Cervicofacial Actinomycosis in the Pediatric Population: Presentation and Management.

BACKGROUND: Infection caused by Actinomyces species is a rare cause of head and neck infection in children. This chronic cervicofacial infection can present with localized swelling, abscess formation, sinus drainage and can be complicated by osteomyelitis. METHODS: Presented are 2 pediatric cases of secondary actinomycosis in the context of congenital lesions: 1 patient with a previously excised preauricular sinus and another with a persistent sublingual mass. A comprehensive literature search was conducted for reported cases of pediatric actinomycosis in the cervicofacial region. RESULTS: Both cases presented were successfully treated with a combination of complete surgical excision of the lesions and prolonged antibiotic therapy. Thirty-four pediatric cases of cervicofacial actinomycosis are reviewed, 2 presented herein, and 32 from the published literature. There was equal gender distribution and the median age was 7.5 years. The most common site for infection was the submandibular area. Four (12%) of cases arose in pre-existing congenital lesions. Most patients were treated with penicillin-based antibiotics for a median duration of 6 months following surgical excision or debridement. CONCLUSIONS: Actinomycosis is a rare infection of the cervicofacial region; secondary infections arising from congenital lesions of the head and neck are even more rare. A previously excised pre-auricular sinus and a sublingual dermoid cyst are not previously reported sites of infection. Actinomycosis should be suspected in chronically draining sinuses of the head and neck region and confirmed through anaerobic culture. Osteomyelitis is a potential complication and magnetic resonance (MR) imaging is warranted. Long-term antibiotic therapy with a penicillin-based antibiotic and surgical excision should be considered.

Pediatric Cervicofacial Actinomycosis: Lessons From a Craniofacial Unit.

Actinomycosis is a rare disease that remains difficult to diagnose and manage. Prompted by 2 recent cases the authors sought evidence-based conclusions about best practice. A systematic review was conducted using standard PRISMA methodology. The study was registered prospectively (PROSPERO: CRD42018115064). Thirty-three children from 23 series are described. The mean age was 8 years (range 3-17). Fifty-five percent were female. Twenty cases involved bone (usually mandible); 13 cases involved cervicofacial soft tissue. Poor dental hygiene and oral trauma were implicated. The median diagnostic delay was 12 weeks (range 1-156 weeks). The median duration of definitive antibiotic therapy was 17 weeks (range 1-130 weeks). Although diagnostic delay did not correlate with number of surgeries, bony involvement was associated with more procedures (P = 0.008, unpaired t test). All (6) cases with residual infection had bony involvement (P = 0.06, Fisher exact test). Neither diagnostic delay nor number of surgeries significantly influenced infection-free outcome which, instead, relies on aggressive surgical debridement and prolonged antibiotic therapy. Mandibular involvement exhibits a higher surgical burden and chronicity in around a third of cases. As dental caries are implicated in mandibular disease, preventative strategies must focus on improving pediatric oral hygiene.

Treatment of cervicofacial actinomycosis: a report of 19 cases and review of literature.

OBJECTIVES: Actinomycosis is a chronic suppurative granulomatous infection caused by the Actinomyces genus. Orocervicofacial actinomycosis is the most common form of the disease, seen in up to 55% of cases. All forms of actinomycosis are treated with high doses of intravenous penicillin G over two to six weeks, followed by oral penicillin V. Large studies on cervicofacial actinomycosis are lacking. Therefore proper guidelines for treatment and treatment duration are difficult to establish. The aim of this study is to establish effective treatment and treatment duration for orocervicofacial actinomycosis. STUDY DESIGN: A Pubmed and Embase search was performed with the focus on treatment and treatment duration for cervicofacial actinomycosis. The hospital records of all patients presenting to our department with head and neck infection from January 2000 to December 2010 were reviewed, retrospectively. The following data were collected: age, gender, clinical presentation, aetiology, duration of symptoms, microbiological findings, treatment, and duration of treatment. The treatment and treatment duration is subsequently compared to the literature. RESULTS: The literature search provided 12 studies meeting the inclusion criteria. All studies were retrospective in nature. Penicillin or amoxicillin/clavulanic acid are the preferred antibiotic regimens found in the literature. Most of our patients were treated with a combination of penicillin G 12 million units/day and metronidazol 500 mg 3/day, most commonly for a duration of 1 - 4 weeks, being shorter than the 3 - 52 weeks reported in the literature. CONCLUSION: When actinomycosis is suspected, our review has shown that a surgical approach in combination with intravenous penicillin and metronidazol until clinical improvement is seen, followed by oral antibiotics for 2 - 4 weeks is generally efficient.

