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J Oral Maxillofac Surg 70:1030-1034, 2012 Endoscopic Removal of Bilateral Supernumerary Intranasal Teeth Toshinori Iwai, DDS,* Noriaki Aoki, DDS, PhD,† Yosuke Yamashita, DDS,‡ Susumu Omura, DDS, PhD,§ Yoshiro Matsui, DDS, PhD, Jiro Maegawa, MD, PhD,¶ and Iwai Tohnai, DDS, PhD# Supernumerary teeth occur in 0.1% to 1% of the general population. 1 Dentists and oral-maxillofacial surgeons (OMSs) sometimes encounter a supernumer- ary tooth, and the most common is the mesiodens; a supernumerary intranasal tooth is very rare. 2-5 Bilat- eral supernumerary intranasal teeth are extremely rare, 2 and the literature contains only a few reports. 3,4 Because most intranasal teeth are found by otolaryn- gologists based on patients presenting with nasal symptoms, dentists and OMSs have less opportunity to treat them. We report a rare case of bilateral su- pernumerary intranasal teeth removed under endo- scopic guidance. Report of Case A 27-year-old man was referred to our department for a foul smell in the nasal cavity. The patient had no history of dentomaxillofacial trauma or surgery, craniofacial anoma- lies or syndromes, or infection, such as syphilis or osteomy- elitis of the maxilla. Clinical examination with a nasal spec- ulum showed hard white masses in the nasal cavities bilaterally. Intraoral examination showed normal dentition and no abnormalities. A panoramic radiograph showed 32 permanent teeth and a supernumerary intranasal tooth (Fig 1), and facial radiographs (posterior-anterior and lateral ra- diographs) and computed tomography (CT) showed a su- pernumerary intranasal tooth in each nasal cavity (Figs 2-5). The patient underwent transnasal endoscopic removal of bilateral supernumerary intranasal teeth under general an- esthesia. First, we placed pharyngeal packing gauze to pre- vent any teeth from dropping to the pharynx after orotra- cheal intubation. We used a 4-mm, 0° rigid endoscope (Olympus Medical Systems, Tokyo, Japan) for good visual- ization and minimally invasive surgical intervention (Figs 6, 7). The intranasal teeth could be removed completely by use of forceps under endoscopic guidance (Figs 8, 9). The right intranasal tooth had a canine crown and dilacerated root; the left intranasal tooth had a conical crown and straight root (Figs 10, 11). The patient’s postoperative course was uneventful, and his symptoms resolved after the teeth were removed. Discussion Ectopic teeth occur in various sites such as the mandibular condyle, 6 coronoid process, 7 maxillary sinus, 8 orbit, 9 palate, 10 facial skin, 11 and nasal cavity. 2-5 Intranasal teeth are uncommon, and septal perforation of an intranasal tooth was reported as an extremely rare case. 12,13 Supernumerary teeth are more commonly seen intranasally than deciduous or permanent teeth. 14-16 Although the etiology of intra- nasal tooth eruption is unclear, the theories include developmental disturbances such as cleft palate, den- tomaxillofacial trauma, cysts, infection (syphilis or osteomyelitis of the maxilla), eruption due to crowd- ing of dentition, persistent deciduous teeth, or dense bone. 4,16-19 However, in our case, an etiologic factor for the supernumerary intranasal teeth has not been identified. Symptoms and signs associated with intranasal teeth include facial pain, 20,21 external nasal deformity, 22 nasal discharge, 5,20 nasal discomfort, 20 foul smell, 12 nasal ob- struction, 5,8,12,14,15,23 rhinorrhea, 8,14,15,21 headache, 23 rhinitis, 14 recurrent epistaxis, 15,21 and oronasal fis- tula. 16 Sometimes an intranasal tooth is asymptom- atic, 1,14,17 and such an intranasal tooth may be inciden- *Assistant Professor, Department of Oral and Maxillofacial Sur- gery, Yokohama City University Graduate School of Medicine, Yo- kohama, Japan. †Chief Doctor, Department of Oral and Maxillofacial Surgery, Saiseikai Yokohamashi Nanbu Hospital, Yokohama, Japan. ‡Staff Doctor, Department of Oral and Maxillofacial Surgery, Saiseikai Yokohamashi Nanbu Hospital, Yokohama, Japan. §Associate Professor, Department of Oral and Maxillofacial Sur- gery, Yokohama City University Medical Center, Yokohama, Japan. Professor and Chairman, Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Kagawa University, Kita-gun, Japan. ¶Associate Professor, Department of Plastic and Reconstructive Surgery, Yokohama City University Hospital, Yokohama, Japan. #Professor and Chairman, Department of Oral and Maxillofacial Surgery, Yokohama City University Graduate School of Medicine, Yokohama, Japan. Address correspondence and reprint requests to Dr Iwai: 3-9 Fukuura, Kanazawa-ku, Yokohama, Kanagawa 236-0004, Japan; e-mail: [email protected] © 2012 American Association of Oral and Maxillofacial Surgeons 0278-2391/12/7005-0$36.00/0 doi:10.1016/j.joms.2011.10.014 1030

