A Case of Actinomycosis of the Minor Salivary Gland in the Buccal Region
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View metadata, citation and similar papers at core.ac.uk brought to you by CORE provided by Elsevier - Publisher Connector Oral Science International, November 2008, p.131-134 Copyright © 2008, Japanese Stomatology Society. All Rights Reserved. A Case of Actinomycosis of the Minor Salivary Gland in the Buccal Region Takayuki Tamura, Kazuo Ryoke, Kazunori Kidani, Kazuko Takubo, Motoki Nakabayashi and Shigeki Amekawa Division of Oral and Maxillofacial Biopathological Surgery, Department of Medicine of Sensory and Motor Organs, School of Medicine, Tottori University Faculty of Medicine Abstract: We report a case of actinomycosis arising in the minor salivary gland in the buccal re- gion. A 71-year-old male presented with a swelling in the left buccal region. The clinical diagnosis was minor salivary gland tumor in the buccal mucosa. Under local anesthesia, the lesion was ex- cised. Histopathological examination showed basophilic amorphous masses of Actinomyces in the dilated excretory duct with squamous metaplasia. A final diagnosis of actinomycosis was made. Its portal of entry was thought to be a disruption of the mucosal barrier after trauma due to mal- adaptation of dentures. There was no sign of recurrence after the surgery. Key words: actinomycosis, buccal salivary gland, gram staining, Grocott staining Introduction Case Report Actinomycosis is a granulomatous inflammatory A 71-year-old male was referred to our depart- disease which shows slow progress and often ment with the chief complaint of a mass at the left arises in the cervico-maxillofacial region. Multiple buccal region in April 2006. The symptoms were abscesses, board-like induration and trismus are first noticed at six months prior to the referral by typical signs of actinomycosis. But recently, such himself, but he did not seek medical attention typical symptoms have decreased in frequency because there was no pain and no change in the and it has become difficult to diagnose the lesion size of the mass. His medical history included clinically1–3. The causative organism of actinomy- well controlled diabetes. Intraoral examination cosis is mainly Actinomyces israelli which is a revealed that a mass, measuring 9 × 8 mm, was gram-positive anaerobic bacterium. In the maxill- located in the left buccal region corresponding to ofacial region, the sites of predilection of actino- the lower left first premolar area (Fig. 1). The mycosis are the mandible (80%) and maxilla (10%). mass was round and movable, elastic and hard, Actinomyces infection in the minor salivary gland and the surface of the mucosa showed normal is uncommon (1%)4. We report a case of actinomy- color. It was not tender. He was dentulous and cosis of the minor salivary gland in the buccal wore a partial denture at the left upper jaw. His region. oral hygiene condition was well controlled. The regional lymph node was not significantly affected. Received 3/05/08; revised 5/22/08; accepted 6/06/08. A diagnosis of minor salivary gland tumor or Requests for reprints: Takayuki Tamura, Division of Oral and inflammation such as lymphoadenitis was sus- Maxillofacial Biopathological Surgery, Department of Medicine of Sensory and Motor Organs, School of Medicine, Tottori University pected clinically. The mass was small. At first a Faculty of Medicine, 36–1 Nishimati, Yonago, Tottori 683–8503, benign minor salivary gland tumor was suspected, Japan, Phone: ＋81–859–38–6687, Fax: ＋81–859–38–6689, E-mail: [email protected] so he was observed for one month without carry- 132 Oral Science International Vol. 5, No. 2 ing out any imaging examination. One month in the submucosal tissues surrounding the minor later, the mass had slightly increased in size to 14 salivary gland (Fig. 2A, B). Gram staining showed × 8 mm. He complained of a yellowish white pus gram-positive organisms (Fig. 3A). Grocott stain- discharge from the mass, so we suspected a sec- ing (Gomori’s methenamin-silver staining) showed ondary infection of the minor salivary gland black-colored filamentous hyphae forming a radi- tumor. Under local anesthesia, we excised the ally arranged complex network (Fig. 3B). In the mass surgically. The mass was multilobular and periphery of the duct, there was fibrous capsule- solid. We administered cefem-type antibiotics for like granulation tissue with lymphocytic infiltra- three days. tion. The final pathological diagnosis was actino- Histopathological examination revealed that mycosis of the minor salivary glands. There has there was a dilated excretory duct with squamous been no recurrence for two years at present. metaplasia. Suppurative exudates and basophilic amorphous masses of bacterial colonies could be Discussion seen in a duct of the minor salivary gland, but not Actinomycosis is a granulomatous inflammatory disease caused mainly by Actinomyces israelli, one of the resident oral flora and a gram-positive anaerobic rod, with mixed infection from other bacteria. It manifests in the form of a suppurative, granulomatous inflammation5. Approximately 