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 Table of Contents  
CASE REPORTS
Year : 2021  |  Volume : 9  |  Issue : 4  |  Page : 149-152

An unusual presentation of primary actinomycosis infection in hard palate of a 15-year-old girl: A case report


Department of Otorhinolaryngology and Head and Neck Surgery, IMS and SUM Hospital, Siksha “O” Anusandhan University, K8, Kalinga Nagar, Bhubaneswar 751003, Odisha, India

Date of Submission10-Dec-2021
Date of Acceptance21-Jan-2022
Date of Web Publication15-Jul-2022

Correspondence Address:
Santosh K Swain
Department of Otorhinolaryngology and Head and Neck Surgery, IMS and SUM Hospital, Siksha “O” Anusandhan University, K8, Kalinga Nagar, Bhubaneswar 751003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/dypj.DYPJ_74_21

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  Abstract 

Actinomycosis is an uncommon infection caused by filamentous Actinomyces, which forms club-shaped filaments arranged in a radiating pattern and rarely infects palate. The clinical presentations are often non-specific, so it posed a challenging situation for clinicians to get diagnosis of actinomycosis. The histopathological study is often helpful for the diagnosis of actinomycosis. The patient needs clinical follow-up for complete healing of the lesion. This case report describes a case of palatal cyst, which was diagnosed as actinomycosis on the basis of histopathological examination. Surgical excision of the cyst with debridement of the granulation tissue is mandatory for such cystic presentation of actinomycosis. This case was undergoing complete excision, followed by amoxycillin and clavulanic administration for 3 months. The bony defect was reconstructed using mucoperiosteal flap of the palate.

Keywords: Actinomycosis, hard palate, oronasal fistula, pediatric patient


How to cite this article:
Swain SK. An unusual presentation of primary actinomycosis infection in hard palate of a 15-year-old girl: A case report. D Y Patil J Health Sci 2021;9:149-52

How to cite this URL:
Swain SK. An unusual presentation of primary actinomycosis infection in hard palate of a 15-year-old girl: A case report. D Y Patil J Health Sci [serial online] 2021 [cited 2022 Aug 8];9:149-52. Available from: http://www.dypatiljhs.com/text.asp?2021/9/4/149/351086




  Introduction Top


Actinomycosis is an uncommon chronic bacterial infection and usually affects head and neck part and abdomen.[1] Actinomycosis is caused by Actinomyces such as Actinomyces israelii and Actinomyces bovis, which may lead to acute or subacute progression of the disease.[1] The commonest species associated with infection is A. israelii. Actinomycosis is a slow-growing uncommon chronic infection caused by anaerobic or microaerophilic, Gram-positive, non-spore forming, non-acid fast bacteria of the genus Actinomyces.[2] Actinomycosis is a Greek word, “Aktino” meaning radiating appearance of sulfur granules and “mykos” meaning mycotic disease.[3] This disease is often difficult to diagnose because of its non-specific symptoms, its atypical course, and difficulty to isolate this organism.[3] This infection is regarded as the most misdiagnosed disease by expert clinicians. There are three clinical varieties of diseases such as cervicofacial, abdominopelvic, and thoracopulmonary with cervicofacial being the commonest type.[1] Palate is extremely rare location for actinomycosis and often results in increase of morbidity of the patient. This article reports a 15-year-old girl presenting with primary and invasive lesion of actinomycosis in the hard palate region.


  Case Report Top


A 15-year-old girl attended the outpatient department of otorhinolaryngology with complaints of swelling in hard palate since 3 months. There was mild tenderness in the swelling of hard palate and size approximately 4 × 5 cm [Figure 1]. She had mild dysphagia for solid food. Computed tomography (CT) scan showed osteolysis in the hard palate region. She was planned for complete excision of the cyst under general anesthesia. During excision of the cyst, there was a bony erosion found at the hard palate, which continued with nasal cavity. The cystic mass was completely excised and sent for histopathological examination. The palatal defect in the hard palate was reconstructed with periosteal graft of the palate. The wound was closed with mucoperiosteal wall of the palate with Ryel’s tube feeding for 5 days. Tissue thick fluid from the cystic mass was sent for bacteriological study, which showed no evidence of any specific bacteria. The histopathological examination showed aggregates of tangled filaments characterized by central eosinophilic mass from which numerous peripheral basophilic rays were extended [Figure 2]. The surrounding tissue was consisting of neutrophils, lymphocytes, and plasma cells. Foreign body type multinucleated giant cells and areas of necrosis were found. In the absence of positive culture, the diagnosis of actinomycosis was made on the basis of the histopathological criteria. However, the palatal defect which was closed by periosteal lining opens up, and oronasal occurred on 7th post-operative day [Figure 3]. The patient was treated with amoxicillin (500 mg) three times daily for 8 weeks, which resulted in complete regression of the palatal lesion. Then the palatal defect was closed with bone graft. After 6-month follow-up, the patient was symptom-free and is kept under observation.
Figure 1: Palatal actinomycosis presenting as a cystic mass

