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ABSTRACTCongenitally missing teeth (CMT) may present as

complete (Anodontia) or partial (Hypodontia or Oligodon-tia) depending on the number of teeth missing. Althoughit has been widely reported as a component of a syndromesuch as Ectodermal Dysplasia, few reports are availableabout its sporadic and non-syndromic occurrence. CMTmay affect and compromise stomatographic functions suchas mastication, speech and esthetics. We report a case ofmanagement and rehabilitation of a 5-year-old girl that pre-sented with 8 congenitally missing primary teeth with noevidence of their succedaneous permanent teeth germ. Fromour investigations, this case was not associated with anyknown syndrome; hence it is a non-syndromic CMT-oli-godontia involving both primary and permanent teeth. Wemanaged both the functional and psychological effect ofit on the child and made efforts to preserve the orofacialstructures by using Flexible Removable Partial Denture asour treatment modality. It was comfortable for the child,she adapted fast to it, and also the functions, esthetics andher psychological well-being were greatly improved.

Keywords: Congenital, Oligodontia, Tooth Germ,Flexible Denture,

INTRODUCTIONIt is rare but not impossible for a tooth to be congeni-

tally missing. Congenitally missing teeth (CMT) is definedas teeth whose germ failed to develop appropriately to al-low further differentiation of the dental tissue [1-3]. Con-genitally missing teeth, which is also referred to as toothagenesis or dental aplasia is a developmental disorder butone of the most common dental anomalies which may besporadic or a part of a hereditary syndrome in occurrence [1-3]. CMT may affect primary or permanent dentition or bothand may manifest on the upper or lower jaw. It could bebilateral or unilateral and may affect the posterior or ante-

rior segment of the jaws or both. Dental agenesis may alsobe total or partial, mild or moderate depending on the numberof teeth missing [1, 4]. When it is total (i.e. no teeth) CMT isdescribed as anodontia and this may be a manifestation ofan hereditary/developmental anomaly such as ectodermaldysplasia- a disorder where there is disturbance in forma-tion and development of tissues of ectodermal origin prob-ably as a result of mutation during embryonic developmentleading to defective formation of teeth, hair, skin, nails andsweat glands [4, 5]. Partial CMT depending on the numberof teeth missing could be hypodontia or oligodontia.Hypodontia is an anomaly where less than five teeth arecongenitally missing while oligodontia is when missingteeth are up to six or more [1, 6-8].

Depending on the number and site of missing teeth,performances of functions like chewing or mastication maybecome a challenge in CMT. CMT may also cause estheticembarrassments, lower self-esteem, communication and pho-nation defects, reduced professional performance, unpleas-ant facial profile secondary to defective alveolar bone growthand hence the overall quality of life may be affected [2, 4].Therefore, there is a need to treat the condition so as torestore both functions and the psychological well-being ofan affected individual.

The cause of CMT is not yet known. However, someetiological factors have been implicated. These multifacto-rial etiological factors include both genetically induced andenvironmental factors. Among the developmental/geneticfactors that have been implicated are: ectodermal dyspla-sia, cleft lip and palate, and Down syndrome. While envi-ronmental factors include: trauma, radiations, drugs likechemotherapy, systemic diseases like rickets and syphilis,local inflammations, infections like rubella virus, hormo-nal disturbance, increased maternal age, low birth weight,multiple births, smoking and exposure of mother to toxinsduring pregnancy [1, 2, 6, 9-12].

The prevalence of CMT varies from one part of the

Case report

NON-SYNDROMIC CONGENITAL, OLIGODON-TIA: MANAGEMENT OF A CASE IN A NIGERIANCHILD.

Enoch Abiodun Idowu1, Rowland Agbara1, Festus Olabode Oladotun2, ObafunkeDenloye3

1) Faculty of Dentistry, College of Health Sciences, University of Jos; Depart-ment of Dental and Maxillofacial Surgery, Jos University Teaching Hospital,Plateau state, Nigeria.2) Department of Dental and Maxillofacial Surgery, Jos University TeachingHospital, Plateau state, Nigeria.3) Department of Child Oral Health, University College Hospital, Ibadan, Oyostate, Nigeria.

