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Actinomycosis in the maxilla of a young asymptomatic patient: unusual case report

Actinomicose em maxila de um paciente jovem assintomático: relato de caso incomum

ABSTRACT

The aim of this study is to report an unusual case of actinomycosis in the maxilla region of an asymptomatic patient. A 21-year-old white man was referred for the analysis of panoramic radiography and cone beam computed tomography, where it was observed the presence of a hypodense lesion on the left side of the maxilla, which extended from the maxillary left central incisor to the region of the maxillary left second premolar. During intraoral examination, a depression was observed in the hard palate mucosa, as well as a fistula in the alveolar mucosa close to maxillary left central incisor, which had pulp vitality confirmed by thermal tests. A fistulography was performed, with periapical radiography, where it was found that the fistulous path did not originate from the tooth mentioned above. An incisional biopsy was performed for diagnostic purposes. However, given the inconclusive microscopic findings, four months later, a new biopsy was performed. The histopathological examination revealed the presence of a colony of microoganisms with filamentous pattern of radiated rosette, surrounded by polymorphonuclear inflammatory cells. Based on the morphological characteristics, the diagnosis of actinomycosis was established. The treatment was based on antibiotic therapy. Six months after treatment of the infection, no signs of recurrence were observed, and the patient remains in follow-up. Actinomycosis in the maxilla is an uncommon infection with a predilection for males between the ages of 20 and 60, whose treatment is antibiotic therapy associated or not with surgical excision.

Indexing terms
Actinomycosis; Bacterial infections; Maxillary diseases; Pathology; Surgery, oral

RESUMO

O objetivo deste estudo é relatar um caso incomum de actinomicose em região de maxila de um paciente assintomático. Um homem de 21 anos, branco, foi encaminhado para análise de radiografia panorâmica e tomografia computadorizada de feixe cônico, onde foi observada a presença de lesão hipodensa do lado esquerdo da maxila, que se estendia da região do incisivo central superior esquerdo ao segundo pré-molar superior esquerdo. Ao exame intrabucal, observou-se depressão na mucosa do palato duro, bem como fístula na mucosa alveolar próxima ao incisivo central superior esquerdo, no qual tinha vitalidade pulpar confirmada pelos testes térmicos. Uma fistulografia com radiografia periapical foi realizada, onde foi observado que o trajeto fistuloso não era originado do dente mencionado anteriormente. Foi realizada biópsia incisional para fins diagnósticos. Porém, diante dos achados microscópicos inconclusivos, quatro meses depois, uma nova biópsia foi realizada. O exame histopatológico revelou a presença de colônia de microrganismos com padrão filamentoso de roseta irradiada, circundado por células inflamatórias polimorfonucleares. Com base nas características morfológicas, foi estabelecido o diagnóstico de actinomicose. O tratamento foi baseado em antibioticoterapia. Seis meses após o tratamento da infecção, não foram observados sinais de recidiva e o paciente permanece em acompanhamento. A actinomicose da maxila é uma infecção incomum, com predileção por pacientes do sexo masculino com idade entre 20 e 60 anos, cujo tratamento é antibioticoterapia associada ou não à excisão cirúrgica.

Termos de indexação
Actinomicose; Infecções bacterianas; Doenças maxilares; Patologia; Cirurgia bucal

