- Email: [email protected]
A rare case presentation of oral actinomycosis
International Journal of Mycobacteriology
2 ( 2 0 1 3 ) 1 8 7 –1 8 9
Available at www.sciencedirect.com
journal homepage: www.elsevier.com/locate/IJMYCO
Case Report
A rare case presentation of oral actinomycosis Davood Yadegarynia a, Muayad A. Merza a b
a,b,*
, Shahnaz Sali a, Ali G. Firuzkuhi
a
Infectious Disease and Tropical Medicine Research Center, Shahid Beheshti University of Sciences, Tehran, Islamic Republic of Iran 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. Ó 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 formations [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 patient had been aware of a whitish discoloration of the mucosal 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 eventually 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 local 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 denied 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: [email protected] (M.A. Merza). 2212-5531/$ - see front matter Ó 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
infectious disease clinic. In the physical examination, viral signs were normal. Intra-oral examination revealed poor dentition 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 hepatosplenomegaly. Laboratory investigations, including complete blood count, blood biochemistry, and urinalysis were within normal values. Purified protein derivative (PPD) test was negative. Chest X-ray (CXR) was normal. Coronal and axial computed 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 granulation and fibrous tissue revealing many actinomycete colonies. Based on the above clinical evidence, particularly histopathological findings, she was diagnosed as having actinomycosis 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 response to the treatment after 4 weeks. Thereafter, follow-up visits were planned on a regular monthly basis for at least 6 months.
Discussion The causative agent of oral actinomycosis is originated from flora of the oropharyngeal mucous membrane. Disruption of the mucosal barrier is the main triggering factor of the infection. 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 for 8 months. The involvement of the hard palate has been described in very few studies [4]. Weese and Smith reported that in 75% of the patients with cervicofacial disease and in 57% of those with primary sites elsewhere symptoms were present for more than six months before referral [4]. Oral actinomycosis manifesting with sinus tract formation should be differentiated from other etiologies, including tuberculosis,
Fig. 1 – Actinomycotic sinus tract of the hard palate.
2 ( 2 0 1 3 ) 1 8 7 –1 8 9
Fig. 2 – Computed tomography (CT) scan of paranasal sinuses. (A) Coronal section and (B) Axial section, both showing cortical disruption of the right side of the hard palate associated with soft tissue mass extending to the inferior meatus of the right nasal fossa. Also, a soft tissue mass on the floor of the right maxillary sinus is seen.
mucormycosis and coccidioides, nocardiosis and neoplasm. Actinomyces are very difficult to grow in culture with <30% diagnostic yield, which limits the usefulness of microbiological identification in such infections [5]. Similarly, other laboratory findings like anemia, leukocytosis, and an increase in ESR are non-specific and are not supportive to establish the diagnosis. In this case, the CT scan result of paranasal sinuses had features suggestive of a tumor, i.e., a soft tissue mass extended 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 recommended 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 histopathological 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 contains colonies of actinomyces upon the microscopic examination [7]. Generally, the disease has a peak incidence in the 4th to 6th decade of life with a slight male predominance [8]. 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, immunosuppressive drugs and diabetes [9]. In this case, the actinomycosis 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 2–6 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 followup of the patient is mandatory. In conclusion, this study reports a case of actinomycosis involving the hard palate, effectively treated with oral penicillin 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) 67–74. [2] D.F. Bennhoff, Actinomycosis: diagnostic and therapeutic considerations and a review of 32 cases, Laryngoscope 94 (1984) 1198–1217. [3] G. Laskaris, Oral manifestations of infectious diseases, Dent. Clin. North Am. 40 (1996) 395–423. [4] W.C. Weese, I.M. Smith, A study of 57 cases of actinomycosis over a 36-year period, Arch. Intern. Med. 135 (1975) 1562–1568. [5] M. Volante, A.M. Contucci, M. Fantoni, R. Ricci, J. Galli, Cervicofacial actinomycosis: still a difficult differential diagnosis, Acta. Otorhinolaryngol. Ital. 25 (2005) 116–119. [6] U. Sakalliog˘lu, G. Ac¸ikgo¨z, T. Kirtilog˘lu, F. Karago¨z, Rare lesions of the oral cavity: case report of an actinomycotic lesion limited to the gingival, J. Oral Sci. 45 (2003) 39–42. [7] J.R. Brown, Human actinomycosis. A study of 181 subjects, Hum. Pathol. 4 (1973) 319–330. [8] M. Miller, A.J. Haddad, Cervicofacial actinomycosis, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 85 (1998) 496–508. [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) 130–133. [10] M. Martin, The use of oral amoxicillin for the treatment of actinomycosis, Br. Dent. J. 156 (1984) 252–254.
