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Non-syndromic occurrence of true generalized microdontia with mandibular mesiodens - a rare case

Head & Face Medicine20117:19

DOI: 10.1186/1746-160X-7-19

Received: 25 May 2011

Accepted: 28 October 2011

Published: 28 October 2011

Abstract

Abnormalities in size of teeth and number of teeth are occasionally recorded in clinical cases. True generalized microdontia is rare case in which all the teeth are smaller than normal. Mesiodens is commonly located in maxilary central incisor region and uncommon in the mandible. In the present case a 12 year-old boy was healthy; normal in appearance and the medical history was noncontributory. The patient was examined and found to have permanent teeth that were smaller than those of the average adult teeth. The true generalized microdontia was accompanied by mandibular mesiodens. This is a unique case report of non-syndromic association of mandibular hyperdontia with true generalized microdontia.

Keywords

Generalised microdontia Hyperdontia Permanent dentition Mandibular supernumerary tooth

Introduction

Microdontia is a rare phenomenon. The term microdontia (microdentism, microdontism) is defined as the condition of having abnormally small teeth [1]. According to Boyle, "in general microdontia, the teeth are small, the crowns short, and normal contact areas between the teeth are frequently missing" [2] Shafer, Hine, and Levy [3] divided microdontia into three types: (1) Microdontia involving only a single tooth; (2) relative generalized microdontia due to relatively small teeth in large jaws and (3) true generalized microdontia, in which all the teeth are smaller than normal. According to these authors, aside from its occurrence in some cases of pituitary dwarfism, true generalized microdontia is exceedingly rare. Microdontia of a single tooth can be further classified into (1) microdontia of the whole tooth, (2) microdontia of the crown of the tooth, and (3) microdontia of the root alone [4].

Involvement of the entire dentition is rare and been reported in radiation or chemotherapeutic treatment during the developmental stage of the teeth [5], pituitary dwarfism [3] and Fanconi's anemia [6]. The syndromes associated with microdontia are Gorlin-Chaudhry-Moss syndrome, Williams's syndrome, Chromosome d/u, 45X [Ullrich-Turner syndrome], Chromosome 13[trisomy 13], Rothmund-Thomson syndrome, Hallermann-Streiff, Orofaciodigital syndrome (type 3), Oculo-mandibulo-facial syndrome, Tricho-Rhino-Phalangeal, type1 Branchio-oculo-facial syndrome.

Supernumerary teeth are defined as any supplementary tooth or tooth substance in addition to usual configuration of twenty deciduous and thirty two permanent teeth [7]. Classification of supernumerary teeth may be based on position or morphology. Positional variations include anterior mesiodens, para-premolars, para-molars and disto-molars. Variations in morphology consist of supplemental and rudimentary types [8].

Supernumerary teeth are common in the maxillary anterior region although supernumerary teeth have been reported in the incisor region of the mandible are very rare. Although supernumerary teeth have been reported in the incisor region of the mandible, they are very rare [914].

Conditions, in which supernumery teeth found, are cleidocranial dysplesia, cleft lip and cleft palate [15]. Syndromes associated with supernumery teeth are Familial adenomatous polyposis [Gardner's], Apert, Klippel-Trenaunay-Weber, Craniometaphyseal dysplasia, Trisomy 21[Down's], Nance-Horan, Orofaciodigital syndrome (type 3), Sturge-weber and Tricho-Rhino-Phalangeal, type1.

In the case described here is a bizarre generalized microdontia involving the entire dentition along with mandibular mesiodens without any other apparent systemic conditions.

Case Report

The patient was a 12 year old boy, only child of consanguineous parents, reported to the department of pedodontics and preventive dentistry with the complaint of small teeth. Parents noted small teeth ever since the eruption of permanent teeth. No abnormalities were reported, however, in their extended family.