Endoscopic Removal of Bilateral Supernumerary Intranasal Teeth

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Page 1: Endoscopic Removal of Bilateral Supernumerary Intranasal Teeth

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J Oral Maxillofac Surg70:1030-1034, 2012

Endoscopic Removal of BilateralSupernumerary Intranasal Teeth

Toshinori Iwai, DDS,* Noriaki Aoki, DDS, PhD,†

Yosuke Yamashita, DDS,‡ Susumu Omura, DDS, PhD,§

Yoshiro Matsui, DDS, PhD,� Jiro Maegawa, MD, PhD,¶ and

Iwai Tohnai, DDS, PhD#

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Supernumerary teeth occur in 0.1% to 1% of thegeneral population.1 Dentists and oral-maxillofacialsurgeons (OMSs) sometimes encounter a supernumer-ary tooth, and the most common is the mesiodens; asupernumerary intranasal tooth is very rare.2-5 Bilat-ral supernumerary intranasal teeth are extremelyare,2 and the literature contains only a few reports.3,4

Because most intranasal teeth are found by otolaryn-gologists based on patients presenting with nasalsymptoms, dentists and OMSs have less opportunityto treat them. We report a rare case of bilateral su-pernumerary intranasal teeth removed under endo-scopic guidance.

Report of Case

A 27-year-old man was referred to our department for afoul smell in the nasal cavity. The patient had no history ofdentomaxillofacial trauma or surgery, craniofacial anoma-lies or syndromes, or infection, such as syphilis or osteomy-elitis of the maxilla. Clinical examination with a nasal spec-

*Assistant Professor, Department of Oral and Maxillofacial Sur-

gery, Yokohama City University Graduate School of Medicine, Yo-

kohama, Japan.

†Chief Doctor, Department of Oral and Maxillofacial Surgery,

Saiseikai Yokohamashi Nanbu Hospital, Yokohama, Japan.

‡Staff Doctor, Department of Oral and Maxillofacial Surgery,

Saiseikai Yokohamashi Nanbu Hospital, Yokohama, Japan.

§Associate Professor, Department of Oral and Maxillofacial Sur-

gery, Yokohama City University Medical Center, Yokohama, Japan.

�Professor and Chairman, Department of Oral and Maxillofacial

Surgery, Faculty of Medicine, Kagawa University, Kita-gun, Japan.

¶Associate Professor, Department of Plastic and Reconstructive

Surgery, Yokohama City University Hospital, Yokohama, Japan.

#Professor and Chairman, Department of Oral and Maxillofacial

Surgery, Yokohama City University Graduate School of Medicine,

Yokohama, Japan.

