Regional odontodysplasia

Regional odontodysplasia A case associated

with asymmetric

maxillary

and mandibular

development

Nikolaos Pandis, DDS, MS,” Cristina Polido, DDS,b and William H. Bell, DDS,c Dallas, Tex. DEPARTMENT

OF SURGERY,

UNIVERSITY

OF TEXAS,

DIVISION

OF ORAL

SOUTHWESTERN

MEDICAL

AND

MAXILLOFACIAL

Regional odontodysplasia is an uncommon condition of variable of unilateral maxillary regional odontodysplasia associated with development and consequent facial asymmetry is reported. (ORAL SURC ORAL MED ORAL PATHOL 1991;72:492-6)

R

egional odontodysplasia is an uncommon condition that involves both the enamel and the dentin of the primary and permanent teeth.’ More than 100 cases have been reported in the literature.2 The term “odontodysplasia” was introduced by Zegarelli et a1.3 and because the abnormality tends to affect only one quadrant, “regional odontodysplasia”4-6 has become the accepted term. The condition has been reported under other names such as “odontogenic dysplasia,“’ “localized arrested tooth development,“* “ghost teeth, ‘r9 “odontogenesis imperfecta,“‘O and “uniiatera1 dental malformation.“’ ’ Regional odontodysplasia is most often expressed unilaterally, unlike amelogenesis imperfecta and dentinogenesis imperfecta, which are generalized. The affected teeth tend to be in a consecutive series that does not cross the midline, although some cases not following this pattern have been documented.9q I2 According to the reported cases, regional odontodysplasia is localized only on one jaw, mainly the maxilla, ’ although involvement of both jaws is possibie.‘O Both sexes are affected, but a higher prevalence has been reported in females.” * Although several factors such as local trauma

3,s. 9, 12-14

infection,s-

9, 13-15

local

ischemia,I6~

17

local vascular defects,6, I8 Rh incompatibility,‘5 irra* diation3 neural damage,18, I9 hyperpyrexia,3 metabolic and nutritional disturbances, heredity,3, 5 and aFormerly Postgraduate tice, Corfu, Greece. bResearch Fellow. CProfessor. 7/16/30616

Orthodontic

Fellow;

now in private

prac-

SURGERY,

THE

CENTER expression asymmetric

and unknown etiology. A case maxillary and mandibular

local somatic mutation3 have been suggested as causes of regional odontodysplasia, the etiology remains uncertain. The histologic characteristics of the condition are well documented.2-4, 8, I33I8720-25 Regional odontodysplasia has been reported in association with failure of eruption of the affected teeth,4 gingival swelling,4y 5, ‘6 24,26-28vascular nevi,‘j* I9 and hydrocephalus. l8 Previous reports have focused mainly on the local radiographic and histologic findings without correlating the dental malformations with possible aberrations of facial structure. A few authors mentioned only facial asymmetry,29 ipsilateral maxillary hypoplasia,30 and maxillary and mandibular ramus hypoplasia3’ associated with the dental dysplasia, without presenting in detail the features of the facial asymmetry and the abnormal jaw development. We report a case of unilateral maxillary odontodysplasia in association with mandibular and maxillary asymmetric development and facial asymmetry with special attention to clinical and radiographic features. CASEREPORT

A 15year-old girl of Spanish origin was seenat our clinic with the chief complaint of missing teeth in the left side of the maxilla and the right side of the mandible. Family and medical histories were noncontributory. Clinical

findings

The patient appeared mesognathic, with normal vertical dimension and slightly retrusive upper lip. From the front the upper part of the face and the eyes were symmetric whereas the lower part of the face was asymmetric, with the chin deviating slightly to the left of the midsagittal plane. The left cheek appeared to be flatter than the right cheek.

Regional odontodysplasia

Volume 72 Number 4

493

3. A, Panoramic radiograph. B, Analysis of tracing from panoramic radiograph. CN, Condylar notch; FM, facial midline; LAgN, left antegonial notch; LCS, left condylar summit; LRHpt, left ramus height point; Pog, pogonion; RAgN, right antegonial notch; RCS, right condylar summit; RRH pt, right ramus height point. Fig.

1. Frontal view of patient. Notice facial asymmetry at left side of face.

Fig.

Fig.

4. Occlusal radiography of area bearing dysplastic

teeth.

Fig. 2. Intraoral views showing occlusal relationships and missing teeth.