Physical examination

Physical growth was within normal limits. The patient was of normal in stature, appearance, height, and weight for his age. Upon examination of the limbs, hands, skin, hair, nails and eyes were all appeared normal. No abnormality was noted in neck, back, muscles, cranium and joints as well. Intellectual and scholastic performance was also normal. His medical history was unremarkable; no other abnormalities were noted in the history apart from the difficult delivery. The child was examined and found to be free of any gross abnormalities.

His blood profile was normal. Serum calcium, phosphorous and alkaline phosphatase levels were also normal. Endocrinological investigation was carried out to rule in or out the possibility of hormonal disorder, and the results were within normal limits.

Intraoral examination

The intraoral soft tissues were healthy, but the teeth were abnormal in size and shape (Figure 1 and 2). Diagnostic casts were obtained to aid in diagnosis (Figure 3). Patient was in permanent dentition, teeth present were small in size. The patient had normal occlusion with excessive spacing between the teeth. Fully erupted mandibular mesiodens was present between the central incisors.
Figure 1

Intra oral view of the upper arch.

Figure 2

Intra oral view of the lower arch.

Figure 3

Diagnostic casts showing the morphology of the teeth.

The anterior teeth lacked normal size in all dimensions. Most of the anterior teeth were "peg-shaped" without the typical variation in mesiodistal and labiolingual dimensions. Almost all the maxillary anterior teeth did not have lingual pits whereas mandibular central and lateral incisors had prominent pits on the lingual surfaces. The posterior teeth were also small and exhibited a short occlusogingival dimension. Overall, the dentition was smaller than that of the average adult (Table 1 and 2). Orthopantomogram or the Intra oral periapical radigraph could not be taken because the patient was not able to afford.
Table 1

Comparison of buccolingual/labiolingual and mesiodistal crown dimensions with an anatomic average* of the right side maxillary and mandibular teeth

Right side

Central incisor

Lateral incisor

Canine

First premolar

Second premolar

First molar

Second molar

Total

Maxillary

MD

LL

MD

LL

MD

LL

MD

BL

MD

BL

MD

BL

MD

BL

MD

LL/BL

Average

8.5

7.0

6.5

6.0

7.5

8.0

7.0

9.0

7.0

9.0

10.0

11.0

9.0

11.0

55.5

61.0

Patient

7.5

6.1

4.7

5.6

6.9

7.4

6.4

7.8

6.5

7.7

9.5

10.4

8.7

10.9

50.2

55.9

Mandible

MD

LL

MD

LL

MD

LL

MD

BL

MD

BL

MD

BL

MD

BL

MD

LL/BL

Average

5.0

6.0

5.5

6.5

7.0

7.5

7.0

7.5

7.0

8.0

11.0

10.5

10.5

10.0

53.0

56.0

Patient

4.4

5.6

5.2

5.8

6.6

7.1

6.7

7.3

6.8

7.7

10.7

10.3

9.9

9.4

50.3

53.2

Measurements in millimeters were taken at widest portion of clinical crown on diagnostic casts. *Anatomic average taken from Wheeler, R. C.: Textbook of Dental Anatomy and Physiology, ed. 7, Philadelphia, 1993, W. B. Saunders Company, pp. 25.

Table 2

Comparison of buccolingual/labiolingual and mesiodistal crown dimensions with an anatomic average* of the left side maxillary and mandibular teeth

Left side

Central incisor

Lateral incisor

Canine

First premolar

Second premolar

First molar

Second molar

Total

Maxillary

MD

LL

MD

LL

MD

LL

MD

BL

MD

BL

MD

BL

MD

BL

MD

LL/BL

Average

8.5

7.0

6.5

6.0

7.5

8.0

7.0

9.0

7.0

9.0

10.0

11.0

9.0

11.0

55.5

61.0

Patient

7.2

5.8

4.5

5.4

7.1

7.5

6.2

7.6

6.3

7.7

9.3

10.2

8.7

10.9

49.3

52.1

Mandible

MD

LL

MD

LL

MD

LL

MD

BL

MD

BL

MD

BL

MD

BL

MD

LL/BL

Average

5.0

6.0

5.5

6.5

7.0

7.5

7.0

7.5

7.0

8.0

11.0

10.5

10.5

10.0

53.0

56.0

Patient

4.2

5.7

5.3

6.1

6.7

7.3

6.7

7.2

6.8

7.7

10.7

10.2

9.6

8.9

50.0

53.1

Measurements in millimeters were taken at widest portion of clinical crown on diagnostic casts. *Anatomic average taken from Wheeler, R. C.: Textbook of Dental Anatomy and Physiology, ed. 7, Philadelphia, 1993, W. B. Saunders Company, pp. 25.