Address correspondence and reprint requests to Dr Iwai: 3-9

Fukuura, Kanazawa-ku, Yokohama, Kanagawa 236-0004, Japan;

e-mail: [email protected]

© 2012 American Association of Oral and Maxillofacial Surgeons

278-2391/12/7005-0$36.00/0

oi:10.1016/j.joms.2011.10.014

1030

ulum showed hard white masses in the nasal cavitiesbilaterally. Intraoral examination showed normal dentitionand no abnormalities. A panoramic radiograph showed 32permanent teeth and a supernumerary intranasal tooth (Fig1), and facial radiographs (posterior-anterior and lateral ra-diographs) and computed tomography (CT) showed a su-pernumerary intranasal tooth in each nasal cavity (Figs 2-5).The patient underwent transnasal endoscopic removal ofbilateral supernumerary intranasal teeth under general an-esthesia. First, we placed pharyngeal packing gauze to pre-vent any teeth from dropping to the pharynx after orotra-cheal intubation. We used a 4-mm, 0° rigid endoscope(Olympus Medical Systems, Tokyo, Japan) for good visual-ization and minimally invasive surgical intervention (Figs 6,7). The intranasal teeth could be removed completely byuse of forceps under endoscopic guidance (Figs 8, 9). Theright intranasal tooth had a canine crown and dilaceratedroot; the left intranasal tooth had a conical crown andstraight root (Figs 10, 11). The patient’s postoperativecourse was uneventful, and his symptoms resolved after theteeth were removed.

Discussion

Ectopic teeth occur in various sites such as themandibular condyle,6 coronoid process,7 maxillaryinus,8 orbit,9 palate,10 facial skin,11 and nasalavity.2-5 Intranasal teeth are uncommon, and septal

perforation of an intranasal tooth was reported as anextremely rare case.12,13 Supernumerary teeth are

ore commonly seen intranasally than deciduous orermanent teeth.14-16 Although the etiology of intra-asal tooth eruption is unclear, the theories includeevelopmental disturbances such as cleft palate, den-omaxillofacial trauma, cysts, infection (syphilis orsteomyelitis of the maxilla), eruption due to crowd-

ng of dentition, persistent deciduous teeth, or denseone.4,16-19 However, in our case, an etiologic factor

for the supernumerary intranasal teeth has not beenidentified.

Symptoms and signs associated with intranasal teethinclude facial pain,20,21 external nasal deformity,22 nasaldischarge,5,20 nasal discomfort,20 foul smell,12 nasal ob-struction,5,8,12,14,15,23 rhinorrhea,8,14,15,21 headache,23

rhinitis,14 recurrent epistaxis,15,21 and oronasal fis-tula.16 Sometimes an intranasal tooth is asymptom-

atic,1,14,17 and such an intranasal tooth may be inciden-
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IWAI ET AL 1031

tally discovered when routine clinical examination isperformed on patients with other symptoms such asotitis.1 Therefore most intranasal teeth are found in

atients presenting for nasal symptoms to otolaryn-ologists, and dentists and OMSs have less opportu-ity to treat this rare condition. However, we shoulde cognizant and knowledgeable about intranasaleeth because routine radiographic examination forental treatment may show a radiopaque lesion in theasal cavity.16,17

FIGURE 1. Panoramic radiograph showed 32 perm

wai et al. Endoscopic Removal of Intranasal Teeth. J Oral Maxi

FIGURE 2. Frontal view of facial radiograph sho

wai et al. Endoscopic Removal of Intranasal Teeth. J Oral Maxillofac S

Because supernumerary intranasal teeth are veryrare, most of the literature is limited to case reportsand case series. Therefore we investigated supernu-merary intranasal teeth in the English-language litera-ture. The review of Kirmeier et al4 between January959 and January 2008 and our further investigationntil July 20115 showed 26 well-documented cases of

supernumerary intranasal teeth, including the presentcase and the case of Kirmeier et al. The age when thesupernumerary intranasal teeth were diagnosed

teeth and a supernumerary intranasal tooth (arrow).

urg 2012.

ilateral supernumerary intranasal teeth (arrows).

anent

wed b

urg 2012.

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1032 ENDOSCOPIC REMOVAL OF INTRANASAL TEETH

ranged from 6 to 61 years (mean, 25.8 � 13.4 years).There were more cases of supernumerary intranasalteeth found in male patients (62%) than in femalepatients (38%), and symptoms associated with super-numerary intranasal teeth occurred in 84.6% of cases.In most cases of supernumerary intranasal teeth, thereis a unilateral single tooth in the nasal cavity,4,14,15 butMartinson and Cockshott24 reported multiple super-

umerary intranasal teeth in 1 nasal cavity as an ex-remely rare case. To our knowledge, there have beennly 2 cases of bilateral supernumerary intranasaleeth,3,4 with our case constituting the third report. Ournvestigation of 25 cases without a median supernumer-ry intranasal tooth (septal perforation)12 showed super-

FIGURE 3. Lateral view of facial radiograph showed bilateralsupernumerary intranasal teeth (arrows).

