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Actinomycotic ulcer of the oral mucosa: an unusual presentation of oral actinomycosis
British Journal of Oral and Maxillofacial Surgery (2000) 38, 121–123 © 2000 The British Association of Oral and Maxillofacial Surgeons DOI: 10.1054/bjom.1997.0373
BRITISH JOURNAL OF ORAL
& M A X I L L O FA C I A L S U R G E RY
Actinomycotic ulcer of the oral mucosa: an unusual presentation of oral actinomycosis F. J. Alamillos-Granados, A. Dean-Ferrer, A. García-López, F. López-Rubio Department of Oral and Maxillofacial Surgery, and Department of Pathology, Reina Sofía Hospital, University of Cordoba, Cordoba, Spain SUMMARY. Although the oral mucosa is often the site of entry of actinomyces into the deeper tissues, actinomycosis in the oral mucosa is extremely rare. Actinomycotic lesions are usually described as either single or multiple abscesses or indurated masses with hard fibrous walls and soft central loculations. Actinomyces israelii is the principal cause of human actinomycosis. We present a rare case of actinomycosis caused by Actinomyces odontolyticus; it presented primarily as a long-standing ulcer of the oral mucosa mimicking a squamous cell carcinoma.
INTRODUCTION Actinomyces is part of the normal human oral flora, but under certain circumstances it may become pathogenic.1 Injury to the oral mucosa allows the organism to penetrate the submucosal tissues.2 Actinomycosis, a chronic suppurative, granulomatous and fibrosing disease, may subsequently develop.3 Although the oral mucosa is often the portal of entry into deeper tissues, actinomycosis localized to the oral mucosa is extremely unusual.4 The microorganism most often responsible for actinomycosis is Actinomyces israelii. Actinomyces odontolyticus is an extremely unusual cause.3 We describe a rare case of actinomycosis caused by A. odontolyticus; it presented primarily as a longstanding ulcer of the oral mucosa mimicking a squamous cell carcinoma.
Fig. 1 – Ulcer on the anterior floor of the mouth, alveolar process, and inferior third of the labial mucosa, with indurated and welldefined borders that clearly separate the ulcer from the healthy mucosa.
Histological examination showed no signs of malignancy or epithelial dysplasia, but numerous colonies of actinomyces. Figure 2 shows a fibrinous exudate on the surface of the ulcer, a fistulous track and some colonies of actinomyces, with a typical palisade organization (Fig. 3). A fragment of the ulcerated oral mucosa was then sent for culture, which grew A. odontolyticus, sensitive to penicillin, cephalosporins, ciprofloxacin, and tetracyclines. The patient was prescribed minocycline 100 mg orally three times a day for 8 weeks, as she said she was allergic to beta-lactam antibiotics. Twenty days later, the ulcer had reduced in size by half and after 40 days the lesion had completely healed. The treatment was continued for a further 2 weeks. After a year there has been no recurrence.
CASE REPORT A 74-year-old insulin-dependent diabetic woman presented with a 5-week history of a painless, indurated oral ulcer. The dentist had extracted the lower incisors, which were mobile, a week previously thinking that they were responsible for the ulcer and which she thought were a periodontal problem. The ulcer measured 3 cm in diameter, and was located in the anterior floor of the mouth, alveolar process, and inferior third of the labial mucosa. It was painless and did not bleed when touched. Its borders were distinct and indurated, separating it clearly from the healthy mucosa (Fig. 1). There were two small painless lumps thought to be enlarged lymph nodes palpable in both submandibular triangles. There was no facial swelling. The lower cuspids were mobile and painful. Orthopantomography showed no sign of mandibular rarefaction. Routine haematological and biochemical investigations were within normal limits. In view of the suspicion of squamous cell carcinoma, an incisional biopsy specimen was taken.
DISCUSSION Actinomycosis is a chronic granulomatous infective disease caused by microaerophilic Gram-positive 121
122
British Journal of Oral and Maxillofacial Surgery
Fig. 2 – Photomicrograph showing a fistulous tract, necrosis, inflammation, and a superficial ulcer. A colony can be seen deep in the tissue. (Haematoxylin and eosin, original magnification × 2.5).
