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Interdisciplinary approach to oral rehabilitation of patient with amelogenesis

imperfecta

Article  in  The New York state dental journal · March 2014

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Burak Yilmaz Ulas Oz

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 restorative dentistry
Interdisciplinary Approach to Oral Rehabilitation
of Patient with Amelogenesis Imperfecta
Burak Yilmaz, D.D.S., Ph.D.; Ulas Oz, D.D.S., Ph.D.; Hasan Guney Yilmaz, D.D.S., Ph.D.
ABSTRACT
Amelogenesis imperfecta is a hereditary condition
that affects the development of enamel, causing
quantity, structural and compositional anomalies
that involve all dentitions. Consequently, the effects
can extend to both the primary and secondary den-
titions. Patients with amelogenesis imperfecta may
present with clinical difficulties, such as insufficient
crown length, tooth sensitivity and orthodontic dis-
crepancies, all of which can be resolved successfully
with an interdisciplinary approach. This case report
describes the interdisciplinary approach to the treat-
ment of a 22-year-old patient with amelogenesis im-
perfecta. The proper alignment of anterior teeth and
gingivo-cervical line was provided with orthodon-
tic and periodontal treatments. All-ceramic crowns
were placed on anterior, and metal-ceramic restora-
tions were placed on posterior teeth to reduce sensi-
tivity and improve esthetics with function. Improved
esthetic appearance, reduced tooth sensitivity and
the resolution of a potentially harmful psychosocial
condition were achieved. Patient remained satisfied
in the 12-month follow-up examination.
Amelogenesis imperfecta (AI) is a group of inherited disorders
characterized by abnormal enamel formation.1,2 The inheritance
patterns of AI include autosomal-dominant, recessive and X-
linked modes of transmission.3 This enamel anomaly affects both
the primary and permanent dentition.4 The classification of AI is
complex, but can be simplified by division into three groups—hy-
poplasia, hypocalcification and hypomaturation—both clinically
and radiographically.1,2 Hypoplastic enamel is often well mineral-
ized and shows such great variation in density that distinguish-
ing it from the underlying dentin may be difficult.5 In patients
with hypoplastic AI, the enamel layer is usually thin and yellow-
brown, and difficulties in tooth eruption may occur.6 The thick-
ness of enamel in people with hypomature AI is within normal
limits, and the enamel has an opaque appearance. In those with
hypocalcified AI, the teeth are severely worn and the enamel de-
taches from the dentin shortly after eruption.7
Clinicians may face problems (poor dental esthetics and
tooth sensitivity caused by rapid wear of the dentition) in treating
patients with AI, regardless of its type.8 A skeletal or dental open
bite is typical of individuals with AI. The cause of open bite is
unknown, but may involve local and/or genetic factors.9-14 In pa-
tients with AI, full-coverage restorations are usually required; and
defects on the teeth are prone to the heavy formation of calculus
that can eventually cover the teeth and cause gingival inflamma-
tion.7 Therefore, it is critical to achieve optimal gingival health
before restorations of the teeth are put in place.
The New York State Dental Journal s MARCH 2014 31
 Figure 1. Pretreatment, frontal view. Figure 2. Pretreatment, intraoral view.
Figure 3. Intraoral view after orthodontic treatment.
It has been shown in previous studies15,16 that having AI is associ-
ated with discernable psychosocial effects. This suggests that den-
tal treatment for AI could be medically necessary because those
effects have a considerably negative impact on the patient’s over-
all health. Patients with AI may require orthodontic, periodontal
and prosthodontic treatment. Therefore, an interdisciplinary ap-
proach may be advantageous in providing the successful evalua-
tion, diagnosis and treatment plan for AI patients. This clinical
report describes an interdisciplinary approach for the treatment
of a patient with AI.