Iwai et al. Endoscopic Removal of Intranasal Teeth. J Oral Max-illofac Surg 2012.

FIGURE 4. CT showed bilateral supernumerary intranasal teeth(arrows).

Iwai et al. Endoscopic Removal of Intranasal Teeth. J Oral Max-

illofac Surg 2012.

umerary intranasal teeth present slightly more often onhe left than the right.

The diagnosis of an intranasal tooth can be con-rmed clinically and radiologically. An intranasalooth is often a hard white mass clinically and isometimes covered completely by nasal mucosa17 and

surrounded by granulation tissue and necrotic de-bris.12,15,16,23 Radiography is useful for the diagnosisbecause intranasal teeth are identified as radiopaquelesions. Although panoramic radiographs can providedetailed information about the condition of dentitionand whether the intranasal tooth is supernumerary,

FIGURE 5. CT showed bilateral supernumerary intranasal teeth(arrows).

Iwai et al. Endoscopic Removal of Intranasal Teeth. J Oral Max-illofac Surg 2012.

FIGURE 6. Endoscopic view of right supernumerary intranasalteeth.

Iwai et al. Endoscopic Removal of Intranasal Teeth. J Oral Max-

illofac Surg 2012.
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IWAI ET AL 1033

deciduous, or a permanent tooth,14 such radiographsre not always sufficient to identify intranasal teeth offhe midline.4 In our case a panoramic radiograph

showed only a supernumerary intranasal tooth. How-ever, bilateral supernumerary intranasal teeth couldbe confirmed by facial (posterior-anterior and lateral)radiographs and CT. Because conventional radiologic

FIGURE 7. Endoscopic view of left supernumerary intranasalteeth.

Iwai et al. Endoscopic Removal of Intranasal Teeth. J Oral Max-illofac Surg 2012.

FIGURE 8. Endoscopic removal of left supernumerary intranasaltooth.

Iwai et al. Endoscopic Removal of Intranasal Teeth. J Oral Max-

illofac Surg 2012. i

examination may not be able to confirm supernumer-ary intranasal teeth precisely, CT is useful to identifysupernumerary intranasal teeth.4,15 Although the dif-erential diagnosis of an intranasal white mass should

FIGURE 9. Endoscopic removal of left supernumerary intranasaltooth.

Iwai et al. Endoscopic Removal of Intranasal Teeth. J Oral Max-illofac Surg 2012.

FIGURE 10. Right supernumerary intranasal teeth.

wai et al. Endoscopic Removal of Intranasal Teeth. J Oral Max-

llofac Surg 2012.
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1034 ENDOSCOPIC REMOVAL OF INTRANASAL TEETH

include nasal foreign body, rhinolith, bony seques-trum, neoplasm, and exostosis,13,14,16 the intranasalooth can be diagnosed with comparative ease by CT.

Supernumerary intranasal teeth should be removeds soon as they are detected because of potentialorbidity.12,20 However, in children the most appro-riate time for removal is when the roots of theermanent teeth have completely formed, to mini-ize the risk of developmental injury to the denti-

ion.15,23 Although some intranasal teeth may beasymptomatic, such intranasal teeth should be re-moved or at least followed radiographically.12,17,25

Although supernumerary intranasal teeth may be re-moved by a transnasal approach8,12,14,21 or intraoralpproach22 according to site of the intranasal teeth,

we should extract supernumerary intranasal teethcarefully to avoid damaging surrounding tissues.20

The transnasal approach is often less invasive, but thisapproach under direct vision with a nasal speculumand head light cannot provide sufficient visualizationto remove supernumerary intranasal teeth in the pos-terior region of the nasal cavity. To overcome theproblem, endoscopy has recently been used to re-move intranasal teeth as a minimally invasive sur-gery.5,14,15,17 Endoscopic removal of intranasal teethcan provide good illumination, better visualization,

FIGURE 11. Left supernumerary intranasal teeth.

wai et al. Endoscopic Removal of Intranasal Teeth. J Oral Max-llofac Surg 2012.

and precise dissection with preservation of surround-

ing tissues compared with a conventional ap-proach.5,14,17 Furthermore, use of endoscopy for re-

oval of such teeth can reduce morbidity, becausehe endoscopic approach is more effective and saferhan a conventional approach.17 We recommend rou-inely using a rigid endoscope for removal of intrana-al teeth.

