PEDIATRIC DENTISTRY V 42 / NO 2 MAR / APR 20

CASE SERIES

Atypical Developmental Defects of Enamel in Primary Incisors Versus Preeruptive

Intracoronal Resorption ( PEIR ) : Case Reports
Camila Palma-Portaro, BSc, MSc1 • Jorge Casián, BSc, MSc2 • Ailín Cabrera-Matta, DDS, MDS3 • Flavia La Rocca, BSc4 • Anna B. Fuks, DDS5

Abstract: Developmental defects of enamel (DDE) in children range from moderate to severe and may predispose to dental fractures and early
childhood caries. The prevalence of DDE in the primary dentition range from 24 to 75 percent, and the right time to diagnose DDE should be
as soon as the teeth erupt. However, early detection of DDE in primary incisors is difficult because parents seldom visit the dentist at this
age and, later on, caries lesions mask the original alteration. The purpose of this paper was to present three cases of unique and similar dental
defects appearing in very young children from different countries—defects that probably share the same etiology. Given the severity of the
defects and the children’s’ age, these could be the first reported cases of preeruptive intracoronal resorption (PEIR) in primary incisors. This
paper also discusses the differentiation between DDE and PEIR. (Pediatr Dent 2020;42(2):146-9) Received August 27, 2019 | Last Revision De-
cember 13, 2019 | Accepted December 19, 2019
KEYWORDS: DEVELOPMENTAL DENTAL DEFECTS, IDIOPATHIC CROWN RESORPTION, PRE-ERUPTIVE INTRACORONAL RESORPTION

Developmental defects of enamel (DDE) in the primary denti- Case 1

tion result from disturbances in the amelogenesis process and
can manifest as quantitative defects (hypoplasia) or qualita-
tive defects (hypomineralization). Hypoplastic enamel presents
alterations in enamel thickness before tooth eruption.
Hypomineralized enamel shows alterations in enamel translu-
cency and opacity, such as demarcated or diffuse opacities. 1-4
Demarcated opacities can display rapid posteruptive changes
or even enamel missing shortly after eruption.5 The prevalence
of DDE in the primary dentition ranges from 24 to 75 per-
cent. 6-10 The ideal time to identify DDE is when the tooth
has recently erupted, before posteruptive breakdown occurs
and caries develops. Reporting DDE prevalence in primary
incisors is a difficult task, as children should be examined
during the first year of life. Recent studies conclude that the
primary second molar is the most affected tooth and demar-
cated opacities are the most prevalent type of defect.6-12
On the other hand, preeruptive intracoronal resorption
(PEIR) could explain a large defect in a newly erupted pri-
mary maxillary incisor not yet submitted to masticatory forces.
The purpose of this paper was to present three unique cases of
asymmetric dental defects of enamel in primary maxillary
central incisors and hypothesize about their origin.
A 10-month-old boy with no remarkable pre- or postnatal
medical history presented to a pediatric dentist practice in
Barcelona, Spain. His mother complained that he had “two
small erupting upper teeth on his right side instead of one.”
The boy was asymptomatic, did not yet walk, and lacked a
history of dental trauma.
Intraoral examination revealed that only the mesial and
distal walls of the primary maxillary right central incisor were
visible, as a gingival ridge covered the middle third of the
tooth (Figure 1). The radiograph revealed a radiopaque layer
above the pulp chamber, compatible with a dentinal bridge.
He was scheduled for a recall appointment two months later
as he awaited complete eruption. During this visit, the defect
was now supragingival and, as the dental pulp was not ex-
posed, the dentist decided to protect the dentin-pulp complex
with a direct composite crown. Under cotton rolls isolation, a
glass ionomer liner (Vitrebond, 3M ESPE, St. Paul, Minn.,
USA) and a flowable resin (Tetric Flow, Ivoclar Vivadent,
Schaan, Liechtenstein) were placed on the floor of the lesion. A

1 Dr. Palma-Portaro is a professor, 3Dr. Cabrera-Matta is a specialist and an associate

professor, and 4Dr. La Rocca is a specialist, Pediatric Dentistry; all at the Peruvian
University Cayetano Heredia, Lima, Perú. 2Dr. Casián is in a pediatric dentistry private
practice, Veracruz, Mexico; and 5Dr. Fuks is a professor emeritus, Hebrew University,
Hadassah School of Dental Medicine, Jerusalem, Israel.
Correspond with Dr. Palma-Portaro at camila.palma.p@upch.pe

HOW TO CITE:
Palma-Portaro C, Casián J, Cabrera-Matta A, La Rocca F, Fuks AB.
Atypical developmental defects of enamel in primary incisors versus
preeruptive intracoronal resorption (PEIR): Case report. Pediatr Dent
2020;42(2):146-9. Figure 1. Severe dental defect in the primary maxillary right central
incisor of a 10-month-old baby.

