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Implants in children with ectodermal dysplasia: a case report and literature review Franz-Josef Kramer Carola Baethge Harald Tschernitschek Authors’ affiliations: Franz-Josef Kramer, Department of Oral and Maxillofacial Surgery, Georg-August-University of Goettingen, Goettingen Germany Carola Baethge, Department of Orthodontics, Charite ` Berlin, Campus Benjamin Franklin, Germany Harald Tschernitschek, Department of Prosthodontics, Medical University of Hanover, Hanover, Germany Correspondence to: Priv. Doz. Dr Dr Franz-Josef Kramer Department of Oral and Maxillofacial Surgery University Hospital Georg-August-University of Goettingen 37099 Goettingen Germany Tel.: þ 49 551 39 2857 Fax: þ 49 551 39 2886 e-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 the Copyright r Blackwell Munksgaard 2006 Date: Accepted 20 February 2005 To cite this article: Kramer F-J, Baethge C, Tschernitschek H. Implants in children with ectodermal dysplasia: a case report and literature review. Clin. Oral Impl. Res. 18, 2007; 140–146 doi: 10.1111/j.1600-0501.2006.01180.x 140

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Page 1: Ectodermal Dysplasia Implants

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

Page 2: Ectodermal Dysplasia Implants

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

Page 3: Ectodermal Dysplasia Implants

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

Page 4: Ectodermal Dysplasia Implants

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

Page 5: Ectodermal Dysplasia Implants

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

Page 6: Ectodermal Dysplasia Implants

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