The simultaneous presence of microdontia and supernumery teeth is been reported in the Cleidocranial dysplasia, Craniometadiaphyseal dysplasia, Dermoodontodysplasia, Hypodontia and nail dysgenesis, Orofaciodigital syndrome type 3 and Tricho-rhino-phalangeal syndrome type 1. However in this case, except for the dental abnormality in the form of generalized microdontia and the presence of fully erupted mandibular mesiodens between the central incisors were found and no other clinical features observed, therefore all the syndrome associated with the simultaneous presence of microdontia and supernumery teeth were ruled out along with Taurodontism, microdontia, and dens invaginatus as well as Distal symphalangism, hypoplastic carpal bones, microdontia, dental pulp stones, narrowed zygomatic arch (Table 3).
Table 3

Comparison of conditions associated with the simultaneous presence of microdontia and supernumery teeth along with taurodontism, microdontia, and dens invaginatus as well as distal symphalangism, hypoplastic carpal bones, microdontia, dental pulp stones, narrowed zygomatic arch.

Taurodontism, microdontia, and dens invaginatus

Cleidocranial dysplasia

Craniometadiaphyseal dysplasia

Dermoodontodysplasia

Hypodontia and nail dysgenesis

Orofaciodigital syndrome type 3

Tricho-rhino-phalangeal syndrome type 1

Distal symphalangism, hypoplastic carpal bones, microdontia, dental pulp stones, narrowed zygomatic arch

Generalized microdontia

Autosomal dominant

Autosomal recessive

Autosomal dominant

Autosomal dominant

Autosomal recessive

Autosomal dominant

Autosomal dominant

Taurodontism of first permanent molars

Chromosome 6

Macrocephaly

Dry skin

Chromosome 4

Thin/hyperconvex/hypoplastic nails

Autosomal recessive

Absent/small nails

Multiple teeth with one or more dens invaginatus

Arm p

Frontal bossing

Ichthyosis

Arm p

Prominent occiput

Chromosome 8

Microdontia

X-linked recessive inheritance

Normal height (with skeletal dysplasia)

Large fontanelle

Thin skin

Dry skin

Frontal bossing

Arm q

Other dental abnormality

 

Short stature - postnatal

Prominent eyes

Pigmented naevi

Fine hair

Round face

Normal height (with skeletal dysplasia)

Abnormal clinical features of the limbs

 

Absent/small nails

Mandibular hyperostosis/sclerosis

Abnormal hair texture

Brittle hair/trichorrhexis nodosa/pili torti

Hypertelorism

Short stature - postnatal

Brachydactyly

 

Macrocephaly

Optic nerve abnormality/atrophy

Sparse/absent scalp hair - localised

Sparse/absent scalp hair - generalised

Down-slanting palpebral fissures

Decreased body hair/hypotrichosis

Irregularities of length/shape of fingers

 

Flat occiput (brachycephaly)

Microdontia

Abnormal nails

Absent/small nails

Other orbital abnormality

Decreased hair pigmentation - general

Syndactyly of fingers

 

Frontal bossing

Abnormal tooth position/malocclusion/open bite

Midface hypoplasia/flat/short midface

Thin/hyperconvex/hypoplastic nails

Paresis of ocular muscles/squint

Decreased hair pigmentation - patchy

Short foot (including brachydactyly)

 

