International Journal of Mycobacteriology 2 ( 2 0 1 3 ) 1 8 7 1 8 9

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Case Report

A rare case presentation of oral actinomycosis

Davood Yadegarynia a, Muayad A. Merza a,b,*

, Shahnaz Sali a, Ali G. Firuzkuhi a

a
Infectious Disease and Tropical Medicine Research Center, Shahid Beheshti University of Sciences, Tehran, Islamic Republic of Iran
b
Azadi Teaching Hospital, School of Medicine, Faculty of Medical Sciences, University of Duhok, Duhok, Kurdistan, Iraq

A R T I C L E I N F O A B S T R A C T

Article history: Actinomycosis is an infectious disease caused by a gram-positive anaerobic or microaero-

Received 22 June 2013 philic Actinomyces species that causes both chronic suppurative and granulomatous
Accepted 29 June 2013 inflammation. The following study reports a 48-year-old Iranian woman presenting
Available online 19 July 2013 with a spontaneous discharging sinus on the hard palate for 8 months. The patient has
no past medical history of note. Laboratory findings were unremarkable. The diagnosis
Keywords: was based on history and clinical evidence of the lesion confirmed by histopathological
Actinomycosis examination. The patient was treated with a regimen of oral ampicillin 500 mg four times
Discharging sinus a day. She had a marked response to the treatment after 4 weeks, and it was planned to
Hard palate continue the treatment for at least 6 months with regular follow-up. To the best of the
researchers knowledge, this is the first report of actinomycotic sinus tract of the hard
palate in Iran.
2013 Asian-African Society for Mycobacteriology. Published by Elsevier Ltd. All rights
reserved.

Introduction Case report

Actinomycosis is a slowly progressive bacterial infection
caused by a gram-positive anaerobic or microaerophilic
Actinomyces species. The disease is characterized by a
chronic suppurative and granulomatous inflammation that
may result in multiple abscesses and sinus tract forma-
tions [1]. Actinomyces is a part of the endogenous flora
of the mouth, the gastrointestinal tract and the female
genital tract. Cervicofacial actinomycosis is the most
common location (55%) of the disease. Although the disease
in this location represents the commonest manifestation,
isolated intra-oral lesions are uncommon [2,3]. Here, the
following reports a rare case of actinomycosis involving
the hard palate. To the best of the researchers knowledge,
this is the first report of hard palate actinomycosis in
Iran.
A 48-year-old Iranian woman presented with a spontaneous
discharging sinus on the hard palate for 8 months. The pa-
tient had been aware of a whitish discoloration of the muco-
sal membrane of her mouth 9 months earlier; however, the
lesion was resolved spontaneously. One week following that,
she developed an indurated area on the hard palate and the
overlying skin became reddish. Abscess-like lesions eventu-
ally draining to the surface developed, which gradually led
to a spontaneous discharging sinus. The discharging fluid
was yellowish in color causing smell impairment but normal
taste. The patient also had post-nasal drip, but she denied lo-
cal pain and bleeding. She also denied fever, chills, loss of
appetite, weight loss and other constitutional symptoms.
The patient has no remarkable past medical history and de-
nied diabetes or any other diseases. She was evaluated in an

* Corresponding author. Address: Department of Internal Medicine, Faculty of Medical Sciences, University of Duhok, Azadi Hospital
Street, Dohuk, Kurdistan, Iraq. Tel.: +964 750 4506172; fax: +964 62 761 8824.
E-mail address: muayadfaily@yahoo.com (M.A. Merza).
2212-5531/$ - see front matter 2013 Asian-African Society for Mycobacteriology. Published by Elsevier Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.ijmyco.2013.06.002
188 International Journal of Mycobacteriology 2 ( 2 0 1 3 ) 1 8 7 1 8 9

