584385

research-article2015

JDRXXX10.1177/0022034515584385Journal of Dental ResearchCrown Therapy and Amelogenesis Imperfecta

Research Reports: Clinical

A Randomized Controlled Trial of

Crown Therapy in Young Individuals
with Amelogenesis Imperfecta

Journal of Dental Research

2015, Vol. 94(8) 10411047
International & American Associations
for Dental Research 2015
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DOI: 10.1177/0022034515584385
jdr.sagepub.com

G. Pousette Lundgren1,2, G.I. Morling Vestlund3, M. Trulsson4,

and G. Dahllf1

Abstract
Amelogenesis imperfecta (AI) is a rare, genetically determined defect in enamel mineralization. Existing treatment recommendations
suggest resin-composite restorations until adulthood, although such restorations have a limited longevity. New crown materials allow
for minimal preparation techniques. The aim of this study was to compare the quality and longevity of 2 crown typesProcera and IPS
e.max Pressin adolescents and young adults with AI. A secondary aim was to document adverse events. We included 27 patients (11
to 22 y of age) with AI in need of crown therapy in a randomized controlled trial using a split-mouth technique. After placing 119 Procera
crowns and 108 IPS e.max Press crowns following randomization, we recorded longevity, quality, adverse events, and tooth sensitivity.
After 2 y, 97% of the crowns in both crown groups had excellent or acceptable quality. We found no significant differences in quality
between Procera and IPS e.max Press crowns. Tooth sensitivity was significantly reduced after crown therapy (P < 0.001). Endodontic
complications occurred in 3% of crowns. The results show that it is possible to perform crown therapy with excellent results and
without severe complications in young patients with AI. The study is registered at http://www.controlled-trials.com (ISRCTN70438627).
Keywords: enamel, ceramics, clinical outcomes, restorative dentistry, patient outcomes, pediatric dentistry

Introduction
Amelogenesis imperfecta (AI) is a rare, genetically determined
defect in enamel mineralization. It is characterized by heterogeneity in its clinical manifestations, histologic appearance,
and genetic pattern. Studies of various populations have
recorded prevalence rates of AI from 1 in 700 to 1 in 14,000
(Bckman and Holm 1986; Witkop 1988). The phenotype of
hypoplastic AI involves quantitative enamel deficiencies, and
that of hypomineralized/hypomatured AI involves qualitative
enamel deficiencies. In the most severely affected patients,
teeth can exhibit rapid loss or fractures of enamel, as well as
alterations in enamel thickness, color, and shape. Several problems are associated with AI, including teeth prone to disintegration or rapid wear, hypersensitivity, problems in masticatory
function, and gingivitis, requiring replacement of restorations
and lifelong, extensive restorative care (Coffield et al. 2005;
Pousette Lundgren and Dahllf 2014).
Existing recommendations suggest covering AI-affected teeth
in the anterior segment with direct or indirect composite restorations and, in the first permanent molars, with stainless-steel
crowns (Crawford et al. 2007; McDonald et al. 2012). Some suggest that direct restorations should be considered temporary, with
multiple replacements anticipated and plans for permanent therapy in adulthood (Malik et al. 2012; Chen et al. 2013). A complicating factor for resin-bonded restorations is that bond strength to
the enamel in permanent teeth affected by hypomineralized/

hypomatured AI is lower than bond strength to normal enamel

(Simmer and Hu 2001; Faria-e- Silva et al. 2011).
Patients with AI often experience negative aesthetic effects
from tooth discoloration and reduced crown size and have
reported significantly higher levels of social avoidance and
distress than subjects without the condition (Coffield et al.
2005). This social impact on education, job satisfaction, and
family building (Coffield et al. 2005), along with the fact that
AI patients would like to receive a permanent therapy at an
earlier stage (Lindunger and Smedberg 2005; Dashash et al.
2013), emphasizes the need for evidence-based therapy.
1

