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Implants in children with ectodermaldysplasia: a case report and literaturereview
Franz-Josef KramerCarola BaethgeHarald Tschernitschek
Authors’ affiliations:Franz-Josef Kramer, Department of Oral andMaxillofacial Surgery, Georg-August-University ofGoettingen, Goettingen GermanyCarola Baethge, Department of Orthodontics,Charite Berlin, Campus Benjamin Franklin,GermanyHarald Tschernitschek, Department ofProsthodontics, Medical University of Hanover,Hanover, Germany
Correspondence to:Priv. Doz. Dr Dr Franz-Josef KramerDepartment of Oral and Maxillofacial SurgeryUniversity HospitalGeorg-August-University of Goettingen37099 GoettingenGermanyTel.: þ49 551 39 2857Fax: þ49 551 39 2886e-mail: [email protected]
Key words: child, dental implants, ectodermal dysplasia, hypodontia, pediatric dentistry
Abstract: The replacement of teeth by implants is usually restricted to patients with
completed craniofacial growth. Implant insertions in children or adolescents are
circumvented due to several unfavorable potential effects including trauma to tooth germs,
tooth eruption disorders and multidimensional restrictions of skeletal craniofacial growth.
Moreover, the functional and esthetic results of the oral rehabilitation are only temporary
acceptable. However, to a small number of pediatric patients suffering congenitally from
severe hypodontia caused by syndromes such as ectodermal dysplasia, conventional
prosthodontic rehabilitations are insufficient. We report the case of a boy with ectodermal
dysplasia who exhibited a severe hypodontia and who was treated with implants inserted
into the anterior mandible at the age of 8 years. The implants were functionally loaded and
resulted in a high patient satisfaction. We recommend the early insertion of dental implants
in children with severe hypodontia. Reviewing the current literature, several aspects of
syndromic hypodontia, patient selection and implant planning are discussed.
Implants inserted into pediatric patients
do not follow the regular growth process
of the craniofacial skeleton and are known
to behave similar to ankylosed teeth, re-
sulting in both functional and esthetic
disadvantages (OpHeji et al. 2003). Addi-
tionally, they can interfere with the posi-
tion and the eruption of adjacent tooth
germs, thus resulting in potential severe
trauma of the patient (Rossi & Andreasen
2003). These and other adverse effects have
resulted in a very restrictive indication for
dental implants in those individuals who
have not completed craniofacial growth yet
(OpHeji et al. 2003).
However, in the last few years, an ex-
ception of this restriction was reported in
those children who suffer from extended
hypodontia or even adontia. While oligo-
dontia with deficiency of only a few teeth
is a relative common finding some popula-
tions, the absence of numerous teeth oc-
curs only rarely (Silverman & Ackerman
1979; Mattheeuws et al. 2004). Most often,
these children suffer from congenital syn-
dromes such as ectodermal dysplasia (ED;
MIM #305100) (Percinoto et al. 2001), a
rare congenital disease characterized by an
aplasia or dysplasia of tissues of ectodermal
origin such as hair, nails, skin and teeth
(Kere et al. 1996). In affected patients, the
extensive lack of both deciduous and per-
manent teeth results in an atrophy and a
reduced growth rate of the affected alveolar
processes (Johnson et al. 2002). Recent
reports suggest that these pediatric patients
can benefit remarkably from an implant-
supported oral rehabilitation already during
childhood (Guckes et al. 2002). The aim of
the present study is to present a report on a
boy with hypodontia caused by hypohydro-
tic ED, who received dental implants at theCopyright r Blackwell Munksgaard 2006
Date:Accepted 20 February 2005
To cite this article:Kramer F-J, Baethge C, Tschernitschek H. Implants inchildren with ectodermal dysplasia: a case report andliterature review.Clin. Oral Impl. Res. 18, 2007; 140–146doi: 10.1111/j.1600-0501.2006.01180.x
140
age of 8 years. Special considerations of
implant insertions in growing individuals
are discussed and the current literature is
reviewed.
