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Jurnal Dentinogenesis Imperfecta
Jurnal Dentinogenesis Imperfecta
Purpose: To demonstrate the field of application and prospects of individually modeled indirect composite restor-
ations for the treatment of children and adolescents based on a case of dentinogenesis imperfecta. Dental malfor-
mations can affect single or multiple teeth. In most cases, direct composite fillings can be placed. However, in
severe cases, these restorations may be more challenging and error-prone, especially when occlusal adjustments
are necessary. Since composite materials do not require a specific lamination strength and are easy to repair, they
can be applied using the indirect technique, enabling conservation of more sound hard tissue than is possible
when conventional restorations are used.
Patient and Methods: A young patient with dentinogenesis imperfecta type II underwent interdisciplinary full-mouth
rehabilitation due to massive tooth wear and loss of vertical occlusion. First, a check bite was taken, and vertical
occlusion was increased using overdentures. Six months later, a construction bite was taken over the existing over-
dentures (focusing on the sagittal dimension) to move the mandibular position more towards the anterior, correct-
ing the skeletal Class II malocclusion. This resulted in a Class I intercuspidation with harmonization of the facial
proportions. After a further six months, all teeth were restored using individually modeled indirect composite restor-
ations, which preserved most of the sound hard tissue and restored esthetics and function.
Conclusion: Indirect composite restorations can be a valuable tool for improving occlusion, esthetics and function
in the treatment of children and adolescents.
Keywords: severe tooth wear, dental malformations, full-mouth rehabilitation, individual lamination technique, fiber
reinforced composite restorations, overdenture, hereditary disorder.
J Adhes Dent 2018; 20: 345–354. Submitted for publication: 10.04.18; accepted for publication: 16.07.18
doi: 10.3290/j.jad.a40991
Shields et al.56 Type I is associated with osteogenesis imper- For the primary dentition, stainless steel crowns and
fecta and originates from a collagen-I defect.35 The degree of composite buildups are recommended for patients with
dental malformation can vary across the primary and the per- enough residual hard tissue.23,44,61 In cases of severe loss
manent dentition, and in some cases the teeth have only of dental hard tissue, overdentures can be made, although
radiographic abnormalities but no clinical damage.35,47,60 In they may hamper oral hygiene and may require further ad-
contrast, DI type II shows severe dental malformations in justments over time.11,55,63
both the primary and the permanent dentition and full pene- The mixed dentition stage includes the most important
tration within a given family.51 The associated malformations and difficult part of therapy, as even partly erupted teeth
are related to a mutation of the dentin sialophosphoprotein begin to wear when they attain occlusion. Ideally, these
gene, which is located on chromosome 4q21.4,7,13,34,36,66,67 teeth should be sealed with composite. Keeping these
DI type III was primarily found in a particular ethnic group in teeth dry during treatment can be difficult, possibly reducing
the USA and described by Witkop et al65 and Hursey et al.22 the longevity of the sealants. Continuous follow-up visits
The deciduous teeth of type III can have a shell-like shape are necessary to extend sealants or repair defective ones
with very short or even missing roots, whereas the perma- as tooth eruption proceeds.61
nent teeth resemble those found in the other types of DI.64 When the CEJ is accessible, the tooth should be covered
Other genetic studies have revealed that Shields type II and completely, preferably with indirect restorations, because
III might be allelic and may represent different severities of they ensure proper occlusal adjustment by the dental tech-
the same disorder.6,13,20 Furthermore, these studies suggest nician, unlike direct composite buildups. As in the present
that dentin dysplasia type II is also a phenotype of this DI. case, ICRs can here be considered a preservative and es-
Whereas DI type I has a greater range of clinical severity, thetically valuable option.14 When applicable, all teeth can
DI types II and III are consistently characterized by amber- be restored consecutively in this way, and repairs can re-
colored dentin, opalescent and bulbous crowns and con- main functional past adolescence. Additionally, orthodontic
strictions at the cementoenamel junction (CEJ). Observa- treatments are easier to perform when the fragile enamel is
tions from radiographs reveal that in addition to the covered. At this point, stainless steel crowns must be men-
typically shortened and narrowed roots, the pulp chamber is tioned as an inexpensive alternative for the permanent pos-
quickly calcified when tooth eruption begins.1,37 Histologi- terior teeth; however, they have drawbacks in terms of es-