146 DDE OR PEIR IN INCISORS?


Figure 2. A significant dental defect in tooth F in an otherwise healthy
eight-month-old boy. Note the pulp exposure.
Figure 3. An evident dentinal bridge is seen below
the zirconia crown four months after a partial
pulpotomy.
Figure 4. A severe notch-like defect seen on the primary maxillary
left central incisor (F) in a 13-month-old boy. Note the dark-red col-
oration in the pulp chamber and the diffuse opacities on mandibular
incisors.
PEDIATRIC DENTISTRY V 42 / NO 2 MAR / APR 20
composite strip crown was then fitted (Filtek™ Z350 XT, 3M
ESPE). Five months later, the radiograph showed no signs of
infection nor resorption. At his last control visit 18 months
later, the patient remained asymptomatic and the pulp re-
mained vital.
Case 2
A healthy eight-month-old boy presented with his mother
to a pediatric dental practice in Veracruz, Mexico, with a
chief complaint of a “weird upper tooth.” The baby´s mother
explained that the tooth had erupted like that, with a darker
color. Clinical examination revealed an atypical DDE with
pulp exposure on the primary maxillary left central incisor
(Figure 2). The contralateral incisor showed diffuse white
opacities on the incisal edge, and his primary mandibular
right central incisor was fused to the lateral incisor.
Due to pulp involvement, a pulpotomy and the placement
of a pediatric zirconia crown was planned. After local anes-
thesia administration and rubber dam isolation, the coronal
pulp tissue was removed with a sterile low-speed round bur.
The pulp chamber was rinsed with saline solution, and a
moist cotton pellet with 1.25 percent sodium hypochlorite
was placed for one minute. The pulp chamber was again
rinsed and filled with mineral trioxide aggregate (NuSmile
®
NeoMTA, NuSmile , Houston, Texas, USA) and covered with
a resin-modified glass ionomer Cement (NuSmile BioCem,
®
NuSmile ).
A week later, the tooth was prepared under local anesthe-
sia and a zirconia crown (NuSmile ZR, NuSmile ) was ce-
mented with a resin-modified glass ionomer cement (NuSmile
®
®
BioCem, NuSmile ). His two-week control visit showed a
correct cervical crown adjustment. At this visit, a diffuse opa-
city was noticed on his newly erupted maxillary right lateral
incisor. During his last control visit four months later, the
patient remained asymptomatic and the periapical radiograph
showed a clear dentinal bridge and continued root development
(Figure 3).
Case 3
A 13-month-old boy presented with his mother to the Pediatric
Dentistry Postgraduate Department at the University Peruana
Cayetano Heredia, Lima, Peru. His mother complained that
he had a newly erupted “black upper tooth” with no history
of dental trauma or pain. On inspection, his maxillary left
central incisor had a notch-like defect near the pulp and an
apparent pulpal inflammation that pigmented the chamber
dark red. The enamel bordering the defect seemed normal. His
mandibular central incisors had demarcated white opacities
(Figure 4). The radiograph showed a lesion close to the pulp,
but no periapical lesion. As there was no referred pain and
the dental pulp was not exposed, a composite strip crown was
fit (Filtek™ Z350 XT, 3M ESPE) under cotton roll isolation.
At a follow-up visit eight months later, a localized abscess
was observed and a pulpectomy was performed through the
composite crown using calcium hydroxide-iodoform paste
(Vitapex, Neo Dental International, Federal Way, Wash., USA).
A glass ionomer lining (Vitrebond™ , 3M ESPE) was placed
at the entrance of the pulp chamber. A year later, the tooth
had a correct clinical appearance and no sign of radiographic
apical lesion.
Table 1 summarizes the characteristics of the three cases.
DDE OR PEIR IN INCISORS? 147
PEDIATRIC DENTISTRY V 42 / NO 2 MAR / APR 20