Case report
The proband (male, white, Caucasian) ex-
hibited the full-blown condition of hypo-
hydrotic ED. At the age of 3 years, clinical
evaluation revealed a hyperactive child
with a dry hypopigmented scaly skin.
The hair was thin, sparse and blond with
sparse eyelashes and eyebrows (Fig. 1).
Intraorally, a severe hypodontia in both
the deciduous and permanent dentition
was found; in the deciduous dentition,
only the central maxillary incisors were
present and in the permanent dentition
only the central incisors and the germ of
the first right mandibular molar was found
(Fig. 2). Following a skin biopsy, hypohy-
drotic ED was diagnosed. At age five,
dentures made of heat-cured acrylic resin
were integrated in both the upper and lower
jaw. During the following years, the man-
dibular rehabilitation became increasingly
difficult due to growth of the mandible and
deficient height of the alveolar processes.
This resulted in recurrent functional insuf-
ficiencies of the lower denture.
At the age of 8 years it was decided
to insert two implants into the anterior
mandible to improve oral functions and to
reduce potential psycho-social handicaps
of the patient. The implant positions were
planned at articulated cast models and
transferred into surgery by an individual
waiver that was fixed at the maxillary
denture (Fig. 3a–c). Following regional an-
esthesia, two implants were placed ad
modum Branemark in the canine region
of the anterior mandible (Fig. 3d–g). De-
spite a remarkable multi-dimensional atro-
phy of the mandibular alveolar process, the
insertion of cylindric screw implants (No-
bel Biocare MK III; diameter 3.75 mm,
Nobel Biocare, Gothenburg, Sweden)
with a length of 13 mm was easily possible
and resulted in safe primary stability
(Fig. 4). After a submerged healing period
of 3 months, the implants were exposed
and abutment connection was performed.
Prosthodontic procedures were started 2
weeks later as soon as the soft tissues
around the abutment cylinder had healed.
An impregum (3M Espe AG, Seefeld, Ger-
many) impression was taken of the lower
jaw with abutment copings and an alginate
impression of the maxillary denture. Mod-
els were made and mounted in an articu-
lator after bite registration.
In the mandible, a bar-based denture was
integrated (Fig. 5a–d). In the edentulous
maxilla, a new conventional denture was
integrated, allowing satisfactory function
(Fig. 5e, f). The boy got easily accustomed
to the new rehabilitation (Fig. 6a–c) and
performed an excellent oral hygiene. The
parents reported that after the insertion of
the restoration, the dietary intake of the
patient had changed significantly. The boy
began to eat meat and apples, food he
avoided before. The denture has now been
in situ for 2 years and has functioned well.
Discussion
Ectodermal Dysplasia
Severe hypodontia or even adontia in chil-
dren are very rare conditions, most often
associated with congenital syndromes such
as Down syndrome (trisomy 21) (Mestro-
vic et al. 1998) or ectodermal dysplasia
(Silverman & Ackerman 1979). There ex-
ist more than 170 clinically distinct her-
editary syndromes in which ED is present
(Kere et al. 1996; Lamartine 2003). They
are caused by an impaired development of
epidermal appendages and are characterized
by a primary defect at least in one of the
following tissues: nails, hair, teeth or sweat
glands (Priolo et al. 2000). EDs are rare
diseases with an estimated incidence of
seven in 100,000 births for all EDs (Itin
& Fistarol, 2004). The pattern of inheri-
tance is different, including Mendelian
modes and sporadic cases. Several classifi-
cations of EDs have been proposed from a
clinical point of view (Pinheiro & Freire-
Maia 1994), with molecular genetic attri-
butes (Priolo et al. 2000) and based on
identified causative genes that most
often are involved in processes of intercel-
lular communication and signalling (La-
martine 2003).