80% of Actinomyces infections occur in the mandi- ble, and around 1% occur in the minor salivary gland, which is extremely rare4. Sometimes actinomycosis occurs at sites in the abdomen, chest and brain. It is presumed that infection of the respiratory organ is caused by mis- swallowing of oral bacteria and that infection of the digestive apparatus is caused by swallowing Fig. 1 Soft tissue mass, measuring 9 × 8 mm, oral bacteria. In the case of mandibular infection, located in the left buccal region. multiple abscesses, board-like induration and tris- A. There are masses of Actinomyces (arrow) with B. Basophilic amorphous masses of Actinomyces (ar- neutrophils infiltration in the dilated excretory row). HE stain. Original magnification ×100 duct with squamous metaplasia. HE stain. Origi- nal magnification ×40 Fig. 2 Basophilic amorphous masses of bacterial colonies detected in the dilated excretory duct. November, 2008 Actinomycosis of Buccal Region 133 A. Gram staining shows positive (arrow). Original B. Grocott staining shows positive. Original magnifi- magnification ×600 cation ×600 Fig. 3 Gram and Grocott staining show positive for the filamentous hyphae forming a radially arranged complex network. mus are typical signs of actinomycosis. When it normal flora of the oral cavity especially in caries extends deeper into the bone, the infected area cavities, periodontal pockets and tonsilla. It has develops into osteomyelitis with sequestrum, and been reported that the cause of infection is surgi- sometimes becomes very serious. As for reporting cal intervention or traumatic invasion such as of actinomycosis with minor salivary gland infec- pericoronitis, tooth extraction, denture bite tion, only a few cases of the lesion have been trauma of the buccal mucosa, and foreign body reported in the buccal4 and labial6 mucosa of invasion. However, there have been a few cases minor salivary glands, and so on. In this case, where the focus of infection could not be specified. which occurred in the buccal mucosa, the only It has been reported that dental infection is the symptom was a sense of incongruity due to mass most common cause of actinomycosis (60%)8–10. In formation. It is hard to distinguish actinomycosis this case, the patient complained of maladaptation from general purulent inflammation for use of of his dentures, so we suspect the cause of infec- antibiotic therapy and monitoring disease progres- tion was denture bite trauma of the buccal sion. The final diagnosis of actinomycosis is done mucosa. Histopathological findings revealed baso- by identifying Actinomyces bacteria through cul- philic amorphous masses of Actinomyces and ture examination or histopathological examina- granulation tissue, and a fibrous capsule with tion7. The causative organism is arduous to culti- lymphocytic infiltration surrounding the dilated vate, which makes diagnosis difficult even when excretory duct with squamous metaplasia, indicat- the condition is strongly suspected. In this case, ing that the inflammatory reaction arose from we did not cultivate the organism, but we could denture bite trauma of the buccal mucosa. make the diagnosis of actinomycosis by histo- Treatment consists of surgical drainage of the pathological examination including gram and Gro- infection and antibiotic therapy. Penicillin has cott staining. Furthermore, we carried out Zeihl- proved to be the drug of choice. There have been Neelsen staining to distinguish Actinomyces from reports that tetracyclines and carbapenems are similar filamentous bacteria of the Nocardia effective for the treatment of actinomycosis11．In genus. Actinomyces are not acid-fast and do not this case, we administered cefem-type antibiotics stain with Zeihl-Neelsen staining but Nocardia do after surgery. The region was not infected after stain well. In this case, Zeihl-Neelsen staining surgery and the clinical course was good. was negative. It is important to distinguish actinomycosis Actinomyces israelli usually exists as part of the arising limited to the soft tissue (sometimes aris- 134 Oral Science International Vol. 5, No. 2 ing as an elastic, hard and unclear borderline 3. Vazquez A.M., Marti C., Renaqa I., and Salavert A.: Actin- mass) from a tumor. Upon clinical diagnosis, MRI omycosis of the tongue associated with human immunode- and US have been reported to gain higher avail- ficiency virus infection: Case report. J Oral Mxillofac Surg ability12, but there are few reports about imaging 55：879–881, 1997. of actinomycosis. At the time of clinical diagnosis, 4. Shinozaki Y., Hoshi K., Jinbu Y., and Kusama M.: A case a comprehensive judgment must be made using of sialolithiasis with actinomycosis in the minor salivary glands. Jpn J Oral Maxillofac Surg 50：589–591, 2004. clinical signs, imaging findings and blood exami- 5. Hasegawa Y., Asada K., Nakagawa Y., Nagashima H., nation data. In this case, we just excised surgi- Usui H., and Ishibashi K.: A clinical study of“ intraoral cally after the final clinical diagnosis of infection actinomycosis”. Jpn J Oral Maxillofac Surg 41：797– of minor salivary gland was made because typical 801, 1995.