2 ( 2 0 1 3 ) 1 8 7 –1 8 9
Available at www.sciencedirect.com
journal homepage: www.elsevier.com/locate/IJMYCO
Case Report
A rare case presentation of oral actinomycosis Davood Yadegarynia a, Muayad A. Merza a b
a,b,*
, Shahnaz Sali a, Ali G. Firuzkuhi
a
Infectious Disease and Tropical Medicine Research Center, Shahid Beheshti University of Sciences, Tehran, Islamic Republic of Iran 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. Ó 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 formations [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 patient had been aware of a whitish discoloration of the mucosal 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 eventually 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 local 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 denied 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: [email protected] (M.A. Merza). 2212-5531/$ - see front matter Ó 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
infectious disease clinic. In the physical examination, viral signs were normal. Intra-oral examination revealed poor dentition 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 hepatosplenomegaly. Laboratory investigations, including complete blood count, blood biochemistry, and urinalysis were within normal values. Purified protein derivative (PPD) test was negative. Chest X-ray (CXR) was normal. Coronal and axial computed 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 granulation and fibrous tissue revealing many actinomycete colonies. Based on the above clinical evidence, particularly histopathological findings, she was diagnosed as having actinomycosis 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 response to the treatment after 4 weeks. Thereafter, follow-up visits were planned on a regular monthly basis for at least 6 months.
Discussion The causative agent of oral actinomycosis is originated from flora of the oropharyngeal mucous membrane. Disruption of the mucosal barrier is the main triggering factor of the infection. 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 for 8 months. The involvement of the hard palate has been described in very few studies [4]. Weese and Smith reported that in 75% of the patients with cervicofacial disease and in 57% of those with primary sites elsewhere symptoms were present for more than six months before referral [4]. Oral actinomycosis manifesting with sinus tract formation should be differentiated from other etiologies, including tuberculosis,
Fig. 1 – Actinomycotic sinus tract of the hard palate.
2 ( 2 0 1 3 ) 1 8 7 –1 8 9
Fig. 2 – Computed tomography (CT) scan of paranasal sinuses. (A) Coronal section and (B) Axial section, both showing cortical disruption of the right side of the hard palate associated with soft tissue mass extending to the inferior meatus of the right nasal fossa. Also, a soft tissue mass on the floor of the right maxillary sinus is seen.
mucormycosis and coccidioides, nocardiosis and neoplasm. Actinomyces are very difficult to grow in culture with <30% diagnostic yield, which limits the usefulness of microbiological identification in such infections [5]. Similarly, other laboratory findings like anemia, leukocytosis, and an increase in ESR are non-specific and are not supportive to establish the diagnosis. In this case, the CT scan result of paranasal sinuses had features suggestive of a tumor, i.e., a soft tissue mass extended 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 recommended 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 histopathological 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 contains colonies of actinomyces upon the microscopic examination [7]. Generally, the disease has a peak incidence in the 4th to 6th decade of life with a slight male predominance [8]. 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, immunosuppressive drugs and diabetes [9]. In this case, the actinomycosis 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 2–6 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 followup of the patient is mandatory. In conclusion, this study reports a case of actinomycosis involving the hard palate, effectively treated with oral penicillin 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) 67–74. [2] D.F. Bennhoff, Actinomycosis: diagnostic and therapeutic considerations and a review of 32 cases, Laryngoscope 94 (1984) 1198–1217. [3] G. Laskaris, Oral manifestations of infectious diseases, Dent. Clin. North Am. 40 (1996) 395–423. [4] W.C. Weese, I.M. Smith, A study of 57 cases of actinomycosis over a 36-year period, Arch. Intern. Med. 135 (1975) 1562–1568. [5] M. Volante, A.M. Contucci, M. Fantoni, R. Ricci, J. Galli, Cervicofacial actinomycosis: still a difficult differential diagnosis, Acta. Otorhinolaryngol. Ital. 25 (2005) 116–119. [6] U. Sakalliog˘lu, G. Ac¸ikgo¨z, T. Kirtilog˘lu, F. Karago¨z, Rare lesions of the oral cavity: case report of an actinomycotic lesion limited to the gingival, J. Oral Sci. 45 (2003) 39–42. [7] J.R. Brown, Human actinomycosis. A study of 181 subjects, Hum. Pathol. 4 (1973) 319–330. [8] M. Miller, A.J. Haddad, Cervicofacial actinomycosis, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 85 (1998) 496–508. [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) 130–133. [10] M. Martin, The use of oral amoxicillin for the treatment of actinomycosis, Br. Dent. J. 156 (1984) 252–254.