Actinomycosis of the Tongue: A Case Report and Review of Literature.
Actinomycosis is an uncommon, chronic, suppurative bacterial infection affecting soft-tissues caused by an anaerobic, filamentous Gram-positive bacterium, Actinomyces israelii. It was recognized for the first time in humans in about 1845 by Von Langenbeck .
Actinomycosis is classified by location as cervicofacial (50-60%), abdominal (25%), or pulmonary (15%) lesions. Cervicofacial actinomycosis is the most frequent clinical form, and the "lumpy jaw syndrome", associated with odontogenic infection, is the most common clinical manifestation . In the oral cavity, the infection commonly occurs in the mandibular area (submandibular region, ramus, and angle) in over 50% of cases [3,4], followed by cheek, parotid gland, tongue, nasal cavity, hypopharynx, gingival and oral space. Actinomycosis is associated with large abscesses and/or mandibular osteomyelitis, with or without the presence of the sinus tract . Actinomyces spp. usually have a low pathogenicity and cause disease in a condition of poor oral hygiene, after surgical procedures and/or dental treatments such as extraction of a mandibular molar or as a complication of maxillofacial trauma.
In 1885, Von Hacker described the first case of actinomycosis of the tongue , which remains a very uncommon lesion of the oral cavity, representing less than 3% of all reported cases of actinomycosis in English literature [6,7]. Although it is a benign condition, if not treated, it can give, in later stages, osteomyelitis with extensive bone destruction [8,9]. Actinomycosis of soft tissues may extend to adjacent bone in up to 15% of cases [10,11].
Actinomycosis especially affects male patients aged 20 to 60 years . The main predisposing factors include diabetes, alcohol abuse, malnutrition, and radiation therapy and immunosuppression . Furthermore, patients affected by human immunodeficiency virus (HIV) had often shown to have poor outcomes [13,14],
The diagnosis of actinomycosis of soft tissues is based on clinical findings and on histological examination and staining of the lesion. The presence of sulphur granules in tissue sample or the pus drainage help during diagnosis, although nocardiosis may also present with sulphur granules [15-17], The clinical diagnosis of tongue actinomycosis may present difficulties because of its ability to mimic other lesions, including both benign and malignant neoplasms. The histopathological examination is decisive to arrive to the final diagnosis and exclude other diseases.
Here, we describe the case of a patient who presented with a tumor-like lingual swelling, then diagnosed as actinomycosis of the tongue. A review of lingual actinomycosis cases is also reported.
2. Case Report
A 52-year-old man was presented at our clinical unit (Oral Medicine Unit, Odontostomatologia II, ASST Santi Paolo e Carlo, Presidio Ospedaliero San Paolo) because of an asymptomatic, not-ulcerated swelling of the anterior part of the tongue. The patient reported a recent occurrence of the lesion. In his clinical history, the patient declared that, about 5 years before, a biopsy was performed approximately at the same location of the current lesion, with a diagnosis of benign squamous papilloma. He did not report any previous history of local trauma or abdominal, thoracic, or cervical abscesses.
Intraoral examination revealed a solid, fixed, bilobed submucosal nodule, located slightly on the right to the midline of the anterior part of the tongue (Figure 1a,b).
The lingual mucosa appeared normal in color, with two flat whitish spots and a small rounded erosion (about 2 mm in diameter). There was no restriction in tongue movement. Extraoral examination showed no evidence of cervical lymphadenopathy.
Hematological examination revealed neutrophilia, monocytosis, and a raised value of c-reactive protein (CRP), with a concentration of 3.5 mg/dL (normal range <0.5 mg/dL).
The clinical diagnosis was of a benign neoplasm, with a differential diagnosis including traumatic lesion, infective lesion or malignant neoplasm.
Under local anesthesia, two incisional biopsies of the lesion were taken for achieving the histopathological diagnosis--the first one of the epithelial layer and the other of the submucosa--in order to obtain a representative section. During the biopsy, a yellowish purulent drainage coming from the lesion was observed.
Three weeks after the biopsy, the patients received systemic antibiotic therapy because of a dental extraction. He was treated with oral intake of clarithromycin for one week (500 mg every 12 h), due to penicillin allergy.
Post-surgical healing was excellent, with the complete resolution of swelling at six weeks follow-up, when the histological result was given to the patient (Figure 2).
Histopathological examination of the biopsy specimen, indeed, showed a nodular mass deep into the muscularis propria of the tongue, showing colonies of actinomyces and a chronic and acute inflammatory process, including suppurative signs (Figure 3).
Lingual actinomycosis is rare. In Table 1, case reports/series available from the literature are reviewed, with particular attention to patient management [4,6,18-30].
In humans, the etiological agent of this disease is mainly Actinomyces israelii, a filamentous Gram-positive bacilli, commensal of the flora of the oropharynx, gastrointestinal tract and urogenital tract. In the oropharynx, it is particularly present in gingival sulcus, tonsillar crypts, dental plaques, and periodontal pockets. Consequently, actinomycosis is considered anend ogenous infection, where the trauma plays a catalytic role in starting the disease [6,7,29].