cal examinations of the dental hard tissue of these patients thetics and fit compared with ICRs. Additionally, once a
reveal regularly structured enamel but aberrations in the stainless-steel crown is applied to a permanent tooth, any
structure of the dentin and the enamel-dentin junction. Gen- future restoration must be a full crown, whereas the use of
erally, the number and size of the dentinal tubules are re- ICRs leaves other options open.14
duced, and the apatite crystallites have an abnormal mor- In later adolescence or adulthood, however, when cranio-
phology, while the water and collagen content is facial growth is complete and the gingiva retracts, the mar-
increased.25,26,30,42 The adherence between the enamel gins of restorations may become visible and further full-
and dentin is compromised and the enamel chips off, re- mouth rehabilitation might be necessary to correct
sulting in severe abrasion and attrition.16,33 Lindau et al30 unacceptable esthetics.24,28 In the treatment of adults, the
ascribed this poor adherence to a malfunction in the inter- literature shows cases of full-mouth rehabilitation using all-
action between the ectodermal and mesenchymal tissues. ceramic restorations43 or full-mouth extractions and im-
This rapidly leads to a loss of vertical occlusion even shortly plant-supported dentures, depending on the individual’s
after tooth eruption begins. Consequently, craniofacial dys- dental chart.50
gnathia, esthetic deficiencies, and disorders in function and The present article focusses on the treatment of the per-
articulation may occur. In many cases, these patients do manent dentition in adolescents by describing the case of
not consult specialists until esthetics are massively com- a young patient with little experience of dental treatment. At
promised or symptoms arise, at which point they show ex- this age, treatment must be carefully planned to consider
tensive tooth destruction.12,17,21,29,59 the loss of dental hard tissue relative to longevity and es-
thetics.31 ICRs seem to be ideal in such cases for two rea-
Therapy Regimes in DI sons. First, thanks to adhesive bonding, no mechanical re-
Numerous case reports exist describing different treatment tention is necessary, which permits the restoration of very
procedures, most of which involve the prosthetic treatment short teeth. In addition, these restorations can be made
of missing teeth.15,19,21,45,50,55,62 Few present early con- with a thickness of only 0.2 mm,14 so that in many cases.
servative non-destructive therapies that can preserve these tooth preparation can be omitted or less invasive. In con-
teeth sustainably.23,53,54,60 trast, commonly used restorations (ie, metal-ceramic or all-
Preventing the early loss of teeth should be the goal of ceramic) require sufficient hard tissue and tooth prepar-
first interventions, as this simplifies later therapies tremen- ation. However, ICRs must be inserted under absolutely dry
dously and avoids the risk of impairing jaw growth and the conditions, which cannot always be guaranteed. Further-
loss of alveolar bone.17,54 This requires continuous visits more, composites are still considered provisional, because
and timely interventions, which can be slowed by the poor of reduced wear resistance68 and a higher rate of stain-
compliancy of children.11,17,61 Depending on the patient’s ing,2,3 although increasing evidence in literature implies
stage of life, different therapies are indicated.5 that ICRs have a longevity comparable to that of ceramic
Fig 2 Negative smile line at the age of Fig 3 Frontal view shows decayed maxil-
12 years. lary incisors and loss of vertical occlusion.
Fig 1 Lateral view shows negative lip pro- Fig 4 Lateral view of the right side. The Fig 5 Lateral view of the left side. The
file at the age of 10 years. first molars are completely worn. first molars are completely worn.
restorations.18,31,38,40 This statement might not fully apply patient’s former dentist covered the exposed dentin with
to patients with dental malformations; for instance, Incici et composite sealants to reduce further tooth wear. Two years
al24 showed that the longevity of any kind of restoration is later, when the patient was 12 years old, orthodontic ther-
compromised in these patients. Nevertheless, a case se- apy was cancelled because of the patient’s poor compli-
ries by Feierabend et al14 revealed satisfactory results with ance. Furthermore, the severe loss of dental hard tissue
ICRs. and the extremely short crowns made it impossible to at-
tach orthodontic brackets. She was then referred to the De-
partment of Operative Dentistry of the University Hospital of
CASE REPORT Würzburg for restorative therapy. At this point, the clinical
findings revealed a negative smile line (Fig 2), reduced verti-
A 10-year-old girl was referred by her dentist to the Depart- cal bite height (Figs 3 to 5) with severely worn and decayed
ment of Orthodontics at the University Hospital of Würzburg incisors, and completely worn first molars (Figs 6 and 7).
