Intranasal supernumerary tooth: two case reports and review of the literature

Abstract

The presence of supernumerary tooth (SNT) in the nasal cavity is a rare condition with limited literature data. We report two cases with a history of nasal obstruction and difficulty breathing. In both cases, clinical and radiological examination confirmed intranasal SNT. Extractions were executed in general anesthesia using Rochester-Pean instruments transnasally. In addition, a literature review of intranasal SNT was performed. The database search retrieved a total number of 50 cases in time period from 1970 to 2020. Mean age of patients was 22.5 years. Most common symptoms were unilateral obstruction of breathing and headache. Surgical extraction of intranasal SNT is recommended to eliminate the symptoms.

Introduction

The presence of the tooth in the nasal cavity is an uncommon clinical finding. Because of its rare occurrence, the literature is limited, usually based on individual case reports or small case series. The cause of this rare anomaly may be either an aberration of the regular dentition or a supernumerary tooth (SNT) (1). Different theories about the intranasal SNT exist, but none have been proven to date (2). It usually deviates morphologically from the biologically normal teeth presenting most frequently as a conical form.

The prevalence of the SNT in the general population is 0.1-1%, without sex and age related predilection (2). It is most commonly ffound on routine examination as a random finding. The true occurrence of intranasal SNT is difficult to predict because of its relative indolent and asymptomatic course. Its anatomical position in the nasal cavity may compress surrounding structures and consequently produce symptoms. The most common symptoms are the unilateral obstruction of breathing, the appearance of unpleasant nasal odors, nasal bleeding and infection, headache, middle-face pain and crusting of nasal mucosa (3). Diagnostic procedures sufficient to set up the diagnosis and to determine the extension of intranasal process include inspection and radiological examination. Differential diagnosis could potentially include foreign body in the nasal cavity, calcifying tumors, cysts and rhinoliths (4).

Different therapeutic methods in the treatment of the intranasal SNT have been noted (5-9). Recent reports put emphasis on endoscopic treatment as an effective replacement of traditional surgery (2). The aims of this paper were to report two new cases of the intranasal SNT and to make a literature review on the existing subject.

Clinical Case Reports

Case report 1

A 46-year-old male presented to our institution with a difficulty in nasal breathing. He reported minor, undefined disturbances in the right nasal cavity which had persisted for several years. His medical history included soft tissue facial trauma in a childhood without any systemic diseases. Family history revealed that his daughter was several times surgically treated for a SNT but insufficient medical documentation was provided for an assessment. The patient had a fixed partial denture in the anterior maxilla for 5 years. MSCT examination revealed SNT extending from the right osseous nasal floor to the area of right nasal cavity. The tooth was surrounded by radio-opaque material. It was decided to treat the patient in general anesthesia. Extraction was performed transnasally using simple instruments (Rochester-Pean forceps) (Figure 1). The postoperative period passed without surgical complications with a full relief of symptoms. A three-year follow-up period was uneventful.

Figure 1.

Clinical features of Case report 1. a,b) MSCT showing SNT extending from the right osseous nasal floor to the area of right nasal cavity. c) Exposure provided by nasal speculum. d) SNT after removal.

Case report 2

A 60-year-old male presented to our institution with difficult left-sided nasal breathing which persisted for two months. His medical history excluded trauma and his family history was unsuspicious. His dentition was clinically normal, without prosthetic rehabilitation or any other dental treatment. MSCT examination revealed a diagonally positioned SNT in the lower nasal corridor. The tooth was surrounded by thick mucosa and multiple calcified, nodular, sharply bound radio-opaque materials. The nasal septum slightly deviated to the left side. It was decided to treat the patient in general anesthesia. Extraction was performed transnasally using simple instruments (Rochester-Pean forceps) (Figure 2). The postoperative period passed without surgical complications with a full recovery. A three-year follow-up period was uneventful.

Figure 2.

Clinical features of Case report 2. a,b) MSCT showing nasal tooth and slight deviation of the nasal septum c) nasal speculum examination d) Tooth and sharply bound radio-opaque material after removal.

Literature Review

The studies and analyses examined in the present paper are a result of text mining and database searching through Medline/PubMed database, as well as individual journal search for all results retrieved by searches of general terms such as ''supernumerary tooth’’, “inverted supernumerary tooth”, “sino-nasal cavity tooth”, “intranasal supernumerary tooth”, “supernumerary teeth” and their combination. PubMed cross-references were used to obtain additional articles. Only English written articles reporting intranasal SNT were analyzed.

The database search retrieved a total number of 50 cases in time period from 1970 to 2020. Mean age of the patients was 22.5 years (ranging from 2 to 64 years) with male sex dominance (33/50, 66%). The majority of patients (66%) had symptoms and the most common symptom was unilateral obstruction of breathing. The nasal floor was the most frequent localization of the SNT in the nasal cavity while the most common side was the left side. Intranasal SNT had also been reported in patients with craniofacial anomalies such as cleft lip, alveolus, and palate. Clinicopathologic characteristics of included patients are presented in Table 1.

Table 1. Clinicopathologic characteristics of reviwed cohort.