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world to the other with contradicting prevalence amongAfricans. While it has been reported that CMT is morecommon among Africans than any other race in the world,another report suggested a lower prevalence of CMT amongAfricans [13, 14]. However, there is no strong evidence tosupport or refute these claims probably as a result of pau-city of information and scanty report of CMT disorders inliterature from many African countries. Presently, there isno available report in the literature about the prevalenceof CMT in Nigeria which is the most populated Africancountry, but in Kenya, a prevalence of 6.30% CMT out-side 3rd molars have been reported [15]. Likewise, a preva-lence of 10.9% in Iran, 8.50% in Japan, 5.9% in Germanyand 8.80% in United States of America have been reported[1, 16, 17].

While CMT disorder has been reported in some stud-ies to have no gender predilection [9, 18], reports from oth-ers indicated higher prevalence among female than male [9,16, 18, 19].

Although unilateral CMT is more widely reportedthan bilateral, the bilateral CMT involving mostly lateralincisors has been widely reported to be more common in themaxillary jaw than mandible [2, 4]. While the report of somestudies showed an equal prevalence of CMT in both jaws,some indicated a higher prevalence in the maxilla than man-dible and others reported higher prevalence in the mandiblethan maxilla [9, 18, 19]. The most widely reported congeni-tally missing tooth with the highest prevalence in manystudies is third molar [19, 20].

The management of CMT is by multi-disciplinaryapproach involving professionals in the field of dentistrysuch as general dental practitioners, pediatric dentists, re-storative dentists and others such as paediatricians and psy-chologists just to mention a few. The major concern of den-tists is to initiate treatment that will restore masticatory func-tions, facial profile and the overall esthetic well-being ofsuch patients. Consequently, appropriate treatment modal-ity must be adopted, well planned and structured with ad-equate consideration for patient’s age and development, theseverity of the condition in terms of number of teeth miss-ing and the possible challenges that may arise as a result oftreatment of choice. Documented treatment modalities inthe literature include; maintenance of primary tooth, place-ment of implants, provision of a full-full denture, remov-able partial denture and fixed prosthesis (bridge) [21-24].Although each of these modalities has its own challenges, itsuffices to say that the condition may be easy to manage inan adult than in children with disabilities, and also amongthe aged.

Report of CMT disorder involving, a set of primaryteeth and their permanent successors, bilaterally on bothupper and lower jaws in an individual is not readily avail-able in the literature; hence, this case report.

CASE REPORTA five-year-old girl in the company of her mother

presented at the Dental and Maxillofacial Surgery Depart-ment of a regional University Teaching Hospital, with acomplaint of missing teeth (Fig.1).

Fig. 1. Clinical picture of the patient showing (a)scalp hair (b) frontal view (c) hand, skin and fingers.

According to the mother, the child who is the firstbornin a family of two children has been emotionally disturbedbecause of the missing teeth. There is no history of suchincidence of missing teeth in both the nuclear and extendedfamily. There was no history of consanguineous marriageamong the direct family or distance relations of either thefather or the mother. According to the mother, she conceivedher when she was 30 years of age and that she complied withantenatal attendance. A review of drug intake during preg-nancy shows she took Fesolate, Folic acid and Calcium sup-plements as part of her routine antenatal managements.

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She was delivered at 38 weeks through caesarean sec-tion at a tertiary health care centre with the child weighing2.65kg at birth. Since birth, the child’s growth has beennormal with no history of any known systemic illness andno history suggestive of intolerance to heat or absence ofsweating. There was no history of trauma to the orofacialstructure.

Extra-oral examination revealed a healthy-lookinggirl with no obvious bossing of the frontal of the bone, nor-mal appearance of scalp hair (Fig 1a) and eyebrows witheyelashes that are not loose. The nose is not saddled, upperand lower lips were not protruded, but the philtrum appearedflattened (Fig 1b). The nails were normal in appearance andthe skin smooth and fresh without abnormal pigmentations(Fig 1c) Intra-oral examination revealed normal clinicalappearance of the tongue, the floor of the mouth, palate,oropharynx and buccal mucosa. The gingivae on the alveo-lar ridges of the edentulous space on both upper and lowerjaws were slightly folded.