Osteomielitis mandibular por actinomices: Reporte de caso / Mandibular osteomyelitis by actinomyces: Case report

Se reporta el caso de un paciente varón de 27 años con antecedente de exodoncia compleja de la tercera molar mandibular inferior derecha que presentaba una tumefacción circunscrita dolorosa en la región submandibular ipsilateral. En el estudio imaginológico, la radiografía panorámica presentaba un proceso osteolítico periapical e interradicular con un patrón osteogénico circundante por lo que se utilizó como complemento una tomografía cone beam, en donde se evidenció compromiso desde la rama mandibular derecha hasta la sínfisis mandibular. Las lesiones a nivel de tejidos blandos y tejidos duros fueron diagnosticadas y manejadas como una actinomicosis cervicofacial y osteomielitis crónica supurativa respectivamente. El conocimiento de las diferentes infecciones bacterianas, un adecuado análisis clínico y estudio imaginológico nos darán la pauta para la correcta terapéutica de las distintas patologías maxilofaciales.

A Case of a 27-years old male with a history of complex extraction of right mandibular third molar that had a painful circumscribed swelling in the ipsilateral submandibular region. In imaging studies, the ortopantograph showed a periapical and interradicular osteolytic process with a osteogenic pattern surrounding, so we used a Cone beam Tomography as complement where we found commitment from the right ramus to the mandibular symphsis. Damage at soft and hard tissue were diagnosed and managed as a cervicofacial actinomycosis and supurative chronic osteomyelitis respectively. The knowledge of different bacterial infections, appropriate clinical analysis and imaging studies will lead us to the correct therapy of various maxillofacial pathologies.

Tongue abscess with actinomycosis.

Actinomycosis of the tongue is uncommon, but without proper treatment, it causes extensive tissue destruction. A mass that may mimic both benign and malignant neoplasms can be seen at clinical presentation and may mislead the diagnosis. Early diagnosis is critical but usually difficult with cultures or imaging. We report a patient who presented with a tumor-like deeply localized primary actinomycosis of the tongue with its magnetic resonance imaging findings.

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.

[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.

Cervicofacial actinomycosis and mandibular osteomyelitis.

Cervicofacial actinomycosis should be included in the differential diagnosis of any soft tissue swelling in the head and neck region, particularly if malignancy or a granulomatous disease is suspected. The diagnosis often is overlooked because of this entity's ability to mimic other conditions. Mandibular osteomyelitis also is underappreciated by many clinicians in their assessment of head and neck infections. Most cases are traced to an odontogenic source, with periapical tooth abscess and posttraumatic or surgical complication as key antecedent events.

Unusual case of resistant actinomycosis following facial trauma.

A case of actinomycosis is reported which appeared as a complication of a ski-pole injury of the face. A 16-year-old girl was treated for 10 years for resistant actinomycosis. She took long-term antimicrobial therapies, underwent repeated excisions, hyperbaric oxygen therapy, repeated skin grafting, immunological treatment, and free flap transfer. The conservative antibiotic treatment was complicated by mycosis and pseudomembranous colitis. After the defect healed up, satisfactory contour of the face was restored with additional free flap transfer. The patient has also been satisfied with the final aesthetic result. The authors highlight the importance of early diagnosis and proper antimicrobial treatment in combination with surgical excisions of granulation tissue preserving vital structures.

[Base tongue actinomycosis]. / Actinomicosis de base de lengua.

Actinomycosis is a bacterial, suppurative chronic infectious disease caused by Actinomyces israelii. Actinomycosis of the tongue is an uncommon form, and only 3% affect the base of the tongue. We report a case with dysphagia from an epiglottis, vallecula and tongue base mass. Diagnosis was made on histologic examination of a tissue biopsy. The disease completely resolved after chirurgical incision and drainage of the abscess and one month of oral amoxicillin therapy. The recurrence of the disease, five years later, made necessary a second surgical procedure and one year of penicillin therapy. We comment the possible causes of this recurrence.

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.

[A classical form of actinomycoses involving the mouth, face bones, orbit and base of the cranium in an African patient]. / Une forme historique d'actinomycose impliquant la cavité buccale, les os de la face, l'orbite et la base du crâne chez un patient africain.

Osteitis due to actinomycosis is now an uncommon clinical entity in industrialized countries. This report describes a classic case of imported actinomycosis involving the buccal cavity, facial bones, orbit and skull base. For two years after the onset, various diagnoses followed by different medical as well as surgical treatments were unsuccessfully attempted first in Senegal then in Italy. Proper diagnosis was finally established in Marseille, France, on the basis of histological findings after extensive surgical biopsy. The patient was treated with high-dose intravenous penicillin G for four weeks followed by daily oral administration of amoxicillin for one year. Surgical reconstruction could not be carried out before the patient's return to Senegal. The pitfalls of diagnosis and treatment of actinomycosis are discussed.