References1. Thawley SE, LaFerriere KA: Supernumerary nasal tooth. Laryn-

goscope 87:1770, 19772. Sokolov M, Jecker P, Roth Y: Nasal teeth associated with

rhinosinusitis. Rhinology 42:167, 20043. Quinn JH, Lewis M: Bilateral inverted supernumerary central

incisors penetrating nasal cavity: Report of case. J Oral SurgAnesth Hosp Dent Surv 17:61, 1959

4. Kirmeier R, Truschnegg A, Payer M, et al: The supernumerarynasal tooth. Int J Oral Maxillofac Surg 38:1219, 2009

5. Sanei-Moghaddam A, Hyde N, Williamson P: Endoscopic re-moval of a supernumerary tooth from the nasal cavity in anadult. Br J Oral Maxillofac Surg 47:484, 2009

6. Gadre KS, Waknis P: Intra-oral removal of ectopic third molarin the mandibular condyle. Int J Oral Maxillofac Surg 39:294,2010

7. Sutton PR: Migrating nonerupted mandibular premolars: A caseof migration into the coronoid process. Oral Surg Oral MedOral Pathol 25:87, 1968

8. Pracy JP, Williams HO, Montgomery PQ: Nasal teeth. J LaryngolOtol 106:366, 1992

9. Sjöberg S, Lörinc P: Intracranial supernumerary tooth. Casereport. Radiologe 24:561, 1984

10. Gans BJ: Ectopic tooth: Report of a case. J Oral Surg AnesthHosp Dent Serv 20:435, 1962

11. Abdin Bey M: Eruption of a third molar through the skin.Quintessence Int (Berl) 1:17, 1970

12. Lee JH: A nasal tooth associated with septal perforation: A rareoccurrence. Eur Arch Otorhinolaryngol 263:1055, 2006

13. Rao AB: Aberrant canine tooth in the nose. J Laryngol Otol67:370, 1953

14. Lee FP: Endoscopic extraction of an intranasal tooth: A reviewof 13 cases. Laryngoscope 111:1027, 2001

15. Lin IH, Hwang CF, Su CY, et al: Intranasal tooth: Report ofthree cases. Chang Gung Med J 27:385, 2004

16. Smith RA, Gordon NC, De Luchi SF: Intranasal teeth. Report oftwo cases and review of the literature. Oral Surg Oral Med OralPathol 47:120, 1979

17. Kim DH, Kim JM, Chae SW, et al: Endoscopic removal of anintranasal ectopic tooth. Int J Pediatr Otorhinolaryngol 67:79,2003

18. Ogisi FO, Odita JC: Ectopic nasal dentition associated withsquamous cell carcinoma of palate in a 12-year-old boy. Br JOral Maxillofac Surg 26:58, 1988

19. Rege SR, Shah KL, Marfatia PT: Osteomyelitis of maxilla withextrusion of teeth in the floor of the nose requiring extraction.J Laryngol Otol 84:533, 1970

20. Chopra SS, Joshi MR: Mesiodens erupted in the nasal cavity.Report of a case. Oral Surg Oral Med Oral Pathol 28:856, 1969

21. Nastri AL, Smith AC: The nasal tooth. Case report. Aust Dent J41:176, 1996

22. Hong CY: Ectopic nasal tooth. Med J Malaysia 30:239, 197623. Murty PS, Hazarika P, Hebbar GK: Supernumerary nasal teeth.

Ear Nose Throat J 67:128, 198824. Martinson FD, Cockshott WP: Ectopic nasal dentition. Clin

Radiol 23:451, 197225. Carver DD, Peterson S, Owens T: Intranasal teeth: A case

report. Oral Surg Oral Med Oral Pathol 70:804, 1990