Scholarly article on topic 'Non-syndromic occurrence of true generalized microdontia with mandibular mesiodens - a rare case'

Non-syndromic occurrence of true generalized microdontia with mandibular mesiodens - a rare case Academic research paper on "Clinical medicine"

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Academic research paper on topic "Non-syndromic occurrence of true generalized microdontia with mandibular mesiodens - a rare case"

HEAD & FACE MEDICINE

CASE REPORT Open Access

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

Seema D Bargale* and Shital DP Kiran

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

* Correspondence: drseemabargale@gmail.com

Department of Pedodontics and Preventive Dentistry, AECS Maruthi Dental College and Research Center, India

[Ullrich-Turner syndrome], Chromosome 13[trisomy 13], Rothmund-Thomson syndrome, Hallermann-Streiff, Oro-faciodigital 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 [9-14].

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-Tren-aunay-Weber, Craniometaphyseal dysplasia, Trisomy 21 [Down's], Nance-Horan, Orofaciodigital syndrome (type 3), Sturge-weber and Tricho-Rhino-Phalangeal, type1.

O© 2011 Bargale and Kiran; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative CGntfäl Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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 pedo-dontics 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 2 Intra oral view of the lower arch.

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.

The simultaneous presence of microdontia and supernumery teeth is been reported in the Cleidocranial dysplasia, Craniometadiaphyseal dysplasia, Der-moodontodysplasia, Hypodontia and nail dysgenesis, Orofaciodigital syndrome type 3 and Tricho-rhino-pha-langeal 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,

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 MD MD MD MD MD MD MD

LL LL LL BL BL BL BL LL/BL

Average 8.5 6.5 7.5 7.0 7.0 10.0 9.0 55.5

7.0 6.0 8.0 9.0 9.0 11.0 11.0 61.0

Patient 7.5 4.7 6.9 6.4 6.5 9.5 8.7 50.2

6.1 5.6 7.4 7.8 7.7 10.4 10.9 55.9

Mandible MD MD MD MD MD MD MD MD

LL LL LL BL BL BL BL LL/BL

Average 5.0 5.5 7.0 7.0 7.0 11.0 10.5 53.0

6.0 6.5 7.5 7.5 8.0 10.5 10.0 56.0

Patient 4.4 5.2 6.6 6.7 6.8 10.7 9.9 50.3

5.6 5.8 7.1 7.3 7.7 10.3 9.4 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.

hypoplastic carpal bones, microdontia, dental pulp stones, narrowed zygomatic arch (Table 3).

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 microdon-tia 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

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 MD MD MD MD MD MD MD

LL LL LL BL BL BL BL LL/BL

Average 8.5 6.5 7.5 7.0 7.0 10.0 9.0 55.5

7.0 6.0 8.0 9.0 9.0 11.0 11.0 61.0

Patient 7.2 4.5 7.1 6.2 6.3 9.3 8.7 49.3

5.8 5.4 7.5 7.6 7.7 10.2 10.9 52.1

Mandible MD MD MD MD MD MD MD MD

LL LL LL BL BL BL BL LL/BL

Average 5.0 5.5 7.0 7.0 7.0 11.0 10.5 53.0

6.0 6.5 7.5 7.5 8.0 10.5 10.0 56.0

Patient 4.2 5.3 6.7 6.7 6.8 10.7 9.6 50.0

5.7 6.1 7.3 7.2 7.7 10.2 CO iol 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.

Taurodontism, microdontia, and dens invaginatus

Cleidocranial dysplasia

Craniometadiaphyseal dysplasia

Dermoodontodysplasia Hypodontia and Orofaciodigital Tricho-rhino- Distal symphalangism, hypoplastic

nail dysgenesis syndrome type 3 phalangeal carpal bones, microdontia, dental pulp

syndrome type stones, narrowed zygomatic arch 1

Generalized microdontia

Taurodontism of first permanent molars

Multiple teeth with Arm p one or more dens invaginatus

X-linked recessive inheritance

Autosomal dominant Chromosome 6

Normalheight (with skeletal dysplasia)

Short stature -postnatal

Autosomalrecessive Macrocephaly

Frontalbossing

Large fontanelle

Prominent eyes

Absent/smallnails Mandibular

hyperostosis/sclerosis

Macrocephaly

Optic nerve abnormality/atrophy

Flat occiput Microdontia

(brachycephaly)

