Bertucci, R.

Scabini, S.

Trunfio, M.

English

Institutional Research Information System University of Turin

Am. J. Trop. Med. Hyg., 98(3), 2018, pp. 645–646doi:10.4269/ajtmh.17-0796Copyright © 2018 by The American Society of Tropical Medicine and HygieneImages in Clinical Tropical MedicineUltrasonography of an Oral Cavity Onchocercidae NoduleMattia Trunfio,* Silvia Scabini, and Roberto BertucciUnit of Infectious Diseases, Amedeo di Savoia Hospital, Department of Medical Sciences, University of Torino, Torino, ItalyA 51-year-old caucasian woman, living in Italy since herbirth, presented with a 6-month history of a left swollencheek and intraoral nodule, unresponsive to antibiotics. Herprevious medical history was positive for seasonal allergiesand left jowl reconstructive surgery with filling for a caraccident 5 years before. Blood tests were unremarkable;physical examination showed an asymptomatic left cheekbulge and a 1 × 0.5 cm nontender noninflammatory sub-mucosal firm nodule located in the left maxillary vesti-bule. An intraoral ultrasound examination was performed(Figure 1). Differential diagnoses of benign and malignantlesions were likely ruled out and preliminary diagnoses ofprobable filariasis or foreign body reaction were made.Filariasis serology and microfilaremia were negative. Thepatient refused antiparasitic treatment, and an excisionalbiopsy was performed; the healing was complicated by asurgical site infection. Microscopy revealed a cystic cavitycontaining a cross section of an onchocercidae worm(Figure 2). Considering the clinical picture, histology, andepidemiology, an ultimate presumable diagnosis of dir-ofilariasis was made.Dirofilaria are mosquito-borne nematodes infecting ca-nines.1 Man is a dead-end host rarely infected by bites ofvector mosquitoes.1 Endemic areas include tropical andsubtropical regions, the United States, Australia, and theMediterranean area.1 Recently, dirofilariasis spread to pre-viously free countries because of climate changes andemerging vectors.2Most of the cases involve the head, neck, or periocular re-gion; oral dirofilariasis is rare.3 Themost commonpresentationis a single symptomless subcutaneous/submucosal nodule.3As in our case, dirofilarias usually affect adult females, andmicrofilaremia, eosinophilia, and immunoglobulin E elevationare absent.3 Serology is not helpful; polymerase chain reactionhas been suggested,2 but it is resource consuming and notwidely available. Because of the wide variety of the differentialdiagnoses, surgical excision is the most used technique toreach the diagnosis.3–5 However, histopathology has limita-tions because the nematode morphology may be alteredby inflammatory responses or surgical manipulations,4 as inour case.To date, scarce data are available about ultrasonographyof dirofilarial nodules; doppler imaging may also be helpful inthe differential diagnosis.4 Older lesions present calcificationand fibrosis,5 whereas younger nodules may show a weaklymoving filiform worm (see Supplemental Video 1).4 In casesFIGURE 1. (A) A cystic lesion approximately of 1 cm in diameter, localized in the submucosa of the left superior maxillary vestibule, containinganechogenic fluid and a solid filiform mobile worm-like structure with echogenic posterior reinforcement (fluent weak movements were videorecorded; see Supplemental Video 1). (B) Color doppler ultrasonography of the same cystic lesion showing suggestive twinkling artifact and notypical nor suspicious vascular pattern. This figure appears in color at www.ajtmh.org.* Address correspondence to Mattia Trunfio, Unit of InfectiousDiseases, Ospedale Amedeo di Savoia, Corso Svizzera 164, 10144Torino, Italy. E-mail: mattia.trunfio@edu.unito.it645of surgical difficulty in reaching sites and controindicationsto diethylcarbamazine, ultrasonography should be consid-ered for a watch and wait strategy. Physicians should beaware of this possibility to prompt the best diagnosticworkup to avoid patient anxiety and invasive or unnecessaryinvestigations.ReceivedOctober 11, 2017. Accepted for publicationNovember 17,2017.Note: Supplemental video appears at www.ajtmh.org.Authors’ addresses:Mattia Trunfio, Silvia Scabini, andRobertoBertucci,Unit of InfectiousDiseases, Amedeodi Savoia Hospital, Department ofMedical Sciences, University of Torino, Torino, Italy, E-mails:mattia.trunfio@edu.unito.it, silviascabini88@gmail.com, and bertucci54@yahoo.it.This is an open-access article distributed under the terms of theCreative Commons Attribution License, which permits unrestricteduse, distribution, and reproduction in any medium, provided theoriginal author and source are credited.REFERENCES1. Simo´n F, Siles-LucasM,Morcho´nR, Gonza´lez-Miguel J,Mellado I,Carreto´n E, Montoya-Alonso JA, 2012. Human and animaldirofilariasis: the emergence of a zoonotic mosaic. Clin Micro-biol Rev 25: 507–544.2. Falidas E, Gourgiotis S, Ivopoulou O, Koutsogiannis I, OikonomouC, Vlachos K, Villias C, 2016. Human subcutaneous dirofilar-iasis caused by Dirofilaria immitis in a Greek adult. J InfectPublic Health 9: 102–104.3. ToEW, TsangWM,ChanKF, 2003.Humandirofilariasis of thebuccalmucosa: a case report. Int J Oral Maxillofac Surg 32: 104–106.4. Latifog˘lu O, Ozmen S, Sezer C, Yavuzer R, Altintas¸ K, Uluog˘lu O,2002. Dirofilaria repens presenting as a premasseteric nodule.Oral Surg Oral Med Oral Pathol Oral Radiol Endod 94: 217–220.5. Tilakaratne WM, Pitakotuwage TN, 2003. Intra-oral Dirofilaria repensinfection: report of seven cases. J Oral Pathol Med 32: 502–505.FIGURE 2. The histology demonstrates an abscess cavity lined with marked fibrosis and a moderate inflammatory response. No calcificationswere observed. At the center of the cavity, the presence of disintegrating cross sections of an adult onchocercidae worm endowed with acharacteristicmuscle layer surrounded by a thick cuticle can be observed (H&Estain, 4x; courtesy of PathologyUnit,Maria VittoriaHospital, Torino,Italy). This figure appears in color at www.ajtmh.org.646 TRUNFIO AND OTHERS

Ultrasonography of an oral cavity onchocercidae nodule

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Ultrasonography of an oral cavity onchocercidae nodule

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