Bony cystic lesion with associated submandibular lymphadenopathy on a background of breast carcinoma: an unexpected case of cervicofacial actinomycosis.

Actinomycosis is an uncommon, chronic suppurative granulomatous infection and needs to be considered as a differential diagnosis. A 56-year-old woman with a background of type 2 diabetes mellitus and breast carcinoma was referred to the Oral and Maxillofacial Surgery 2-week wait clinic, regarding a tender sublingual mass and firm erythematous swelling in the right submandibular and submental region. This was slowly progressive and had not responded to oral co-amoxiclav. An orthopantomogram showed a well-defined radiolucency and smaller radiolucent lesions throughout the edentulous right body of the mandible. A contrast-enhanced CT confirmed a right submandibular abscess communicating with cavitating lesions. The differentials included osteomyelitis, bony metastases, multiple myeloma or other cystic lesions. The patient underwent incision and drainage of the abscess, alongside biopsies, and intravenous co-amoxiclav was given. Microbiology cultures confirmed the presence of Actinomyces israelii and a diagnosis of cervicofacial actinomycosis with mandibular osteomyelitis. The patient was successfully treated with prolonged antibiotics.

Facial Actinomycosis Mimicking a Cutaneous Tumor.

Actinomycosis is a chronic granulomatous infection that commonly occurs in the cervicofacial region. Although Actinomcyes is an element of the normal oral flora, infections of the facial skin are very rare because of the entirely endogenous habitation of the organism. The authors report a case of facial actinomycosis, which mimicked a cutaneous tumor both clinically and surgically in a 44-year-old woman with chronic renal failure and Hepatitis C viral infection. The majority of cases can be treated with long-term antibiotics. However, a treatment-resistant abscess, a fistula, or postsurgical excision of the mass formation that are infected can be treated with antibiotics as soon as possible, and recurrence of infection is prevented. The treatment should consist of conservative surgery to obtain a firm histological diagnosis and to drain any collections.

Cervical spinal cord compression in a child with cervicofacial actinomycosis.

Actinomycosis is a rare, chronic, suppurative, and slowly progressive granulomatous disease caused by a group of filamentous gram-positive anaerobic bacteria belonging to the normal flora of the oral cavity, gastrointestinal, and genitourinary tracts. It may involve several organs; however, spinal cord compression has rarely been reported. We report a 7-year-3-month-old girl who suffered from neck pain with restricted movement, poor appetite, body weight loss, mild right limbs weakness, and a non-tender neck mass. Histopathology of the neck mass revealed sulfur granules of actinomyces. Since actinomycosis was strongly suspected, she was treated with high dose of parenteral penicillin G followed by oral penicillin with complete recovery. The unusual features of our case suggested that actinomycosis should be considered as a differential diagnosis of neck mass and cervical spinal cord compression.

Carotid artery rupture and cervicofacial actinomycosis.

Cervicofacial actinomycosis is an uncommon, progressive infection caused by bacilli of the Actinomyces genus. Actinomyces are common commensal saprophytes in the oral cavity which may have medical importance as facultative pathogens. Subsequent to local injuries to the oral mucosa, they may penetrate the deep tissues and be responsible for suppurative or granulomatous infections. We herein report a case of a 65-year-old man who underwent surgery followed by chemotherapy and radiotherapy for a tonsillar carcinoma. An ulcerous lesion in the base of the tongue developed and spread to the carotid artery wall. The man died of a massive hemorrhage due to left carotid artery rupture. Postmortem computed tomography angiography performed prior to autopsy allowed the precise localization of the source of bleeding to be detected. Postmortem biochemical investigations confirmed the presence of inflammation associated with local bacterial infection. Histological investigations revealed the rupture of the left carotid artery surrounded by numerous colonies of Actinomyces. Acute and chronic inflammation with tissue necrosis as well as post-actinic, fibrotic changes were also found in the tissues surrounding the ruptured artery wall.

Mandibular Actinomyces osteomyelitis complicating florid cemento-osseous dysplasia: case report.