INTRODUCTION

Actinomycosis is a subacute to chronic infection caused by filamentous Gram-positive bacteria, anaerobic [11 Suhasini K, Madhusudhana K, Suneelkumar C, Lavanya A. Unknown to a rare known: a case report of apical actinomycosis. SRM J Res Dent Sci. 2016;7(4):248-251. http://dx.doi.org/10.1590/10.4103/0976-433X.195639
https://doi.org/10.1590/10.4103/0976-433...
,22 Rao K, Babu SG, Shetty U, Castelino RL, Tilak G. Actinomycosis involving the periapical region and mimicking a dentoalveolar abscess: an unusual presentation. Eur J Ther. 2017;23(2): 91-94. https://doi.org/10.5152/EurJTher.2017.02017
https://doi.org/10.5152/EurJTher.2017.02...
], non-acidic [11 Suhasini K, Madhusudhana K, Suneelkumar C, Lavanya A. Unknown to a rare known: a case report of apical actinomycosis. SRM J Res Dent Sci. 2016;7(4):248-251. http://dx.doi.org/10.1590/10.4103/0976-433X.195639
https://doi.org/10.1590/10.4103/0976-433...
,22 Rao K, Babu SG, Shetty U, Castelino RL, Tilak G. Actinomycosis involving the periapical region and mimicking a dentoalveolar abscess: an unusual presentation. Eur J Ther. 2017;23(2): 91-94. https://doi.org/10.5152/EurJTher.2017.02017
https://doi.org/10.5152/EurJTher.2017.02...
] and without spores. This disease has, clinically, four types: thoracic, abdominopelvic, cerebral and cervicofacial [22 Rao K, Babu SG, Shetty U, Castelino RL, Tilak G. Actinomycosis involving the periapical region and mimicking a dentoalveolar abscess: an unusual presentation. Eur J Ther. 2017;23(2): 91-94. https://doi.org/10.5152/EurJTher.2017.02017
https://doi.org/10.5152/EurJTher.2017.02...
]. The latter usually affects the body of the jaw, followed by the mental region and angle of the jaw, but rarely affects the maxilla or the temporomandibular joint [33 Baldawa P, Shirol P, Kulkarni D, Koshti S, Mishra N. Actinomycotic osteomyelitis of palate masquerading periapical pathology: A rare case report. Indian J Dent Res. 2018 Mar-Apr;29(2):247-251. https://doi.org/10.4103/ijdr.IJDR_68_17
https://doi.org/10.4103/ijdr.IJDR_68_17...
].

Actinomycosis in the maxilla accounts for only 0.5% to 9% of all head and neck cases [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
], with periapical actinomycosis being one of the rarest forms of actinomycosis occurring in the maxillofacial region [55 Pasupathy SP, Chakravarthy D, Chanmougananda S, Nair PP. Periapical actinomycosis. BMJ Case Rep. 2012;2012:bcr2012006218. https://doi.org/10.1136/bcr-2012-006218
https://doi.org/10.1136/bcr-2012-006218...
]. The pathogenesis of cervicofacial actinomycosis is not exactly known, however, dental caries and trauma seem to be favorable factors to the development of the disease [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
,66 Valour F, Sénéchal A, Dupieux C, Karsenty J, Lustig S, Breton P, et al. Actinomycosis: etiology, clinical features, diagnosis, treatment, and management. Infect Drug Resist. 2014;7:183-97. https://doi.org/10.2147/IDR.S39601
https://doi.org/10.2147/IDR.S39601...
]. Loss of mucosal integrity caused by tooth extractions, periodontal disease [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
,66 Valour F, Sénéchal A, Dupieux C, Karsenty J, Lustig S, Breton P, et al. Actinomycosis: etiology, clinical features, diagnosis, treatment, and management. Infect Drug Resist. 2014;7:183-97. https://doi.org/10.2147/IDR.S39601
https://doi.org/10.2147/IDR.S39601...
], local anesthesia, bone or dental fractures, tooth eruption, pulp exposure or even endodontic treatment [66 Valour F, Sénéchal A, Dupieux C, Karsenty J, Lustig S, Breton P, et al. Actinomycosis: etiology, clinical features, diagnosis, treatment, and management. Infect Drug Resist. 2014;7:183-97. https://doi.org/10.2147/IDR.S39601
https://doi.org/10.2147/IDR.S39601...
] and non-vital teeth can function as a gateway for these bacteria, and thus, start the infection [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
].

The most common clinical presentation of actinomycosis is soft tissue abscess and drainage of cervical fistulas [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
,77 Oostman O, Smego RA. Cervicofacial Actinomycosis: diagnosis and management. Curr Infect Dis Rep. 2005;7(3):170-174. https://doi.org/10.1007/s11908-005-0030-0
https://doi.org/10.1007/s11908-005-0030-...
]. The skin that covers the abscess is red to purplish and hardened on palpation [77 Oostman O, Smego RA. Cervicofacial Actinomycosis: diagnosis and management. Curr Infect Dis Rep. 2005;7(3):170-174. https://doi.org/10.1007/s11908-005-0030-0
https://doi.org/10.1007/s11908-005-0030-...
]. The discovery of “holes” or large defects of the jaw bone within the oral cavity is a relatively unusual presentation of cervicofacial actinomycosis, when it results in osteomyelitis [33 Baldawa P, Shirol P, Kulkarni D, Koshti S, Mishra N. Actinomycotic osteomyelitis of palate masquerading periapical pathology: A rare case report. Indian J Dent Res. 2018 Mar-Apr;29(2):247-251. https://doi.org/10.4103/ijdr.IJDR_68_17
https://doi.org/10.4103/ijdr.IJDR_68_17...
,44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
].

