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Actinomycosis of the Tongue: A Case Report and Review of Literature.

1. Introduction

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 [1].

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 [2]. 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 [2]. 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 [5], 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 [12]. The main predisposing factors include diabetes, alcohol abuse, malnutrition, and radiation therapy and immunosuppression [3]. 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).

3. Discussion

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 [31]. 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 [4], 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 [2]. Typical microscopic findings include identification of Actinomyces spp. colonies and sulphur granules that are made of Gram-positive conglomeration of bacteria trapped in biofilm [2]. 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 [29]. The drug of choice is penicillin. The addition of beta-lactamase inhibitors or metronidazole gives benefits with recurrent and polymicrobial Actinimycosis infections [32]. 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 [30], 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.

4. Conclusions

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.

doi: 10.3390/antibiotics9030124

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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[14.] Chaudhry, S.I.; Greenspan, J.S. Actinomycosis in HIV infection: A review of a rare complication. Int. J. STD AIDS 2000, 11, 349-355. [CrossRef]

[15.] Park, J.K.; Lee, H.K.; Ha, H.K.; Choi, H.Y.; Choi, C.G. Cervicofacial actinomycosis: CT and MR imaging findings in seven patients. Am. J. Neuroradiol. 2003, 24, 331-335.

[16.] Volante, M.; Contucci, A.M.; Fantoni, M.; Ricci, R.; Galli, J. Cervicofacial actinomycosis: Still a difficult differential diagnosis. Acta Otorhinolaryngol. Ital. 2005, 25, 116-119.

[17.] Balatsouras, D.G.; Kaberos, A.K.; Eliopoulos, P.N.; Kandiloros, D.; Economou, C.N. Cervicofacial actinomycosis presenting as an acute upper respiratory tract obstruction. J. Laryngol. Otol. 1994, 108, 801-803. [CrossRef]

[18.] Ahmed, S.; Ali, M.; Adegbite, N.; Vaidhyanath, R.; Avery, C. Actinomycosis of tongue: Rare presentation mimicking malignancy with literature review and imaging features. Radiol. Case Rep. 2018, 14, 190-194. [CrossRef]

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[20.] Sodagar, R.; Kohout, E. Actinomycosis of tongue as pseudotumor. Laryngoscope 1972, 82, 2149-2152. [CrossRef]

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[26.] Rocha, O.K.M.S.; de Franga, G.M.; Oliveira, P.T.; Santos, Miguel, M.C.C.; Silveira, E.J.D. Unusual presentation of oral actinomycosis in the tongue: Case report. J. Bras. Patol. Med. Lab. 2017, 53, 330-333. [CrossRef]

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[28.] Aneja, T.; Bhat, N.; Chawla, G.; Negi, M. Actinomycosis of tongue: Case report of an atypical presentation. IOSR J. Dental Med. Sci. 2017, 16, 84-86.

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[32.] Smith, A.J.; Hall, V.; Thakker, B.; Gemmell, C.G. Antimicrobial susceptibility testing of Actinomyces species with 12 antimicrobial agents. J. Antimicrob. Chemother. 2005, 56,407-409. [CrossRef]

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; fiura@hotmail.it (F.D.); rohfranchini@gmail.com (R.F.)

(2) UOC Anatomia Patologica, Citogenetica e Patologia Molecolare; ASST Santi Paolo e Carlo--Presidio Ospedaliero San Paolo, 20142 Milano, Italy; laura.moneghini@asst-santipaolocarlo.it

(3) Dipartimento di Scienze Biomediche, Chirurgiche ed Odontoiatriche, Universita degli Studi di Milano, 20142 Milano, Italy; niccolo.lombardi@unimi.it (N.L.); giovanni.lodi@unimi.it (G.L.); andrea.sardella@unimi.it (A.S.)

