A 57-year-old man with painful ophthalmoplegia and cavernous sinus involvement: Why this is not Tolosa-Hunt syndrome.

Tolosa-Hunt syndrome (THS) is an idiopathic condition included in the differential diagnosis of painful ophthalmoplegia. Although this was once a common diagnosis, the increasing availability of tests reveals an alternative etiology in many cases. Exclusion of treatable disorders is important, because the prognosis may otherwise be poor. We here describe a patient who presented with painful ophthalmoplegia with an infiltrating lesion in the cavernous sinus. Initially suspected of THS, he had a fatal evolution, and postmortem evaluation revealed cervicocephalic actinomycosis. Actinomycosis diagnosis is often missed, and still represents a challenge to the clinician. We highlight pearls and pitfalls to establish a proper diagnosis to avoid missing a treatable condition in patients with suspected THS.

A case of endobronchial actinomycosis as a challenging cause of lung atelectasis.

BACKGROUND: Primary endobronchial actinomycosis is exceptionally uncommon and can be misdiagnosed as unresolving pneumonia, endobronchial lipoma, bronchogenic carcinoma or foreign body. Predisposing factors are immunosuppressive conditions, chronic lung diseases, poor oral hygiene or foreign body aspiration. CASE PRESENTATION: We reported a case of 88-year old woman with a 4 days history of mild exertional dyspnea, productive cough with purulent sputum and fever up to 37.8 °C, who developed left sided endobronchial actinomycosis in absence of any pre-existent risk conditions; endobronchial de-obstruction and specific antibiotic treatment were performed with success, achieving a full resolution of the disease, with bronchoscopy playing a key role in the diagnosticand therapeutic pathways. CONCLUSIONS: This case raises the necessity for increased awareness in the management of endobronchial lesions and in cases of suspected endobronchial actinomycosis; bronchoscopy plays a key role in the diagnostic and therapeutic process; prompt recognition of this entity can expedite proper treatment and recovery.

Giant sialolith associated with Actinomyces infection, suggesting a new etiopathogenic correlation: a case report.

Sialolithiasis is a disorder of the salivary glands characterized by the presence of mineralized structures. The aim of this article is to contribute to the understanding of the etiopathogenesis of sialolithiasis based on data reported in the literature and the interpretation of scanning electron microscopic (SEM), histopathologic, fluorescence, and energy-dispersive X-ray spectroscopic (EDX) studies. The results of these analyses in a single case are presented. Fluorescence microscopy captured saliva flowing inside a giant sialolith, EDX analysis enabled the quantification of samples of minerals and characterized the chemical composition in the internal areas of the sialolith, and SEM images captured the external communications of the sialolith (ostia). Many theories have been proposed to explain sialolithiasis development, but few consider the role of saliva in its dynamic form. The authors propose that the triple synergism of microbiota, biofilm, and salivary dynamics plays a fundamental role in the development and intrinsic architecture of sialoliths, creating a cyclic sialolithic mineralization. This hypothesis is supported by the results of the laboratory studies in the present case.

Abdominal Wall Actinomycosis Associated with Foreign Body Perforation.

Abdominal wall actinomycosis is a very rare infection caused by anaerobic Gram-positive bacteria Actinomyces. We present a case of a 72-year-old female with chronic pain located in the right hypochondriac region and anterior abdominal wall mass which had developed six months before. An ultrasonography (USG) and computed tomography (CT) scan of the abdomen were performed and showed an inflammatory change with a strong internal linear reflection in the right upper abdomen. The tumor was located inside the rectus abdominis muscle and connected with internal organs and subcutaneous tissue. The patient qualified for surgery. En block tumor excision was made with partial resection of the transverse colon. Postoperative study revealed fishbone-associated inflammatory actinomycosis tumor. The patient was successfully managed postoperatively with penicillin and discharged on the 11th day after the surgery.

Actinomyces spp. bloodstream and deep vein thrombus infections in people who inject drugs.

INTRODUCTION: Actinomyces spp. cause several well-described syndromes including cervicofacial and pelvic infections. Actinomyces spp. infection as an opportunistic infection among people who inject drugs has rarely been described with few case reports published. METHODS AND RESULTS: Here we describe four people who inject drugs admitted with Actinomyces spp. infections, all with an overlapping syndrome and who presented a challenge to both diagnose and to manage. DISCUSSION: This case series highlights the potential to overlook Actinomyces spp. infection in people who inject drugs and aims to increase clinician awareness of diagnosis, empirical and directed treatment, and potential complications of this infection.

Actinomycotic osteomyelitis of a long bone in an immunocompetent adult: a case report and literature review.

