Non-syndromic multiple supernumerary teeth: meta-analysis

Non-syndromic multiple supernumerary teeth:meta-analysis

Joaquın Alvira-Gonzalez1, Cosme Gay-Escoda

1,2,3

1Oral Surgery and Implantology Department, Faculty of Dentistry, University of Barcelona, Barcelona, Spain; 2IDIBELL Institute,Barcelona, Spain; 3Oral and Maxillofacial Surgeon, Teknon Medical Center, Barcelona, Spain

BACKGROUND: Non-syndromic multiple supernumer-

ary teeth (NSMST) is a condition with a very low preva-

lence and which is opportunely described in the literature

by case reports or group of cases. This represents a

problem when it comes to describing and discussing the

characteristics of this disorder.

MATERIALS AND METHODS: A search was conducted

in PubMed, Web of Knowledge, Science Direct, and

Scopus databases to locate and identify cases and series

of cases about articles on multiple supernumerary teeth

not associated with syndromes that have been reported

so far. The following data were recorded: age, gender,

total number of supernumerary teeth, number of teeth in

both the maxilla and mandible, respectively, and number

of teeth in each dental series (incisor, canine-premolar,

and molar). Bilaterality in dental series and its localiza-

tion in either maxilla or mandible were also determined.

RESULTS: A total of 46 cases including patients of both

genders with a mean age of 19 years (SD = 6.826) and a

ratio of 2.14:1 were identified. A total of 393 supernu-

meraries were reported; 43.26% (n = 170) were located in

the maxilla and 56.74% (n = 223) in the mandible. No

significant differences were found between genders with

respect to the total number, involvement of the dental

series, or distribution of supernumerary teeth in each of

the arches.

CONCLUSION: Although NSMST is a condition that has

a higher incidence in the male gender, no differences

have been found as to their number and localization in

both genders. Bilaterality seems to be a common char-

acteristic of this disorder.

J Oral Pathol Med (2011)

Keywords: diagnosis; non-syndromic multiple supernumerary

teeth; supernumerary teeth; treatment

Introduction

Multiple hyperdontia, or non-syndromic multiple super-numerary teeth (NSMST), is diagnosed, according todifferent authors, because of the presence of five or moresupernumerary teeth (1–5) or by the occurrence ofsupernumerary teeth in more than one dental series (6,7). There is no pathological association in the casesdescribed here, as opposed to multiple hyperdontiaassociated with syndromes, such as cleidocranialdysostosis, cleft lip and palate, and Gardner syndrome(1, 2, 6).

Non-syndromic multiple supernumerary teeth is alow-prevalence condition that has opportunely beendescribed in the literature by isolated cases or series ofcases. Most authors seem to agree as to their greaterincidence in the male gender, while the premolar seriesare the most affected (1, 2, 5, 8). However, data arelimited because of the small number of cases and thescarce available information provided, on many occa-sions, by authors.

The aim of this study was to record all NSMST casesreported in the literature to analyze and describe itscharacteristics.

Materials and methods

A search was conducted in PubMed, Web of Knowl-edge, Science Direct, and Scopus databases to locate andidentify cases and series of cases about articles onmultiple supernumerary teeth not associated with syn-dromes that had been reported until May of 2011.NSMST reported cases that described the presence offive or more supernumerary teeth, regardless they wereaffecting more than one dental series.

The following data were recorded: age, gender, totalnumber of supernumerary teeth, number of teeth inboth the maxilla and the mandible, respectively, as wellas the number of teeth in each dental series (incisor,canine-premolar and molar). Bilaterality in dental seriesand its localization in either maxilla or mandible werealso determined. All variables were recorded by thesame examiner and data were entered into the Micro-soft Access database and processed by the Statistical

Correspondence: Dr. Cosme Gay-Escoda, Teknon Medical Center,C ⁄Vilana 12, 08022 Barcelona, Spain. Tel: +34 93 402 42 74, Fax:+34 93 393 30 70,E-mail: [email protected] for publication October 14, 2011

doi: 10.1111/j.1600-0714.2011.01111.x

J Oral Pathol Med

ª 2011 John Wiley & Sons A/S Æ All rights reserved

wileyonlinelibrary.com/journal/jop

Journal of

Oral Pathology & Medicine

Package for Social Science� v.12.0; (SPSS, Chicago, IL,USA). A descriptive, bivariate (Student’s t-test and chi-square test), and multivariate (ANOVA) analysis of thevariables recorded were performed.

Results

A total of 46 cases of NSMST describing the presence offive or more supernumeraries were recorded, while thosewith insufficient information for the statistical studywere dismissed (Table 1).