Actinomycosis is termed “masker” in the head and neck region due to its unusual presentations [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
], because it often mimics a malignant lesion or a granulomatous lesion [88 Moturi K, Kaila V. Cervicofacial actinomycosis and its management. Ann Maxillofac Surg. 2018;8(2):361-364. https://doi.org/10.4103/ams.ams_176_18
https://doi.org/10.4103/ams.ams_176_18...
]. Therefore, the final diagnosis is usually reached only after surgical removal of the lesion and the histopathological examination of the sample. Long-term high-dose penicillin is needed for treatment [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
,66 Valour F, Sénéchal A, Dupieux C, Karsenty J, Lustig S, Breton P, et al. Actinomycosis: etiology, clinical features, diagnosis, treatment, and management. Infect Drug Resist. 2014;7:183-97. https://doi.org/10.2147/IDR.S39601
https://doi.org/10.2147/IDR.S39601...
], which can vary from four weeks to one year, based on the severity of the disease.

Given the above, the objective of the present study is to report an unusual case of actinomycosis in the maxilla region of an asymptomatic young patient.

CASE REPORT

A 21-year-old white man was referred by an orthodontist for the analysis of cone-beam computed tomography, in which significant imaging alterations were observed. During the anamnesis, the patient reported that there had been no previous use of bone modifying agents. The extraoral physical examination did not reveal any changes. On intraoral physical examination, a soft and resilient depression was observed in the mucosa of the hard palate, in addition to a parulis in the alveolar mucosa at the height of the maxillary left central incisor, which had pulp vitality confirmed by thermal tests. A fistulography was performed, with periapical radiography, where it was found that the fistulous path did not originate from the tooth mentioned above.

In cone beam computed tomography, a hypodense image was observed, extending from the region of the maxillary left central incisor to the region of maxillary left second premolar, with regular margins and well-defined limits. In addition, it was also possible to observe the withdrawal of the roots of maxillary left central incisor, maxillary left lateral incisor and maxillary left canine, in absence of root resorption (figure 1A); compression of the anterior wall of the maxillary sinus (figure 1B) and expansion and rupture of the vestibular and palatal cortex of the incisive canal (figure 1C).

Figure 1
A: Cone beam computed tomography in panoramic reconstruction. To observe hypodense image in the maxilla, on the left side, of teeth 21 to 25. B: Cone beam computed tomography, in axial section, showing the extent of the lesion. C: Axial section on a cone beam computed tomography showing a rupture of the incisive canal cortex.

Under the diagnostic hypothesis of a cyst of odontogenic nature, an intraosseous incisional biopsy was chosen to be performed, with access through the palatal region. In view of the inconclusive microscopic findings, monitoring of the patient for four months was recommended, to then perform a new biopsy, which was performed after evaluation of a tomographic examination, which revealed the stagnation of the lesion, without increasing the extent of bone destruction. In the second biopsy, a partial resection of the lesion was performed. At this time, a Partsch type incision was made in the alveolar mucosa, vestibule background, followed by a bone window in the left hemimaxila, to access the lesion (figures 2A and 2B). Then, the bone cavity was curetted, and fragments of the lesion were removed followed by the overlying mucosa suture (figures 2C and 2D).

Figure 2
A: Incision in the mucosa. B: Opening the bone window. C: Access to the injury. D: Suture (3-0 silk thread).

Histopathological analysis of soft tissue fragments showed the presence of connective tissue which varied in density, with collagen fibers randomly arranged, permeated by fusiform and ovoid fibroblasts, in addition to inflammatory infiltrate, predominantly lymphoplasmacytic, and blood vessels, of various calibers and congested by red blood cells, with hemorrhagic areas. In the focal area, there was a microorganism colony with a filamentous pattern of radiated rosette, whose central portions were basophilic and the peripheral ones, eosinophilic, surrounded predominantly by polymorphonuclear cells (figure 3A).

Figure 3
A: Photomicrograph showing normal salivary glandular parenchyma (Hematoxylin and eosin – H/ E, 40x). B: Colony of microorganisms with filamentous pattern of radiated rosette, with central basophilic and peripheral eosinophilic portions, predominantly surrounded by polymorphonuclear cells (H/ E, 40x). C: Intense presence of foamy macrophages, endothelial cells, and rare multinucleated giant cells (H/ E, 400x). D: Photomicrograph demonstrating macrophage immunopositivity for CD68 (100x).