Wide sutures/delayed fusion of sutures

Missing permanent teeth/retained deciduous teeth

Micrognathia/agnathia/retrognathia

Dysplastic/grooved/thick/discoloured nails

Other eye movement disorder

Fine hair

Syndactyly (other than minimal 2nd and 3rd toes)

 

Large fontanelle

Anodontia/oligodontia

Microdontia

Depressed premaxillary region

Broad/bulbous nasal tip

Brittle hair/trichorrhexis nodosa/pili torti

Irregular length or shape of toes

 

Facies significantly abnormal

Natal/neonatal teeth

Anodontia/oligodontia

Midface hypoplasia/flat/short midface

Cleft soft palate/bifid uvula/submucous cleft

Sparse/absent scalp hair - generalised

Other skull abnormality

 

Small face

Supernumerary teeth

Supernumerary teeth

Micrognathia/agnathia/retrognathia

Microdontia

High hairline - front

Absent/small/hypoplastic carpals

 

Hypertelorism

Dental caries

Other dental abnormality

Absent/decreased eyebrows/lateral thinning

Abnormal tooth position/malocclusion/open bite

Thin/hyperconvex/hypoplastic nails

Symphalangism

 

Prominent supraorbital ridges

Low set ears

Simian creases

Absent/decreased lashes

Supernumerary teeth

Dysplastic/grooved/thick/discoloured nails

Cone shaped epiphyses

 

Depressed premaxillary region

Scoliosis

Dislocated hip

Everted/protruding lips

Cleft/notched tongue

Broad/bifid nails

Symphalangism

 

Midface hypoplasia/flat/short midface

Bowed limbs

 

Tooth crown shape abnormality

Hamartoma/other tumours of the mouth

Frontal bossing

Cone-shaped epiphyses of middle phalanges

 

Prognathism

Mental retardation of any degree

 

Microdontia

Other abnormality of tongue/gingivae/mucosa

High forehead

 
 

Depressed nasal bridge

Boney sclerosis of any type

 

Abnormal tooth position/malocclusion/open bite

Low set ears

Facies significantly abnormal

 
 

Paramedian/lateral cleft lip (uni/bilateral)

Multiple fractures/increased boney fragility

 

Delayed eruption of teeth

Tragus abnormal

Long face

 
 

Cleft soft palate/bifid uvula/submucous cleft

Enchondroma/radiolucencies - localized

 

Anodontia/oligodontia

Pectus excavatum (funnel chest)

Grooved/dimpled chin

 
 

High vaulted and narrow palate

Lytic/lucent lesions of bone

 

Supernumerary teeth

Abnormally placed nipples

Micrognathia/agnathia/retrognathia

 
 

Microdontia

Fibrous dysplasia of bone

  

Thoracolumbar general kyphosis

Medial flare of eyebrows

 
 

Developmental defect of enamel

Wide diaphyses (undertubulation)

  

Irregularities of length/shape of fingers

Absent/decreased eyebrows/lateral thinning

 
 

Tooth discolouration

Submetaphyseal undermodelling/expansion

  

Syndactyly of fingers

Absent/decreased lashes

 
 

Delayed eruption of teeth

Thin cortex of diaphyses

  

Polydactyly - postaxial (ulnar)/type unspecified

Long/large nose

 
 

Missing permanent teeth/retained deciduous teeth

Bowing of long bones

  

Abnormal palmar dermatoglyphics/skin creases

Broad nasal bridge (see telecanthus)

 
 

Supernumerary teeth

Cartilage tongues of metaphyses - localized

  

Polydactyly of feet - postaxial/type unspecified

High nasal bridge

 
 

Dental cysts/tumours

Hyperostosis/thickened/sclerotic calvarium

  

Syndactyly (other than minimal 2nd and 3rd toes)

Broad/bulbous nasal tip

 
 

Deafness - conductive

Absent/abnormal sinuses

  

Cranial nerve/nuclei

Hypoplastic/small nostrils

 
 

Other hearing abnormality

Wormian bones

  

Mental retardation - moderate/severe

Abnormal columella

 
 