Age, gender, and location (maxilla and mandible)A total of 30 men and 14 women (two patients did notspecify gender) with NSMST and a mean age of 19 years

(SD = 6.826) were included in the study, with a ratioestablished at 2.14:1 (Table 1). A total of 393 supernu-merary teeth (range 5–21) were found; 43.26%(n = 170) of which were located in the maxilla and56.74% (n = 223) in the mandible. Forty-two (91.3%)patients presented simultaneously supernumerary teethin both jaws, with rare involvement of the mandible(n = 3) or maxilla (n = 1) alone (Table 1).

There were no statistically significant differences innumber with respect to gender (total: P = 0.83; maxilla:P = 0.82; mandible: P = 0.58) or according to agegroups (total: P = 0.29; maxilla: P = 0.19; mandible:P = 0.83). Sixty-three percent of cases (n = 29) werediagnosed in the second decade of life, followed infrequency by those detected in the third decade (n = 11)

Table 1 Distribution of non-syndromic multiple supernumerary teeth cases

Case (author) Gender Age Number Maxilla MandibleAnterior(incisal)

Premolar(canine-premolar) Molar

Ferriol et al. (7) F 12 14 5 9 2 8 4Ferriol et al. (7) M 20 12 3 3 0 12Acikgoz et al. (1) M 27 8 4 4 1 7Acikgoz et al. (1) M 20 7 4 3 7Acikgoz et al. (1) M 17 5 2 3 2 3Acikgoz et al. (1) M 33 6 2 4 1 5Acikgoz et al. (1) M 20 6 1 5 6Acikgoz et al. (1) M 22 5 3 2 2 3Ruhlman and Neely (22) M 14 9 6 3 6 3Manrique Mora (12) M 12 5 1 4 5King et al. (14) F 13 6 3 3 6King et al. (14) M 18 6 4 2 6King et al. (14) M 30 8 3 5 8Leslie (23) M 25 6 2 4 3 3Rosenthaler and Beideman (24) F 26 6 6 4 2Yusof and Awang (15) F 22 10 3 7 7 3Sharma (25) F 12 11 7 4 5 6Mopager et al. (26) - 13 7 4 3 7Acton (27) M 24 7 4 3 1 6Mason et al. (28) M 15 16 6 10 2 10 4Mason et al. (28) F 12 5 3 2 4 1Mason et al. (28) M 15 5 2 3 5Rizzuti and Scotti (29) M 10 21 10 11 8 10 3Hopcraft (18) M 18 10 5 5 8 2Heling and Shekter (30) - 20 7 2 5 3 4Dıaz et al. (9) F 20 17 12 5 1 2 1Batra et al. (2) F 17 11 3 8 3 8Batra et al. (2) M 20 8 8 4 4Barnett (31) M 12 6 2 4 2 4Finkel et al. (32) M 24 9 3 6 6 3Stevenson and McKechnie (33) M 10 11 6 5 5 6Shusterman et al. (34) F 7 6 4 2 4 2Reichart (35) M 18 7 4 3 7Yusof and Awang (15) M 24 16 5 11 11 5Yucel (36) M 22 6 1 5 6Desai and Shah (3) M 25 7 5 2 4 3Desai and Shah (3) M 36 16 9 7 3 6 7Ng’ang’a et al. (37) M 14 8 4 4 8Ng’ang’a et al. (37) F 13 7 2 5 5 2Hyun et al. (38) M 13 6 2 4 6Hyun et al. (38) F 20 5 5 5Hyun et al. (38) M 17 5 5 5Inchingolo et al. (39) F 17 5 3 2 5Srivatsan and Aravindha Babu (40) F 19 10 2 8 2 6 2Kaya et al. (4) F 39 5 1 4 6Sivapathasundharam and Einstein (41) M 20 14 5 9 2 12

M, male; F, female.

Non-syndromic multiple supernumerary teeth

J Alvira-Gonzalez and C Gay-Escoda

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(Figs 1 and 2). Most of the cases (76%) had between fiveand 10 supernumerary teeth and also reported nostatistically significant differences in gender.

Dental seriesTable 2 describes the distribution of supernumeraryteeth according to their dental series and position. Atotal of 380 supernumerary teeth were found. As to acase in which a patient was diagnosed with 17 supernu-merary teeth, Dıaz et al. (9) do not describe to whichdental series the group of 13 erupted teeth belonged, andonly reported information concerning the toothimpacted. Approximately 70% (n = 264) of the super-numerary teeth were found in the canine-premolarseries, while the canine-premolar region of the mandiblewas the most affected location, with 46.84% (n = 178)of all supernumeraries. Molar and incisal series reportedsimilar rates (15.79% and 14.73%, respectively). Nostatistically significant differences were found betweengenders with respect to the total number and distribu-tion of teeth in dental arches according to each dentalseries (Fig. 2). However, the amount of supernumeraryteeth in an arch varied with regard to the dental series,

while it was in the mandible where a major involvementcould be found by the canine-premolar series, whereasthe maxilla was the most affected by the molar andincisal series (Fig. 2).