BACKGROUND: Actinomycosis is a rare, chronic granulomatous disease caused by Gram-positive anaerobic bacteria that colonize the oral cavity. Cervicofacial actinomycosis is the most frequent clinical presentation of actinomycosis, but hematogenous osteomyelitis at distant sites can occur in rare instance in immunocompromised or pediatric patients, only a few cases have been reported in healthy patients. Here we described a new case of distal femur osteomyelitis caused by Actinomyces in an adult patient who was immunocompetent and had no predisposing factors. CASE PRESENTATION: A woman aged 52 years with no history of trauma presented with severe pain, swelling, and increased local heat in the proximal area of the right knee 3 weeks after she first noticed discomfort. Magnetic resonance imaging showed persistent osteomyelitis of the distal metaphysis and diaphysis of the femur with a multifocal intraosseous abscess pocket. An incision and drainage of the abscess were conducted. The tissue culture, fungus culture, acid fast bacillus (AFB) culture, AFB smear, and tuberculosis polymerase chain reaction test results were negative. A pathologic examination confirmed the presence of actinomycosis. The patient was successfully treated with intravenous penicillin G for 8 weeks followed by oral amoxicillin-clavulanate for 6 weeks with repeated surgical debridement and drainage. After a 5-year follow up, the patient had no signs of recurring infection or complications and she had full range of movement in the affected knee. CONCLUSIONS: Although rare, actinomycotic osteomyelitis can occur in healthy people. Furthermore, actinomycotic osteomyelitis is easily misdiagnosed as tuberculosis in areas with a high prevalence of tuberculosis. To detect and identify the bacteria accurately, pathologic examination should be performed as well as culture tests, because the probability for culture confirmation of actinomycosis is quite low. The initial treatment is vital to a successful outcome without ostectomy or amputation.

Presentation of pulmonary tuberculosis and actinomyces co-infection as a lung mass: a literature review and unique case report.

Parenchymal lung infections occasionally present with clinical symptoms and radiological findings similar to lung malignancy. Pulmonary actinomycosis is a rare condition of its own right, let alone in coexistence with tuberculosis. We report a case of a man presenting with hemoptysis alongside a chest computed tomography compatible with lung cancer. The diagnosis, after removal of a large endobronchial mass with flexible bronchoscopy and cryon, was a concomitant infection with Mycobacterium tuberculosis and Actinomyces odontoliticus. In the literature, there are few reported cases with concomitant tuberculosis and actinomycosis. To our knowledge, such radical treatment without surgical intervention has not been reported in the past.

Case report of Actinomyces turicensis meningitis as a complication of purulent mastoiditis.

BACKGROUND: Central nervous system (CNS) infections caused by Actinomyces spp. including brain abscess, actinomycoma, subdural empyema and epidural abscess are well described, however reports of Actinomyces-associated meningitis are scarcely reported. CASE REPORT: We present the case of a 43-year-old Hungarian male patient with poor socioeconomic status who developed acute bacterial meningitis caused by Actinomyces turicensis originating from the left side mastoiditis. The bacterial cultures of both cerebrospinal fluid (CSF) and purulent discharge collected during the mastoid surgery showed slow growing Gram-positive rods that were identified by automated systems (API, VITEK) as A. turicensis The bacterial identification was confirmed by 16S rRNA PCR and subsequent nucleic acid sequencing. No bacterial growth was detected in blood culture bottles after 5 days of incubation. Hence, multiple antibacterial treatments and surgical intervention the patient passed away. CONCLUSIONS: Anaerobes are rarely involved in CNS infections therefore anaerobic culture of CSF samples is routinely not performed. However, anaerobic bacteria should be considered as potential pathogens when certain risk factors are present, such as paranasal sinusitis, mastoiditis in patients with poor socioeconomic condition. To the best of our knowledge, our case report is the first description of A. turicensis meningitis that has been diagnosed as consequence of purulent mastoiditis.

Actinomyces cavernous sinus infection: a case and systematic literature review.

A 63-year-old man presented with a 2-month history of progressive right-sided exophthalmos, painful ophthalmoplegia and fevers. As more features developed, he was diagnosed with giant cell arteritis, then Tolosa-Hunt syndrome, and transiently responded to corticosteroids. A bland cerebrospinal fluid and highly metabolically active brain (18F)-fluoro-D-glucose-positron emission tomography suggested lymphoma. Biopsy of the mass showed sulphur granules with Gram-positive filamentous bacteria with Actinomyces-like colonies. Actinomyces cavernous sinus infections are rare and indolent. They often mimic non-infective causes including other inflammatory and infiltrative conditions, vascular and neoplastic causes, particularly lymphoma. Clinicians should consider infective cavernous sinus syndromes in people with a fluctuating painful ophthalmoplegia that responds poorly to corticosteroids. The term Tolosa-Hunt syndrome is problematic and should be retired or used only with reservation.