due to an Angle Class II malocclusion. The primary disease The radiographic findings revealed no periapical lesions but
was diagnosed as DI type II. Severe tooth wear had caused presented the typical narrow, short roots with calcified root
a loss of vertical dimension and insufficient occlusion. The canals and bulb-shaped crowns (Fig 8).
vertical loss was accompanied by skeletal Class II maloc-
clusion, Angle Class II occlusion, and an increased overjet Pretreatment
(8.0 mm). Extraorally, a negative lip profile was noticeable Planning and therapy were performed in collaboration with
(Fig 1). When the patient was 10 years old, functional orth- the orthodontist. First, two plaster models of each jaw were
odontic therapy with a Class-II bionator was attempted to made and a check bite was performed using wax. The first
stimulate the growth of the mandible. At the same time, the pair of the models was mounted in an articulator (Protar 7
Fig 9 Checkbite with vertically increased Fig 10 Diagnostic wax-up of all teeth. Fig 11 Representative silicone mold (ther-
occlusion. After analysis of this situation, moformable splint is not shown in this
the incisal pin was lowered by 1 mm. image).
Articulator, KaVo; Biberach, Germany) using a face bow with struction in oral hygiene and was advised to wear the
the check bite in maximal intercuspidation (Fig 9). The inter- overdentures for at least 16 h a day but to leave them out
maxillary space, intercuspidation, and required space for the while eating to prevent fractures. The incisor length was
prospective restorations were analyzed and the future adjusted during return visits to improve esthetics and pro-
lengths of the incisors were estimated. Subsequently, we nunciation. Overall, the first phase of treatment took six
lowered the incisal pin of the articulator by 1 mm to achieve months until the patient felt comfortable with her bite and
an increase in the vertical intermaxillary distance of 2 mm at pronunciation.
the molars and 4 mm at the incisors. A diagnostic wax-up The second phase of treatment started with another
was performed on the casts (Fig 10) and the wax-up was analysis of intercuspidation and the proportion of the skull
cast with silicone to produce a mold for overdenture prosthe- since the patient’s growth proceeded. The clinical findings
ses (Fig 11). The second pair of plaster models was revealed a slightly shortened lower face, a persistent neg-
mounted in the articulator in the same manner used for the ative lip profile (Fig 13), and a persistent Angle Class II
first models, and thermoformable splints (Erkodur 1,0 mm malocclusion with distal occlusion of half the premolar
hard, Erkodent; Pfalzgrafenweiler, Germany) were adapted size. To improve the intercuspidation and the facial propor-
and cut back to half of each occlusal tooth surface. Using tions, we performed another silicone check bite (Imprint
the silicone molds, composite (Biodent, DeguDent; Hanau, Bite, 3M Oral Care; St Paul, MN, USA) of the overden-
Germany) was polymerized on the thermoformable splints to tures: A construction bite was made with the mandible in
produce the overdenture prostheses (Fig 12). The fit of the a forward position (by a half-premolar size) resulting in a
overdentures and the length of the incisors were checked. bilateral Class I occlusion. Figure 14 shows balanced fa-
No adhesive paste was needed to fix the overdentures, cial proportions of the patient with inserted overdentures
since their buccal margins were extended to the undercut and the new construction bite.