Authors	Number of cases	Gender	Age	Symptoms	Approach
Kohli and Verma, 1970.5	1	Male	13	Yes	Nasal speculum
Arora et al., 1973.6	1	Male	14	Yes	Nasal speculum
Sood and Kakar, 1975.7	1 of 2	Male	28	No	Not reported
	2of 2	Male	12	Yes	Not reported
Thawley et al., 1977.3	1	Female	25	No	Patient denied operation
Smith et al., 1979.8	1 of 2	Male	14	No	Nasal speculum
	2 of 2	Female	34	Yes	Patient denied operation
Murty et al., 1988.9	1	Male	30	Yes	Nasal speculum
Pracy et al., 1992.10	1	Male	30	Yes	Nasal speculum
Nastri ett Smith, 1996.11	1	Female	18	Yes	Nasal speculum
Chamyal PC, 1997.12	1	Male	19	Yes	Maxillary approach
Chen et al., 2002.13	1	Male	8	Yes	Endoscope
Kim et al., 2003.14	1	Male	12	No	Endoscope
Kuroda et al., 2003.15	1	Male	27	Yes	Nasal Speculum
Lin et al., 2004.16	1of 3	Female	16	Yes	Endoscope
	2 of 3	Male	21	Yes	Endoscope
	3 of 3	Female	16	Yes	Endoscope
Sokolov et al., 2004.17	1 of 2	Female	22	Yes	Not reported
	2 of 2	Female	36	Yes	Microscope
Lee, 2006.18	1	Male	61	Yes	Endoscope
Kirmeier et al., 2009.1	1	Female	49	Yes	Microscope
Janardhan et al., 2012.19	1	Male	30	Yes	Endoscope
Iwai et al., 2012.4	1	Male	27	Yes	Endoscope
Krishnans et al., 2013.20	1	Female	13	Yes	Endoscope
Mohebbi et al., 2013.21	1	Male	19	Yes	Not reported
Van Essen and Van Rijswijk, 2013.22	1	Male	26	Yes	Nasal speculum
Dhaferi et al., 2014.23	1	Male	22	Yes	Endoscope

Discussion

The presence of an intranasal tooth is a rare clinical finding. It is important to distinguish SNT from the aberrations of normal deciduous or permanent teeth which present a separate entity. As an aberration, teeth or a tooth, usually present in the cranio-facial or systemic anomalies such as cleft palate, Gardner's syndrome and cleidocranial dysostosis (3, 10-33). There are some reports where the presence of multiple supernumerary teeth is non-syndrome associated (33, 34). Early childhood orofacial trauma can also be a consequence of the intranasal tooth persistence (2). The evolutionary theories about SNT are debatable due to largely unexploited biological and genetic mechanisms. The atavistic theory that assumes the existence of the third dental lamina in the region of premaxilla is abandoned because there have not been any pathological and clinical confirmations (35). Some authors consider the hypothesis that a possible etiological factor for the development of a SNT might entail origination from an additional local splinting or independent hyperactivity of the dental lamina (3). The inverted mesiodens that has erupted in nasal cavity as a SNT was discussed by some authors (36, 37). There are reports presenting the embryonic theory of lagging in the migration of the fronto-nasal neural crest cells before the end of the fifth embryonic week causing ectopic development of tooth germs in premaxilla (38, 39). Our first case report suggests, through the family history, that hereditary factors could be involved. A similar consideration was reported by Shafer, W.C., Hine, M.K. and Levy, B. M as well as by Anthonappa RP, King NM, Rabie AB (38, 39).

In 1886, Marshal was the first to describe typical symptoms of an intranasal tooth such as nasal obstruction and strong headache (40). Meanwhile, different symptoms and signs indicating the presence of supernumerary teeth had been reported, including unilateral obstruction, chronic nasal discharge, crusting of nasal mucosa, nasal pain, epistaxis, facial pain and headache, perforation of nasal septum (17, 19, 23). However, intranasal SNT may be asymptomatic and only incidentally recognized during routine clinical or radiologic examination. Differential diagnoses of an intranasal dense radiopaque shadow include a foreign body, bony sequestrum, neoplasm, exostosis and rhinolith.

The diagnosis of nasal tooth is mainly based on clinical examination and imaging methods. Considering an optimal time for SNT removal, it seems reasonable to do it when the roots of the permanent tooth have completely formed in order to avoid their injury. While surgical extraction of SNT is a standard method of treatment, no recommendations exist regarding optimal surgical approach. The most frequent approach described in literature is extraction of the tooth transnasally with a speculum or with an endoscopic assistance. Some authors advocate extraction of intranasal SNT endoscopically as a standard procedure because the endoscope enables a clear visualization of the tooth’s insertion and allows a precise dissection (2, 29, 31).

In conclusion, intranasal SNT is a rare condition with headache and nasal obstruction as leading symptoms. A differential diagnosis is important to exclude possible temporary nasal obstacles and neoplasms. Clinical examination and radiography are sufficient diagnostic methods. Minimally invasive surgical extraction with an endoscopic assistance transnasally is an optimal method in the treatment of intranasal SNT.