Regarding the analysis of the histological sections of the bone pieces, it was possible to verify, on the periphery of the specimen, trabrecular bone with normal aspect. In addition, there was an intense presence of foamy macrophages, endothelial cells, and few multinucleated giant cells. In these areas, cells with altered morphology and some mitosis figures were observed (figures 3B and 3C). In order to rule out the possibility of being a neoplastic lesion, the immunohistochemical marker CD68 was used, which demonstrated immunopositivity for foamy macrophages (figure 3D).

The diagnosis of actinomycosis was established, and the chosen treatment was antibiotic therapy with Amoxicillin (875mg) associated with Clavulanic Acid (125mg) (Generic Medication Law No. 9,787, 1999), for 30 days, one tablet every 12 hours. The antibiotic time was reduced as a result of the surgical intervention performed previously. After finishing the drug therapy, a panoramic radiography was performed, indicating slight regression of the lesion. Therefore, the patient was instructed to attend periodically.

Upon patient’s return, four months later, it was observed that the maxillary left central incisor had extensive provisional restoration and compromised marginal sealing, as well as the maxillary left lateral incisor, which presented unsatisfactory restoration. Sensitivity tests were performed to evaluate the pulp vitality of both teeth, and both showed a negative response to the tests. Periapical radiography was performed, and the lesion was located at the root apex of these two teeth. In view of this, endodontic treatment of both teeth was requested, and, after the endodontic treatments were completed, total lesion regression was observed (figure 4). Six months after the infection treatment, no clinical or radiographic signs of recurrence were observed, and the patient remains in follow-up.

Figure 4
Panoramic radiograph showing total regression of the lesions that were at the apexes of teeth 21 and 22.

DISCUSSION

Actinomycosis is a rare granulomatous suppurative disease caused by Actinomyces spp [33 Baldawa P, Shirol P, Kulkarni D, Koshti S, Mishra N. Actinomycotic osteomyelitis of palate masquerading periapical pathology: A rare case report. Indian J Dent Res. 2018 Mar-Apr;29(2):247-251. https://doi.org/10.4103/ijdr.IJDR_68_17
https://doi.org/10.4103/ijdr.IJDR_68_17...
,44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
], originated from the Greek word “Aktino”, which means radiant appearance of sulfur granules, and “Mykos”, which labels the condition as mycotic disease [11 Suhasini K, Madhusudhana K, Suneelkumar C, Lavanya A. Unknown to a rare known: a case report of apical actinomycosis. SRM J Res Dent Sci. 2016;7(4):248-251. http://dx.doi.org/10.1590/10.4103/0976-433X.195639
https://doi.org/10.1590/10.4103/0976-433...
,33 Baldawa P, Shirol P, Kulkarni D, Koshti S, Mishra N. Actinomycotic osteomyelitis of palate masquerading periapical pathology: A rare case report. Indian J Dent Res. 2018 Mar-Apr;29(2):247-251. https://doi.org/10.4103/ijdr.IJDR_68_17
https://doi.org/10.4103/ijdr.IJDR_68_17...
]. However, actinomycetes are now grouped as bacteria, due to their composition in the cell wall, lack of nuclear membrane and lack of growth inhibition by antifungal agents [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
]. Like fungi, these bacteria form a mycelial network of branched filaments, but like bacteria, they are thin, have cell walls containing muramic acid and are susceptible to antibiotics [33 Baldawa P, Shirol P, Kulkarni D, Koshti S, Mishra N. Actinomycotic osteomyelitis of palate masquerading periapical pathology: A rare case report. Indian J Dent Res. 2018 Mar-Apr;29(2):247-251. https://doi.org/10.4103/ijdr.IJDR_68_17
https://doi.org/10.4103/ijdr.IJDR_68_17...
].