The microorganism is unable to penetrate healthy tissue, while, to become invasive, it requires the interruption of mucosal barrier to gain access to the submucosal tissue . Actinomycosis of the tongue is an uncommon disease since lingual epithelial layer has a structure particularly resistant to local trauma and infection. The keratinized mucosal lining, vascular-rich parenchyma, great mobility, and mechanical cleansing by saliva make difficult for bacteria to adhere and multiply.
Lingual actinomycosis is generally localized on the anterior two-thirds of the tongue, lateral to the medline , as occurred in this patient. When, after the trauma, such as self-biting (Table 1), the microorganism spreads deeply into the tissues and produces a massive fibrotic reaction surrounding the center of the lesion. Clinically, lingual actinomycosis appears as a hard nodular mass or swelling, slightly mobile on the adjacent layers, which can be rarely ulcerated and associated with necrotic tissue (Table 1). Although we could not retrieve specific information regarding recent traumas of the dorsum of the tongue, the patient reported a previous biopsy (with a diagnosis of squamous papilloma) at the same site of the current nodular lesion, which might have played a role in triggering the actinomycosis.
Pain, dysphagia, speech impairment, difficulty in moving the tongue can be reported by the patient (Table 1), although, in this report, the lesion was asymptomatic.
The gold standard for the final diagnosis is the histological examination including the histological staining to detect Actinomyces spp. colonies, whenever possible, also performed on purulent material. Bacterial culture is not recommended because it remains sterile in just about 50% of cases . Typical microscopic findings include identification of Actinomyces spp. colonies and sulphur granules that are made of Gram-positive conglomeration of bacteria trapped in biofilm . In our case, the diagnosis was achieved by the clinical findings and the identification of Actinomyces spp. colonies within the epithelial specimen.
The differential diagnosis should include other infections (lingual abscess, nocardiosis, botryomycosis), granulomatous lesions, infected cyst, pyogenic abscess, and benign and malignant neoplasms [29-31].
The preferred treatment remains administration of antibiotics with surgical excision or incision of the lesion. The drainage of abscess or surgical excision, when the lesion is small, largely enhance the efficacy of antibiotic therapy . The drug of choice is penicillin. The addition of beta-lactamase inhibitors or metronidazole gives benefits with recurrent and polymicrobial Actinimycosis infections . Other therapies include administration of third-generation cephalosporin or, in case of patients allergic to amoxicillin, macrolides and clindamycin [2,3]. In literature, there is no agreement on the ideal duration of therapy. Some studies suggested the need of long therapies, from weeks to months. Recurrence may occur after the cessation of the antibiotic , especially when the therapy was incomplete or with insufficient duration. No studies described local or distant recurrence of lingual actinomycosis after being treated successfully with drainage and a complete cycle of antibiotics (Table 1). In this case, the patient showed complete resolution of the clinical picture, lasting one month from the biopsy with pus drainage and antibiotic 1-week treatment with clarithromycin.
Although lingual actinomycosis infections is a rare event, early diagnosis and treatment are pivotal to avoid severe and life-threatening cases. Because of its ability to mimic other diseases, especially neoplasms, actinomycosis can be a challenging problem for the clinician and always requires diagnostic investigations, including biopsy.
This infection must be considered in the differential diagnosis of any cervicofacial mass. Treatment should always include pus drainage and systemic antibiotic therapy.
Received: 11 February 2020; Accepted: 11 March 2020; Published: 16 March 2020
Author Contributions: Conceptualization, E.M.V. and F.D.; investigation, R.F., F.D., L.M.; writing--original draft preparation, F.D.; writing--review and editing, E.M.V., N.L., L.M., A.S.; supervision, A.S. and G.L. All authors have read and agreed to the published version of the manuscript.
Funding: This research received no external funding.
Conflicts of Interest: The authors declare no conflict of interest.
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Fiorella D'Amore (1), Roberto Franchini (1), Laura Moneghini (2), Niccolo Lombardi (3) [ID], Giovanni Lodi (3), Andrea Sardella (3) and Elena M. Varoni (3), * [ID]
(1) UOC Odontostomatologia II, ASST Santi Paolo e Carlo--Presidio Ospedaliero San Paolo, 20142 Milano, Italy; firstname.lastname@example.org (F.D.); email@example.com (R.F.)
(2) UOC Anatomia Patologica, Citogenetica e Patologia Molecolare; ASST Santi Paolo e Carlo--Presidio Ospedaliero San Paolo, 20142 Milano, Italy; firstname.lastname@example.org
(3) Dipartimento di Scienze Biomediche, Chirurgiche ed Odontoiatriche, Universita degli Studi di Milano, 20142 Milano, Italy; email@example.com (N.L.); firstname.lastname@example.org (G.L.); email@example.com (A.S.)
