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

PEREIRA, José Lucas dos Santos Henrique; SILVA, Diego Filipe Bezerra; SOUZA, Rebeca Cecília Vieira de; SILVA FILHO, Tiago João da; DIAS, Isabella Jardelino; GOMES, Daliana Queiroga de Castro

doi:10.1590/1981-86372023002720220034

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RGO - Revista Gaúcha de Odontologia

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CLINICAL • Rev Gaúch. Odontol. 71 • 2023 • https://doi.org/10.1590/1981-86372023002720220034 copy

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

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

Authorship SCIMAGO INSTITUTIONS RANKINGS

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

Authors/ year	Age (yr)/ sex	Duration	Condition	Signs and symptoms	Treatment
Main & Macphee [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.]	16/ Male	3 weeks	Normal	Mass of hyperplastic tissue buccal to the upper second molar and detachable from the tooth. This tooth was slightly mobile but in full functional use.	Excisional biopsy and 600,000 units of penicillin, administered 1 hour prior to operation; phenoxymethylpenicillin, one 250 mg. tablet every 4 hours.
Musser et al. [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... ]	16/ Female	-	Normal	Clinical examination disclosed a nontender, spongy swelling in the anterior maxilla that appeared to involve soft, tissue. The area extended from the right lateral incisor to the Ieft canine in the maxilla.	Surgery; tetracycline.
Fergus & Savord [¹⁵15 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.]	23/ Male	1 year	-	A raised yellow swelling in the anterior left maxilla, which was tender to palpation.	Surgery and prophylactic dose of 250 mg. phenoxymethyl penicillin four times a day for 1 week.
Rubin & Krost [¹⁶16 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 https://doi.org/10.1016/0278-2391(95)901... ]	58/ Male	-	Cocaine snorting	Nasal congestion, yellow sputum, and purulence	Intravenous aqueous penicillin + oral clindamycin; intravenous penicillin
Herman et al. [¹⁷17 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... ]	65/ Female	2 weeks	Chronic lymphocytic leukemia	Pain, malaise, generalized prostration, difficulty eating and swallowing	Intravenous penicillin
Tarner et al. [¹⁸18 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.]	49/ Male	-	Vasculitis involving the lungs and brain	Mucosal defect of the palate representing the orifice of a fistula between the oral and nasal cavities was found in the area of tooth 23 and 24, which had been extracted without antibiotic prophylaxis 6 months earlier.	-
Crossman & Herold [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... ]	85/ Male	-	Normal	Bony sequestra bilaterally affecting the upper premolar regions	Oral clindamycin
De et al. [²⁰20 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... ]	32/ Male	2 years	Normal	-	Intravenous crystalline penicillin G
Yadegarynia et al. [²¹21 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... ]	48/ Female	8 months	Normal	An indurated area on the hard palate and the overlying skin became reddish. Abscess-like lesions eventually draining to the surface of her developed, which gradually led to a spontaneous discharging sinus. The patient also had post-nasal drip. A discharging sinus on the mid-line of the anterior hard palate with slight deviation to the right.	Biopsy and oral ampicillin 500 mg four times a day. It was planned to continue the treatment for 6 months.
de Andrade et al. [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... ]	46/ Female	4 days	Diabetes mellitus	Dysphagia and pain	penicillin
Jamshidi et al. [²³23 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.]	40/ Female	More than a year	Normal	Persistent sinus tract discharge. In clinical examination, the cutaneous lesion expressed a purulent discharge	Osteotomy and apicoectomy of mesiobuccal, distobuccal, and palatal roots of maxillary right first molar were performed and retropreparations were made
Suhasini et al. [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... ]	19/ Female	2 years	History of trauma in the region	Oblique fracture involving incisal third toward distal side seen with slight discoloration in relation to maxillary right central incisor. Swelling measuring about 3 cm × 3 cm in the palatal aspect of maxillary right central incisor-maxillary right second premolar, extending anteroposterior and mediolaterally 0.5 cm crossing away the mid-palatine raphe and 1 cm away from the marginal gingiva of premolar (palatal) was seen.	Apical surgery and Systemic antibiotics (amoxicillin; Amox 500 mg 3 times daily 5 days) and analgesics (ibuprofen; brufen 400 mg twice daily for 3 days)
Klein et al. [²⁴24 Klein M, Carrard VC, Munerato MC. Cervicofacial actinomycosis of the maxilla and HIV infection: a case report. Otolaryngol (Sunnyvale) 2017;7(1):288.]	31/ Male	2 months	HIV-positive diagnosis with secondary syphilis, pneumocystis pneumonia, and retinitis caused	A far-reaching destructive lesion on the right maxilla. Necrotic gingiva and alveolar mucosa exposed bone and roots of maxillary right canine and maxillary right first molar.	5,000,000 IU of penicillin G crystalline IV for three weeks supplemented with amoxicillin 500 mg orally administered every 8 h for six months.
Dahiya et al. [²⁵25 Dahiya V, George BR, Ramachandran K, Peter S. Actinomycosis hard palate: a rare presentation. Int J Otorhinolaryngol Clin. 2019;11(3):73-75.]	79/ Female	10 months	Diabetic and hypertensive; history of carcinoma of breast detected 1½ years back with postoperative renal failure needing dialysis.	A large 2 × 2 cm ulcer with bone erosion involving anterior half of midline of the hard palate more on the right side with oroantral and oronasal communications. It was covered with slough at the edges and blackish necrotic tissue was present in the surrounding areas	Crystalline penicillin and oral amoxicillin 500mg thrice daily for 12 months
Gurbanov & Torul [²⁶26 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... ]	30/ Female	1.5 months	Previous exodontics in the region	Painful, bleeding prone area with necrotic bone in the left side of the hard palate	Excision of the necrotic bone and surrounding soft tissues; Penicillin and analgesic.
Jangla et al. [²⁷27 Jangla SM, Machhi BS, Bhandarkar PD. Invasive actinomycosis of maxilla - an unusual case report. JKIMSU. 2019;8(2):90-93.]	78/ Female	6 months	Trauma to left side tooth which was self-inflicted using a wooden stick.	A lump on the left side of the upper jaw and difficulty in opening the mouth; ulcer in the mouth at the same site of the trauma with whitish discharge	Biopsy and amoxycillin-clavulanic acid for eight days; ampicillin-sulbactam and doxycycline for four weeks
Ahmedbrahim et al. [²⁸28 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.]	70/ Male	6 months	Smoker and had been diagnosed with epidermoid carcinoma of the lung 1 year previously, which was in remission after radio chemotherapy treatment.	Extensive ulcerative lesion on the hard palate that measured approximately 4 cm in diameter with yellow pseudomembrane deposit and posterior erythema on the soft palate.	Oral penicillin
Samanta & Paul [²⁹29 Samanta A, Paul NR. Actinomycosis affecting the maxilla: a case report of rare occurrence. JMSCR. 2020;8(6):487-490.]	36/ Male	3 years	-	Maxillary left first molar was having grade III with furcation involvement on the buccal aspect along with distobuccal root exposure.	Tooth extraction and histopathological analysis of the soft tissue involved the tooth; Amoxycillin (875 mg) plus clavulanic acid (125 mg) twice a day for a period of 6 weeks, Metronidazole (400 mg) thrice a day for 5 days along with analgesics for 5 days, which was further prolonged for another continuous five weeks.
Present case	21/ Male	-	Normal	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	Resection of the lesion and Amoxicillin (875mg) associated with Clavulanic Acid (125mg), for 30 days, one tablet every 12 hours.

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[1] Correspondence to: DQC Gomes. E-mail: dqcgomes@hotmail.com.