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Figure 2: Histopathological picture of the palatal actinomycosis

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Figure 3: Post-operative disruption of the palatal defect

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  Discussion Top


Actinomycosis is a subacute-to-chronic bacterial infection caused by a non-motile, non-spore forming Gram-positive anaerobic microaerophilic filamentous bacterium.[3] It is characterized by granulomatous inflammation with contagious spread and forms multiple abscesses and sinus tracts which may discharge sulfur granules.[2]Actinomyces microorganisms are usually commensal in the oral cavity with low potential for virulence. Other bacteria act as synergistic ecosystem for their proliferation. The disruption of the mucosal lining is prerequisite for establishment of this infection. The predisposing factors include oral trauma and tooth extraction or dental abscess.[4] One study stated that the Actinomyces are more prevalent in elderly age groups and females with predilection in the anterior part of maxilla.[4] Sinus tracts are usually associated with actinomycosis patients. In this case, there was a sinus tract between the nasal cavity and the oral cavity. Anatomically and clinically, actinomycosis is classified into three types such as cervicofacial, pulmonary, and abdominopelvic, with the first type being the most common. One study showed that non-diabetic patients can prevent bacterial invasion much more effectively, whereas the diabetic patients are more prone to succumb to the bacterial infections. In addition to this, it is known, although not understood well, that diabetes mellitus hampers the wound healing. The classical actinomycosis infection is usually chronic in nature; however, it may be atypical with acute or subacute clinical presentations. There are three distinct presentations of this disease such as acute painful swelling with duration lesser than 1 month; chronic long-standing infection with duration more than 3 months; and unsuspected microbiologically proven actinomycotic lesion.[4] Actinomycosis affecting the hard palate is rarely seen, and only few cases have been reported in the literature [Table 1].[5],[6],[7] The present case has young age in comparison to other previous cases reported and also with no evidence of immunodeficiency.
Table 1: Different clinical cases of actinomycosis affecting the hard palate

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The clinical diagnosis of actinomycosis is often confusing and difficult as its onset is not specific and has wide ranges of differential diagnosis.[8] The diagnosis of actinomycosis on admission of the patient is correct in lesser than 10% of the cases.[8] There are several pathological entities which mimic actinomycosis. These pathological entities include necrotizing sialometaplasia, a benign reactive inflammatory process which affects minor salivary glands of the hard palate.[9] The commonly proposed pathophysiology for this lesion is ischemia, so clinical history is very important for its diagnosis. The palate is the common site for the development of primary salivary gland tumors like malignant salivary gland neoplasm, which is also considered as a differential diagnosis in the present case.[10] The malignant salivary gland tumors of the palate are mucoepidermoid carcinoma, adenoid cystic carcinoma, and polymorphous low-grade adenocarcinoma.[5],[11] However, more aggressive neoplasms such as salivary duct carcinoma should be thought for differential diagnosis as this tumor extends toward bone and causes bone destruction. The diagnosis is usually confirmed by imaging like CT scan and biopsy. The diagnosis of the actinomycosis is often challenging for prompt treatment. Actinomyces are usually difficult to isolate, as cultures are negative in around 70% of the cases.[12] The diagnosis is based on histopathological examination of surgically excised tissue.[13] The histopathological examination shows two characteristic features such as yellowish actinomycotic granule (sulfur granule), consisting of rounded formation, in the center of which Actinomyces forms a dense, wheel-spoke mass of filaments; and actinomycotic follicle, corresponding to granulomatous reaction surrounding the granules.[14]