The clinical diagnosis of hypohidrotic
EDs in the neonatal period and in early
infancy is sometimes difficult because
sparse hair and absent teeth are often
Fig. 1. Patient at the age of 3 years. The phenotype
of ectodermal dysplasia included a severe hypodon-
tia in both the mandible and the maxilla.
Fig. 2. Patient at the age of 3 years. The phenotype of ectodermal dysplasia included a severe hypodontia in
both the mandible and the maxilla.
Kramer et al . Implants in ectodermal dysplasia
141 | Clin. Oral Impl. Res. 18, 2007 / 140–146
normal findings at this stage. Dysmorphic
facial features such as prominent supor-
aorbital ridges, frontal bossing and a de-
pressed nasal bridge might be interpreted
as normal variants. However, as seen in
the presented case, the diagnosis becomes
more easy during childhood based on the
medical history and clinical examination.
Sparse hair growth and deficient teeth
become increasingly recognized, probably
associated with hypoplastic mucous mem-
branes and a decreased mucous production
in the aerodigestive tract due to absent
mucous glands. This can lead to chronic
upper respiratory tract infections, otitis,
dysphagia, hoarseness, bronchitis and
sometimes hemoptysis (Siegel & Potsic
1990). Facial symptoms become increas-
ingly manifest and include maxillary
hypoplasia, ‘saddle’ nose formation, pro-
minent lips and linear wrinkles around the
eyes. The hair is fine, dry, brittle and
sparse, and the skin is thin and dry with
hypohidrosis. Absent or decreased sweat-
ing in patients with anhidrotic, respec-
tively, hypohydrotic ED is caused by
absence of sweat glands. Affected children
have difficulty controlling fevers and al-
ready mild illness may produce extremely
high fevers because of absent temperature
regulation by sweating. Affected adults are
unable to tolerate a warm environment
and require special measures to maintain
a normal body temperature (Pinheiro &
Freire-Maia 1994). The diagnosis of hypo-
hydrotic ED is usually performed by a
biopsy of the skin that shows absent or
hypoplastic sweat glands. Additionally, for
some subtypes of EDs genetic testing is
available (Vincent et al. 2001). Currently,
there is no causative treatment available.
Intraorally, beside mucous symptoms,
patients with hypohydrotic ED suffer
from both an altered shape and a reduced
number of teeth. In clinical examinations
(Glavina et al. 2001) and experimentally in
knock-out organisms representing the phe-
notype of ED (Kristenova et al. 2002), the
teeth were often found to be smaller in size
and exhibited a conical peg-like shape.
Hypo-respectively adontia related to ED
can affect both the deciduous and perma-
nent dentition (Nunn et al. 2003). The
most conserved teeth in hypohydrotic ED
include in the permanent dentition the
maxillary central incisors, maxillary first
molars, mandibular first molars and max-
illary canines (Guckes et al. 1998).
It was reported that especially in mild
forms of ED, the most common complaint
of childhood and adolescence is concern
about the dental anomalities and facial
appearance (Siegel & Potsic 1990). Conse-
quently, the dentist, orthodontist or the
maxillofacial surgeon are probably the first
medical professionals to be confronted with
complaints of EDs.
Implant positioning
The conventional prosthodontic treatment
of patients with severe hypodontia pre-
sents considerable problems (Ekstrand &
Thomsson 1988; Boj et al. 1993). The
irregular distribution and abnormal shape
of teeth often limit the integration of
bridges or crowns in patients with ED
(Nunn et al. 2003). Especially, the hypo-
dontic mandible of children with ED ex-
hibits underdeveloped alveolar ridges
(Rashedi 2003) and is therefore an area in
which it is very difficult to gain adequate
retention and support for conventional
prostheses (Oesterle 2000). Concerning
Oesterle, a prudent clinician should al-
ways attempt to use a conventional proth-
esis to gather functional and esthetic
information to aid in the design of the
final prosthesis and to allow as much
growth as possible before initiating the
implant-assisted phase of treatment (Oes-
terle 2000).