Case Report
A 22-year-old woman was referred to the Department of Prost-
hodontics with the chief complaints of tooth sensitivity and the
unesthetic appearance of her anterior teeth. The patient’s detailed
medical and dental histories were obtained, and radiographs and
photographs of her teeth were taken. The patient said that al-
though her parents did not have any intraoral problems similar
to hers, her grandparents and sisters had the same complaints
(esthetics and sensitivity) and tooth deformations. The patient’s
intraoral appearance showed features characteristic of AI, such as
worn, sensitive teeth, and an anterior open bite. The distribution
of the diastemas in the maxillary and mandibular anterior region
was irregular. The right maxillary first molar was apically and
periodontally compromised. The results of radiologic and clinical
examinations suggested that the patient probably suffered from
32 MARCH 2014 s The New York State Dental Journal
Figure 4. Intraoral view after gingivectomy and gingivoplasty.
a hypoplastic type of AI. The patient said she had difficulty smil-
ing because of the unaesthetic appearance of her teeth (Figures
1 and 2).
Diagnostic casts were made and mounted in centric relation
on a semi-adjustable articulator (Hanau H2; Teledyne Hanau, Buffalo, NY)
with the help of a face-bow transfer. The extraction of the right
maxillary first molar was planned after analysis of the radiographs.
A treatment plan was devised with the aims of improving the pa-
tient’s dental esthetics and reducing the sensitivity of her teeth.
Before prosthetic rehabilitation was initiated, an orthodontic
treatment plan, designed to correct the horizontal and vertical
overlap, was prepared. The proper alignment of the maxillary an-
terior teeth and the optimal horizontal and vertical overlap were
provided with orthodontic treatment. The vertical overlap was –4
mm, and the horizontal overlap was +4 mm. The orthodontic
treatment was accomplished in seven months with the use of an
edgewise fixed appliance with maxilla-mandibular anterior ver-
tical elastics. When the casts made after the orthodontic treat-
ment were analyzed, it was noted that a +2 mm vertical overlap
and a +3 mm horizontal overlap had been provided and that the
orthodontic treatment distributed the diastemas properly in the
anterior region (Figure 3).
When the discrepancies in the gingivocervical region of the
anterior teeth and the inadequate crown lengths were considered,
gingivectomy and gingivoplasty procedures were considered nec-
essary to ensure satisfactory esthetic results.17 These procedures
were performed after the optimal health of gingival tissues was Discussion
achieved via scaling and root planing (Figures 4 and 5). After The treatment of a patient with AI who has high esthetic demands
healing of gingival tissues had occurred four weeks after those is a challenge for the clinician. The abnormal form of the enamel,
procedures, full-crown restorations were considered as the first orthodontic deficiencies, the potential for periodontal disease and
treatment option for the patient’s tooth sensitivity and the the psychological effect of that anomaly should be considered before
grooves and defects on her tooth surfaces, all of which may lead treatment is initiated; treatment options may vary because of those
to the propagation of caries, gingival inflammation and esthetic limitations. In the case presented here, the patient had an anterior
problems. Because of the patient’s high esthetic expectations, it
was decided to fabricate all-ceramic crowns for maxillary and
mandibular incisors, canines and first premolars. The second pre-
molars and the mandibular right, left and maxillary left molars
were targeted for restoration with metal-ceramic crowns.
For replacement of the missing right maxillary first molar, a
metal-ceramic fixed partial denture between the maxillary right
second premolar and the second molar was chosen. The anterior
teeth were prepared with diamond rotary cutting instruments
(Diamir; Resia, Italy), creating 1.5-mm chamfer cervical finish lines.
Gingival retraction was performed with a Roeko Comprecord
(Coltène/Whaledent, Inc., Cuyahoga Falls, OH), and definitive impressions
of the prepared teeth were made with an elastomeric impres-
sion material (Impregum; 3M Espe Dental Products, St. Paul, MN). The Figure 5. Intraoral view after one month of healing.
