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Actinomycosis of the Tongue: a Case Report and Review of Literature

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Actinomycosis of the Tongue: a Case Report and Review of Literature antibiotics Article Actinomycosis of the Tongue: A Case Report and Review of Literature Fiorella D’Amore 1, Roberto Franchini 1, Laura Moneghini 2, Niccolò Lombardi 3 , Giovanni Lodi 3, Andrea Sardella 3 and Elena M. Varoni 3,* 1 UOC Odontostomatologia II, ASST Santi Paolo e Carlo—Presidio Ospedaliero San Paolo, 20142 Milano, Italy; fi[email protected] (F.D.); [email protected] (R.F.) 2 UOC Anatomia Patologica, Citogenetica e Patologia Molecolare; ASST Santi Paolo e Carlo—Presidio Ospedaliero San Paolo, 20142 Milano, Italy; [email protected] 3 Dipartimento di Scienze Biomediche, Chirurgiche ed Odontoiatriche, Università degli Studi di Milano, 20142 Milano, Italy; [email protected] (N.L.); [email protected] (G.L.); [email protected] (A.S.) * Correspondence: [email protected]; Tel.: +39-025031017; Fax: +39-025031041 Received: 11 February 2020; Accepted: 11 March 2020; Published: 16 March 2020 Abstract: Background: Actinomycosis of the tongue is an uncommon, suppurative infection of lingual mucosa, caused by actinomyces. The clinical diagnosis may present serious difficulties because of its ability to mimic other lesions, including both benign and malignant neoplasms. Methods: Here, we describe the case of a 52-years-old patient affected by an asymptomatic, tumor-like tongue swelling, then diagnosed as actinomycosis. A review of tongue localization of actinomycosis is also reported, with emphasis on clinical findings and therapy. Results and Conclusion: Early diagnosis and treatment, with pus drainage and systemic antibiotic therapy, are pivotal to avoid severe and life-threatening complications. Keywords: tongue actinomycosis; oral actinomycosis; tongue lesion; oral infection; oral lesion 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]. Antibiotics 2020, 9, 124; doi:10.3390/antibiotics9030124 www.mdpi.com/journal/antibiotics Antibiotics 2020, 9, 124 2 of 8 ActinomycosisAntibiotics 2020, 9, x especially affects male patients aged 20 to 60 years [12]. The main predisposing2 of 8 factors include diabetes, alcohol abuse, malnutrition, and radiation therapy and immunosuppression [3]. Actinomycosis especially affects male patients aged 20 to 60 years [12]. The main predisposing Furthermore, patients affected by human immunodeficiency virus (HIV) had often shown to have poor factors include diabetes, alcohol abuse, malnutrition, and radiation therapy and immunosuppression outcomes[3]. Furthermore, [13,14]. patients affected by human immunodeficiency virus (HIV) had often shown to have Thepoor diagnosisoutcomes [13,14]. of actinomycosis of soft tissues is based on clinical findings and on histological examinationThe anddiagnosis staining of actinomycosis of the lesion. of The soft presence tissues is ofbased sulphur on clinical granules findings in tissue and on sample histological or the pus drainageexamination help during and staining diagnosis, of the although lesion. The nocardiosis presence of may sulphur also presentgranules within tissue sulphur sample granules or the pus [15 –17]. The clinicaldrainage diagnosis help during of tonguediagnosis, actinomycosis although nocardiosis may present may also diffi prcultiesesent with because sulphur of its granules ability [15– to mimic other17]. lesions, The clinical including diagnosis both benignof tongue and actinomycosis malignant neoplasms.may present Thedifficulties histopathological because of its examination ability to is decisivemimic to arriveother tolesions, the final including diagnosis both and benign exclude and othermalignant diseases. 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 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. diagnosed as actinomycosis of the tongue. A review of lingual actinomycosis cases is also reported. 2. Case2. Case Report Report A 52-year-oldA 52-year-old man man was was presented presented at at our our clinical clinical unit unit (Oral(Oral Medicine Unit, Unit, Odontostomatologia Odontostomatologia II, ASSTII, Santi ASST Paolo Santi e Paolo Carlo, e PresidioCarlo, Presidio Ospedaliero Ospedalie Sanro Paolo) San Paolo) because because of an of asymptomatic, an asymptomatic, not-ulcerated not- swellingulcerated of the swelling anterior of part the ofanterior the tongue. part of Thethe tongue. patient The reported patient a reported recent occurrence a recent occurrence of the lesion. of the In his clinicallesion. history, In his the clinical patient history, declared the patient that, about declared 5 years that, about before, 5 years a biopsy before, was a performedbiopsy was performed approximately at theapproximately same location at of the the same current location lesion, of withthe current a diagnosis lesion, of with benign a diagnosis squamous of papilloma.benign squamous He did not reportpapilloma. any previous He did history not report of local any pr traumaevious orhistory abdominal, of local trauma thoracic, or abdominal, or cervical thoracic, abscesses. or cervical abscesses. Intraoral examination revealed a solid, fixed, bilobed submucosal nodule, located slightly on the Intraoral examination revealed a solid, fixed, bilobed submucosal nodule, located slightly on the right to the midline of the anterior part of the tongue (Figure1a,b). right to the midline of the anterior part of the tongue (Figure 1 a and b). (a) (b) FigureFigure 1. Tongue 1. Tongue nodular nodular lesion, lesion, as as observed observed atat firstfirst examination: examination: the the localized localized swelling swelling on the on lingual the lingual dorsumdorsum was was covered covered by by normal normal mucosa. mucosa. ( a(a)) FrontalFrontal view view of of the the lesion, lesion, (b) ( blateral) lateral view view of the of lesion. the lesion. Antibiotics 2020, 9, x 3 of 8 The lingual mucosa appeared normal in color, with two flat whitish spots and a small rounded Antibiotics 2020, 9, 124 3 of 8 erosion (about 2 mm in diameter). There was no restriction in tongue movement. Extraoral examination showed no evidence of cervical lymphadenopathy. The lingual mucosa appeared normal in color, with two flat whitish spots and a small rounded Hematological examination revealed neutrophilia, monocytosis, and a raised value of c-reactive erosion (about 2 mm in diameter). There was no restriction in tongue movement. Extraoral examination protein (CRP),showed with no evidence a concentration of cervical lymphadenopathy. of 3.5 mg/dl (normal range <0.5 mg/dl). The clinicalHematological diagnosis examination was of revealeda benign neutrophilia, neoplasm, monocytosis, with a anddifferential a raised value diagnosis of c-reactive including traumaticprotein lesion, (CRP), infective with a lesion concentration or malignant of 3.5 mg /neoplasm.dL (normal range <0.5 mg/dL). Under localThe clinical anesthesia, diagnosis two was ofincisional a benign neoplasm, biopsies with of a dithefferential lesion diagnosis were taken including for traumatic achieving the lesion, infective lesion or malignant neoplasm. histopathological diagnosis—the first one of the epithelial layer and the other of the submucosa—in Under local anesthesia, two incisional biopsies of the lesion were taken for achieving the order to histopathologicalobtain a representative diagnosis—the section. first one During of the epithelialthe biopsy, layer a and yell theowish other purulent of the submucosa—in drainage coming from theorder lesion to was obtain observed. a representative section. During the biopsy, a yellowish purulent drainage coming Threefrom weeks the lesion after was the observed. biopsy, the patients received systemic antibiotic therapy because of a dental extraction.Three weeksHe was after treated the
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