Delayed Exfoliation of Primary Teeth Due to Second Pathoses:
Case Series Study
ABSTRACT
The delayed exfoliation of primary teeth among
children is a common and frequent dental problem
whose most sited cause is misalignment of the crown
of the successional permanent tooth with the root
apex of the primary tooth. Treatment is often
extraction of the over retained primary teeth and
reassurance of the guardian that the normal tongue
movements will push the misaligned permanent
successional teeth into line in cases of mild
misalignment or orthodontic treatment in severe
cases of misalignment. However, meticulous history
taking and careful clinical oral examination may
yield an underlying pathosis as the three cases in this
study illustrate. Underlying second pathoses may
exert local and systemic effects on the normal
exfoliative mechanism resulting in over retention of
primary teeth despite proper alignment of the crown
of the permanent successional tooth with the root
apex of the primary tooth.
INTRODUCTION
The aetiology of over retention of primary teeth is
often misalignment of the crown of the successional
permanent tooth as it erupts relative to the root of the
primary tooth. This explanation may be satisfactory
to the ‘concerned’parent or guardian but as the three
cases in this study illustrate, there may be a second
pathosis causing the over retention.
Case 1.
A seven-year old black female presented with over
retention of 51, 52, 61, 62, 71 and 81 with ectopic
eruption of the successional permanent teeth 11, 21,
22, 31 and 41
. Poor oral hygiene was clinically
evident as arrested and progressive carious lesions
on all primary and permanent teeth except the
permanent sixes. No periapical abscesses were
evident radiographically nor draining sinuses and a
diagnosis of rampant caries was made. Treatment
rendered was oral health instruction, extraction
under local anaesthesia of over retained primary
teeth and restorations on the remainder.
Interestingly, roots of the over retained 51, 52, 62
and 81 were intact showing no resorption as seen on
the palatal and lingual aspects of 61 and 71
respectively.
Case 2.
A six year-old black female presented with a
progressive swelling of the mandible of one-year
duration. Extraoral examination showed mandibular
prognathism and macrocheilia of the lower lip.
Intraoral examination showed marked alveolar
expansion of the anterior mandible on the labial
aspect and overretention of primary teeth 71, 72, 81
and 82. The corresponding permanent successional
teeth 31, 32, 41 and 42 had erupted on the lingual
aspect of the deciduous teeth (Fig. 1.).
Fig. 1. Case 2: Labial alveolar expansion and over
retention of anterior incisors of mandible and
secondary mandibular prognathism in 6-year-old
black female.
Radiographic investigation showed a well
demarcated unilocular radiolucent lesion of the
anterior mandible. Laboratory investigations of
serum found 48 U/L of alkaline phosphatase
(normal 20-125 U/L), 1.03 mmol/L of calcium
(normal 2.12-2.57mmol/L and 9 g/L of albumin
(normal 35-50g/L). A biopsy of the lesion under
local anaesthesia was submitted for histological
examination, which showed fibrous connective
tissue stroma lined by 3 to 8 epithelial cell layers
with occasional intraluminal heaping of the
epithelial cells. Epithelial cells devoid of nuclei the
so-called ‘ghost cells’were evident in the superficial
epithelial layers and the intraluminal aggregates.
These features were interpreted as those of a
calcifying odontogenic cyst. The case was
subsequently referred to the oral and maxillofacial
surgery clinic for further management.
Case 3.
A 12 year-old black female presented with a 5-year
history of a painless right maxillary swelling that
was noted on extraoral examination as facial
asymmetry. Intraoral examination found a hard
bony swelling limited to the buccal aspect of the
right maxilla covered by normal coloured mucosa.
Over retained primary teeth were present in the
swollen right maxilla namely 52, 53, 54 and 55
(Fig.2) as well as labial to the successional
permanent teeth in the mandible namely 73, 72, 71,
81, 82 and 83 (Fig.3). Laboratory investigations of
serum gave 393 U/L of alkaline phosphatase
(normal 20-125 U/L), 2.49 mmol/L of calcium
(normal 2.12-2.57 mmol/L) and 43 g/L of albumin
(normal 35-50g/L). Radiographic examination
showed a diffuse ‘ground glass’ radiopacity and
expansion of the right maxilla. Asubsequent biopsy
under local anaesthesia of the alveolar bone of the
swollen right maxilla was submitted for
histological examination that showed irregular
trabeculae of woven bone in fibrous connective
tissue stroma; features that were diagnostic of
monostotic fibrous dysplasia. Treatment rendered
included extraction of the over retained primary
teeth whose roots were intact and referral to an oral
and maxillofacial surgery clinic for further
management of the bony expansion and possible
orthodontic exposure of the unerupted permanent
teeth.