The lips were canted in such a way that the left commissure was higher than the right (Fig. 1). The distance of the left commissure from the orbital plane was 5 mm shorter than on the right side. The left and right mandibular bodies were at different levels vertically, and the antegonial notches were in different planes sagittally. The facial asymmetry and the lip asymmetry become more severe during anima-

tion. Intraorally, no teeth were visible clinically in the maxillary left quadrant and in the mandibular right first and second premolar areas (Fig. 2). After questioning the patient and the mother we determined that the primary teeth had never erupted in the left maxillary quadrant. The maxillary dental midline, compared with the face, deviated to the left (the side without erupted teeth), whereas the lower dental midline deviated to the right of the facial midline. The molar and canine dental relationship corresponded to Angle’s Class II malocclusion. The mandibular occlusal plane was canted such that it was higher on the affected side and the lower dentition impinged on the alveolar crest of the edentulous maxillary

494

Pandis, Polido, and Bell

ORAL

SURG

ORAL

MED

ORAL PATHOL October 199 1

6. A, Submentovertex radiograph. B, Analysis of tracing from submentovertex radiograph. FM, Facial midline; LCC, left condyle center; MdMD, mandibular dental midline; Peg, pogonion; RCC, right condyle center. Fig.

Fig. 5. A, Posteroanterior skull radiography. B, Analysis of tracing from posteroanterior skull radiography. FM, Facial midline; LAgN, left antegonial notch; KS, left condylar summit; LJ, left jugular point; OP, occlusal plane; PP, palatal plane; RAgN, right antegonial notch; RCS, right condylar summit; RJ, right jugular point.

area. Periodontal health and hygiene were good. The temporomandibular joint, muscle, and mandibular movements were normal. Radiographic

findings

Panoramic radiography showed that the mandibular right premolars were formed and ready to erupt (Fig. 3). In the maxillary left quadrant the teeth from the midline to the

second molar were severelymalformed and the second molar root was behind in development when compared with the right side. The teeth were irregularly shaped and smaller, Only a thin mineralized border was present, without clear distinction between enamel and dentin (Fig. 4). The affected teeth had wide pulp chambers and open apical foramina. Panoramic radiography. The condylar height, which is the distance from the coronoid notch to the line perpendicular to the facial midline and tangent to the condylar summit, were equal for the left and right condyles (Fig. 3). The ramus height point on the left and right sides (RRH pt and LRH pt) of the panoramic view were the intersections between the line perpendicular to the facial midline and tangent to the condylar summits and the line perpendicular to the first line, originating from the antegonial notches. The left ramus height, the distance between the left ramus height point and the left antegonial notch, was 60

Regional odontodysplasia

Volume 72 Number 4 mm (affected side). The right ramus height, the distance between the right ramus height point and the right antegonial notch, was 70 mm. The left mandibular body length, the distance between the left antegonial notch and pogonion, was 85 mm (affected side). The right mandibular body length, the distance between the right antegonial notch and pogonion, was 77 mm. Posteroanterior skull radiography. The left ramus height, defined differently on the frontal view, is the distance between the left condylar summit and the line connecting the left and the right antegonial notches. This was 49 mm (affected side). The right ramus height, the distance between the right condylar summit and the line connecting left and right antegonial notches, was 56 mm (Fig. 5). The chin was approximately coincident with the facial midline. The palatal plane, defined by connecting the left and right jugular points was canted such that the left side was higher than the right. The mandibular dental midline deviated to the right of the facial midline, and dental compensations were indicated by the discrepancy between the mandibular apical base and dental midline. The mandibular occlusal plane was tilted, with the left side higher than the right. Submentovertex radiography. The distance between the center of the left condyle to the pogonion was 95 mm; the distance between the center of the right condyle to pogonion was also 95 mm (Fig. 6). The dental midline deviated to the right of the facial midline. The distance between the center of the left condyle and the mandibular dental midline was 85 mm. The distance between the center of the right condyle and the mandibular dental midline was 80 mm. DISCUSSION

The radiographic analysis in this case clearly shows that the left ramus of the mandible was shorter vertically than the right. However, no discrepancy was found in the condylar height between the left and the right sides, thus excluding right condylar hyperplasia or left condylar hypoplasia as causes for this facial asymmetry. The mandibular body on the affected side (left) was unexpectedly longer than the right side, and this could well be true to compensatory growth. It is postulated that the maxillary vertical asymmetry found during radiographic examination could be related to the dental hypoplasia and the subsequent underdevelopment of the alveolar bone on the affected side. The canting of the mandibular occlusal plane was most probably due to supereruption of the mandibular left posterior teeth. Supereruption of the opposing teeth in cases of regional odontodysplasia with no eruption on the affected side has been reported elsewhere.30, 3’ The patient will undergo comprehensive treatment that will address the dental, skeletal, and esthetic problems. A team approach that involves the orth-

odontist, the oral and maxillofacial prosthodontist is planned.

495

surgeon, and the

REFERENCES

1. Lustmann J, Kline H, UlmanskyM. Odontodysplasia:report of two casesand reviewof the literature. ORAL SURG ORAL MED ORAL 2. 3.

PATHOL

1975;39:781-2.

Crawford PJ, Aldred MJ. Regionalodontodysplasia:a bibliography.J Oral Pathol Med 1989:18:251-63. Z&&eIli EV,KutscherAH, ApplebaumE, Archard HO. Odontodysplasia.ORAL SURG ORAL MED ORAL PATHOL 1963; 16:187-93.

4.