* Correspondence: elena.varoni@unimi.it; 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
[24]
ANEJA, 2017      Tongue                 Nodular mass
[28]

JAT, 2017        Tongue                 Nodular mass
[27]
ROCHA, 2017      Tongue                 Necrotic tissue with purulent
[26]                                    right lateral tongue
                                        developed after sclerosing
                                        agent injection to treat
                                        vascular lesion
ESCODA, 2013     Tongue                 Two ulcerated lesions
[23]

KURTARAN,        Tongue                 Solid mass
2011
[22]
HABIBI, 2008     Tongue                 Solid mass
[31]
ATESPARE, 2006   Tongue                 solid mass
[7]

VAZQUEZ, 1997    Tongue                 Swelling
[29]
GERBINO, 1996    Tongue                 Nodules
[4]              (n = 2)
FICARRA, 1993    Tongue                 Swelling
[31]
ISALSKA, 1991    Tongue                 Swelling
[21]
BRIGNALL, 1989   Tongue                 Swelling
[19]

KUEPPER, 1979    Tongue                 Mass
[6]
UHLER, 1972      Tongue                 Nodular mass
[25]
SODAGAR, 1972    Tongue                 Solid mass
[20] *

Reference        Symptoms           Triggering Factors

SADEGHI, 2018    No                 No
[24]
ANEJA, 2017      No                 Tongue bite one month
[28]                                before

JAT, 2017        No                 No
[27]
ROCHA, 2017      No                 No
[26]

ESCODA, 2013     Pain               No
[23]

KURTARAN,        Pain               Dental prosthesis
2011                                problem 1 month before
[22]
HABIBI, 2008     No                 Tongue bite
[31]
ATESPARE, 2006   No                 No
[7]

VAZQUEZ, 1997    No                 No
[29]
GERBINO, 1996    NA                 NA
[4]
FICARRA, 1993    No                 No
[31]
ISALSKA, 1991    Local discomfort   No
[21]
BRIGNALL, 1989   Acute discomfort   Accidental self-inflicted
[19]                                bite to the tongue 6
                                    months previously
KUEPPER, 1979    NA                 NA
[6]
UHLER, 1972      NA                 NA
[25]
SODAGAR, 1972    NA                 NA
[20] *

Reference        Other Associated Signs

SADEGHI, 2018    Dysphagia and speech
[24]             impairment
ANEJA, 2017      Difficulty in speech
[28]

JAT, 2017        Poor oral hygiene and
[27]             dental caries
ROCHA, 2017      No
[26]

ESCODA, 2013     Erosive lichen
[23]             asymptomatic

KURTARAN,        Dysphagia and speech
2011             impairment
[22]
HABIBI, 2008     No
[31]
ATESPARE, 2006   Speech disturbance
[7]

VAZQUEZ, 1997    No
[29]
GERBINO, 1996    NA
[4]
FICARRA, 1993    No
[31]
ISALSKA, 1991    No
[21]
BRIGNALL, 1989   Lost normal movement
[19]             and dysphagia

KUEPPER, 1979    NA
[6]
UHLER, 1972      NA
[25]
SODAGAR, 1972    NA
[20] *

Reference        Treatment

SADEGHI, 2018    Penicillin for 22 weeks
[24]
ANEJA, 2017      Excision and
[28]             amoxicillin-clavulanic acid for
                 2 weeks
JAT, 2017        Excision. Doxycycline for 3
[27]             weeks
ROCHA, 2017      Local debridement
[26]

ESCODA, 2013     Clyndamicin (unknown
[23]             posology) and chlorhexidine
                 digluconate for 1 month
KURTARAN,        Excision and combination of
2011             amoxicillin-clavulanic acid and
[22]             metronidazole for 5 weeks
HABIBI, 2008     Excision and intravenus
[31]             penicillin for 3 weeks
ATESPARE, 2006   Excision and
[7]              amoxicillin-clavulanic acid for
                 3 weeks
VAZQUEZ, 1997    Amoxicillin for 4 weeks
[29]
GERBINO, 1996    Penicillin V for 1 month
[4]
FICARRA, 1993    Penicillin for 2 weeks
[31]
ISALSKA, 1991    Amoxycillin for 6 month
[21]
BRIGNALL, 1989   Henoxy-methyl-penicillin for 3
[19]             month

KUEPPER, 1979    Penicillin for 1 month
[6]
UHLER, 1972      Excision and penicillin for 6
[25]             months
SODAGAR, 1972    Excision
[20] *

* 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
Publication:Antibiotics
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Date:Mar 1, 2020
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