Colonic obstruction secondary to Actinomyces.

Actinomycosis is an uncommon granulomatous infection by Gram-positive anaerobic bacteria of the genus Actinomyces. A. israelii is a major human pathogen. The most frequent locations for colonization are cervicofacial (50%), abdominal (20%) and thoracic (15-20%). The abdominal actinomycosis predisposing factors include recent surgery, trauma and neoplasias. Certain cases have been associated with the intrauterine contraception device (IUD).

Cardiac Tamponade Due to Actinomyces odontolyticus Originating From a Dentigerous Cyst.

This report describes a rare case of pericardial effusion owing to Actinomyces odontolyticus in a 52-year-old woman that originated from a dentigerous cyst, which developed on the distal aspect of a lower left third molar. The cyst had remained asymptomatic for a long period, with no specific functional complications. This is the first case report of a patient with acute pericarditis in which the same strain of A odontolyticus was detected in an asymptomatic dentigerous cyst and in the pericardial fluid.

Periodontitis-associated septic pulmonary embolism caused by Actinomyces species identified by anaerobic culture of bronchoalveolar lavage fluid: a case report.

BACKGROUND: Periodontal disease is a less common but important cause of septic pulmonary embolism (SPE). However, the pathogens causing periodontal disease-associated SPE (PD-SPE) have been poorly understood. Actinomyces species are resident microbiota in the oral cavity. Here we report a case of PD-SPE caused by Actinomyces species, which was identified by anaerobic culture of bronchoalveolar lavage fluid (BAL). CASE PRESENTATION: A 64-year-old Asian man, complicated with severe chronic periodontitis, was admitted with chest pain and fever. Chest CT revealed multiple bilateral pulmonary nodules located subpleurally. We diagnosed the case as SPE associated with periodontitis. Although blood cultures were negative for the usual 5-day incubation, anaerobic culture of the BAL fluid sample yielded Actinomyces species. Antibacterial therapy alone did not ameliorate the symptoms; however, additional dental treatment, including tooth extraction, promptly did. The patient was discharged 23 days after admission. The 3-month follow-up revealed no recurrence of the symptoms and complete resolution of the lung lesions. CONCLUSION: This case demonstrated that Actinomyces species can cause PD-SPE. Additionally, clinicians should consider performing appropriate anaerobic culture of BAL fluid to identify the pathogen of SPE, and to ordering dental treatment, if necessary, in addition to antibiotics for the initial management of PD-SPE.

Actinomyces osteomyelitis in bisphosphonate-related osteonecrosis of the jaw (BRONJ): the missing link?

Bisphosphonate-related osteonecrosis of the jaw (BRONJ) is a rare complication of bisphosphonate treatment characterized by the development of exposed, necrotic bone in the jaw with inflammatory signs. The pathogenesis of BRONJ is not yet fully understood. This review analyzes the evidence supporting the hypothesis that BRONJ may be considered as a bisphosphonate-induced Actinomyces infection of the jaw according to the modified Koch's postulates. The main arguments relies on the following factors: (1) the high prevalence of isolation of Actinomyces from bone BRONJ lesions (73.2 % in retrospective series); (2) the similar pathological appearance of BRONJ and Actinomyces osteomyelitis in most studies, although BRONJ lesions without inflammation have been reported; (3) the high incidence of events that disrupt the normal mucosal barrier as a necessary trigger to develop BRONJ in bisphosphonate-exposed patients; (4) the predilection of bisphosphonate-induced osteonecrosis for the bones of the jaws; and (5) the favorable response of BRONJ on treatment that is active on Actinomyces. If BRONJ confirms to be a bisphosphonate-induced Actinomyces osteomyelitis of the jaw, this has major consequences for the prevention and treatment of this condition.

Acute primary actinomycosis involving the hard palate of a diabetic patient.

Actinomycosis is a relatively rare infection caused by saprophytic bacteria of the oral cavity and gastrointestinal tract that can become pathogenic. The chronic hyperglycemia of diabetes mellitus induces events that promote structural changes in various tissues and are associated with problems in wound healing. This infection remains largely unknown to most clinicians because of its different presentations, and palatal involvement is extremely rare. This report describes the case of a 46-year-old woman who was diagnosed with actinomycosis involving the hard palate. The main clinical, histopathologic, and therapeutic characteristics and differential diagnosis of actinomycosis are reviewed. To date, 3 cases of actinomycosis involving the hard palate have been reported.