areas of the natural teeth, providing sufficient friction. A face-bow was then applied over the maxillary over-
Initially, the patient complained of myofacial pain in the denture and both jaws were then cast with alginate along
temporal muscle, which was alleviated with physical ther- with inserted overdentures. New plaster models were
apy, speech therapy, and occlusal adjustments of the over- made jointly with the overdentures and mounted in the
dentures at the follow-up visits. The patient received in- articulator using the new bite registration. To minimize
effort, the overdentures were then remodeled according Treatment – Restorative Stage
to the design of an orthopedic twinblock appliance:8 Tilts First, the incisors and premolars were restored. To simplify
were polymerized on each of them to inhibit retrusion of the checkbite procedure after the prospective tooth prepar-
the mandible (Fig 15). Speech therapy and physical ther- ation, a resin splint (Biodent, Dentsply; York, PA, USA) was
apy were continued until the patient felt comfortable with created using new plaster models, which were mounted in
the new maxillomandibular relationship, and the restora- the articulator in the same manner used in the overdenture
tive stage began six months later. This long second wait- remodeling procedure described above. The maxillary model
ing period was due more to the time-consuming corre- was mounted using a face-bow appliance, the mandible
spondence with the patient’s medical insurance, which model was mounted against it, and the overdentures were
initially refused to assume therapy costs, rather than to placed on both models. These were joined together by a
medical reasons. silicone check bite before they were mounted (Fig 15), al-
lowing the current intraoral relationship of the bite to be
transferred to the future resin splint (Fig 16). The splint with
Fig 17 Pre-preparation of the incisors with inserted and silica- Fig 18 Composite buildups and chamfer preparation of the central
coated parapulpal retentive pins. Enamel of the lateral incisors was incisors. Lateral incisors with removed undercuts and residual
preserved since only undercuts were removed. enamel.
Fig 19 Tooth preparation of the mandibular incisors and premolars Fig 20 Fine-impression by means of the resin splint and provi-
showing removed undercuts with residual enamel and intact inter- sional cement. The splint was cut back at the maxillary incisor re-
proximal contacts. gion since buildups were made.
Fig 21 The maxillary overdenture was cut back and fixed with ad- Fig 22 Completed individually modeled indirect composite restor-
hesive paste. The incisors received conventional provisional crowns. ations.
its impressions provided guidance for later registration after teeth were prepared just by removing undercut areas and
the tooth preparation was completed. smoothing sharp edges to conserve as much enamel as pos-
As all premolars and the mandibular incisors showed suf- sible. However, the maxillary incisors showed a strong de-
ficient amounts of residual enamel (Figs 6 and 7), these gree of destruction, having no clinical crown height and only
Fig 24 Smile at 2-year post-treatment visit. Fig 25 Frontal view at 2-year visit.
Fig 26 Lateral view right side at 2-year visit. Fig 27 Lateral view left side at 2-year visit.
overbite, we additionally protruded the mandible forward sumed that this young patient would require further full-
with an incorporated bite plane. One year after all the res- mouth rehabilitation treatment during her lifetime,9,24,28,31
torations had been inserted, however, the bite relapsed to saving as much enamel as possible had first priority.
an Angle Class II malocclusion, but the intercuspidation Initially, we considered bleaching the teeth, as there is
was still sufficient (Figs 25 to 27). Dysgnathia surgery was one case report in the literature that describes successful
considered but rejected by the patient and her mother, bleaching in a patient with DI.10 However, we made no at-
since the patient was satisfied with her situation. tempt to bleach our patient’s teeth, since it had not been
successful in her brother, in whom we had attempted it
Treatment shortly before.
In the present case, treatment was performed with indirect The histological structure of the enamel in patients with
composite restorations created using an individual lamina- DI has proven to be regular,33 so a reliable bond between
tion technique. These restorations can be manufactured composite and enamel can be ensured. In contrast, the
with a thickness of 0.2 mm;14 a retentive preparation is not histological structure of the dentin shows less mineraliza-
needed, as the restorations are bonded. This allows more tion, more collagen, and lower hardness. Therefore, re-
healthy hard tissue to be preserved than when convention- duced bond strength between composite and dentin is
ally cemented restorations are used. However, thin material likely.16 As some of our patient’s teeth had little or no re-
layers can reduce the masking ability of the restorations, sidual enamel, we did not want to rely on adhesion alone.
which can be a concern in some cases. In our patient, Therefore, we performed a conventional chamfer prepar-
some restorations had cervical margins with a thickness of ation for additional mechanical retention in these teeth. To
0.2 mm as a result of the compromise between saving hard assure the best possible bond strength among dental hard
tissue and the masking effect. Different thicknesses were tissue and composite, an etch-and-rinse adhesive system
previously tested using the diagnostic wax-up, and thicker was used. Furthermore, the ICRs and the direct composite
restoration margins would either have lead to more bulbous buildups were silica coated and silanized, respectively,
crowns or greater loss of hard tissue. As it must be as- which is intended to deliver the best adhesion.57 However,
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