Fig. 2. Clinical photograph showing congenitallymissing primary teeth

Histological report of skin biopsy was unremarkable

Fig. 3. Micrograph showing histological sections of(a) the skin with normal skin epithelium (b) sweat gland.

Teeth Present

Missing Teeth are

EDC CDEEDC CDE

BA ABBA AB

Histological section of skin tissue harvested showednormal anatomy (Fig. 3).Radiographic investigations re-vealed congenitally missing tooth germs of upper and lowerprimary incisors, their permanent successors, and mandibu-lar second and third molars (Fig. 4). Rehabilitation usingFlexible Removable Partial Denture (FRPD) was the treat-ment modality adopted in the management of this case. Af-ter explaining this option to the parents, we obtained theirconsent before starting the procedure. The first step takenwas oral hygiene measures through scaling and polishing ofthe standing teeth. Impressions of the upper and lower jawswere then taken with appropriate stock trays using alginate(Figure 5a).

Impressions were cast in stone (Figure 5b) and thejaw models were used for designing the flexible RP. Thedenture was designed and set up of trimmed adult teeth done(Figure 6).

Fig. 4. Orthopantomograhic view of the patient jawswith congenitally absent tooth germs of upper and lowerdeciduous incisors and their permanent successors, andmandibular second and third molars.

Fig. 5a. Pictures of upper and lower impression takenin alginate.

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Fig. 5b. Stone model on which dentures were fabri-cated.

Fig. 7. Dentures on delivery to patients.

Fig. 6. Wax pattern of the removable partial dentureframework.

The trying-in was carried out, and after the motherand child were satisfied with the colour and the sizes of theteeth, relevant laboratory processes were carried out and thedefinitive FRPD replacing congenitally missing teeth onmaxilla and mandible were produced. The material used forthe construction of denture base was Biosoft Flexible Resin-a derivative of Nylon. Flexible RPD was delivered to thepatient (Figure 7), and both the patient and the mother weretrained on how to insert, withdraw and maintain the den-tures. The patient was recalled two days post-delivery, andexcept for mild pains on mastication from a sharp sport onthe denture, which warranted minor trimming, the patientwas observed to be satisfied. The patient was later recalledafter 2 and then after 4 weeks, and we observed she wascoping well without any challenges. Her last visit to theclinic was 8 weeks post-delivery of dentures, and we ob-served she was satisfied with improved facial appearance,masticatory functions, speech and self-esteem. The patientis now presently on a bi-monthly appointment.

DISCUSSIONThis is a case of non-syndromic CMT (oligodontia)

with 8 primary teeth missing, and absence of tooth germs ofpermanent successors that will replace 8 missing temporaryteeth and those for the upper and lower second and thirdpermanent molars. It is non-syndromic disorder because noother anomaly was found associated with the CMT. It is welldocumented that for ectodermal dysplasia (ED) which is thecommonest cause of syndromic CMT to be diagnosed atleast two of the following features must be well established;Abnormal dentition, Trichodysplasia (Abnormal hair),Onchodysplasia (Abnormal nail) and Dyshydrosis (Abnor-mal or missing sweat gland) [5]. There was no evidence ofthe presence of any of the last three in this case. The scalphair was not scanty, eyebrow with eyelashes appeared nor-mal, the fingernails were growing well, and skin biopsy wasnot suggestive of any abnormality of the sweat gland. Clini-cal history did not reveal intolerant to heat or absence ofsweat. However, genetic screening is a possibility, althoughthis is not readily available in this environment.

