CASE REPORT Apparent hypodontia: A case of misdiagnosis

Jonathan Alexander-Abt, BDS, FDS RCS(Eng)a Hitchin, Herts, UK The case of a 12-year-old girl is reported, whose pretreatment radiograph demonstrated agenesis of two premolars and a canine and slow development of the contralateral premolars. A follow-up radiograph taken 1 year later showed initial mineralization of a tooth germ in the site of one of the apparently missing premolars. The cause, diagnosis, and treatment planning implications of delayed mineralization and slow development of second premolars are discussed with reference to the literature. (Am J Orthod Dentofacial Orthop 1999;116:321-3)

At what point during the process of dental development can a definitive diagnosis of developmental hypodontia be made? A knowledge of average mineralization times can be helpful in determining whether a developing tooth, not visible on radiographs, can be considered developmentally absent.1 A differential diagnosis should include the possibility of delayed mineralization, given the significant individual variation in the timing of dental development.2 Although radiographic evidence of second premolar mineralization is usually visible by 5 years of age, these teeth can be very late in developing, especially in the maxilla.3 When a second premolar is diagnosed as missing, 1 solution is timely interceptive extraction of the overlying deciduous second molar to allow space closure by drift of the adjacent teeth.4 Alternatively, where planned extraction of the deciduous molar to provide space for relief of crowding is not anticipated, the tooth can be retained on the understanding that later it may become infraoccluded, necessitating coronal build-up.5 If retained, the tooth may later become lost through exfoliation or forced extraction, necessitating replacement prosthetically, by autotransplantation or an implant.6 Therefore, the unexpected late development of a second premolar may complicate the initial treatment plan or even go undiagnosed if appropriate followup radiographs are not taken.
CASE REPORT

ectopically, E/ E were infraerupted by 2mm, and E/CE was retained. Both developing 5/ and /5 had not yet developed roots (suggesting slow development compared with the rest of the dentition) and appeared asymmetric in the stages of their development. In all other respects, the dental age corresponded to the chronologic age. All third molars showed signs of initial tooth formation. Treatment began with the extraction of all remaining deciduous teeth to encourage earlier establishment of the permanent dentition.7 Removable appliances were provided to correct the crossbite and maintain space for the ectopically erupting /5 while allowing spontaneous closure of the E/E extraction spaces. Thirteen months later (at the age of 13 years), the progress panoral radiograph (Fig 2) showed ini tial crown formation of 5/, which was impacting between 6/ and 4/. The patient was referred for surgi cal extraction of 5/, /5, and 5/ before space closure with fixed appliances.
DISCUSSION

A girl (aged 11 years 11 months) received treatment for a Class I malocclusion with a crossbite associated with a functional shift. The panoral radiograph (Fig 1) showed agenesis of /3, /5 and 5/ ; /5 was erupting

aIn Private Practice Reprint requests to: Jonathan Alexander-Abt, 47 Bancroft, Hitchin, Herts SG5 1LA, United Kingdom. Copyright 1999 by the American Association of Orthodontists. 0889-5406/99/$8.00 + 0 8/4/95061

This case illustrates a group of features hypothesized to arise from a common cause, namely delayed mineralization of a second premolar, slow development and asymmetry in the stages of formation of the contralateral second premolars, and developmental hypodontia.8 The cause of developmental hypodontia is largely genetic,9 transmitted most commonly in an autosomal dominant pattern with incomplete penetrance and variable expressivity.10 It is hypothesized that delayed formation of the second premolars might be a milder expression of developmental hypodontia.8 The frequency of occurrence of some or all of these features is reported to range from 0.1%8 to 9%11 in children with no cleft palate and as much as 30% in children with cleft palate.12 The delay in mineralization and development of second premolars has been shown to be constant throughout subsequent tooth for321

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American Journal of Orthodontics and Dentofacial Orthopedics September 1999

Fig 1. Pretreatment panoral radiograph of patient aged 11 years 11 months shows agenesis of 13, 15, and 5/; /5 erupting ectopically.

Fig 3. Magnified retrospective examination of the devel oping 5/ area (arrowed).