casts were poured with a type V dental stone (Jade Stone; Whip Mix
Corp, Louisville, KY) and were mounted in an articulator (Hanau H2;
Teledyne Hanau) with a face-bow transfer. The restorations were
fabricated with IPS Empress 2 (Ivoclar Vivadent; Schaan, Liechtenstein)
all-ceramic material according to the manufacturer’s instruc-
tions. Lithium disilicate reinforced core structures were evalu-
ated for marginal fit, and veneering porcelain was applied in
the laboratory. The restorations were glazed after trial placement
had been performed to confirm fit, retention, marginal integ-
rity and anterior guidance. The all-ceramic crowns were luted
with dual-polymerizing composite resin cement (Rely X ARC; 3M
Espe Dental Products) according to the manufacturer’s instructions. Figure 6. Intraoral view of definitive restorations in maximum intercuspation 12
Photopolymerization was performed with a light-polymerizing months after placement.
unit (Litex 692S LED; Dentamerica, City of Industry, Calif.) for 40 seconds
on all surfaces.
The posterior teeth were prepared; and impressions were
made with an elastomeric material (Impregum; 3M Espe Dental Products)
after gingival retraction (Roeko Comprecord; Coltène/Whaledent, Inc.). For
fabrication of the posterior metal-ceramic restorations, the casts
were mounted on an articulator. Trial placements were performed
intraorally to confirm fit, retention and marginal integrity and to
determine the need for occlusal refinement. After the glazing of
the ceramic material (Vita Omega; Vita Zahnfabrik, Bad Sackingen, Germany)
had been performed, the restorations were luted with zinc-poly-
carboxylate cement (Poly F Plus; Dentsply DeTrey GmbH, Konstanz, Germany)
according to the manufacturer’s instructions. Oral hygiene in-
structions were given to the patient.
At the 12-month follow-up evaluation it was noted that she
was satisfied with the esthetics and function of the restorations Figure 7. Post-treatment, frontal view.
and that her tooth sensitivity had resolved (Figures 6 and 7).

The New York State Dental Journal s MARCH 2014 33

open bite, which is a characteristic associated with AI. Schulze13 re-
ported the occurrence of an anterior open bite in three families with a
hypoplastic variety of AI and suggested that this anomaly is a skeletal
type arising from a dysgnathia that might result from abnormal skull
development. Rowley et al.10 stated that the association was due less to
local factors and more to a genetic abnormality of craniofacial devel-
opment, which would include skeletal open-bite features. Rao et al.14
appear to disagree with that hypothesis; they attribute the frequent
association of anterior open bite and AI to a locally acting mechanism.
The authors assumed that thermally sensitive rough teeth may lead
to a tongue-thrusting habit that, in turn, acts as a local impediment
to alveolar growth and, ultimately, produces an anterior open bite.
When she was asked about tongue thrusting, our patient reported
having practiced that behavior since childhood, a habit that may have
contributed to the development of her open bite.
The esthetics of gingival tissues and tooth crown length associ-
ated with the smile line can be altered via periodontal surgical tech-
niques. An internal bevel gingivectomy can improve the appearance
of the patient’s smile. The clinician can affect the appearance of the
smile by correcting tooth length relative to the maxillary lip line and
by also correcting right-to-left asymmetries.17 Because the maxillary
gingival tissues of the patient presented here were extremely thick
and improperly aligned, gingivectomy and gingivoplasty procedures
were deemed necessary to achieve satisfactory esthetic results.
34 MARCH 2014 s The New York State Dental Journal
All-ceramic restorations offer considerable advantages because of
their esthetic and biocompatible structures. The IPS Empress 2 (Ivo-
clar Vivadent, Schaan) framework material is a lithium disilicate glass
ceramic. The bending strength of IPS Empress 2 is three-times
higher than that of IPS Empress; this improvement was achieved by
increasing the percentage of the crystal content.18 The final restora-
tion, which is made of a lithium disilicate framework ceramic and
a fluoroapatite layering ceramic, offers clinical benefits in terms of
machinability, polishability, reduced wear of the opposing tooth
structure, and control of optical properties such as translucency,
brightness and light scattering of the layering material.19 The use
of all-ceramic restorations in patients with AI has been reported
previously;7 therefore, that type of restoration for the anterior teeth
of the esthetic-conscious patient presented here was selected.