Fig. 3 – A colony of actinomyces with its typical palisade organization and peripheral inflammatory reaction. (Haematoxylin and eosin, original magnification × 25).
bacteria of the genus actinomyces. Several species have been isolated from the oral cavity of humans, including A. israelii, A. viscosus, A. naeslundii and A. odontolyticus.3 Actinomycosis usually involves the cervicofacial region, and actinomycosis localized in the oral mucosa is extremely rare.4 Although actinomyces is a normal inhabitant of the oral cavity, clinical infection is relatively rare. It is assumed that it develops as an endogenous infection and that injuries, such as wounds of the oral mucosa, tooth extraction,1,2,5 or fractures, are necessary to initiate the disease. Necrotic dental pulp may also be important.2 Our patient had periodontal disease, but no tooth had been extracted before the onset of the ulcer. However, the lower incisors were extracted because the periodontal disease was initially considered to be the cause of the lesion. Debilitating states such as diabetes (as in this patient), malignancy, or immunosuppression may contribute to the development of the disease.6 Actinomycotic lesions are usually described as single or multiple abscesses or indurated masses with hard fibrous walls and soft central loculations containing white or yellow pus.1 There is an inflammatory swelling at the site of inoculation that grows slowly.
This is typically painless and hard, and so may mimic a malignant tumour.6,7 Cervicofacial actinomycosis often involves the parotid, submandibular triangle and mandible. Rarely actinomycosis has involved the hard palate,2 tongue,7 lips,8 hypopharynx,9 and gingiva.10 In these cases the disease usually presented as a swelling suggestive of an abscess or mimicking a neoplasm. In our case the actinomycotic lesion presented instead as a chronic ulcer. Watkins et al. reported a case of a non-healing extraction site in a patient with AIDS.5 We could not find any reports in English of a case of actinomycosis presenting as a chronic ulcer of the oral mucosa, so our case is of interest in being both a rare presentation, and because we were led to suspect that it might be a squamous cell carcinoma. The microorganism most often responsible for actinomycosis is A. israelii. A. odontolyticus is an extremely unusual cause of the disease.3 The growth of A. odontolyticus may be an important finding in the context of this unusual case. The diagnosis of actinomycosis depends largely on the presence of sulphur granules in the exudate, on the culture, and on the histological examination of the biopsy specimen.3 However, macroscopic sulphur granules may not be evident in the exudate1 and
Actinomycotic ulcer of the oral mucosa
paradoxically may also be present in non-actinomycotic infections.2 Culture techniques may be problematic because the cultures are usually contaminated with oral flora.9 In addition, there must be some degree of clinical suspicion because the actinomyces may easily be missed.9 In the review by Brown, less than 30% of the cultures yielded actinomycetes.1 Treatment of actinomycosis consists of surgical debridement of the lesion and giving a long course of an antibiotic. Penicillins, erythromycin, streptomycin, lincomycin, vancomycin, cephalosporins, chloramphenicol, clindamycin, and tetracyclines have all been used with success.3,6 We used a tetracycline because of reported allergy to the beta-lactam group and also because of the generally good tolerance to tetracyclines. After 6 weeks of treatment the lesion had cleared and it had not reappeared after a year. References 1. Brown JR. Human actinomycosis: a study of 181 subjects. Hum Pathol 1973; 4: 319–330. 2. Weese WC, Smith IM. A study of 57 cases of actinomycosis over a 36-year period. Arch Intern Med 1975; 135: 1562–1568. 3. Topazian RG, Goldberg MH. Oral and maxillofacial infections, 2nd edn. Philadelphia: WB Saunders, 1987: 403–407. 4. Pindborg JJ. Atlas de enfermedades de la mucosa oral, 4th edn. Barcelona: Salvat, 1986: 28. 5. Watkins KV, Richmond AS, Langstein IM. Nonhealing extraction site due to Actinomyces naeslundii in patient with AIDS. Oral Surg Oral Med Oral Pathol 1991; 71: 675–677.
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6. Emmanuelli JL. Infectious granulomatous diseases of the head and neck. Am J Otolaryngol 1993; 14: 155–167. 7. Ficarra G, Di Lollo S, Pierleoni F, Panzoni E. Actinomycosis of the tongue: a diagnostic challenge. Head Neck 1993; 15: 53–55. 8. Appiah-Anane S, Tickle M. Actinomycosis – an unusual presentation. Br J Oral Maxillofac Surg 1995; 33: 248–249. 9. Hughes RA, Paonessa DF, Conway WF. Actinomycosis of the larynx. Ann Otol Rhinol Laryngol 1984; 93: 520–524. 10. Schoeman RJ, Rose DL. Destructive gingival mass. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1995; 79: 407–409.