Fig. 2. Case 3: Firm bony expansion covered by
normal coloured mucosa and over retention of
primary teeth of right maxilla in 12-year-old black
female. 
Fig.3. Case 3. Over retained mandibular incisors in
12-year-old black female with monostotic fibrous
dysplasia of the right maxilla.
DISCUSSION
Disturbed primary or deciduous tooth exfoliation
can either be premature or delayed. This study
illustrates the possible roles of second pathoses in
the delayed exfoliation or over retention of primary
teeth. The exact mechanism of primary tooth
exfoliation could involve pressure resorption of the
deciduous root invoked by the erupting
successional tooth and or differentiation of
monocytes of the periodontal ligament into
odontoclasts. The odontoclasts then resorb the
deciduous root in a similar manner to osteoclasts
during bone remodeling or resorption with absence
of an inflammatory response
1
. The factor(s) that
trigger this process remain unknown. The majority
of carious deciduous teeth undergo normal
exfoliation without restorations2
. It is postulated
that a cumulative and quantitative effect of rampant
caries may delay the start of the root resorptive
process as case 1 shows with only 2 out of the 6 over
retained teeth showing evidence of root resorption.
Over retention of a deciduous tooth (84) due to the
presence of a COC and an intraluminal
adenomatoid odontogenic tumour has been
reported 3
. Case 2 of this study documents a second
case of COC involvement in over retention of four
deciduous teeth 71, 72, 81 and 82. The COC of case
2 could have locally affected the exfoliative
mechanism of deciduous teeth in the presence of
reduced serum levels of calcium and albumin. The
exact mechanism could have been the misalignment
of successional teeth relative to the deciduous roots
due to the cortical and alveolar cystic expansion,
reduced serum levels of calcium and albumin or
both. However, alveolar cystic expansion was the
most likely explanation as it disturbed the integrity
of the periodontal ligament (PDL) where the
monocytes that differentiate into odontoclasts are
located besides the physical misalignment.
Monostotic fibrous dysplasia (MFD) is a result of
postnatal mutation of the guanine nucleotidebinding protein, alpha-stimulating activity
polypeptide 1 (GNAS1) gene
4
. Case 3 had three
times the normal alkaline phosphatase level while
calcium and albumin were within normal ranges.
Interestingly, the MFD of case 3 exerted both local
and systemic effects during disruption of the
mechanism of deciduous root resorption in the right
maxilla whereas only the systemic effects could
explain the over retention of the mandibular
incisors. The bone trabeculae of fibrous dysplasia
are considered to arise by metaplasia4
and in this
case, the local effect could have been the metaplasia
of alveolar bone of the right maxilla disturbing the
integrity of the PDL. It is theorized that systemic
effects in both jaws but more so in the mandible
could be the arrest of monocyte differentiation into
odontoclasts, failure of the odontoclasts to attach to
the deciduous roots’surface and lacunae formation,
odontoclast failure to release the odontolytic
enzymes into the lacunae or inactivation of released
odontolytic enzymes due to the high serum alkaline
phosphatase levels. Bone growth causes an agedependent rise in alkaline phosphatase normal
values particularly in children <2 yrs and
adolescents. Thereafter, alkaline phosphatase
activity declines reaching normal adult levels after a
growth spurt during adolescence 5
. Three times the
normal values of alkaline phosphatase in case 3 of
the 12-year-old female cannot be adequately
explained by early onset of puberty as the patient did
not have physical signs of puberty at presentation.
Chronic malnutrition reflected by a stunted growth
pattern has been associated with delayed exfoliation
of deciduous teeth 6
. The three children comprising
this study showed no signs of chronic malnutrition
and consequently malnutrition was ruled out in the
differential diagnosis of the aetiology for the over
retention of primary or deciduous teeth. In view of
the unique aetiology for over retention of primary
teeth and the female gender preponderance in this
study, the number of cases does not support a factual
female predominance of second pathoses delaying
exfoliation of primary or deciduous teeth.
R
Dentistry 4 lovers
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