Actinomyces can live in the oropharynx, gastrointestinal tract and urogenital tract as a normal microbiota [99 Asgary S, Roghanizadeh L. Rapid bone healing after intentional replantation of a molar with apical actinomycosis. Iran Endod J. 2018;13(1):135-138. https://doi.org/10.22037/iej.v13i1.19369
https://doi.org/10.22037/iej.v13i1.19369...
], acting as commensals [11 Suhasini K, Madhusudhana K, Suneelkumar C, Lavanya A. Unknown to a rare known: a case report of apical actinomycosis. SRM J Res Dent Sci. 2016;7(4):248-251. http://dx.doi.org/10.1590/10.4103/0976-433X.195639
https://doi.org/10.1590/10.4103/0976-433...
]. The most important species involved in clinical infection include Actinomyces viscosus, Actinomyces naeslundi, Actinomyces odontolyticus e Actinomyces israelii [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
], the latter being the most prevalent [99 Asgary S, Roghanizadeh L. Rapid bone healing after intentional replantation of a molar with apical actinomycosis. Iran Endod J. 2018;13(1):135-138. https://doi.org/10.22037/iej.v13i1.19369
https://doi.org/10.22037/iej.v13i1.19369...
]. Typically, these bacteria have low potential for pathogenicity or invasion [11 Suhasini K, Madhusudhana K, Suneelkumar C, Lavanya A. Unknown to a rare known: a case report of apical actinomycosis. SRM J Res Dent Sci. 2016;7(4):248-251. http://dx.doi.org/10.1590/10.4103/0976-433X.195639
https://doi.org/10.1590/10.4103/0976-433...
], but they become pathological when gaining access to subcutaneous tissues. In addition, the infection is polymicrobial, with up to five to 10 other bacterial species present [1010 Sharma S, Hashmi MF, Valentino III DJ. Actinomycosis. [Updated 2020 Feb 13]. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2020 Jan-. Available from: https://www.ncbi.nlm.nih.gov/books/NBK482151/
https://www.ncbi.nlm.nih.gov/books/NBK48...
], like Staphylococcus and Streptococcus [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
]. These associated bacteria seem to increase the low pathogenic potential of actinomycetes [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
], working synergistically to form a specific ecosystem with low oxidation reduction potential favorable to anaerobic growth [1111 Gannepalli A, Ayinampudi BK, Baghirath PV, Reddy GV. Actinomycotic osteomyelitis of maxilla presenting as oroantral fistula: a rare case report. Case Rep Dent. 2015;2015:689240. https://doi.org/10.1155/2015/689240
https://doi.org/10.1155/2015/689240...
].

In cases of actinomycosis in the apical region, the source of infection can be intraradicular biofilm or extraradicular bacterial aggregations, such as sulfur granules [99 Asgary S, Roghanizadeh L. Rapid bone healing after intentional replantation of a molar with apical actinomycosis. Iran Endod J. 2018;13(1):135-138. https://doi.org/10.22037/iej.v13i1.19369
https://doi.org/10.22037/iej.v13i1.19369...
]. In addition to local risk factors for the development of infections, diabetes mellitus, alcohol use disorders, malnutrition, malignancies, HIV+ patients, transplantation of solid organs such as lungs and kidneys, biological agents such as infliximab and acute lymphoblastic leukemia treated with chemotherapy are other known risk factors [1010 Sharma S, Hashmi MF, Valentino III DJ. Actinomycosis. [Updated 2020 Feb 13]. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2020 Jan-. Available from: https://www.ncbi.nlm.nih.gov/books/NBK482151/
https://www.ncbi.nlm.nih.gov/books/NBK48...
]. In the case reported here, the patient did not have any systemic factors that could trigger an infection, and one of the teeth associated with the lesion, confirmed with fistulography, showed positive response to pulp sensitivity tests. In addition, the patient had no history of trauma or soft tissue lesion.

Cervicofacial actinomycosis is a relatively rare condition worldwide, without any predilection for age, race [66 Valour F, Sénéchal A, Dupieux C, Karsenty J, Lustig S, Breton P, et al. Actinomycosis: etiology, clinical features, diagnosis, treatment, and management. Infect Drug Resist. 2014;7:183-97. https://doi.org/10.2147/IDR.S39601
https://doi.org/10.2147/IDR.S39601...
] and sex. In the study by Pulverer et al. [1212 Pulverer G, Schütt-Gerowitt H, Schaal KP. Human cervicofacial actinomycoses: microbiological data for 1997 cases. Clin Infect Dis. 2003;37(4):490-497. https://doi.org/10.1086/376621
https://doi.org/10.1086/376621...
], aiming to evaluate microbiological and clinical data from 1997 cases of human cervicofacial actinomycosis, a predisposition of male patients was observed, which varied with age and it seemed to be especially pronounced in patients aged from 20 to 60 years, with a higher incidence found in female patients aged from 11 to 40 years and in male patients aged from 21 to 50 years old. This infection usually involves tissues around the maxilla or mandible, including the mandible itself in approximately 50% of cases, buccal mucosa (15%), mentum (15%) and branch and angle of the mandible (10%) [66 Valour F, Sénéchal A, Dupieux C, Karsenty J, Lustig S, Breton P, et al. Actinomycosis: etiology, clinical features, diagnosis, treatment, and management. Infect Drug Resist. 2014;7:183-97. https://doi.org/10.2147/IDR.S39601
https://doi.org/10.2147/IDR.S39601...
]. Few cases in the literature report maxillary involvement [11 Suhasini K, Madhusudhana K, Suneelkumar C, Lavanya A. Unknown to a rare known: a case report of apical actinomycosis. SRM J Res Dent Sci. 2016;7(4):248-251. http://dx.doi.org/10.1590/10.4103/0976-433X.195639
https://doi.org/10.1590/10.4103/0976-433...
,1111 Gannepalli A, Ayinampudi BK, Baghirath PV, Reddy GV. Actinomycotic osteomyelitis of maxilla presenting as oroantral fistula: a rare case report. Case Rep Dent. 2015;2015:689240. https://doi.org/10.1155/2015/689240
https://doi.org/10.1155/2015/689240...
,1313 Main JH, Macphee IT. Actinomycosis of the maxilla in relation to a periodontal abscess. Oral Surg Oral Med Oral Pathol. 1964;17:299-304.