The treatment of palatal actinomycosis consists of combination of surgery and long-term antibiotic therapy. The surgical treatment helps to reduce the bacterial load through mechanical debridement and produce aerobic environment.[6] Penicillin is still considered as first-line antibiotic for actinomycosis, despite the absence of consensus regarding the exact dose regimen.[14] Treatment of the actinomycosis consists of oral administration of penicillin.[6] After full course of antibiotic, the lesion usually regresses completely. In the case of osteomyelitis, gallium scintigraphy can be useful in the evaluation of treatment duration of actinomycosis. This examination should be repeated every 3–4 weeks during treatment by antibiotics, which should be continued for 1 week after normal scintigraphy. Follow-up scintigraphy is helpful to ensure the absence of subclinical recurrence. In the case of allergy to penicillin, the patient may use other antibiotics such as macrolides, rifampicin, tetracyclines, clindamycin, or chloramphenicol.[14]


  Conclusion Top


Actinomycosis is a rare and often severe disease when the diagnosis is missed. In the absence of classical symptoms, actinomycosis is a diagnosis of exclusion. The histopathological study is the most reliable for diagnosis and considered as gold standard for the definitive diagnosis of actinomycotic infection. Inadequate treatment results in substantial morbidity and mortality. Clinicians should be aware about the possibility of the actinomycosis in the palatal region and its adequate treatment in time. In this present case, the actinomycosis of the palate was unusually aggressive. Although the diagnosis of actinomycosis is difficult, it must be suspected in the context of palatal-infected cyst or chronic wound. Histopathological examination often confirms the diagnosis and eliminates the other differential diagnoses. Treatment is based on combination of surgery and antibiotics.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
D’Amore F, Franchini R, Moneghini L, Lombardi N, Lodi G, Sardella A, et al. Actinomycosis of the tongue: A case report and review of literature. Antibiotics 2020;9:124.  Back to cited text no. 1
    
2.
Paulo CO, Jordão S, Correia-Pinto J, Ferreira F, Neves I Actinomycosis, a lurking threat: A report of 11 cases and literature review. Rev Soc Bras Med Trop 2018;51:7-13.  Back to cited text no. 2
    
3.
Karanfilian KM, Valentin MN, Kapila R, Bhate C, Fatahzadeh M, Micali G, et al. Cervicofacial actinomycosis. Int J Dermatol 2020;59:1185-90.  Back to cited text no. 3
    
4.
Kuklani RM, Bhattacharyya I, Nair MK Radiographic evaluation of periapical lesions with and without biopsy-proven actinomyces: A pilot study. Quintessence Int 2011;42:301-6.  Back to cited text no. 4
    
5.
Herman WW, Whitaker SB, Williams MF, Sangueza OP Acute actinomycosis presenting as an ulcerated palatal mass. J Oral Maxillofac Surg 1998;56:1098-101.  Back to cited text no. 5
    
6.
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;65:1235-6.  Back to cited text no. 6
    
7.
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;72:537-41.  Back to cited text no. 7
    
8.
Carinci F, Polito J, Pastore A Pharyngeal actinomycosis: A case report. Gerodontology 2007;24:121-3.  Back to cited text no. 8
    
9.
Krishna S, Ramnarayan BK Necrotizing sialometaplasia of palate: A case report. Imaging Sci Dent 2011;41:35-8.  Back to cited text no. 9
    
10.
Swain SK, Das A, Sahu MC An unusual presentation of pleomorphic adenoma of the soft palate in a 13-year-old boy—A case report. Pediatria Polska 2016;91:265-8.  Back to cited text no. 10
    
11.
Swain SK, Debta P, Sahoo S, Samal S, Sahu MC, Mohanty JN A rare site for oral cavity schwannoma: A case report. Indian J Public Health 2019;10:1022-25.  Back to cited text no. 11
    
12.
Sobol SE, Samadi DS, Wetmore RF Actinomycosis of the temporal bone: A report of a case. Ear Nose Throat J 2004;83:327-9.  Back to cited text no. 12
    
13.
Gazzano E, Chanteret C, Duvillard C, Folia M, Romanet P A case of actinomycosis of the middle ear and a review of the literature. Int J Pediatr Otorhinolaryngol Extra 2010;5:70-3.  Back to cited text no. 13
    
14.
Oukessou Y, Ait Elkerdoudi M, Abada RL, Mahtar M Complicated actinomycosis of the temporal bone: A historical case report. Eur Ann Otorhinolaryngol Head Neck Dis 2015;132:227-9.  Back to cited text no. 14
    


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