Department of Dental Medicine, Division of Pediatric Dentistry,

Karolinska Institutet, Stockholm, Sweden
2
Department of Pediatric Dentistry, Public Dental Service, Dalarna
County, Falun, Sweden
3
Department of Prosthetic Dentistry, Public Dental Service, Dalarna
County, Falun, Sweden
4
Department of Dental Medicine, Division of Prosthetic Dentistry,
Karolinska Institutet, Stockholm, Sweden
A supplemental appendix to this article is published electronically only at
http://jdr.sagepub.com/supplemental.
Corresponding Author:
G. Pousette Lundgren, Karolinska Institutet, Department of Dental
Medicine, Division of Pediatric Dentistry, Box 4064, SE-141 04
Huddinge, Sweden.
Email: gunilla.pousette-lundgren@ki.se

1042

Journal of Dental Research 94(8)

and IPS e.max Press, the latter does not

have a zirconia inner coping as described
in the registration document.)
From May 2009 to March 2012, we
enrolled patients with AI who had been
referred to the Department of Pediatric
Dentistry in Dalarna County, Sweden.
Each patient received an oral and written
explanation of the study protocol from the
clinical examiner and patients, and his or
her parents provided informed consent. To
be included, patients needed a clinically
verified AI diagnosis, confirmed by an
anamnestic family history or histologic
examination. We excluded patients with
fluorosis, molar incisor hypomineralization, other oral developmental disturbances, and systemic disorders, as well as
patients who were unable to provide
informed consent. Of the primary study
sample of 82 patients with AI (40 males
Figure 1. Flowchart showing patient selection, randomization, and follow-up examination. AI,
and 42 females aged 6 to 25 y), 27 patients
amelogenesis imperfecta.
(12 males and 15 females, aged 11 to 22 y)
In recent years, the development of all-ceramic restorations
needed prosthetic therapy. All of the patients selected for proshas made it possible to make crown restorations with quality
thetic therapy agreed to participate in the study. Following the
and longevity comparable to metal-ceramic crowns (Pelaez
classification system of Sundell and Koch (1985), 15 patients
et al. 2012; Esquivel-Upshaw et al. 2013). Recently, high-termed
received a diagnosis of hypoplastic AI and 12, hypomineralpressed lithium disilicate glass crowns (IPS e.max Press)
ized/hypomatured AI. In cases of mixed forms of AI, we
showed similar clinical outcomes to presintered zirconium
recorded the most dominant form (Fig. 1).
dioxide covered by porcelain (Procera AllCeram) and metalceramic crowns (Etman and Woolford 2010). Procera and IPS
Randomization
e.max Press have different properties. In Procera, the tendency
for chipping is higher, transparency lower, and the need for
We used a randomized split-mouth design and a patient-blind
thickness of material higher, as more tooth material needs to be
data acquisition protocol. We used the number generator table
removed (Al-Amleh et al. 2010; Al-Amleh et al. 2014). Yet,
from http://random.org for the randomization process, selectIPS e.max Press has a transparency that can give color probing the first crown material in the side section of the jaw by the
lems when restoring dark or yellow teeth and is less tested in
upcoming number in the table, which stipulated the type of
long-term studies (Etman and Woolford 2010).
material for the whole section. We then used the other material
Therefore, the aim of this study was to test the hypothesis that
on the opposite side of the jaw, creating a split-mouth
treatment with Procera crowns (zirconia dioxide coping with
method. In the front sections (13-23 and 33-43), the randomVita porcelain) cemented with Rely X ARC results in a better
ization process decided the material to be used for the entire
clinical outcome than that of IPS e.max Press crowns (lithium
front section and for each jaw separately. The type of crown
disilicate glass-ceramic) cemented with Rely X ARC in chilwas blinded to the patient and to the external examiner during
dren, adolescents, and young adults with AI. A secondary aim
the first control examination. Thereafter, radiographs taken
was to document complications associated with the therapy.
during the 1- and 2-y controls made it impossible to blind
crown type to the examiner.
In most cases, due to the eruption pattern of the teeth, restoMaterials and Methods
rations began in the upper jaw, and most patients received
crowns in the incisors first and then premolars and canines in
The Regional Ethics Review Board in Uppsala (Daybook
the second stage. After randomization, we made 119 Procera
2008/108) approved this study. The study followed the
and 108 IPS e.max Press crowns (Fig. 1).
Declaration of Helsinki guidelines and is a single-center
double-blind randomized controlled trial with a split-mouth
design. It was conducted at the Centre for Oral Rehabilitation,
Protocol
Department of Pediatric Dentistry, Falun, Sweden, and is registered at http://www.controlled-trials.com (ISRCTN704386
All cases followed the same therapy protocol: the clinical exam27). (Note: Of the 2 crown types included in the study, Procera
ination included an anamnestic family history to exclude