Narrow/sloping shoulder/hypermobile shoulders

Sclerotic/hyperostotic facial bones

  

Hypotonia

Thin lips

 
 

Pectus excavatum (funnel chest)

Other skull abnormality

  

Movement disorder - dystonia/chorea/tremor/spasm

Long philtrum

 
 

Bell-shaped chest

Hyperostotic/wide clavicle

  

EEG abnormality

Deeply grooved philtrum

 
 

Thoracolumbar general kyphosis

Abnormal rib structure including fusion

  

Short sternum

Microdontia

 
 

Gibbus/localised kyphosis

Widened ribs

   

Abnormal tooth position/malocclusion/open bite

 
 

Scoliosis

Irregular shape of pubic and ischial bones

   

Supernumerary teeth

 
 

Hyperextensible/hypermobile joints

Absent/hypoplastic/short femur

   

Anteverted/prominent/bat ears

 
 

Small hand

Femora short/deformed/bowed

   

Long/large ear

 
 

Brachydactyly

Other abnormal femur

   

Pectus carinatum (pigeon chest)

 
 

Seizures of any type

Bow legs - genu varum

   

Thoracolumbar general kyphosis

 
 

Hypotonia

    

Scoliosis

 
 

Imperforate anus/anal stenosis

    

Hyperextensible/hypermobile joints

 
 

Horseshoe/fused/ectopic kidneys

    

Small hand

 
 

Hypospadias/epispadias

    

Brachydactyly

 
 

Undescended/ectopic testes

    

Clinodactyly of 5th finger

 
 

Wilms tumour

    

Terminal hypoplasia fingers

 
 

Delayed skeletal maturation

    

Spindle shaped/tapered fingers

 
 

Poorly ossified calvarium/Soft skull

    

Ulnar deviation of fingers

 
 

Absent/abnormal sinuses

    

Other hand abnormality

 
 

Wormian bones

    

Mental retardation of any degree

 
 

Platybasia/basilar impression

    

Abnormal cardiovascular structure/function

 
 

Enlarged foramen magnum

    

Winged/other abnormal scapula (See Shoulder)

 
 

Small/absent scapula

    

Coxa vara

 
 

Winged/other abnormal scapula (See Shoulder)

    

Cone shaped epiphyses

 
 

Absent/hypoplastic clavicles

    

Small femoral head epiphyses

 
 

Pseudarthrosis of clavicle

    

Flat femoral head epiphyses

 
 

Short ribs (circumferential)

    

Deformed/irregular femoral head epiphyses

 
 

Under-/unossified sternum

    

Broad femoral neck

 
 

Hypoplastic/absent ribs

    

Cone-shaped epiphyses of proximal phalanges

 
 

Dorsal wedging of vertebral bodies

    

Some phalanges short and deformed

 
 

Narrow/trapezoid iliac wings (lack of flare)

    

Cone-shaped epiphyses of middle phalanges

 
 

Horizontal/flat acetabular roof

    

Cone-shaped epiphyses of distal phalanges

 
 

Delayed ossification of pubic and ischial bones

      
 

Open pubic symphysis in adults

      
 

Coxa valga

      
 

Coxa vara

      
 

Dislocated hip

      
 

Cone shaped epiphyses

      
 

Fibulae a-/hypoplastic/under-/unossified

      
 

Cone-shaped epiphyses of proximal phalanges

      
 

Cone-shaped epiphyses of middle phalanges

      
 

All middle phalanges short/deformed

      
 

Cone-shaped epiphyses of distal phalanges

      
 

All distal phalanges short/deformed

      

A diagnosis of non-syndromic occurance of true generalized microdontia with mandibular mesiodens was made as no systemic condition was observed. The fully erupted mandibular mesiodens was extracted under local anesthesia in order to correct midline and to facilitate the orthodontic treatment.