The Authors F. J. Alamillos-Granados MD A. Dean-Ferrer MD Consultant Oral and Maxillofacial Surgeons A. García-López MD Resident, Oral and Maxillofacial Surgery Unit F. López-Rubio DMD Consultant Pathologist Reina Sofía Hospital University of Cordoba Cordoba Spain Correspondence and requests for offprints to: Dr F.J. Alamillos Granados, Servicio de Cirugía Oral y Maxilofacial, Hospital Universitario Reina Sofía, Avda Menéndez Pidal sn, 14004 Córdoba, Spain Paper received 29 September 1997 Accepted 27 November 1997
BRITISH JOURNAL OF ORAL
& M A X I L L O FA C I A L S U R G E RY
Actinomycotic ulcer of the oral mucosa: an unusual presentation of oral actinomycosis F. J. Alamillos-Granados, A. Dean-Ferrer, A. García-López, F. López-Rubio Department of Oral and Maxillofacial Surgery, and Department of Pathology, Reina Sofía Hospital, University of Cordoba, Cordoba, Spain SUMMARY. Although the oral mucosa is often the site of entry of actinomyces into the deeper tissues, actinomycosis in the oral mucosa is extremely rare. Actinomycotic lesions are usually described as either single or multiple abscesses or indurated masses with hard fibrous walls and soft central loculations. Actinomyces israelii is the principal cause of human actinomycosis. We present a rare case of actinomycosis caused by Actinomyces odontolyticus; it presented primarily as a long-standing ulcer of the oral mucosa mimicking a squamous cell carcinoma.
INTRODUCTION Actinomyces is part of the normal human oral flora, but under certain circumstances it may become pathogenic.1 Injury to the oral mucosa allows the organism to penetrate the submucosal tissues.2 Actinomycosis, a chronic suppurative, granulomatous and fibrosing disease, may subsequently develop.3 Although the oral mucosa is often the portal of entry into deeper tissues, actinomycosis localized to the oral mucosa is extremely unusual.4 The microorganism most often responsible for actinomycosis is Actinomyces israelii. Actinomyces odontolyticus is an extremely unusual cause.3 We describe a rare case of actinomycosis caused by A. odontolyticus; it presented primarily as a longstanding ulcer of the oral mucosa mimicking a squamous cell carcinoma.
Fig. 1 – Ulcer on the anterior floor of the mouth, alveolar process, and inferior third of the labial mucosa, with indurated and welldefined borders that clearly separate the ulcer from the healthy mucosa.
Histological examination showed no signs of malignancy or epithelial dysplasia, but numerous colonies of actinomyces. Figure 2 shows a fibrinous exudate on the surface of the ulcer, a fistulous track and some colonies of actinomyces, with a typical palisade organization (Fig. 3). A fragment of the ulcerated oral mucosa was then sent for culture, which grew A. odontolyticus, sensitive to penicillin, cephalosporins, ciprofloxacin, and tetracyclines. The patient was prescribed minocycline 100 mg orally three times a day for 8 weeks, as she said she was allergic to beta-lactam antibiotics. Twenty days later, the ulcer had reduced in size by half and after 40 days the lesion had completely healed. The treatment was continued for a further 2 weeks. After a year there has been no recurrence.
CASE REPORT A 74-year-old insulin-dependent diabetic woman presented with a 5-week history of a painless, indurated oral ulcer. The dentist had extracted the lower incisors, which were mobile, a week previously thinking that they were responsible for the ulcer and which she thought were a periodontal problem. The ulcer measured 3 cm in diameter, and was located in the anterior floor of the mouth, alveolar process, and inferior third of the labial mucosa. It was painless and did not bleed when touched. Its borders were distinct and indurated, separating it clearly from the healthy mucosa (Fig. 1). There were two small painless lumps thought to be enlarged lymph nodes palpable in both submandibular triangles. There was no facial swelling. The lower cuspids were mobile and painful. Orthopantomography showed no sign of mandibular rarefaction. Routine haematological and biochemical investigations were within normal limits. In view of the suspicion of squamous cell carcinoma, an incisional biopsy specimen was taken.
DISCUSSION Actinomycosis is a chronic granulomatous infective disease caused by microaerophilic Gram-positive 121
122
British Journal of Oral and Maxillofacial Surgery
Fig. 2 – Photomicrograph showing a fistulous tract, necrosis, inflammation, and a superficial ulcer. A colony can be seen deep in the tissue. (Haematoxylin and eosin, original magnification × 2.5).
Fig. 3 – A colony of actinomyces with its typical palisade organization and peripheral inflammatory reaction. (Haematoxylin and eosin, original magnification × 25).
bacteria of the genus actinomyces. Several species have been isolated from the oral cavity of humans, including A. israelii, A. viscosus, A. naeslundii and A. odontolyticus.3 Actinomycosis usually involves the cervicofacial region, and actinomycosis localized in the oral mucosa is extremely rare.4 Although actinomyces is a normal inhabitant of the oral cavity, clinical infection is relatively rare. It is assumed that it develops as an endogenous infection and that injuries, such as wounds of the oral mucosa, tooth extraction,1,2,5 or fractures, are necessary to initiate the disease. Necrotic dental pulp may also be important.2 Our patient had periodontal disease, but no tooth had been extracted before the onset of the ulcer. However, the lower incisors were extracted because the periodontal disease was initially considered to be the cause of the lesion. Debilitating states such as diabetes (as in this patient), malignancy, or immunosuppression may contribute to the development of the disease.6 Actinomycotic lesions are usually described as single or multiple abscesses or indurated masses with hard fibrous walls and soft central loculations containing white or yellow pus.1 There is an inflammatory swelling at the site of inoculation that grows slowly.