14 Musser LB, Tulumello TN, Hiatt WR. Actinomycosis of the anterior maxilla. Oral Surg Oral Med Oral Pathol Oral Radiol. 1977;44(1):21-24. https://doi.org/10.1016/0030-4220(77)90236-5
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15 Fergus HS, Savord EG. Actinomycosis involving a periapical cyst in the anterior maxilla. Report of a case. Oral Surg Oral Med Oral Pathol. 1980;49(5):390-3.

16 Rubin MM, Krost BS. Actinomycosis presenting as a midline palatal defect. J Oral Maxillofac Surg. 1995;53(6):701-3. https://doi.org/10.1016/0278-2391(95)90176-0
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17 Herman WW, Whitaker SB, Williams MF, Sangueza OP. Acute actinomycosis presenting as an ulcerated palatal mass. J Oral Maxillofac Surg. 1998;56(9):1098-101. https://doi.org/10.1016/s0278-2391(98)90264-6
https://doi.org/10.1016/s0278-2391(98)90...

18 Tarner IH, Schneidewind A, Linde HJ, Schubert T, Schölmerich J, Glück T, et al. Maxillary actinomycosis in an immunocompromised patient with longstanding vasculitis treated with mycophenolate mofetil. J Rheumatol. 2004; 31(9):1869-71.

19 Crossman T, Herold J. Actinomycosis of the maxilla - a case report of a rare oral infection presenting in general dental practice. Br Dent J. 2009;206(4):201-202. https://doi.org/10.1038/sj.bdj.2009.115
https://doi.org/10.1038/sj.bdj.2009.115...

20 De D, Dogra S, Kanwar AJ, Saikia UN. Actinomycosis presenting as a destructive ulcerated plaque on the palate and gingiva. J Am Acad Dermatol. 2011;65(6):1235-6. https://doi.org/10.1016/j.jaad.2009.02.043
https://doi.org/10.1016/j.jaad.2009.02.0...

21 Yadegarynia D, Merza MA, Sali S, et al. A rare case presentation of oral actinomycosis. Int J Mycobacteriol. 2013; 2(3):187-9. https://doi.org/10.1016/j.ijmyco.2013.06.002
https://doi.org/10.1016/j.ijmyco.2013.06...

22 de Andrade AL, Novaes MM, Germano AR, Luz KG, de Almeida Freitas R, Galvão HC. Acute primary actinomycosis involving the hard palate of a diabetic patient. J Oral Maxillofac Surg. 2014;72(3):537-541. https://doi.org/10.1016/j.joms.2013.08.006
https://doi.org/10.1016/j.joms.2013.08.0...

23 Jamshidi D, Moazami F, Sobhnamayan F, Taheri A. Clinical and histopathologic investigation of periapical actinomycosis with cutaneous lesion: a case report. J Dent (Shiraz). 2015;16(3 Suppl):286-90.

24 Klein M, Carrard VC, Munerato MC. Cervicofacial actinomycosis of the maxilla and HIV infection: a case report. Otolaryngol (Sunnyvale) 2017;7(1):288.

25 Dahiya V, George BR, Ramachandran K, Peter S. Actinomycosis hard palate: a rare presentation. Int J Otorhinolaryngol Clin. 2019;11(3):73-75.