Discussion

The initiating factor or factors responsible for microdontia remain obscure. Mutation in developmental regularity genes are known to cause variety of dental defects [16]. Both genetic and environmental factors are involved in the complex etiology of microdontia. Genetic factors probably play a role in the formation of microdontia. Although the proband was the only child, the presence of consanguinity in the form of both parents being maternal first cousins could suggest recessive or polygenic inheritance.

The development of a tooth has been shown to have ectodermal, mesodermal, and neural crest contributions. The variation in size of a particular tooth arises during the period when the form of the tooth is being determined by the enamel organ and the sheath of hertwig at the bell stage of enamel organ. The determination of the form of the crown is thought to be related to different regions of the oral epithelium or to the ectomesenchyme. Studies have shown that different regions of the oral epithelium rather than the underlying ectomesenchyme are initially responsible for the shape of the crown [17]. Bones dating from the Middle Ages which were excavated at Alborg, Denmark proved evidence for generalized microdontia resulting from intrauterine growth retardation [18].

On the basis of visual documentation, the patient in the current case seems to have been more severely affected in all his teeth which exhibited aberrant morphology and all were smaller than normal. MEDLINE search in the English dental literature for true generalized microdontia revealed zero search results. Although child's mother had difficult delivery, it was insignificant and neither microdontia nor mesiodens has been reported in the literature.

The prevalence of mesiodens varies between 0.09 and 2.05% in different studies. In permanent dentition, a 0.15 to 3.8% incidence of mesiodens has been reported [19]. Erupted supernumerary teeth in the mandible are rare, is about 0.01% which indicated marked low value [20]. Supernumerary teeth in the mandible anterior region in this case is fully erupted which is unusual.

Sexual dimorphism is reported by most authors with males being more commonly affected. Hogstrum and Andersson [21] reported a 2:1 ratio of sex distribution. A study of supernumerary teeth in Asian school children found a greater male to female distribution of 6.5:1 for Hong Kong children [22] which indicates that supernumery teeth is more common in males than females which is consistent in our case.

Non-syndromic multiple supernumerary teeth occur most frequently in the mandible region especially premolar region followed by molar and anterior region [9]. Few cases of non-syndrome multiple supernumery teeth have been reported [23, 24] however in the present case non-syndromic single supernumerary tooth was observed in the mandibular anterior region.

Evidence regarding etiology of mesiodens indicates that genetic susceptibility together with environmental factors might increase the activity of dental lamina leading to formation of the extra tooth/teeth [19]. A number of theories have been proposed as regards the causes of the occurrence of supernumerary teeth: 1] Atavism theory [8, 24, 25] 2] Independent hyperactivity of the dental lamina [24, 25] and 3] Dichotomy of the tooth bud are also suggested as a possible etiological factors [8, 25]. However, none of these theories alone offers a sufficient explanation for this phenomenon.

Since mesiodens may interfere with normal occlusal development, in the present case an early diagnosis could have prevented the lower diastema formation. Early diagnosis and treatment of patients with supernumerary teeth are important to prevent or minimize complications.

As the patient did not show any abnormal systemic manifestations, all the syndrome associated with the dental anomalies were ruled out. The simultaneous presence of supernumerary teeth and the generalized microdontia is very rare. To our knowledge, this is the first such case of non-syndromic occurance of true generalized microdontia in association with mandibular mesiodens. Such unusual nature of dental anomaly has not been reported so far in the literature.

Conclusion

The dental finding seen in this case is certainly rare. The case is also sporadic, with no positive family history. The wide variation in clinical manifestations in cases of non-syndromic occurrence of dental anomalies is challenging and is an area for further research. Mesiodens are familiar to pediatric dentists and orthodontists as one of the more common anomalies to affect the developing dentition and it demands a multidisciplinary assessment.

Consent

Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Declarations

Acknowledgements

The authors also wish to thank the patient and their family for their contribution to this article. Written consent for publication was obtained from the patient's parent.

Authors’ Affiliations

(1)
Department of Pedodontics and Preventive Dentistry, AECS Maruthi Dental College and Research Center

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© Bargale and Kiran; licensee BioMed Central Ltd. 2011

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