BACKGROUND: Apart from neoplastic processes, chronic disfiguring and destructive diseases of the mandible are uncommon. CASE PRESENTATION: We report, perhaps for the first time, the simultaneous occurrence of two such conditions in one patient, in a case that emphasizes the importance of bone biopsy in establishing the correct diagnosis. Florid cemento-osseous dysplasia (FCOD) is a chronic, disfiguring condition of the maxillofacial region. This relatively benign disease is primarily observed in middle-aged women of African ancestry. Cervicofacial actinomycosis is an uncommon and progressive infection caused by bacilli of the Actinomyces genus that typically involves intraoral soft tissues but may also involve bone. The accurate diagnosis of actinomycosis is critical for successful treatment. A diagnosis of osteomyelitis caused by Actinomyces bacteria was diagnosed by bone biopsy in a 53 year-old African-American woman with a longstanding history of FCOD after she presented with a new draining ulcer overlying the mandible. CONCLUSIONS: Clinicians should be aware of the possibility of actinomycosis arising in the setting of FCOD, and the importance of bone biopsy and cultures in arriving at a definitive and timely diagnosis.

The pathogenesis of bisphosphonate-related osteonecrosis of the jaw: so many hypotheses, so few data.

Bisphosphonate-related osteonecrosis of the jaw (BRONJ) has generated great interest in the medical and research communities yet remains an enigma, given its unknown pathogenesis. The goal of this review is to summarize the various proposed hypotheses underlying BRONJ. Although a role of the oral mucosa has been proposed, the bone is likely the primary tissue of interest for BRONJ. The most popular BRONJ hypothesis-manifestation of necrotic bone resulting from bisphosphonate--induced remodeling suppression--is supported mostly by indirect evidence, although recent data have shown that bisphosphonates significantly reduce remodeling in the jaw. Remodeling suppression would be expected, and has been shown, to allow accumulation of nonviable osteocytes, whereas a more direct cytotoxic effect of bisphosphonates on osteocytes has also been proposed. Bisphosphonates have antiangiogenic effects, leading to speculation that this could contribute to the BRONJ pathogenesis. Compromised angiogenesis would most likely be involved in post-intervention healing, although other aspects of the vasculature (eg, blood flow) could contribute to BRONJ. Despite infection being present in many BRONJ patients, there is no clear evidence as to whether infection is a primary or secondary event in the pathophysiology. In addition to these main factors proposed in the pathogenesis, numerous cofactors associated with BRONJ (eg, diabetes, smoking, dental extraction, concurrent medications) could interact with bisphosphonates and affect remodeling, angiogenesis/blood flow, and/or infection. Because our lack of knowledge concerning BRONJ pathogenesis results from a lack of data, it is only through the initiation of hypothesis-driven studies that significant progress will be made to understand this serious and debilitating condition.

Coexistence of actinomycosis and sialolithiasis in the submandibular gland.

Actinomycosis of the submandibular gland is unusual. A 55-year-old male presented with a right, painless submandibular mass of four-year duration. Physical examination revealed a 4 x 3-cm mass in the submandibular region. Ultrasonography-guided fine needle aspiration from the lesion showed microorganisms compatible with actinomyces colonies in the glandular tissue. The diagnosis was made as actinomycosis of the right submandibular gland accompanied by sialolithiasis. The patient was treated with penicillin G (10 million U/day) for three months, followed by 3 g/day oral penicillin for six months. After this medication, physical examination showed no decrease in the size of the mass and the clinical features remained unchanged. Submandibular gland excision under general anesthesia was performed.

Apical actinomycosis as a continuum of intraradicular and extraradicular infection: case report and critical review on its involvement with treatment failure.