Fig 2. Progress panoral radiograph 13 months into treat ment; initial crown fromation of 5/ is now apparent.

mation so that their ultimate size and shape are within normal limits.12 Although a link between developmental absence of third molars and delayed mineralization of posterior teeth has been demonstrated,13 such a link is not evident in this nor in other reported cases,14,15 because all 4 third molars were developing. To reduce the chance of misdiagnosis, radiographs that indicate developmental absence of a second premolar should be scrutinized with a magnifying glass to screen for the presence of an unmineralized tooth germ.2 The radiographic appearance of a circumscribed homogenous area in the usual site of second premolar odontogenesis is indicative of a tooth germ before mineralization; (bony) trabeculation indicates developmental absence.2 A retrospective examination of the pretreatment radiograph in this case does reveal a circumscribed homogenous area in the bone directly beneath

the bifurcation area of E/, providing evidence of very early /5 odontogenesis (Fig 3). The radiographic diagnosis of the developmental absence of second premolars can be assumed to be correct when the patient is 8 to 9 years of age4,16 because relatively few second premolars develop after this age.3 This case, in which a lower second premolar showed unambiguous radiographic evidence of initial mineralization after 12 years of age, should therefore be considered unusual. The optimal timing (to encourage maximum space closure by bodily drift of the adjacent teeth) for interceptive extraction of the retained mandibular deciduous second molar is the period between 8 years of age and the completion of root development of the first permanent molar and first premolar.4 It is probable that only in a few cases, such as the one reported, does a conflict arise between the agerelated demands of correct diagnosis of the developmental absence of the mandibular second premolar and optimal timing for interceptive extraction of the overlying deciduous tooth. It is not routine practice to screen for the late development of teeth during orthodontic treatment.17 However, the presence of developmental hypodontia coupled with slow and asymmetric development of second premolars should alert the clinician to the possible presence of a not yet visible unmineralized tooth germ. In these circumstances, consideration should be given to taking follow-up radiographs, especially after interceptive extractions or before orthodontic space closure.
SUMMARY

Second premolar mineralization and development can be delayed by as much as 7 years (or more).

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Delayed mineralization results in the radiographic phenomenon of apparent hypodontia. Published evidence suggests an etiologic relationship between delayed mineralization, slow and asymmetric development of the contralateral second premolars, and developmental hypodontia. Therefore, developmental hypodontia coupled with slow and asymmetric development of contralateral premolars should alert the clinician to the possible presence of a not yet visible, unmineralized tooth germ. To reduce the chance of misdiagnosis, correctly taken radiographs should be scrutinized for early indications of a tooth germ before mineralization. The possibility of delayed tooth development should be considered when deciding on the need for follow-up radiographs, especially after interceptive extractions or before orthodontic space closure.
REFERENCES 1. Mitchell L. An introduction to orthodontics. Oxford: Oxford University Press; 1996. 2. Moyers RE, Riolo ML. Early treatment. In: Moyers RE, editor. Handbook of orthodontics. 4th ed. Chicago: Year Book Medical Publishers Inc; 1988. p. 343-431. 3. Ravn JJ, Nielsen, HG. A longitudinal radiographic study of the mineralization of 2nd premolars. Scan J Dent Res 1977;85:232-6.

4. Lindqvist B. Extraction of the deciduous second molar in hypodontia. Eur J Orthod 1980;2:173-81. 5. Evans RD, Briggs PFA. Restoration of an infra-occluded primary molar with an indirect composite onlay: a case report and literature review. Dental Update 1996;23:52-4. 6. Fields HW. Treatment of nonskeletal problems in preadolescent children. In: Profitt WR, editor. Contemporary orthodontics. 2nd ed. St Louis (MO): Mosby Year Book; 1993. p. 376-422. 7. Ronnerman A. The effect of early loss of primary molars on tooth eruption and space conditions: a longitudinal study. Acta Odont Scand 1977;35:229-39. 8. Ranta R. Hypodontia and delayed development of the second premolars in cleft palate children. Eur J Orthod 1983;5:145-8. 9. Kirdelan JD, Rysieck;G, Childs WP. Hypodontia: Genotype or environment? A case report of monozygotic twins. Br J Orthod 1998;25:175-8. 10. Graber LW. Congenital absence of teeth: a review with emphasis on inheritance patterns. J Am Dent Assoc 1978;96:266-75. 11. Kahl B, Schwarze CW. Late mineralization of premolars in relation to orthodontic diagnosis and therapy [German]. Fortschritte der Kieferorthopadie 1986;47:234-44. 12. Ranta R. Developmental course of 27 late-developing second premolars. Proc Finn Dent Soc 1983;79:9-12. 13. Garn SM, Lewis AB, Vicinus JH. Third molar polymorphism and its significance to dental genetics. J Dent Res 1963;42:1344-63. 14. Coupland MA. Apparent hypodontia. Br Dent J 1982;152: 388. 15. Uner O, Yucel-Eroglu E, Karaca I. Delayed calcification and congenitally missing teeth: case report. Aust Dent J 1994;39:168-71. 16. Rolling S. Hypodontia of permanent teeth in Danish school children. Scand J Dent Res 1980;88:365-9. 17. Wisth PJ, Thunold K, Boe OE. Frequency of hypodontia in relation to tooth size and dental arch width. Acta Odont Scand 1974;32:201-6. 18. Cochrane SM, Clark JR, Hunt NP. Late developing supernumerary teeth in the mandible. Br J Orthod 1997;24:293-6.

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