Actinomicosis cervicofacial / Cervicofacial actinomycosis

Hemos considerado oportuna la presentación de 4 casos clínicos de Actinomicosis Cervicofacial vistos en la Cátedra de Dermatología de la UNT en el período 1999-2000, dada la baja frecuencia de presentación y la confusión con fístulas dentarias. Además queremos destacar la importancia del trabajo multidisciplinario (Odontólogo, Dermatólogo y Micólogo) para solucionar en totalidad el problema.

Actinomicosis cervicofacial / Cervicofacial actinomycosis

Hemos considerado oportuna la presentación de 4 casos clínicos de Actinomicosis Cervicofacial vistos en la Cátedra de Dermatología de la UNT en el período 1999-2000, dada la baja frecuencia de presentación y la confusión con fístulas dentarias. Además queremos destacar la importancia del trabajo multidisciplinario (Odontólogo, Dermatólogo y Micólogo) para solucionar en totalidad el problema. (AU)

Actinomicosis. Consideraciones clínicas, diagnósticas y terapéuticas. A propósito de dos casos / Actinomycosis. Clinical, diagnostic and therapeutic considerations. About two cases

Las infecciones por actinomices afectan de forma prioritaria a la región cervicofacial, según las series del 32-55 por ciento del total se desarrollan a ese nivel. Las especies que con más frecuencia las provocan son Actinomyces israelii (78 por ciento) y Arachnia propiónica. Clásicamente se describen formas de actinomicosis de presentación aguda o de desarrollo crónico. Presentamos dos casos de actinomicosis desarrollados en pacientes varones de edad media, provenientes de medio rural. Describimos en primer lugar un caso provocado por el Actinomyces meyeri, en la forma clínica de presentación aguda. Se manifiesta bajo la forma de absceso laterocervical del lado derecho. El segundo caso clínico está localizado en el dorso de lengua y suelo de boca, provocado por el Actinomyces odontolíticus. Su desarrollo acontece lentamente simulando un tumor en ambas localizaciones. Aunque ambos casos corresponden a prototipos clínicos de la actinomicosis los actinomices que las provocan raramente son citados en la literatura médica al respecto (AU)

[Report of two cases of actinomycosis of the neck, one acute and one chronic].

We report two cases of actinomycosis of the neck, one acute and one chronic. The acute actinomycosis patient was a 63-year-old woman who complained of high fever, sore throat, and swelling of her neck on her first visit. Plain CT revealed gas-formation in the soft tissues of the left parapharyngeal space, the hyoid area and the cartilage thyroid area. We performed emergency tracheotomy and surgically drained her neck. No actinomycotic bodies were found microscopically in the surgically resected material, but cultures of the neck pus grew out actinomyces. A diagnosis of actinomycosis was made, and the patient was treated with administration of antibiotics, an ASPC drip infusion for about 7 weeks and oral BAPC for about 6 months, and the lesion was improved. The patient has been symptom-free for 4 years since the operation. The chronic actinomycosis patient was a 61-year-old woman who complained of swelling in the right submandibular region. Surgical resection was performed to exclude the possibility of a malignant tumor. Actinomycotic bodies were detected microscopically in the surgically resected material. The patient was treated with oral AMPC for about 6 months, and she has been symptom-free for 1 year since the operation.

Cervicofacial actinomycosis after partial mandibular resection: case report.

An unusual case of cervicofacial actinomycosis in a 60-year old male Nigerian is reported. The patient had had partial mandibular resection for plexiform ameloblastoma one year earlier with insertion of Kirschner wire to serve as a splint. The authors highlight the possible pathophysiology of this condition. It is suggested that clinicians be aware of the various modes of presentation and natural history of the disease to facilitate prompt diagnosis and appropriate treatment. This will assist in forestalling the numerous complications that may result from actinomycosis.

Cervicofacial actinomycosis with paravertebral spread: a case report.

We report a case of cervicofacial actinomycosis with paravertebral extension in a 60-year-old man who presented with recurrent neck masses. Diagnosis was confirmed on culture and histology of pus and debris obtained from surgical drainage. He improved only after lengthy in-hospital high dose penicillin therapy. He is currently well and is on maintenance doxycycline therapy for 6 months following the high dose penicillin therapy.

[Mucormycosis-actinomycosis and caseous dental sinusitis associated with sinusal foreign body]. / Mucormycose-actinomycose et sinusite caséeuse dentaire avec corps étranger intra-sinusien.

The authors report an uncommon case of caseous dental sinusitis with infection or superinfection by mocorales and actinomycetes. The patient was healthy, without diabetes mellitus, but was treated for lymphoma 18 years before. This observation leads to a discussion of nosologic, diagnostic and therapeutic features of caseous sinusitis, dental or not, fungal or not, which are often or too often called aspergillosis sinusitis. The recovery of the patient was complete after surgery, amphotericin B and hyperbaric oxygen.