Fig. 3. (a–g) Surgical procedure of implant position-
ing and implant insertion.
Kramer et al . Implants in ectodermal dysplasia
142 | Clin. Oral Impl. Res. 18, 2007 / 140–146
The insertion of dental implants in
children or adolescents before completion
of craniofacial growth is related to several
problems. Experimentally, it was shown
that endosseous implants placed in
young pigs have imitated the effects of
ankylosed teeth (Sennerby et al. 1993).
Placed in alignment with adjacent teeth,
the implants did not participate in growth
processes, resulting in an infraocclusion
and multidimensional dislocation when
compared with the developing teeth
(Sennerby et al. 1993). Additionally, adja-
cent tooth germs exhibited morphologic
changes and disorders of eruption (Thilan-
der et al. 1992). In the nearly anodontic
child, however, these problems can be
neglected.
Several aspects of craniofacial skeletal
growth seem relevant for implant inser-
tions in hypodontic children. Both the
maxilla and the mandible are dynamically
changing during childhood (Bjork 1963,
1977; Skieller et al. 1984; Enlow 1990).
Behaving similar to ankylosed teeth
implants cannot participate with the
maxillary growth processes of drift and
displacement (Enlow 1990), resulting in
unpredictable implant dislocations during
growth or, if implants are fixed together,
maxillary growth disturbances. Because
of the resorptive aspects of maxillary
growth at the nasal floor and the
anterior surface of the maxilla, unpredict-
Fig. 4. Orthopantomographic view after implant insertion.
Fig. 5. (a–f) Concept of prosthodontic rehabilitation.
Fig. 6. (a–c) Extra- and intraoral appearance of the
patient with the implant-supported rehabilitation.
Kramer et al . Implants in ectodermal dysplasia
143 | Clin. Oral Impl. Res. 18, 2007 / 140–146
able implant dislocations in vertical and
anteroposterior direction can occur and
even implant losses have to be expected.
Transversal growth of the maxilla occurs
mostly at the midpalatal suture. Conse-
quently, fixed implant constructions
crossing the midpalatal suture will result
in a transversal growth restriction of the
maxilla. All in all, the insertion of im-
plants in the growing maxilla should be
avoided until early adulthood (Cronin &
Oesterle 1998).
In the mandible, however, the transver-
sal skeletal or alveolo-dental changes are
less dramatic as in the maxilla. In the
posterior mandible, growth changes occur
predominantly in late childhood with large
amounts of anteroposterior, transverse and
vertical growth (Enlow 1990). Addition-
ally, the mandible undergoes rotational
growth, resulting particularly in vertical
alterations (Skieller et al. 1984; Enlow
1990). When several teeth are present,
vertical growth is a major aspect of dental
height increase and results in anteroposter-
ior compensatory changes in the dentition.
Consequently, implants would remain in
an infraocclusal position and would prob-
ably be displaced in the anteroposterior
direction (Oesterle 2000). To our knowl-
edge, there exist in the literature no reports
on implant insertions in the posterior
mandible in pediatric patients.
In the anterior mandible, however, al-
veolar growth seems relatively small when
teeth are missing (Oesterle 2000). The
majority of transversal growth of the
mandible occurs quite early in childhood;
the anteroposterior growth occurs mainly
at the posterior mandible (Skieller et al.
1984). The use of implants to replace
single teeth in the anterior mandible is
not advisable due to the compensatory
anteroposterior and the vertical growth in
this area, however, in children with severe
hypodontia, the anterior mandible might
represent probably the most suitable site
of implant placement (Oesterle 2000).