BilateralityThe percentage of patients with supernumerary teethinvolving the same dental series in contralateral quad-rants was 71.73% (n = 33). Thirty (90.9%) patientspresented bilaterality in the canine-premolar series,compared with 9 (27.27%) in the molar region; sixcases also had bilateral teeth in both series simulta-neously (Fig. 3). There were no statistically significantdifferences between genders in terms of bilateralism inthe dental series (canine-premolar: P = 0.42; molar:P = 0.53, premolar and molar series simultaneously:P = 0.78).

About half of the cases (n = 17) reported bilateralsupernumerary teeth in both arches simultaneously,regardless of the dental series involved, while 39.39%(n = 13) and 9% (n = 3) of patients had bilateralsupernumerary teeth in the mandible or in the maxilla,respectively. Similarly, when analyzing the bilaterallocation of each dental series, it was observed that theinvolvement of both jaws is a common factor in both thecanine-premolar and the molar series, a feature that wasseen in 50% (n = 15) and 44.44% (n = 4) of cases,respectively (Fig. 3).

Discussion

Non-syndromic multiple hyperdontia, or NSMST, is adisorder that describes an excessive amount of teethcompared with 20 and 32 teeth in the primary andpermanent dentition, respectively, without being part ofanother disease condition, such as cleidocranial dyspla-sia, Gardners syndrome, or cleft lip and palate (6, 7).

The exact etiology of supernumerary teeth isunknown; however, several theories have been proposed

Table 2 Distribution of supernumerary teeth according to the dentalseries in which they were located

Range Minimum Maximum Total Mean SD

Total number 16 5 21 393 8.54 3.903Maxilla 12 0 12 170 3.70 2.493Mandible 11 0 11 223 4.85 2.530

Anterior-incisal 8 0 8 56a 1.22 1.931Maxilla 6 0 6 39 0.85 1.475Mandible 4 0 4 17 0.37 0.903

Canine-premolar 12 0 12 264a 5.74 2.679Maxilla 5 0 5 86 1.87 1.485Mandible 9 0 9 178 3.87 2.104

Molar 7 0 7 60a 1.30 1.931Maxilla 5 0 5 37 0.80 1.240Mandible 3 0 3 23 0.50 0.888

SD, standard deviation.aDıaz et al. (9) do not clarify, in their article, the exact location of 13supernumerary erupted teeth; they are not included in the distributionof dental series.

Figure 1 Distribution of the mean number (total, in the maxilla andin the mandible) of supernumerary teeth in different age groups.

Figure 2 Distribution of the mean number (total, in the maxilla andmandible) of supernumerary teeth in each dental series (anterior-incisor, canine-premolar and molar) with respect to gender.



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to explain their presence, and the following are the mostaccepted: phylogenetic theory, as a throwback to theapes, whose dental formula had more teeth; an autoso-mal recessive inheritance pattern, or link to X-chromo-some; an abnormal reaction to a traumatic local event;environmental factors; and the dichotomy of the toothgerm and the theory of hyperactivity of the dentallamina(9). As suggested by Batra et al. (2), the occur-rence of multiple supernumerary teeth in individuals ofthe same family may suggest an autosomal dominantinheritance pattern. However, although there is morelikelihood of finding a case of NSMST in patients whoserelatives also had supernumerary teeth, it seems clearthat inheritance does not follow a defined Mendelianpattern.Although most authors (1, 2, 8) define NSMST as the

presence of five or more supernumeraries, other authors,such as Ferriol et al. (7) and Yague-Garcıa et al. (6),describe it as the involvement of two or more dentalseries by supernumerary teeth. This entails a bias whenstudying the casuistry of this disease that results from thelack of unanimity in diagnoses, a factor that could castdoubt on the diagnosis of cases. This fact becomesevident when examining the prevalence of the disease,with values ranging from 1% (10) to 11.1% (11).However, authors agree with only 1% of cases, as itcan be concluded from publications that describe five, ormore, supernumerary teeth (5, 12). Because most articlesrefer to the number (five or more supernumerary teeth)and not the location, this should be one of the maincriteria for the diagnosis of this condition to unify thefeatures of this entity and provide new data in the future.Results of this study show the incidence of this

condition in the male gender, a feature already describedin previous publications (5, 13). However, the fact thatthere are no differences in number according to gendershows that this disorder occurs in both genders on anequal basis, a fact that should be taken into accountbecause in some publication no woman was registered(5). Similarly, no differences in location were found withrespect to gender, whereas its simultaneous occurrencein both arches seems to be the most prevalent location

compared with the occurrence of this condition in themaxilla or in the mandible alone.