Frontal bossing

Wide sutures/ delayed fusion of sutures

Large fontanelle

Facies significantly Natal/neonatalteeth abnormal

Smallface

Supernumerary teeth

Hypertelorism Dentalcaries

Autosomaldominant Dry skin

Ichthyosis

Thin skin

Pigmented naevi

Abnormalhair texture

Sparse/absent scalp hair - localised

Abnormal nails

Abnormaltooth position/malocclusion/ open bite

Missing permanent teeth/retained deciduous teeth Anodontia/oligodontia Microdontia

Midface hypoplasia/flat/ short midface

Micrognathia/agnathia/ retrognathia

Anodontia/oligodontia Supernumerary teeth

Other dental abnormality

Autosomal dominant Chromosome 4

Autosomal recessive

Autosomal dominant

Thin/hyperconvex/ Autosomal hypoplastic nails recessive

Autosomal dominant Absent/small nails

Prominent occiput Chromosome 8 Microdontia

Dry skin

Fine hair

Brittle hair/ trichorrhexis nodosa/pili torti Sparse/absent scalp hair -generalised

Absent/smallnails

Frontal bossing Arm q

Round face

Hypertelorism

hyperconvex/ hypoplastic nails

Dysplastic/ grooved/thick/ discoloured nails

Depressed

premaxillary

region

Midface

hypoplasia/flat/

short midface

Micrognathia/

agnathia/

retrognathia

Absent/decreased

eyebrows/lateral

thinning

Down-slanting palpebralfissures

Other orbital abnormality

Paresis of ocular muscles/squint

Other eye

movement

disorder

Broad/bulbous

nasaltip

Cleft soft palate/ bifid uvula/ submucous cleft Microdontia

Abnormal tooth position/

malocclusion/open bite

Normal height (with skeletal dysplasia)

Short stature -postnatal

Decreased body hair/hypotrichosis

Decreased hair pigmentation -general

Decreased hair pigmentation -patchy

Fine hair

Brittle hair/ trichorrhexis nodosa/pili torti

Sparse/absent scalp hair -generalised High hairline -front

hyperconvex/ hypoplastic nails

Other dental abnormality

Abnormalclinicalfeatures of the limbs

Brachydactyly

Irregularities of length/shape of fingers

Syndactyly of fingers

Short foot (including brachydactyly)

Syndactyly (other than minimal2nd and 3rd toes)

Irregular length or shape of toes

Other skullabnormality

Absent/small/hypoplastic carpals

Symphalangism

Prominent Low set ears Simian creases

supraorbitalridges

Depressed Scoliosis Dislocated hip

premaxillary

region

Midface Bowed limbs

hypoplasia/flat/

short midface

Prognathism Mentalretardation of

any degree

Depressed nasalBoney sclerosis of any bridge type

Paramedian/lateralMultiple fractures/ cleft lip (uni/ increased boney fragility

bilateral)

Cleft soft palate/ Enchondroma/ bifid uvula/ radiolucencies -

submucous cleft localized

High vaulted and Lytic/lucent lesions of narrow palate bone

Microdontia Fibrous dysplasia of

DevelopmentalWide diaphyses defect of enamel(undertubulation)

Tooth Submetaphyseal

discolouration undermodelling/ expansion

Delayed eruption Thin cortex of diaphyses of teeth

Missing Bowing of long bones

permanent teeth/

retained

deciduous teeth

Supernumerary Cartilage tongues of teeth metaphyses - localized

Dentalcysts/ Hyperostosis/thickened/

tumours sclerotic calvarium

Absent/decreased Supernumerary lashes teeth

Everted/ Cleft/notched

protruding lips tongue

Dysplastic/ Cone shaped epiphyses

grooved/thick/

discoloured nails

Broad/bifid nails Symphalangism

Tooth crown shape

abnormality Microdontia

Abnormaltooth position/ malocclusion/ open bite

Delayed eruption of teeth

Anodontia/ oligodontia

Supernumerary teeth

Hamartoma/other tumours of the mouth

Other abnormality of tongue/ gingivae/mucosa Low set ears

Frontal bossing

High forehead

Facies

significantly

abnormal

Abnormally placed nipples

Thoracolumbar generalkyphosis Irregularities of length/shape of fingers

Syndactyly of fingers

Cone-shaped epiphyses of middle phalanges

Tragus abnormalLong face

Pectus excavatum Grooved/dimpled (funnel chest) chin

Micrognathia/

agnathia/

retrognathia

Medialflare of

eyebrows

Absent/decreased

eyebrows/lateral

thinning

Absent/decreased lashes

Polydactyly - Long/large nose postaxial (ulnar)/ type unspecified

Abnormal palmar Broad nasal

dermatoglyphics/ bridge (see

skin creases telecanthus)