One of the patient’s complaints was tooth sensitivity; therefore,
the cement selection was important for preventing post-treatment
sensitivity. The zinc polycarboxylate cement was used for cementing
the metal ceramic restorations because of its good biocompatibility,
which is due to the low intrinsic toxicity and also to the rapid rise of
the cement pH toward neutrality; localization of the polyacrylic acid
and limitation of diffusion by its molecular size and ion binding to
dentinal fluid and proteins; and the minimal movement of fluid in
the dentinal tubules in response to the cement. Low irritation of the
pulp is also an advantage of this cement.20
According to a previous study, AI negatively affects the relation-
ships and self-esteem of nearly all patients with the disorder.16
The negative psychosocial effects of AI were evident in the patient
described in this report. Before treatment, her smile was limited
because of her contracted lip muscles and she did not smile read-
ily. However, after successful interdisciplinary dental treatment,
those muscles relaxed and her smile line was extended.
Conclusion
This case report describes the interdisciplinary approach to treatment of
a 22-year-old patient with amelogenesis imperfecta. The proper align-
ment of anterior teeth and gingivo-cervical line was provided with orth-
odontic and periodontal treatments. All-ceramic crowns were placed on
anterior, and metal-ceramic restorations were placed on posterior teeth
to reduce sensitivity and improve esthetics with function. The patient
remained satisfied in the six-month follow-up examination. 
Queries about this article can be sent to Dr. Hasan Yilmaz at guneyyilmaz@
hotmail.com.
REFERENCES
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enamel hypoplasia: a case report. J Esthet Restor Dent 2002;14:340-348.
2. Holroyd I, Lench N, Winter GB. Amelogenesis imperfecta in triplets: a unique family re-
cord. Br Dent J 1995;178:465-468.
3. Hart PS, Hart TC, Simmer JP, et al. A nomenclature for X-linked amelogenesis imperfecta.
Arch Oral Biol 2002;47:255-260.
4. Aldred MJ, Savarirayan R, Crawford PJ. Amelogenesis imperfecta: a classification and cata-
logue for the 21st century. Oral Dis 2003;9:19-23.
5. Collins MA, Mauriello SM, Tyndall DA, et al. Dental anomalies associated with amelogen-
esis imperfecta: a radiographic assessment. Oral Surg Oral Med Oral Pathol Oral Radiol
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6. Fritz GW. Amelogenesis imperfecta and multiple impactions. Oral Surg Oral Med Oral
Pathol 1981;51:460.
7. Kostoulas I, Kourtis S, Andritsakis D, et al. Functional and esthetic rehabilitation in amelogen-
esis imperfecta with all-ceramic restorations: a case report. Quintessence Int. 2005;36:329-38.
8. Seow WK. Clinical diagnosis and management strategies of amelogenesis imperfecta vari-
ants. Pediatr Dent 1993;15:384-93.
9. Persson M, Sundell S. Facial morphology and open bite deformity in amelogenesis imper-
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11. Cartwright AR, Kula K, Wright TJ. Craniofacial features associated with amelogenesis im-
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12. Bäckman B, Adolfsson U. Craniofacial structure related to inheritance pattern in amelogen-
esis imperfecta. Am J Orthod Dentofacial Orthop 1994;105:575-82.
13. Schulze C. Developmental Abnormalities of the Teeth and Jaws. In: Gorlin RJ, Goldman
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14. Rao S, Witkop CJ Jr. Inherited defects in tooth structure. Birth Defects Orig Artic Ser 1971;7:153-184.
15. Coffield KD, Phillips C, Brady M, et al. The psychosocial impact of developmental dental de-
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16. Lindunger A, Smedberg JI. A retrospective study of the prosthodontic management of pa-
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17. Townsend CL. Resective surgery: an esthetic application. Quintessence Int 1993;24:535-42.
18. Zawta C. Fixed partial dentures with an all-ceramic system: a case report. Quintessence Int
2001;32:351-58.
19. Wright JT, Roberts MW, Wilson AR, Kudhail R. Tricho-dento-osseous syndrome. Features of
the hair and teeth. Oral Surg Oral Med Oral Pathol 1994;77:487-93.
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