This is typically painless and hard, and so may mimic a malignant tumour.6,7 Cervicofacial actinomycosis often involves the parotid, submandibular triangle and mandible. Rarely actinomycosis has involved the hard palate,2 tongue,7 lips,8 hypopharynx,9 and gingiva.10 In these cases the disease usually presented as a swelling suggestive of an abscess or mimicking a neoplasm. In our case the actinomycotic lesion presented instead as a chronic ulcer. Watkins et al. reported a case of a non-healing extraction site in a patient with AIDS.5 We could not find any reports in English of a case of actinomycosis presenting as a chronic ulcer of the oral mucosa, so our case is of interest in being both a rare presentation, and because we were led to suspect that it might be a squamous cell carcinoma. The microorganism most often responsible for actinomycosis is A. israelii. A. odontolyticus is an extremely unusual cause of the disease.3 The growth of A. odontolyticus may be an important finding in the context of this unusual case. The diagnosis of actinomycosis depends largely on the presence of sulphur granules in the exudate, on the culture, and on the histological examination of the biopsy specimen.3 However, macroscopic sulphur granules may not be evident in the exudate1 and
Actinomycotic ulcer of the oral mucosa
paradoxically may also be present in non-actinomycotic infections.2 Culture techniques may be problematic because the cultures are usually contaminated with oral flora.9 In addition, there must be some degree of clinical suspicion because the actinomyces may easily be missed.9 In the review by Brown, less than 30% of the cultures yielded actinomycetes.1 Treatment of actinomycosis consists of surgical debridement of the lesion and giving a long course of an antibiotic. Penicillins, erythromycin, streptomycin, lincomycin, vancomycin, cephalosporins, chloramphenicol, clindamycin, and tetracyclines have all been used with success.3,6 We used a tetracycline because of reported allergy to the beta-lactam group and also because of the generally good tolerance to tetracyclines. After 6 weeks of treatment the lesion had cleared and it had not reappeared after a year. References 1. Brown JR. Human actinomycosis: a study of 181 subjects. Hum Pathol 1973; 4: 319–330. 2. Weese WC, Smith IM. A study of 57 cases of actinomycosis over a 36-year period. Arch Intern Med 1975; 135: 1562–1568. 3. Topazian RG, Goldberg MH. Oral and maxillofacial infections, 2nd edn. Philadelphia: WB Saunders, 1987: 403–407. 4. Pindborg JJ. Atlas de enfermedades de la mucosa oral, 4th edn. Barcelona: Salvat, 1986: 28. 5. Watkins KV, Richmond AS, Langstein IM. Nonhealing extraction site due to Actinomyces naeslundii in patient with AIDS. Oral Surg Oral Med Oral Pathol 1991; 71: 675–677.
123
6. Emmanuelli JL. Infectious granulomatous diseases of the head and neck. Am J Otolaryngol 1993; 14: 155–167. 7. Ficarra G, Di Lollo S, Pierleoni F, Panzoni E. Actinomycosis of the tongue: a diagnostic challenge. Head Neck 1993; 15: 53–55. 8. Appiah-Anane S, Tickle M. Actinomycosis – an unusual presentation. Br J Oral Maxillofac Surg 1995; 33: 248–249. 9. Hughes RA, Paonessa DF, Conway WF. Actinomycosis of the larynx. Ann Otol Rhinol Laryngol 1984; 93: 520–524. 10. Schoeman RJ, Rose DL. Destructive gingival mass. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1995; 79: 407–409.
The Authors F. J. Alamillos-Granados MD A. Dean-Ferrer MD Consultant Oral and Maxillofacial Surgeons A. García-López MD Resident, Oral and Maxillofacial Surgery Unit F. López-Rubio DMD Consultant Pathologist Reina Sofía Hospital University of Cordoba Cordoba Spain Correspondence and requests for offprints to: Dr F.J. Alamillos Granados, Servicio de Cirugía Oral y Maxilofacial, Hospital Universitario Reina Sofía, Avda Menéndez Pidal sn, 14004 Córdoba, Spain Paper received 29 September 1997 Accepted 27 November 1997