We chose to begin transitional treatment at this age(5 years) in order to boost the patient’s self-esteem and psy-chological well-being, improve functions and facilitate betterfacial appearance by normalizing perioral muscle and basalbone growth. Although cases of CMT have been managedin the past with fixed or removable prosthesis, implant andimplant supported prosthesis, orthodontic movement of teethor crowning of well rooted primary teeth with no permanentsuccessor. In this case, flexible, removable partial denturewas our treatment of choice for rehabilitating this patientbecause of the following reasons: flexible, removable par-tial denture is non rigid with lesser possibility of trauma tothe periodontium of the adjacent teeth, residual ridges andadjacent soft tissues during functions, insertion and with-drawal; adaptation is better with flexible than rigid remov-able acrylic dentures; FRPD is lighter compared to the rein-

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1. Sheikhi M, Sadeghi MA,Ghorbanizadeh S. Prevalence of con-genitally missing permanent teeth inIran. Dent Res J (Isfahan). 2012Dec;9(Suppl 1):105-11. [PubMed]

2. Rakhshan V. Congenitally miss-ing teeth (hypodontia): A review ofthe literature concerning the etiology,prevalence, risk factors, patterns andtreatment. Dent Res J (Isfahan). 2015Jan-Feb;12(1):1-13. [PubMed]

3. Moyers RE, Riolo ML. Earlytreatment. Handbook of orthodontics,4th ed. Chicago: Year Book edicalPublishers; 1988. p. 348 53.

4. Jahanimoghadam F, Torabi M,Rostami S. Case Report: CongenitallyMissing Teeth. ASJ. 2015; 12(1):45-50. [Internet]

5. Ralph E, McDonald RE, AveryDR, Developmental and morphologyof primary teeth. In: Donald J, editor.Dentistry for the child and Adoles-cent. 9th ed. Philadelphia: Mosby;2011. p. 52 69.

6. Arte S, Nieminen P, Pirinen S.Gene defect in hypodontia: Exclusionof EGF, EGFR, and FGF3 as candidategenes. J Dent Res. 1996; 75:1346 52

7. Schalkvan der WY, Steen WH,Bosman F. Distribution of missingteeth and tooth morphology in pa-tients with oligodontia. ASDC J DentChild. 1992; 59:133 400

forced rigid acrylic dentures; it will be easy to adjust as thechild passes through the growing phases; it is a means ofencouraging good oral hygiene practices at the early child-hood stage as it is easy to clean. Therefore, the use of rigidconnector which can interfere with the growth of basal bonein a young patient [5] especially when it crosses the midlinewas avoided in this case. We equally considered the place-ment of implant supported prosthesis as not being appropri-ate because the jaw bones are still growing. Although it iswell documented that implant can be placed on anteriormandible as early as 7-12 years according to Guidelines ofNational Foundation for ED [19], the report some studieshave suggested placement of implants after maximum bonegrowth [6, 9, 13]. The reason may be due to the fact thatimplants do grow and it may, therefore, interfere and im-pede both the vertical and horizontal growth of jaw bones,it may also be dislocated, become exposed, ankylosed orcause infraoclussion thereby leading to both functional andesthetic problems especially when used with rigid connec-tors across the midline in fixed prosthesis.

CONCLUSIONThis case report focused on the management and re-

habilitation of a 5-year-old girl that presented with 8 con-genitally missing primary teeth with no evidence of theirsuccedaneous permanent teeth germ. The choice of flex-ible, removable partial denture which is lighter and morecomfortable to wear than a rigid acrylic denture for the re-habilitation of this patient was based on our conviction thatthis child will find it easy to tolerate better than rigid re-movable partial dentures. This treatment modality has im-proved the appearance of the child, masticatory functions,speech and her psychological well-being. The flexible, re-movable partial denture will also give room for future pe-riodic adjustment without interfering with the continuousand progressive growth of jaw bones before definitive treat-ment later in life. It will also boost the child oral hygieneawareness, care and practices early in life.

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Corresponding Author:Dr Agbara Rowland,Faculty of Dentistry, College of Health Sciences, University of Jos/Department of Dental and Maxillofacial Surgery, Jos University TeachingHospital, Plateau state, Nigeria.E-mail: row_prof@yahoo.com

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Please cite this article as: Idowu EA, Agbara R, Oladotun FO, Denloye O. Non-Syndromic Congenital, Oligodontia:Management of a Case in a Nigerian Child. J of IMAB. 2019 Oct-Dec;25(4):2862-2867.DOI: https://doi.org/10.5272/jimab.2019254.2862

Received: 04/02/2019; Published online: 23/12/2019