was unilateral, no plaque was visible, and no caries risk factors

Table 1. SUMMARY OF THE CHARACTERISTICS OF THREE
could be identified. ECC was also ruled out because the chil-
CLINICAL CASES*
dren were very young for that magnitude of dental destruction;
Characteristics Case 1 Case 2 Case 3 also, the mothers reported that the teeth had erupted with
these defects.
Patient’s age at the first visit 10 months 8 months 13 months
The suspicion that this asymmetrical entity could be pre-
Gender Male Male Male eruptive intracoronal resorption is based on the significant size
Country of origin Spain Mexico Peru of the defects. It is also unlikely that hypoplasia could cause
such a severe posteruptive loss of dentin in such a short time,
Affected tooth E F F
especially considering that these incisors had just emerged and
Notch-like asymmetric defect Yes Yes Yes had not yet been subject to masticatory forces.
Did it erupt like that? Yes Yes Yes PEIR is a well-circumscribed radiolucent lesion located in
History of dental trauma No No No the coronal dentin, just beneath the enamel-dentin junction of
unerupted teeth.17-20 The enormous size of the defect relative
History of pain/difficulty eating No No No
to the short time the tooth has been in the mouth should
History of complications during No No No alert the clinician that the lesion is unlikely to be caries.21 The
pregnancy or birth origin of the resorption cells, as well as their pathway of entry,
History of illness or fever during No No No is not well understood. It is speculated that the cells arise from
the first 6 months undifferentiated cells of the developing dental follicle. These
History of hospitalization during No No No resorption cells probably enter the dentin through a break in
the first 6 months the enamel surface, such as hypoplastic pits or lamellae.21 The
Pulp diagnosis Reversible Reversible Irreversible trigger factors for the resorption are also unknown.22 The pro-
pulpitis pulpitis pulpitis gress of the lesion is stopped short of the pulp by the predentin
DDE on other teeth No Yes Yes layer. The resorptive process rapidly progresses through the
dentin but will resorb the enamel at a much slower rate, prob-
Dental anomalies on other teeth No Yes (fusion) No
ably due to its much higher calcified structure. When the
Caries lesions on other teeth No No No tooth erupts or when its crown is exposed surgically, the lesion
Caries risk factors detected or No No No loses its nutrient supply line and its vital elements necrose,
reported by mother leaving an inert and dead tissue within the crown of the tooth.
Restorative treatment Glass- MTA Pulpectomy This tissue is harmless, but it may later be secondarily affected
ionomer Pulpotomy + composite by dental caries. For this reason, it needs to be treated soon
lining + + zirconia strip crown after eruption.23
composite crown PEIR prevalence in the permanent dentition has been
strip crown estimated to occur in approximately one to six percent of chil-
dren.18,20,24 PEIR in primary teeth has been described twice in
* DDE=development defects of enamel; MTA=mineral trioxide aggregate. the dental literature.19,25 It could be argued that the etiology
of the defects in these three cases was PEIR due to the lack of
clinical symptoms (in spite of deep dentinal involvement),
asymmetry of the lesions, and disproportionate size of the
Discussion dental destruction considering the age of the babies and the
DDE in primary teeth, although capable of rapid posteruptive time since dental eruption. The pulpal involvement in two of
changes,5 are rarely as severe and as asymmetrical in recently the cases described was probably due to the penetration of
erupted primary incisors as seen in these three cases. Unilateral bacteria through a microscopic defect in enamel once the teeth
notch-like defects in maxillary central incisors at these early were exposed to the oral cavity. The authors could also hypo-
ages have not been previously reported in the dental literature. thesize that the presence of a DDE predisposes it to PEIR.
DDE are infrequent in primary central incisors,6 although Perhaps a quantitative enamel defect (hypoplasia) could
they could be considered as a differential diagnosis. Moreover, facilitate the entry of resorption cells into the dentin, favoring
in two cases, diffuse and demarcated opacities could be distin- rapid coronal destruction as seen in these cases. However, the
guished on other teeth. Considering that, in all of the cases authors do not possess enough clinical, histological, or genetic
presented here, the mothers reported that the teeth already information to confirm this possibility.
erupted with the defect, it seems feasible that the lesion was Although all three children were very young and the
present prior to eruption. clinical appearance was similar, they were restored differently,
DDE severity is related to the magnitude and duration of depending on the clinicians’ judgement. Due to the young age
the disturbance during tooth formation.13-15 In these three pa- of these patients and the poor root development of the affected
tients, the unilateral lesions are severe. Considering this, a incisors, the aim of the restorative treatment was to avoid bac-
report of a significant event during the prenatal or perinatal terial penetration to the pulp, maintain pulp vitality, and,
period would be expected. However, mothers reported no therefore, allow normal root development. This is the first work
history of systemic or local factors. Genetic alterations have also to show PEIR cases in primary incisors, likely due to the fact
been reported as etiological factors for DDE, although mul- that radiographs of unerupted primary teeth are seldom
tiple affected teeth are the common pattern in those cases.4 justifiable.
Early childhood caries (ECC) was also ruled out, as Based on the cases presented here, it can be concluded
carious lesions typically present along the gingival margin of that, due to the rapid progression of defects like these in
maxillary incisors.16 In the cases presented here, the pattern maxillary incisors, it is crucial that parents be aware of the

148 DDE OR PEIR IN INCISORS?


importance of the first dental visit before age one in order to
preserve coronal and pulp integrity as much as possible. Addi-
tionally, pediatric dentists must be able to diagnose and
promptly manage atypical dental defects like the ones presented
in this article. Lastly, although the three cases come from dif-
ferent countries, they probably share the same etiology,
although it remains unclear if this is confirmation of PEIR or
a new clinical identity.
Acknowledgment
The authors wish to thank Kim Seow, BDS, MDSc, PhD,
DDS, professor, School of Dentistry, The University of Queens-
land, Brisbane, Queensland, Australia, for her review and intel-
lectual contribution to the paper discussion.
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