1043

Crown Therapy and Amelogenesis Imperfecta

possible differential diagnoses and childhood diseases (Gadhia
et al. 2012), then an evaluation of caries (Amarante et al. 1998),
gingival bleeding on mesial and distal surfaces (recorded before
and after therapy) on the teeth selected for crown therapy
(Ainamo and Bay 1975), previous trauma history, quality of restoration (Ryge and Snyder 1973; California Dental Association
1977; Ryge and DeVincenzi 1983), and endodontic diagnoses
(rstavik et al. 1986). The examination also included a panoramic radiograph if one was not available. We recorded tooth
sensitivity for the whole dentition on a visual analog scale (VAS;
Price et al. 1983; Berge 1988). Orthodontic consultation was
made if necessary and if we anticipated future problems with the
occlusal curve. After receiving information that included therapy risk factors, the patient had to wait 3 mo before making the
final decision for crown therapy. During this period, we helped
the patient optimize oral hygiene, provided behavioral management training (including injection), and evaluated the patients
ability to cooperate with therapy. After this waiting period, the
patients had to confirm their decision for prosthetic therapy.
Evaluation of possible endodontic complications was scheduled after 1 mo and then at the 1-y follow-up examination to
maintain patient security. Four patients received preprosthetic
orthodontics (with the intention not to close the interdental
spaces); 8 patients had had previous orthodontic treatment for
other reasons; and 1 had undergone orthognathic surgery.
The detailed treatment procedure can be seen in the
Appendix.

Follow-up
Control examinations occurred after 1 mo, 1 y, and 2 y. A nontreating dentist evaluated the treatment. At the follow-up examination, we evaluated the quality of the restoration, comparing
anatomic form, marginal integrity, surface, and color according
to the 1977 California Dental Association guidelines (Ryge and
Snyder 1973; Ryge and DeVincenzi 1983) as well as caries, gingival bleeding, trauma history, and endodontic problems. At the
2-y follow-up, we took apical radiographs. Patients underwent
the 2-y examination when 2 y had elapsed since placement of the
last crown. The follow-up time was 24 to 35 mo in 77 crowns, 36
to 47 mo in 65 crowns, and 48 to 60 mo in 84 crowns.

Statistical Analysis
The chi-square trend test and Fisher exact test compared the
quality of restorations among groups. We drew Kaplan-Meier
survival curves for patients with AI, subgroups of AI, and type
of crown material, comparing survival curves within groups
using the log-rank test and right censoring to analyze the survival of restorations. Independent-sample t test and Wilcoxon
signed-rank test were used to compare different outcome of
gingivitis and VAS score. Multilevel Cox regression analyses
helped identify factors related to the survival of restorations.
Spearman rank correlation was used to test bivariate correlations to change in VAS score. A P value <0.05 was considered
as statistically significant. We used SPSS 20 and 21 (IBM,
Chicago, IL, USA) to perform these analyses.