infectious disease clinic. In the physical examination, viral

signs were normal. Intra-oral examination revealed poor den-
tition and acceptable oral hygiene, but no signs of gingival
inflammation and the rest of the head and neck examination
were normal. However, there was a discharging sinus on the
mid-line of the anterior hard palate with slight deviation to
the right (Fig. 1). There was no lymphadenopathy or hepato-
splenomegaly. Laboratory investigations, including complete
blood count, blood biochemistry, and urinalysis were within
normal values. Purified protein derivative (PPD) test was neg-
ative. Chest X-ray (CXR) was normal. Coronal and axial com-
puted tomography (CT) scan evaluation of paranasal sinuses
revealed cortical disruption of the right side of the hard palate
associated with a soft tissue mass extending to the inferior
meatus of the right nasal fossa. Further, there was soft tissue
density on the floor of the right maxillary sinus (Fig. 2).
Thereafter, a biopsy specimen was advised to identify the
exact etiology. Hence, under general anesthesia, a biopsy was
performed and the result demonstrated squamous-lined
mucosal tissue underlined by chronically inflamed granula-
tion and fibrous tissue revealing many actinomycete colonies.
Based on the above clinical evidence, particularly histopa-
thological findings, she was diagnosed as having actinomyco-
sis of the hard palate. The patient was treated with a regimen
of oral ampicillin 500 mg four times a day. It was planned to
continue the treatment for 6 months. She had a marked re-
sponse to the treatment after 4 weeks. Thereafter, follow-up
visits were planned on a regular monthly basis for at least
6 months.
Fig. 2 Computed tomography (CT) scan of paranasal
sinuses. (A) Coronal section and (B) Axial section, both
Discussion
showing cortical disruption of the right side of the hard
palate associated with soft tissue mass extending to the
The causative agent of oral actinomycosis is originated from
inferior meatus of the right nasal fossa. Also, a soft tissue
flora of the oropharyngeal mucous membrane. Disruption of
mass on the floor of the right maxillary sinus is seen.
the mucosal barrier is the main triggering factor of the infec-
tion. Actinomycosis of the oral cavity can be present as a
mass or abscess or ulcerative lesion or sinus [2]. The present
case report describes a discharging sinus of the hard palate mucormycosis and coccidioides, nocardiosis and neoplasm.
for 8 months. The involvement of the hard palate has been Actinomyces are very difficult to grow in culture with <30%
described in very few studies [4]. Weese and Smith reported diagnostic yield, which limits the usefulness of microbiologi-
that in 75% of the patients with cervicofacial disease and in cal identification in such infections [5]. Similarly, other labo-
57% of those with primary sites elsewhere symptoms were ratory findings like anemia, leukocytosis, and an increase in
present for more than six months before referral [4]. Oral acti- ESR are non-specific and are not supportive to establish the
nomycosis manifesting with sinus tract formation should be diagnosis. In this case, the CT scan result of paranasal sinuses
differentiated from other etiologies, including tuberculosis, had features suggestive of a tumor, i.e., a soft tissue mass ex-
tended to the inferior meatus of the right nasal fossa and a
soft tissue density on the floor of the right maxillary sinus.
Hence, a histopathological examination was highly recom-
mended to make a definitive diagnosis. It has been shown
that ruling out other causes through histopathology is the
most reliable diagnostic tool [6]. The result of the histopathol-
ogical examination was consistent with actinomycosis. It is
well-known that pathological investigation is crucial in the
diagnosis of actinomycosis since it can reveal an outer area
of granulation tissue and an inner area of necrosis that con-
tains colonies of actinomyces upon the microscopic examina-
tion [7].
Generally, the disease has a peak incidence in the 4th
to 6th decade of life with a slight male predominance [8].
Fig. 1 Actinomycotic sinus tract of the hard palate. Similarly, this patient was of middle age, but in contrast she
 International Journal of Mycobacteriology
was a female. Although this case was immunocompetent,
actinomycosis has a predilection for causing infection in
immunocompromised hosts such as malignancy, immuno-
suppressive drugs and diabetes [9]. In this case, the actinomy-
cosis lesion was limited to the mouth, particularly the hard
palate, which describes the rarity of the infection in this
location. In the available literature, intra-oral actinomycosis
usually is accompanied by cervicofacial disease [3].
The patient was treated with oral penicillin, which is the
drug of choice. The treatment was planned to be continued
for at least 6 months. Generally, serious infection and bulky
disease should be treated with intravenous penicillin for
26 weeks followed by oral penicillin for 12 months, and low
bulk oral disease can be treated with oral agents for a shorter
period [10]. In patients allergic to penicillin, tetracycline,
erythromycin and clindamycin are reasonable alternatives
[4]. Overall, antibiotic treatment should be continued for a
long duration until the wound is healed completely. Relapse
is a common feature of the disease; thus long-term follow-
up of the patient is mandatory.
In conclusion, this study reports a case of actinomycosis
involving the hard palate, effectively treated with oral penicil-
lin therapy. It is important to consider actinomycosis in the
differential diagnosis of indurated lesions or sinus tracts of
the mouth.
2 ( 2 0 1 3 ) 1 8 7 1 8 9 189
R E F E R E N C E S
[1] V.T. De Montpreville, N. Nashashibi, E.M. Dulmet,
Actinomycosis and other bronchopulmonary infections with
bacterial granules, Ann. Diagn. Pathol. 3 (1999) 6774.
[2] D.F. Bennhoff, Actinomycosis: diagnostic and therapeutic
considerations and a review of 32 cases, Laryngoscope 94
(1984) 11981217.
[3] G. Laskaris, Oral manifestations of infectious diseases, Dent.
Clin. North Am. 40 (1996) 395423.
[4] W.C. Weese, I.M. Smith, A study of 57 cases of actinomycosis
over a 36-year period, Arch. Intern. Med. 135 (1975) 15621568.
[5] M. Volante, A.M. Contucci, M. Fantoni, R. Ricci, J. Galli,
Cervicofacial actinomycosis: still a difficult differential
diagnosis, Acta. Otorhinolaryngol. Ital. 25 (2005) 116119.
[6] U. Sakallioglu, G. Acikgoz, T. Kirtiloglu, F. Karagoz, Rare
lesions of the oral cavity: case report of an actinomycotic
lesion limited to the gingival, J. Oral Sci. 45 (2003) 3942.
[7] J.R. Brown, Human actinomycosis. A study of 181 subjects,
Hum. Pathol. 4 (1973) 319330.
[8] M. Miller, A.J. Haddad, Cervicofacial actinomycosis, Oral Surg.
Oral Med. Oral Pathol. Oral Radiol. Endod. 85 (1998) 496508.
[9] T.T. Kingdom, T.A. Tami, Actinomycosis of the nasal septum
in a patient infected with the human immunodeficiency
virus, Otolaryngol. Head Neck Surg. 111 (1994) 130133.
[10] M. Martin, The use of oral amoxicillin for the treatment of
actinomycosis, Br. Dent. J. 156 (1984) 252254.