26 Gurbanov V, Torul D. Isolated Actinomycosis of the Hard Palate Incurred Subsequent to Local Anesthesia. J Craniofac Surg. 2019;30(7):e645-e646. https://doi.org/10.1097/SCS.0000000000005701
https://doi.org/10.1097/SCS.000000000000...

27 Jangla SM, Machhi BS, Bhandarkar PD. Invasive actinomycosis of maxilla - an unusual case report. JKIMSU. 2019;8(2):90-93.

28 Ahmedbrahim A, Medelhafed R, Hammouda Y, et al. A rare presentation of hard palate actinomycosis mimicking a neoplasm: case report. Otolaryngol Open Access J. 2020;5(2):1-3.
-2929 Samanta A, Paul NR. Actinomycosis affecting the maxilla: a case report of rare occurrence. JMSCR. 2020;8(6):487-490.] (table 1). In the present case, the patient was male, 21 years old and the lesion was present in the anterior region of the maxilla. Although the patient in the present case was quite young, two cases of 16-year-old patients have already been reported [1313 Main JH, Macphee IT. Actinomycosis of the maxilla in relation to a periodontal abscess. Oral Surg Oral Med Oral Pathol. 1964;17:299-304.,1414 Musser LB, Tulumello TN, Hiatt WR. Actinomycosis of the anterior maxilla. Oral Surg Oral Med Oral Pathol Oral Radiol. 1977;44(1):21-24. https://doi.org/10.1016/0030-4220(77)90236-5
https://doi.org/10.1016/0030-4220(77)902...
].

Table 1
Cases of actinomycosis involving the maxilla reported in the literature.

Clinically, cervical actinomycosis presents as a significant increase in volume associated with external drainage ulcers, fistulas and, occasionally, sulfur granules [1010 Sharma S, Hashmi MF, Valentino III DJ. Actinomycosis. [Updated 2020 Feb 13]. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2020 Jan-. Available from: https://www.ncbi.nlm.nih.gov/books/NBK482151/
https://www.ncbi.nlm.nih.gov/books/NBK48...
]. In addition, it is asymptomatic, hardened on palpation and may present a reddish to purplish color, which evolves to multiple abscesses [1010 Sharma S, Hashmi MF, Valentino III DJ. Actinomycosis. [Updated 2020 Feb 13]. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2020 Jan-. Available from: https://www.ncbi.nlm.nih.gov/books/NBK482151/
https://www.ncbi.nlm.nih.gov/books/NBK48...
]. When located in the maxilla region, the authors report, in their cases, as ulcerated areas involving bone destruction [1111 Gannepalli A, Ayinampudi BK, Baghirath PV, Reddy GV. Actinomycotic osteomyelitis of maxilla presenting as oroantral fistula: a rare case report. Case Rep Dent. 2015;2015:689240. https://doi.org/10.1155/2015/689240
https://doi.org/10.1155/2015/689240...
,2222 de Andrade AL, Novaes MM, Germano AR, Luz KG, de Almeida Freitas R, Galvão HC. Acute primary actinomycosis involving the hard palate of a diabetic patient. J Oral Maxillofac Surg. 2014;72(3):537-541. https://doi.org/10.1016/j.joms.2013.08.006
https://doi.org/10.1016/j.joms.2013.08.0...
], which may be associated with areas of multiple bone sequestrations exposed in the oral cavity [1919 Crossman T, Herold J. Actinomycosis of the maxilla - a case report of a rare oral infection presenting in general dental practice. Br Dent J. 2009;206(4):201-202. https://doi.org/10.1038/sj.bdj.2009.115
https://doi.org/10.1038/sj.bdj.2009.115...
]. In its occurrence, when symptomatic, in the periapical form, it presents as a persistent and recurrent drainage fistula in the periapical region [55 Pasupathy SP, Chakravarthy D, Chanmougananda S, Nair PP. Periapical actinomycosis. BMJ Case Rep. 2012;2012:bcr2012006218. https://doi.org/10.1136/bcr-2012-006218
https://doi.org/10.1136/bcr-2012-006218...
]. In the present case, the patient did not present painful symptoms or any increase in volume, despite the extent of the lesion. On the other hand, a parulis was observed in the region above maxillary left central incisor, which was vital by pulp sensitivity tests.