The location as well as the involvement of dentalseries in NSMST cases seems to be the clinical charac-teristics shared by most authors, which coincides withthe results obtained in this study. Different authors (5, 8,14) have outlined that in non-syndromic multiplehyperdontia cases (five or more teeth), supernumeraryteeth are more often seen in the mandible, in thepremolar region to be more specific, as opposed to one-to-four supernumerary teeth, which are located in theanterior-superior and molar region of the maxilla.However, despite the mandible was the most prevalentlocation, similar percentages were obtained from bothdental arches (56.74% in the mandible and 43.26% inthe maxilla), in comparison with figures reported byYusof (8) and Acikgoz et al. (1), figures that roughlyrepresent twice the number of supernumeraries in themandible with respect to the maxilla.

Another very important feature of this condition is,just as Acikgoz et al. (1) have noted, the bilaterality ofsupernumerary teeth in cases of NSMST. As previouslydescribed, the occurrence of supernumerary teeth indental series was diagnosed in 71.73% of cases in thecontralateral quadrants of the same arch. Also, morethan half of the bilateral cases of supernumerary teethoccurred in both the maxilla and the mandible simul-taneously, regardless of the dental series involved. Thisfeature becomes even more relevant when analyzing thebilateral location of each dental group, because thesimultaneous involvement of both arches becomes acommon factor for both the premolar and the molarseries, respectively.

The diagnosis of this condition is mainly achievedthrough a clinical and radiological study that revealedthe presence of five or more supernumerary teeth,regardless of their location. It is also essential to ruleout any concomitant abnormality that could explain thischange in the tooth count, either localized in the alveolarcleft lip and palate, or as part of another more complexdisease (cleidocranial dysplasia and Gardner syndrome)(2, 5, 6, 8, 15).

According to Acikgoz et al. (1), 75% of the supernu-merary teeth are asymptomatic and are mostly diag-nosed as the result of an accidental radiological findings;however, sometimes the formation of these teeth is notfree of complications, as for example, the presence ofdiastemas, any alteration in the eruption of permanentteeth, cyst formation, the rotation, and tilting of theadjacent teeth (8, 16). Some authors such as Nazif et al.(17) have associated the presence of supernumerarieswith 30% of anomalies, while Hopcraft (18) and Asaumiet al. (10) reported the development of cysts in associ-ation with this condition in 9% and 11% of cases,respectively.

Although supernumerary teeth begin to developbefore teeth of the dental series to which they arerelated, some authors describe the formation of latersupernumerary teeth (6, 19, 42). This factor, and the factthat this condition is asymptomatic, makes difficult togive a correct diagnosis. It is therefore advisable to

Figure 3 Distribution of bilateral cases (total, premolar and molarseries) according to location.



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conduct periodic radiological examinations to rule outthe formation of new supernumerary teeth in a sus-pected case of NSMST.

The management of supernumerary teeth, andNSMST in particular, varies according to the differentpublications and range from the extraction of teeth –from the prophylactic point of view, while benefitingfrom the eruptive power of adjacent teeth – to anabstention procedure accompanied by regular clinicaland radiological follow-up visits. There is no unanimityon the ideal age for surgery, especially for NSMSTlocated in the pre-maxilla; some authors (20, 21)propose an immediate extraction after diagnosis, whileothers suggest to wait until the age of 8–10 years whenthe apical closure of the maxillary central and lateralincisors has occurred (7).

Meanwhile, abstention would be a possibility, pro-vided it is justified, as for example, when teeth haveerupted properly, there is no associated pathology,orthodontic treatment is not necessary, and in situationswhen an extraction could compromise the vitality ofadjacent teeth (1, 20).

Conclusions

The NSMST or non-syndromic multiple hyperdontia isan alteration with a very low prevalence that isdiagnosed by the presence of five or more supernumer-ary teeth, regardless of its location, and the absence ofany concomitant abnormality that could explain thischange in the tooth count. The involvement of thecanine-premolar series, for the most part, and thesimultaneous occurrence of supernumerary teeth inboth arches represent common features of NSMSTcases. Despite its high incidence in men, no differences ingender were found as to the number or location.Bilaterality of supernumerary teeth seems to be acommon characteristic of this disorder, which demandsconducting a complete clinical and radiological study,and arranging regular follow-up visits to exclude apossible occurrence of late supernumerary teeth.

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Conflict of interest and source of funding

The authors declare that they do not have any conflict of interest. This

study was performed by the �Dental and Maxillofacial Pathology and

Therapeutic’ research group of the UB-IDIBELL Institute, with the

economic support of an educational–clinical agreement in Oral Surgery

between the University of Barcelona, General Health Consortium, and the

Catalan Health Service of the Autonomous Government of Catalonia.



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Non-syndromic multiple supernumerary teeth: meta-analysis