This article reports a case of extraradicular actinomycosis that clearly formed a continuum with the intraradicular infection. Histobacteriologic and histopathologic analyses of the root tip and attached apical periodontitis lesion obtained by surgery from a tooth with persistent disease were performed to look for the possible reasons for persistent disease. Although no stainable bacteria were observed in the apparently well-treated main canal, apical ramifications were clogged with dense bacterial biofilms that were contiguous to extraradicular actinomycotic aggregates. A critical review of the literature revealed that there is no clear evidence that apical actinomycosis is indeed an independent entity leading to persistent apical periodontitis lesions.

Dual pathology: cervicofacial actinomycosis and nicorandil-induced oral ulceration.

INTRODUCTION: Oral ulceration has many causes and is a common presenting symptom in otolaryngology. CASE REPORT: This article presents an unusual case of dual pathology oral ulceration in an elderly patient. Oral malignancy was initially suspected, but the history, examination and investigation showed that the oral ulceration was caused by actinomycosis infection and by nicorandil use. DISCUSSION: Cervicofacial acinomycosis is a rare, suppurative bacterial disease in which abscesses can form in the tissues and break through the skin, creating pus-discharging lesions. Nicorandil is a potassium channel blocker used in the treatment of ischaemic heart disease. It has been recently recognised as a cause of persistent ulcerative stomatitis. CONCLUSION: This case highlights the importance of a high index of suspicion for unusual and reversible causes of oral ulceration, and of dual pathology as a cause. Such vigilance enables early recognition and treatment of potentially reversible conditions.

[Clinical symptoms and treatment of cervicofacial actinomycosis. Literature survey and case report]. / Az actinomycosis cervicofacialis klinikai jellemzoi es korszeru terápiája. Irodalmi osszefoglaldó es esetközlés.

Actinomycosis is a chronic, specific inflammation which is characterized by suppuration, abscess formation, tissue fibrosis and granuloma formation. Actinomycosis has three main forms (cervicofacial, which is the most frequent, approximately 60%, pulmonary and abdominal), but other regions of the body can be involved, too (e.g. neck, ovaries, bones), that is why its differential diagnosis becomes more and more relevant. Regarding its treatment, the majority of authors recommends the combination of surgical and antibiotic treatment. The authors of this article present a typical case of cervicofacial actinomycosis, in which the authors used the combination of surgical and antibiotic treatment. As a result of the treatment the healing process was completed successfully and without complications.

[Osteonecrosis of the jaws associated with bisphosphonates: five cases]. / Ostéonécrose des maxillaires sous bisphosphonates: cinq cas.

INTRODUCTION: Bisphosphonates have been recently associated with bone necrosis of jaws. PATIENTS AND METHOD: Five patients were treated, two males and three females. All patients received bisphophonates and chemotherapeutic agents, two for multiple myeloma and three for breast cancer with bone metastasis. Bisphophonates were discontinued. None of the patients had received radiotherapy to the head and neck. The localization was mandibular in four patients and the maxillary area in one with oro-antral fistula. Histological and microbiological studies were performed. RESULTS: Histopathology demonstrated osteonecrosis and ruled out detectable neoplastic disease of the jaws. Four biopsies showed Actinomycosis. Four patients underwent sequestrectomy, but the mucosa did not heal. DISCUSSION: Two mechanisms are involved: the antiangiogenic effect of bisphophonates has recently been described in vivo; as bone mineralization increases, accumulation of microdamage is observed in animals studies. Only the jaws are affected by bone necrosis, suggesting a possible trophism for bisphosphonate uptake? Other risks factors have been found: cancer, use of chemotherapeutic agents or corticosteroids, renal failure, Actinomycosis infection. The only effective treatment is dental prevention before starting treatment.

[Bisphosphonates and maxillo-mandibular osteo(chemo)necrosis]. / Bisphosphonates et ostéo(chimio)nécrose maxillo-mandibulaire.