Table. Quality Assessment According to California Dental Association

Criteria in 27 Patients with Amelogenesis Imperfecta at the 2-y
Follow-up.
Crowns, n
Quality after 2 y
Excellent
Acceptable
In need of adjustment
Immediate danger to tooth
Total

Procera

IPS e.max Press

108
8
3
0
119

94
11
2
1
108

Results
Out of 82 AI patients aged 6 to 24 y, 9 patients were too young
for prosthetic therapy due to the eruption pattern of their teeth,
and 46 patients had mild or moderate forms of AI and did not
need prosthetic therapy at the moment. We selected 27 patients,
aged 11 to 22 y, for prosthetic therapy.
In total, we made 227 crowns (105 in males, 122 in females;
151 in hypoplastic AI, 76 in hypomineralized/hypomatured AI).
Made after randomization, these included 119 Procera crowns
and 108 IPS e.max Press crowns: 80 Procera in the hypoplastic
AI group, with 39 in the hypomineralized/hypomatured AI
group, and 71 IPS e.max Press in the hypoplastic AI group, with
37 in the hypomineralized/hypomatured AI group (Fig. 1).
Mean age at crown therapy was 17.9 3.4 y. We found no significant difference in age at therapy between the different AI
types. The tooth had to be fully erupted before the start of crown
therapy. This made it impossible to make all crowns in the dentition at the same time during adolescence. Because of this, the
final observation period ranged between 24 and 60 mo. The
Appendix Figure shows the distribution of teeth in the dentition
with crown therapy. Eighty teeth in the lateral segments could
be evaluated using a strict split-mouth design.

Quality Comparison between Procera and IPS

e.max Press
When comparing the Procera and IPS e.max Press groups, we
found no differences with regard to age at crown therapy, type of
AI, sex, traumatic history, or apical status. Based on the
California Dental Association quality criteria, there were no significant differences in quality between Procera and IPS e.max
Press crowns after 2 y (Table). Figure 2 shows the advantages of
performing crown therapy early when it is possible to use the
interdental spaces. The crowns can be made in a natural size and
form that avoid the crowding problems seen when crown therapy is performed in late adolescence or early adulthood.

Longevity of Crowns
A Kaplan-Meier plot shows no significant differences in longevity of Procera and IPS e.max Press crowns in AI patients
(Fig. 3). It also indicates a high success rate of both crown
types during the follow-up time of 2 to 5 y. After 2 y, 97% of
the crowns in both crown groups were in excellent

1044

Journal of Dental Research 94(8)

Figure 2. Advantages of early prosthetic therapy in adolescents with amelogenesis imperfecta. (A, B) A 13-y-old patient diagnosed with hypoplastic
type of amelogenesis imperfecta who had crown therapy between 14 and 17 y of age. At this age, spaces between the teeth allow for an ideal anatomic
form of crowns and normal interdental spaces. (C, D) Crown therapy was performed at 19 y of age in a patient with hypoplastic form of amelogenesis
imperfecta. The crowding did not allow for an ideal anatomic form of the crowns.

Figure 4. Tooth sensitivity before and 2 y after crown therapy in 27

patients with amelogenesis imperfecta. VAS, visual analog scale.

between the 2 crown types with regard to longevity and quality. After a follow-up of 48 to 60 mo for 84 crowns, no additional failures were diagnosed.
Figure 3. Longevity (in months) of Procera and IPS e.max Press
crowns (cemented in patients between 11 and 22 y) with amelogenesis
imperfecta.

Gingivitis

or acceptable condition. Evaluating the 80 crowns in lateral

segments with a corresponding tooth (strictly split-mouth
group; Appendix Table 1), we found no significant difference

The mean number of surfaces with gingivitis decreased from

1.3 0.9 surfaces per tooth before therapy to 0.9 0.9 after
crown therapy (P < 0.001). We found no significant differences
between the 2 AI groups or between the different crown types.

Crown Therapy and Amelogenesis Imperfecta

Sensitivity
We evaluated sensitivity problems before and 2 y after crown
therapy. Figure 4 shows that 24 of 27 patients had a reduction
in sensitivity 2 y after crown therapy. Three patients (1 with
pulpitis, 1 with apical periodontitis, and 1 without endodontic
complications) showed increased sensitivity after porcelain
crown therapy. The median VAS score was reduced from 5.2 (0
to 8.4) to 0.6 after 2 y (0 to 5.5; P < 0.001). Bivariate correlations between changes in sensitivity and the variables AI type,
age, sex, and crown type found no significant correlations.

