Chronic rhinorrhea revealing an actinomycotic rhinolithiasis with ectopic tooth

Abstract

Intranasal ectopic tooth is a rare nidus for a rhinolith where local infection may be concomitant. No description of the triple association ‘actinomycotic rhinolithiasis ectopic tooth’ could be found in the medical literature. Classically, the Actinomyces species are sensitive to regimens of penicillin from 6 to 12 months or longer. Immunocompetent patients can benefit from shorter courses of antibiotic therapy, such as ciprofloxacin, with a favourable outcome. The authors describe the case of a 25-year-old man who presented with an actinomycosis chronic discharge revealing actinomycosis associated with rhinolithiasis and ectopic tooth. They attempt to explain the likely mechanism of occurrence of this triple association.

Rhinolithiasis is a disorder formed from salt deposition on an intranasal endogenous or exogenous material. Actinomycosis is a chronic disease caused by non-spore-forming species of the genus Actinomyces , which occurs mostly in the cervicofacial region as a chronic granulomatous infection. The authors describe a case of actinomycosis involving the nasal cavity in association with rhinolithiasis and ectopic tooth and attempt to explain the likely mechanism of occurrence of this triple association.

Case report

This 25-year-old immunocompetent man was referred to the authors’ clinic for one-sided nasal obstruction, chronic nasal discharge unresponsive to treatment with multiple antibiotics, and episodic headache with epistaxis. His general medical history was unremarkable, there was no history of foreign body insertion into his right nasal cavity.

Anterior rhinoscopy and nasal endoscopy revealed mucosal inflammation with purulent rhinorrhea with a greyish, hard and irregularly surfaced mass in the right nasal cavity. The mass was attached to the nasal mucosa and surrounded by a layer of granulation tissue and normal nasal mucosa. On intraoral examination there was neither an oral–nasal fistula nor a missing tooth.

In order to obtain better localization of the lesion and determine its relationship with the surrounding anatomical structures, a maxillofacial CT scan was performed. It revealed dystrophic and homogeneous calcification (15 mm in diameter) in the right nasal cavity that could not be distinguished from the floor of the nasal cavity, with septal adhesion ( Fig. 1 ). There was a significant deviation of nasal septa to the left. There was no destruction or perforation of structures near the lesion. An infectious rhinolithiasis was suspected. The patient was not aware of any foreign mass in his nose and could not recall a prior maxillofacial trauma.

Fig. 1
Maxillofacial CT scan on axial (A) and coronal (B) views demonstrated a heterogeneous calcified lesion in the right nasal cavity between the right inferior turbinate and nasal septum. Note the mass adhesion to the floor of nasal cavity.

Endoscopic removal of the rhinolith was performed readily with rigid nasal endoscopy under topical anaesthesia via an anterior approach using forceps and suction. An intranasal canine was identified ( Fig. 2 ). Histological examination revealed that the specimen consisted of tooth and granulation tissue, calcification and bacteria colonies including the presence of Actinomyces species. Postoperatively, a 6-week regimen of ciprofloxacin was given with a good outcome.

Fig. 2
Maxillofacial CT scan on coronal (B) view showing ectopic tooth (right maxillary canine) (arrow).

Discussion

The pathogenesis of rhinoliths is not clear. They may develop on an endogenous nidus such as teeth, sequestra, dried blood clots, dried pus, desquamated epithelium, leukocytes, bone fragments and rarely ectopic tooth. Foreign bodies, such as fruit, seeds, beads, buttons, dirt, pebbles, sand, peas, parasites, wood, glass, and the remains of a gauze tampon are examples of exogenous sources . These nasal concretions can be surrounded by granulation tissues and debris as in this case.

The exact pathogenesis of intranasal teeth is not completely understood, but some theories have been proposed. These include the displacement of teeth by trauma to the oral cavity region, maxilla osteomyelitis, incomplete union of an embryonic process as in cleft palate and space restriction caused by crowding of dentition . The current theory explaining the aetiology of ectopic teeth is trauma . After mucous injury, a tooth may lodge into the nasal cavity. In the present case, the patient did not recall any history of a nasal trauma.

The most common presenting symptoms of rhinolithiasis and intranasal tooth are foetid purulent rhinorrhea and unilateral nasal obstruction that have persisted for many months or years, as in the present case. Other findings include a foul smell, pain, epistaxis, swelling of the nose or face, and headache.

The Actinomyces species are found worldwide, they are Gram-positive bacteria and commonly filamentous organisms that are facultative anaerobes in patients with predisposing factors (oral trauma, leukaemia, diabetes mellitus, renal insufficiency, steroid treatment, immunosuppression) . Actinomyces reside in the oral cavity of the normal hosts and are thought to become pathogenic by inoculation of the submucosal tissues following mucosal injury .

Actinomyces organisms are often underestimated as causes of infectious diseases because they are not routinely sought and are difficult to detect . A history and physical examination are always required for diagnosis, but the presenting symptoms of actinomycosis are confusing because they often mimic other disease processes, and a high index of suspicion is required to make an accurate and timely diagnosis. The diagnosis is usually confirmed by the isolation of Actinomyces in culture. Actinomyces can be difficult to isolate, especially when synergistic organisms are present. Occasionally, the diagnosis must be established on the basis of the presence of sulphur granules and the histologic findings on the removed specimen . In this case, actinomycosis and rhinolithiasis appeared to be related to a breach in the nasal mucosa by the ectopic canine.

Classically, Actinomyces species are sensitive to longer regimens of penicillin, from 6 to 12 months or more, depending on the site and severity of infection . Immunocompetent patients can receive a shorter course (2 month) of ciprofloxacin antibiotic therapy with a favourable outcome .

In conclusion, actinomycosis and rhinolithiasis should be considered in a patient with a long standing unilateral nasal cavity infection that is unresponsive to antibiotics, especially when associated with an ectopic tooth. Immunocompetent patients could benefit from a shorter course of antibiotic therapy, such as ciprofloxacin, but further studies will be needed to consider this treatment in a broader population.

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Jan 26, 2018 | Posted by in Oral and Maxillofacial Surgery | Comments Off on Chronic rhinorrhea revealing an actinomycotic rhinolithiasis with ectopic tooth
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