INTRODUCTION: Maxillo-mandibular osteonecrosis is exceptional outside a context of cervico-facial radiotherapy. Bisphosphonates are non-metabolized pyrophosphate analogues which inhibit osteoclastic activity. Bisphosphonates are prescribed for the treatment of malignant hypercalcemia, osteolysis associated with metastatic bone disease, Paget's disease and osteoporosis. Maxillomandibular osteonecrosis with bisphosphonates can be observed in 1/10000 patients, but is probably underestimated due to lack of dental examination. MATERIAL AND METHODS: We describe six cases of mandibular necrosis associated with bisphosphonates: five of them as part of their treatment regimen for a neoplastic condition and one for osteoporosis. RESULTS: Two patients developed spontaneous bone necrosis. In two others, tooth extraction preceded the onset of osteonecrosis. In the last two patients, we noted a preexisting dental infection. All the histopathological examinations showed necrotic bone colonized by Actinomyces. DISCUSSION: Bisphosphonate-induced osteonecrosis is only found in the maxillomandibular area because the jaws are the only bone in the skeleton exposed to the external environment. The mandible is rendered particularly prone to necrosis even after minor trauma because of its terminal vascularization. Careful oral examination is recommended before prescribing bisphosphonate therapy.

[Internal carotid artery vasculitis originated by cervicofacial actinomycosis as a predisponing factor to stroke]. / Vasculitis de la arteria carótida interna originada por actinomicosis cervicofacial como factor predisponente de infarto cerebral.

Actinomycosis is a chronic, suppurative, fibrotic infection produced by a gram-positive anaerobic bacteria of the normal mouth flora, the infection having tendency to fistulate and to extend without respecting tissue layers. Initial diagnosis is usually delayed due to its painless evolution. Three clinical forms are known: cervicofacial, abdominal and thoracic. Occasionally, there is vascular involvement which is mainly associated with venous thrombotic phenomena with low clinical expresion and hematogenous dissemination that can result in a formation of abscess or meningitis in the central nervous system. We report the case of a 28 year old female patient with type I diabetes and microangiopathyc complication with a internal carotid artery stenosis adjacent to mandibular angle actinomicosis. A few months later and after a miscarriage she suffered from ischemic stroke. The anticardiolipin antibody were positive at the time of stroke with post-operative period and over the next two years. To our knowledge (Medline, 1984-2004), it is the first report of internal carotid vasculitis originated by cervicofacial actinomicosis. We discuss the neuroradiological aspects of our case.

Periapical actinomycosis: a review.

Actinomycosis has increasingly been recognized as a cause of persistent or recurrent periapical disease associated with endodontically treated teeth. This case report shows the classic clinical picture of periapical actinomycosis: persistent periapical disease with recurrent sinus tracts. Although there was no pain or swelling after clinically acceptable initial endodontic treatment, a periapical lesion developed. After retreatment, the periapical lesion persisted, and a sinus tract developed. The sinus tract healed with antibiotic therapy but recurred within a few months. This cycle of sinus tract to antibiotic therapy to recurrence of the sinus tract repeated several times over a period of 5 years. Upon biopsy, periapical actinomycosis was diagnosed, where classic "sulfur granules" were demonstrated in the histologic examination of the periapical lesion. Antibiotic therapy for a period of 6 weeks was prescribed subsequent to the histologic diagnosis because of the possibility of spread of the actinomycotic infection into the maxillary sinus. Considerable healing was evident within 5 months of surgical and antibiotic treatment.

Actinomycosis of tonsil masquerading as tumour in a 12-year old child.

A case of unusual presentation of actinomycosis of tonsil causing massive unilateral enlargement, mimicking a tumor in a 12-year old girl is reported.

Atypical presentations of pediatric actinomycosis: report of a case.

It is well recognized that cervicofacial actinomycosis is rare in children, especially at maxilla. Actinomycosis involving the maxilla usually is seen as a localized intraoral infection in contrast to classical cervicofacial actinomycosis. In this article, we describe an 8-year-old patient who had actinomycosis involving the bone at the left maxillary lateral incisor region. The diagnosis was based on histologic report because of location and development of the lesion with unusual history. The treatment of choice was removal of the soft and hard tissues with concomitant prolonged penicillin administration. In cases of persistent oral infection the diagnosis of actinomycosis should be actively attempted through microbiologic and histologic examination.