* Correspondence: firstname.lastname@example.org; Tel.: +39-025031017; Fax: +39-025031041
Caption: Figure 1. Tongue nodular lesion, as observed at first examination: the localized swelling on the lingual dorsum was covered by normal mucosa, (a) Frontal view of the lesion, (b) lateral view of the lesion.
Caption: Figure 2. Complete resolution of the lesion at six weeks, after incisional biopsy with pus drainage and one-week antibiotic therapy.
Caption: Figure 3. Histopathological examination of the lesion. At small magnification (20x; H&E) the oral mucosa shows acute and chronic intense inflammatory infiltration and, at one side of the sample, two aggregates (a) better assessed at high magnification (60x) (b) and after PAS staining (c) consistent with Actinomyces spp. colonies. The colonies are composed of Actinomyces spp. filaments, called "sulfurs granules", surrounded by neutrophilic granulocytes.
Table 1. Studies reporting cases of actinomycosis of the tongue. Reference Oral Site Clinical Appearance (Number of Patients) SADEGHI, 2018 Tongue Macroglossia  ANEJA, 2017 Tongue Nodular mass  JAT, 2017 Tongue Nodular mass  ROCHA, 2017 Tongue Necrotic tissue with purulent  right lateral tongue developed after sclerosing agent injection to treat vascular lesion ESCODA, 2013 Tongue Two ulcerated lesions  KURTARAN, Tongue Solid mass 2011  HABIBI, 2008 Tongue Solid mass  ATESPARE, 2006 Tongue solid mass  VAZQUEZ, 1997 Tongue Swelling  GERBINO, 1996 Tongue Nodules  (n = 2) FICARRA, 1993 Tongue Swelling  ISALSKA, 1991 Tongue Swelling  BRIGNALL, 1989 Tongue Swelling  KUEPPER, 1979 Tongue Mass  UHLER, 1972 Tongue Nodular mass  SODAGAR, 1972 Tongue Solid mass  * Reference Symptoms Triggering Factors SADEGHI, 2018 No No  ANEJA, 2017 No Tongue bite one month  before JAT, 2017 No No  ROCHA, 2017 No No  ESCODA, 2013 Pain No  KURTARAN, Pain Dental prosthesis 2011 problem 1 month before  HABIBI, 2008 No Tongue bite  ATESPARE, 2006 No No  VAZQUEZ, 1997 No No  GERBINO, 1996 NA NA  FICARRA, 1993 No No  ISALSKA, 1991 Local discomfort No  BRIGNALL, 1989 Acute discomfort Accidental self-inflicted  bite to the tongue 6 months previously KUEPPER, 1979 NA NA  UHLER, 1972 NA NA  SODAGAR, 1972 NA NA  * Reference Other Associated Signs SADEGHI, 2018 Dysphagia and speech  impairment ANEJA, 2017 Difficulty in speech  JAT, 2017 Poor oral hygiene and  dental caries ROCHA, 2017 No  ESCODA, 2013 Erosive lichen  asymptomatic KURTARAN, Dysphagia and speech 2011 impairment  HABIBI, 2008 No  ATESPARE, 2006 Speech disturbance  VAZQUEZ, 1997 No  GERBINO, 1996 NA  FICARRA, 1993 No  ISALSKA, 1991 No  BRIGNALL, 1989 Lost normal movement  and dysphagia KUEPPER, 1979 NA  UHLER, 1972 NA  SODAGAR, 1972 NA  * Reference Treatment SADEGHI, 2018 Penicillin for 22 weeks  ANEJA, 2017 Excision and  amoxicillin-clavulanic acid for 2 weeks JAT, 2017 Excision. Doxycycline for 3  weeks ROCHA, 2017 Local debridement  ESCODA, 2013 Clyndamicin (unknown  posology) and chlorhexidine digluconate for 1 month KURTARAN, Excision and combination of 2011 amoxicillin-clavulanic acid and  metronidazole for 5 weeks HABIBI, 2008 Excision and intravenus  penicillin for 3 weeks ATESPARE, 2006 Excision and  amoxicillin-clavulanic acid for 3 weeks VAZQUEZ, 1997 Amoxicillin for 4 weeks  GERBINO, 1996 Penicillin V for 1 month  FICARRA, 1993 Penicillin for 2 weeks  ISALSKA, 1991 Amoxycillin for 6 month  BRIGNALL, 1989 Henoxy-methyl-penicillin for 3  month KUEPPER, 1979 Penicillin for 1 month  UHLER, 1972 Excision and penicillin for 6  months SODAGAR, 1972 Excision  * * Full-text not available, only abstract; NA = not available information.
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|Author:||D'Amore, Fiorella; Franchini, Roberto; Moneghini, Laura; Lombardi, Niccolo; Lodi, Giovanni; Sardella|
|Date:||Mar 1, 2020|
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