Adverse Events
Seven patients experienced adverse events in 12 teeth. The
adverse events involved development of apical periodontitis
(3% of crowns), and all cases but 1 were related to the experience of dental trauma (Appendix Table 2). Difficulties with
impressions or cementation had resulted in suboptimal design
of crowns or shortage of cement. In 1 case, chipping was diagnosed, also related to dental trauma.

Discussion
The results of this study show that crown therapy can be performed with excellent results in children, adolescents, and
young adults with severe forms of AI. Furthermore, this study
found that tooth sensitivity is significantly reduced and that
adverse events during the 2-y follow-up period were few. These
results are encouraging for this group of patients since the longevity of composite restorations is limited and the number of
replaced restorations is high (Pousette Lundgren and Dahllf
2014). Many authors recommend postponing prosthetic therapy
until adulthood (Crawford et al. 2007; Markovic et al. 2010;
Malik et al. 2012; McDonald et al. 2012), mostly due to a risk
of endodontic complications and the risk for exposure of the
disturbing margins of crowns. This study shows that there are
many advantages to patients receiving permanent therapy at an
early age. Not only can the number of dental appointments and
replacements of resin-composite restorations be minimized
(Pousette Lundgren and Dahllf 2014), but aesthetic problems
can also be solved and sensitivity problems decreased. It is also
possible to use the interdental spaces that exist before mesial
movement of the teeth to maintain a normal size for the restored
crowns with minimal removal of tooth substance. This may also
contribute to an increased quality of life in AI patients, since
most patients ask for permanent-quality restorations at an earlier age (Lindunger and Smedberg 2005; Krieger et al. 2009).
An important factor for the success of prosthetic therapy in
children and adolescents is to educate and prepare the patient
before therapy. The preprosthetic phase involving oral hygiene
training and assessing the patients ability to cooperate was
found to be important, not only for behavior management and
caries prevention, but for achieving excellent periodontal
health (Lindunger and Smedberg 2005; Malik et al. 2012). The
patient also had time to reflect on the suggested treatment plan
and decide for oneself.

1045
Patients with disturbances in tooth mineralization often have
problems with pain related to dental procedures and dental fear
and anxiety (Klingberg et al. 1995; Jlevik and Klingberg 2002).
To achieve treatment without pain, we used a combination of
analgesic drugs, local anesthesia, fluoride varnish, and nitrous
oxygen sedation (Coffield et al. 2005; Crawford et al. 2007;
McDonald et al. 2012; Dashash et al. 2013).
Due to the eruption process in adolescence, all the crowns
cannot be placed at the same time. It is possible to start crown
therapy in the incisors first and then place crowns in premolars
and canines in a second session; so, it is important to plan for the
occlusal curve throughout treatment. In some cases, an open bite
appeared (Ravassipour et al. 2005). Some teeth without occlusion seemed to overerupt during growth, with development
resulting in visible crown margins. The risk for aesthetic problems with visible margins in the growing bite has been mentioned
as a problem (Malik et al. 2012; McDonald et al. 2012). When
using porcelain crowns, none of the patients mentioned this as a
problem, and the smile line was undisturbed. In patients without
a tendency toward an open bite, we found no visible margins.
Longevity of crown therapy is reported to be good
(Lindunger and Smedberg 2005; Pjetursson et al. 2007). A
study on patients without AI found loss of vitality, followed by
caries, to be the 2 most common biological complications
(Pjetursson et al. 2007). In AI patients, however, a 10-y followup found aesthetic problems to be the main reason for crown
replacement (Krieger et al. 2009). A 3-y follow-up found that
IPS e.max Press crowns demonstrated clinical performance
comparable to Procera, AllCeram, and metal-ceramic crowns,
while IPS e.max Press performed better with regard to crack
propagation and wear resistance in patients without AI (Etman
and Woolford 2010). It seems that new porcelain restoration
materials fulfill the demands for quality and longevity, as well
as aesthetic demands. In our study, 97% of crowns were of
excellent or acceptable quality after 2 y. We found no differences in longevity or quality among the different groups of AI
and no loss of crown. The decreased bonding strength in the
hypomineralized/hypomatured types of AI (Faria-e-Silva et al.
2011) seemed to be of minor importance when full coverage
crowns were used with mechanical retention.
We evaluated 80 teeth in the lateral segments with a strict
split-mouth randomization. Our study used a split-mouth
method for the lateral segments while using 1 material for the
whole front segments. There were no significant differences in
quality of crowns between the crown types in the strict splitmouth selection and between teeth without a corresponding
tooth or teeth in the frontal segments. For aesthetic reasons, it
is not possible to use a split-mouth design for the frontal region.
There are few randomized studies using split-mouth design in
lateral segments. Federlin et al. (2010) found that the 5.5-y survival rate of partial ceramic crowns was 89% compared to 93%
in partial-cast gold crowns. We have previously also reported a
similar lower survival rate of partial ceramic crowns (Pousette
Lundgren and Dahllf 2014).
Concerning oral hygiene, gingivitis decreased after crown
therapy, maybe as a result of decreased tooth sensitivity or
because of a now smooth surface with less plaque retention.

