenopalatine foramen and may cause bone erosion and displacement of This article is an open access article adjacent structures [5–10]. There is accumulating evidence that a morphologically similar distributed under the terms and con- ditions of the Creative Commons At- angiofibroma, namely extranasopharyngeal angiofibroma (ENA), should be considered as tribution (CC BY) license (https:// a distinct subtype. In fact, it is generally characterized by occurrence in a wider age range, creativecommons.org/licenses/by/ localization outside the nasopharyngeal pterygopalatine fossa, frequent involvement of 4.0/). Healthcare 2021, 9, 33. https://doi.org/10.3390/healthcare9010033 https://www.mdpi.com/journal/healthcare Healthcare 2021, 9, x 2 of 10 Healthcare 2021, 9, x 2 of 10 Healthcare 2021, 9, 33 2 of 10 (ENA), should be considered as a distinct subtype. In fact, it is generally characterized by (ENA), should be considered as a distinct subtype. In fact, it is generally characterized by occurrence in a wider age range, localization outside the nasopharyngeal pterygopalatine occurrence in a wider age range, localization outside the nasopharyngeal pterygopalatine fossa, frequent involvement of the nasal septum and the maxillary sinus, and usually thefossa, nasal frequent septum involvement and the maxillary of the sinus,nasal andseptum usually and showsthe maxillary a more attenuatedsinus, and clinicalusually shows a more attenuated clinical course, with low propensity for recurrence [11]. course,shows a with more low attenuated propensity clinical for recurrence course, with [11]. low propensity for recurrence [11]. IntraoralIntraoral localization localization of of ENA ENA has has been been occasionally occasionally reported reported in in the the lip, lip, palate, palate, tonsil, tonsil, Intraoral localization of ENA has been occasionally reported in the lip, palate, tonsil, tonsillartonsillar pillar, pillar, cheek, cheek, retro-molar retro-molar area area and and gingiva gingiva [12– [12–31].31]. We reportWe report on a caseon a ofcase extrana- of ex- tonsillar pillar, cheek, retro-molar area and gingiva [12–31]. We report on a case of ex- sopharyngealtranasopharyngeal angiofibroma angiofibroma (ENA) with(ENA) localization with localization to the adherent to the ad maxillaryherent maxillary gingiva and gin- tranasopharyngeal angiofibroma (ENA) with localization to the adherent maxillary gin- withoutgiva and maxillary without sinusmaxillary involvement. sinus involvement. The diagnostic The diagnostic work-up and work-up the surgical and the manage- surgical giva and without maxillary sinus involvement. The diagnostic work-up and the surgical mentmanagement by preoperative by preoperative diode laser diode transmucosal laser transmucosal photocoagulation, photocoagulation, and subsequent and removal subse- management by preoperative diode laser transmucosal photocoagulation, and subse- ofquent the lesion removal by conventionalof the lesion by scalpel conventional surgery, arescalpel accurately surgery, described. are accurately described. quent removal of the lesion by conventional scalpel surgery, are accurately described. InIn addition, addition, all all previously previously reported reported cases cases of of ENAs ENAs were were selected selected from from the the literature, literature, In addition, all previously reported cases of ENAs were selected from the literature, collectedcollected and and discussed. discussed. All All angiofibromas angiofibromas arising arising from from or or involving involving the the nasal/maxillary nasal/maxillary collected and discussed. All angiofibromas arising from or involving the nasal/maxillary cavitiescavities were were excluded, excluded, as as well well as as cases cases with with primary primary manifestation manifestation in in the the cheek cheek as as an an cavities were excluded, as well as cases with primary manifestation in the cheek as an extensionextension of of a a tumor tumor arising arising elsewhere, elsewhere, in in order order to to better better define define the the clinico-pathological clinico-pathological extension of a tumor arising elsewhere, in order to better define the clinico-pathological featuresfeatures of of ENA ENA with with exclusive exclusive localization localization in in the the oral oral cavity. cavity. features of ENA with exclusive localization in the oral cavity. 2.2. Case Case Presentation Presentation 2. Case Presentation TheThe patient patient was was a a 17-year-old 17-year-old Caucasian Caucasian male, male referred, referred to to our our attention attention for for persistent persistent The patient was a 17-year-old Caucasian male, referred to our attention for persistent gingivalgingival swelling. swelling. His His medical medical history history was was unremarkable, unremarkable, with with the the exception exception of of massive massive gingival swelling. His medical history was unremarkable, with the exception of massive bleedingbleeding during during an an attempt attempt to to surgically surgically remove remove the the same same gingival gingival lesion lesion performed performed by by bleeding during an attempt to surgically remove the same gingival lesion performed by hishis general general practitioner practitioner two two months months earlier. earlier. The The patient patient referred referred no no previous previous bleeding bleeding at at his general practitioner two months earlier. The patient referred no previous bleeding at otherother sites sites and and his his blood blood tests tests were were within within normal normal limits. limits. other sites and his blood tests were within normal limits. AtAt intra-oral intra-oral examination, examination, a a soft, soft, firm firm and and painless painless swelling swelling of of the the adherent adherent gingiva gingiva At intra-oral examination, a soft, firm and painless swelling of the