Hintz CS, PetersRA. Odontodysplasia:report of an unusual caseand a reviewof the literature. ORAL SURG ORAL MED

5.

GardnerDC, SappJP.Regionalodontodysplasia. ORAL

ORAL ORAL 6.

PATHOL

1972;35:744-50.

MED ORAL

PATHOL

Walton JL, Witkop CJ, Walker‘PO.Odontodysplasia:report of three caseswith vascularnevioverlyingthe adjacentskinof the face. ORAL SURG ORAL 84.

7.

MED ORAL

PATHOL

1978;46:676-

SibleyLC, ZimmermannER.Odontogenicdysplasia:reportof a case. 1370-3.

8.

SURG

1973:35:351-6.

ORAL

SURG

ORAL

MED

ORAL

PATHOL

1962;15:

SuherT, Jump EB, Landis RL. Localizedarrestedtooth development. ORAL SURG ORAL MED ORAL PATHOL 1953; 6:1305-14.

Rushton MA. Odontodysplasia:“ghost teeth.” Br Dent J 1965;119:109-13. 10. ChaudhryAP, Wittich HC, StickelFR, Holland MR. Odontogenesisimperfecta:report of a case.ORAL SURG ORAL MED 9.

ORAL

PATHOL

1963;14:1099-103.

11. Billet J, Kerebel B, Lumineau JP, Schmidt J. Une exceptionelle anomalie dentaire “la dent fantom” ou “odontodysplasie.”Rev Stomatol Chir Maxillofac 1975;76:23-31. 12. AbramsAM, GroperJ. Odontodysplasia: report of threecases. ASDC J Dent Child 1966;33:352-62. 13. BergmanG, LysellL, PindborgJJ. Unilateral dental malformation: report of two cases.ORAL SURG ORAL MED ORAL PATHOL

1963;16:48-60.

14. AlexanderWN, Lilly GE, Irby WB. Odontodysplasia.ORAL SURG ORAL

MED ORAL

PATHOL

1966;22:8

14-20.

15. Herman NG, Moss S. Odontodysplasia:report of a case.J Dent Child 1977;44:52-4. 16. Burch MS, BesleyKW, SamuelsHS. Regionalodontodysplasia with associatedmandibular cyst: report of case.J Oral Maxillofac Surg 1973;31:44-8. 17. Lunin M, DevoreD. The etiologyof regionalodontodysplasia [Abstract]. J Dent Res 1976;55:B109. 18. Dahllof G, LindskogL, Theorell K, UssisooR. Concomitant regional odontodysplasiaand hydrooephalus.ORAL SURG ORAL 19. 20.

21. 22.

MED ORAL

PATHOL

1987;63:354-7.

SlootwegPJ, MeuwissenPRM. Regionalodontodysplasiain epidermalnevussyndrome.J Oral Pathol 1985;14:256-62. Sapp JP, Gardner DG. Regional odontodysplasia:an ultrastructuraland histochemicalstudy of the soft tissuecalcifications.ORAL SURG ORAL MED ORAL PATHOL 1973;36:383-92. GardnerDG. The dentinalchangesin regionalodontodysplasia. ORAL SURG ORAL MED ORAL PATHOL 1974,38:887-97. Lustmann J, UlmnaskyM. Structural changesin odontodysplasia.ORAL SURG ORAL MED ORAL PATHOL 1976;41:193202.

23.

KerebelLM, KerebelB. Soft tissuecalcificationsof the dental follicle in regional odontodysplasia:a structural and ultrastructural study. ORAL SURG ORAL MED ORAL PATHOL

24.

Neupert EA, Wright JM. Regionalodontodysplasiapresent-

1983;56:396-404.

496

Pandis, Polido, and Bell

ORAL

SURG ORAL

MED

ORAL

October

25.

26. 27. 28. 29.

ing as a soft tissue swelling. ORAL SURG ORAL MED ORAL P~THOL 1989;67:193-6. Kerebel B. Kerebel LM. Heron D. Le Cabellec LT. Reeional odontodysplasia: new histopathological data. J Biol Buccale 1989;17:121-8. Miller WA, Seymore HR. Odontodysplasia. Br Dent J 1968;i 25:56-9. Gould AR, Farman AG, Marks ID. Pericoronal features of regional odontodysplasia. J Oral Med 1984;39:236-42. Lowe 0, Duperon DF. Generalized odontodysplasia. J Pediatr 1985;9:233-43. Schmid-Meier E. Unilateral odontodysplasia with ipsilateral

PATHOL

1991

hypoplasia of the mid-face: a case report. J Maxillofac Surg 1982;10:119-22. 30. Gibbard PO, Lee KW, Winter GB. Odontodysplasia. Br Dent J 1973;135:525-32. 31. Pruhs RJ, Simonsen CR, Sharma PS, Fodor B. Odontodysplasia. J Am Dent Assoc 1975;91:1057-66. _ Keprint requests: Nikolaos Pandis, DDS, MS 11 Mitropolitou Athanasiou Corfu 49 100, Greece