In the last few years, several case reports
of implant insertions in the anterior
mandible of children have been published
(Bergendal et al. 1991; Bergendal 2001;
Kargul et al. 2001; Giray et al. 2003);
most of the authors agree that the man-
dibular anterior area seems to hold the
greatest potential for very early use of
an implant-supported prosthesis. In a
monocentric prospective study, the survi-
val rate of implants placed in the anterior
mandible of pediatric patients with ED
was reported with 91% (Guckes et al.
2002). Interestingly, some reports have
demonstrated that craniofacial morphol-
ogy did not differ significantly between
implant-treated and non-treated children
with ED, suggesting that treatment with
intraosseous dental implants did not ne-
cessarily interrupt normal craniofacial
growth as assumed before (Johnson et al.
2002). In the long run, implants located
at the anterior mandible probably seem
affected by the mandibular growth rota-
tion, which can result in a change in
implant angulation (Becktor et al. 2001).
Implant timing
The finding of the ideal time of implant
treatment in children seems quite difficult
because many different aspects have to be
considered while finding the best indivi-
dual treatment strategy. It seems clear that
the placement of implants in children
with ED must be a team effort consisting
of a surgeon, prosthodontist, orthodontist
and periodontist. In younger ages, addi-
tionally, a pediatric dentist might also be
required. First, the medical diagnosis of
ED must be confirmed to avoid mistreat-
ment. Profound dental clinical and radio-
graphic examination to determine the
extent of hypodontia is fundamental for
further planning. The role of the dental
team includes the preservation of the ex-
isting dentition, especially in patients
with xerostomia, and maintenance of an
adequate oral hygiene. If some teeth are
present, their prognosis might be esti-
mated. The orthodontist might use the
intended implants as an orthodontic an-
chor to obtain the best position for the
existing dentition. Particularly important
is an adaequate prosthodontic rehabilita-
tion in order to ensure functional, esthetic
and psychological well-being of the pedia-
tric patient. Interim restorations may be
indicated before definitive care and ortho-
dontic treatment may be necessary to
optimize dental position and axis. To
date, there is only little research of the
impact of extensive hypodontia on a
young person (Hummel & Guddack
1997). It was reported that children with
disabilities realize at the age of 9 years
their specific conditions when they com-
pare themselves with other children (Hog-
berg et al. 1986), probably resulting in a
state of depression (Nussbaum & Carrel
1976). Therefore, the dental team also has
to support the child in coping with issues
of attractiveness during the formative
years of childhood (Hogberg et al. 1986).
There are reports that implant treat-
ment should be ended before puberty
for optimum functional and psychosocial
development (Giray et al. 2003). Never-
theless, reports in the literature describe
placement of implants as early as 3
years (Guckes et al. 1997) or 5 years of
age (Smith et al. 1993). From the ortho-
dontic view the safest time to place im-
plants seems to be during the lower
portion of the declining adolescent growth
curve at or near adulthood that can be
determined by cephalographic radio-
graphs, serial measure of stature or hand-
wrist radiographs (Oesterle 2000). Other
relevant aspects to consider include the
individual status of the existing dentition,
the functional status of mastication and
phonetics, esthetic aspects and emotional/
psychological well-being (Nunn et al.
2003). Finally, both the parents and the
child have to be compliant to implant
treatment and implant hygiene (Kearns
et al. 1999).
Conclusion
Following the presented case and the re-
viewed literature, we recommend the in-
sertion of implants in those pediatric
patients who suffer from extended syndro-
mal hypodontia such as seen in ectodermal
dysplasia. The most suitable site of inser-
tion seems to be the anterior mandible;
insertions in the maxilla should be avoided
or at least should not cross the midline. In
order to determine the optimal individual
time point of implant insertion, the status
of skeletal growth, the degree of hypodon-
tia and extension of related psychosocial
stress should be taken into account in
addition the status of the existing dentition
and dental compliance of the pediatric
patient.
Kramer et al . Implants in ectodermal dysplasia
144 | Clin. Oral Impl. Res. 18, 2007 / 140–146
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