1046
Sensitivity was evaluated prior to crown therapy and at the
2-y follow-up. This is one of the important results of this study.
The most common problems after ceramic crown therapy in
patients without AI are increased sensitivity and gingivitis
(Pihlaja et al. 2014). In a randomized controlled study with
split-mouth design comparing different cements, Selz et al.
(2013) found severe hypersensitivity that led to endodontic
treatment in 7.4% of abutment teeth in adults without AI.
Patients with AI report a high level of tooth sensitivity before
treatment. As seen in this study, crown therapy resulted in
decreased tooth sensitivity. Of 27 patients, only 3 reported
increased sensitivity: 1 was a patient with no apical complications; another was a patient with pulpitis; and the third was a
patient with apical periodontitis.
Regarding adverse events, we found no difference between
the 2 crown types or between the AI types. A history of dental
traumatic injury prior to or after crown therapy seemed to be an
important contributing factor to endodontic complications.
Endodontic complications appeared in late adolescence, 18 to 19 y
of age. It seems that the risk of endodontic problems in young
teeth with large pulp chambers is overestimated (McDonald
et al. 2012). Of the 227 teeth, 5 had an endodontic diagnosis, and
2 were under observation. With 7 endodontic complications, the
prevalence of endodontic complications was 3% after 2 y. The
estimated rate of loss of vitality in adult patients without AI after
crown therapy is 6.1% (4.9% to 7.6%; Pjetursson et al. 2007).
The results must be interpreted with caution since 2 y may be too
short to document all late complications in a group of growing
individuals, although the continuous follow-up after 4 y in 80
crowns did not contribute any additional failures.

Conclusions
After 2 y, 97% of the crowns in both crown groups had excellent or acceptable quality.
We found no significant differences between Procera and IPS
e.max Press crowns with regard to quality and longevity. Crown
therapy also resulted in decreased sensitivity problems in young
AI patients. It seems to be possible to perform early crown therapy without severe complications in young patients with AI.

Author Contributions
G. Pousette Lundgren, contributed to conception, design, data
acquisition, analysis, and interpretation, drafted and critically
revised the manuscript; G.I. Morling Vestlund, contributed to conception, data acquisition, and interpretation, critically revised the
manuscript; M. Trulsson, contributed to design and data interpretation, drafted and critically revised the manuscript; G. Dahllf, contributed to conception, design, data analysis, and interpretation,
drafted and critically revised the manuscript. All authors gave final
approval and agree to be accountable for all aspects of the work.

Acknowledgments
This study was supported by grants from the Center for Clinical
Research and Public Dental Service, Dalarna County, and the
Emelie and Gotthard Thourn Foundation, Karolinska Institutet.
We also thank the dental technicians sa Bulai Appelgren and

Journal of Dental Research 94(8)

Anna Stobnicka for their high-quality work. The authors declare
no potential conflicts of interest with respect to the authorship and/
or publication of this article.

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