The imaging findings are nonspecific and do not contribute to the diagnosis of the disease but will help to assess the degree of involvement of soft tissues and bones. Periapical radiographs are useful in assessing apical abscesses [1010 Sharma S, Hashmi MF, Valentino III DJ. Actinomycosis. [Updated 2020 Feb 13]. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2020 Jan-. Available from: https://www.ncbi.nlm.nih.gov/books/NBK482151/
https://www.ncbi.nlm.nih.gov/books/NBK48...
]. In the case reported here, panoramic radiography and cone beam computed tomography were performed for orthodontic purposes, being the lesion discovered accidentally. A fistulography was performed, with periapical radiography, where it was found that the fistulous path did not originate from the tooth mentioned above. The other imaging exams were essential for knowing the extent of the lesion and planning the surgery.

The diagnosis of infection is made based on clinical manifestations, associated with microbiological and histopathological findings [3030 Gomes NR, Diniz MG, Pereira TD, Estrela C, de Macedo Farias L, de Andrade BA, et al. Actinomyces israelii in radicular cysts: a molecular study. Oral Surg Oral Med Oral Pathol Oral Radiol. 2017;123(5):586-590. https://doi.org/10.1016/j.oooo.2017.02.006
https://doi.org/10.1016/j.oooo.2017.02.0...
]. In the case reported here, clinical, imaging, and histopathological findings were used to close the diagnosis due to the initial diagnostic hypotheses. A fact also to be considered is the difficulty of conducting the cultivation of bacterial species in an anaerobic environment for subsequent microbiological analysis.

Microscopically, it is possible to observe a zone of granulation tissue that consists of collagen fibers around central purulent loculations containing abundant neutrophils [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
,2222 de Andrade AL, Novaes MM, Germano AR, Luz KG, de Almeida Freitas R, Galvão HC. Acute primary actinomycosis involving the hard palate of a diabetic patient. J Oral Maxillofac Surg. 2014;72(3):537-541. https://doi.org/10.1016/j.joms.2013.08.006
https://doi.org/10.1016/j.joms.2013.08.0...
]. In the central part, bacterial colonies are observed, with small basophilic heads called conidiophores towards the center and thin eosinophilic hyphae radiating outwards towards the periphery [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
]. In the present case, this characteristic pattern was observed and to discard the hypothesis of neoplastic lesion, the immunohistochemical marker CD68 was used, which was positive for foamy macrophages.

For the treatment of actinomycosis, antibiotic therapy with 6 to 12 million IU penicillin is recommended, which can vary from four weeks to one year, based on the severity of the disease [88 Moturi K, Kaila V. Cervicofacial actinomycosis and its management. Ann Maxillofac Surg. 2018;8(2):361-364. https://doi.org/10.4103/ams.ams_176_18
https://doi.org/10.4103/ams.ams_176_18...
]. In addition, drainage of abscesses, debridement and surgical excision of the sinus tract is recommended to increase the penetration of antibiotics [44 Vinay BH, Mohan A, Haritha P, Lakshmi KR. A rare coexistence of aspergillosis with actinomycosis. J Oral Maxillofac Pathol. 2017;21(2):277-281. https://doi.org/10.4103/jomfp.JOMFP_66_17
https://doi.org/10.4103/jomfp.JOMFP_66_1...
].

Surgical treatment without antibiotic therapy is associated with recurrence. Although good responses have been reported with combined surgical treatment and short-term antibiotic therapy for cervicofacial actinomycosis, surgical treatment is associated with morbidity, especially with extensive lesions [88 Moturi K, Kaila V. Cervicofacial actinomycosis and its management. Ann Maxillofac Surg. 2018;8(2):361-364. https://doi.org/10.4103/ams.ams_176_18
https://doi.org/10.4103/ams.ams_176_18...
], therefore, the need for surgical treatment associated with antibiotic therapy must be carefully considered. In the present case, curettage of the lesion associated with the prescription of 30 days of amoxicillin (875 mg) associated with clavulanic acid (125 mg) was performed. After this period, no signs of recurrence were observed, and the patient remains in follow-up. However, even though surgery was effective in the treatment and essential to reduce the time of antibiotic therapy, probably led to necrosis of the involved teeth.

It is concluded that the present case was of a young patient with actinomycosis in the maxilla without clear evidence of infection, whose treatment of choice was surgery associated with antibiotic therapy. Surgical treatment favored a reduction in the time of antibiotic use and this association promoted an excellent recovery. Despite this, the surgery led to necrosis of the involved teeth and endodontic treatments were performed.

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Edited by

Assistant editor: Luciana Butini Oliveira

Publication Dates

  • Publication in this collection
    07 July 2023
  • Date of issue
    2023

History

  • Received
    09 May 2022
  • Accepted
    15 Dec 2022
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