Polydactyly of feet High nasalbridge

- postaxial/type

unspecified

Syndactyly (other Broad/bulbous

than minimal2nd nasaltip and 3rd toes)

Deafness -conductive

Other hearing abnormality

Narrow/sloping shoulder/ hypermobile shoulders

Pectus excavatum (funnelchest)

Absent/abnormal sinuses

Wormian bones

Sclerotic/hyperostotic facialbones

Other skullabnormality

Cranialnerve/ Hypoplastic/small

nuclei nostrils

Mental retardation Abnormal - moderate/severe columella

Bell-shaped chest Hyperostotic/wide clavicle

Thoracolumbar Abnormalrib structure generalkyphosis including fusion

Gibbus/localised Widened ribs kyphosis

Scoliosis Irregular shape of pubic

and ischialbones

Hyperextensible/ Absent/hypoplastic/ hypermobile joints short femur

Hypotonia

Movement disorder -dystonia/chorea/ tremor/spasm EEG abnormality

Short sternum

Smallhand

Brachydactyly

Seizures of any type

Hypotonia

Imperforate anus/ anal stenosis

Horseshoe/fused/ ectopic kidneys

Hypospadias/ epispadias

Undescended/ ectopic testes

Wilms tumour

Femora short/ deformed/bowed

Other abnormal femur Bow legs - genu varum

Thin lips

Long philtrum

Deeply grooved philtrum

Microdontia

Abnormaltooth position/ malocclusion/ open bite

Supernumerary teeth

Anteverted/

prominent/bat

Long/large ear

Pectus carinatum (pigeon chest)

Thoracolumbar generalkyphosis

Scoliosis

Hyperextensible/

hypermobile

joints

Small hand

Brachydactyly

Clinodactyly of 5th finger

Terminal

hypoplasia fingers

Delayed skeletal maturation

Poorly ossified calvarium/Soft skull

Absent/abnormal sinuses

Wormian bones

Platybasia/basilar impression

Enlarged foramer magnum

Small/absent scapula Winged/other abnormal scapula (See Shoulder)

Absent/

hypoplastic

clavicles

Pseudarthrosis of clavicle

Short ribs (circumferential)

Under-/unossified sternum

Hypoplastic/ absent ribs

Dorsal wedging of vertebral bodies

Narrow/trapezoid iliac wings (lack of flare)

Horizontal/flat acetabular roof

Spindle shaped/ tapered fingers

Ulnar deviation of fingers

Other hand abnormality Mental

retardation of any degree

Abnormal

cardiovascular

structure/function

Winged/other abnormal scapula (See Shoulder)

Coxa vara

Cone shaped epiphyses

Smallfemoral head epiphyses

Flat femoralhead epiphyses

Deformed/ irregular femoral head epiphyses

Broad femoral neck

Cone-shaped epiphyses of proximal phalanges

Some phalanges short and deformed

Cone-shaped epiphyses of middle phalanges

Cone-shaped epiphyses of distal phalanges

Delayed ossification of pubic and ischial bones

Open pubic symphysis ir adults

Coxa valga

Coxa vara

Dislocated hip

Core shaped epiphyses

Fibulae a-/

hypoplastic/

under-/unossified

Core-shaped epiphyses of proximal phalanges

Core-shaped epiphyses of middle phalanges

Allmiddle phalanges short/ deformed

Cone-shaped epiphyses of distal phalanges

All distal

phalanges short/ deformed

difficult delivery, it was insignificant and neither micro-dontia 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 supernum-ery 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.

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' contributions

SB and SK drafted the manuscript paper, analysed the patient's history and contributed to the writing of the finalversion as wellas extracted the mesiodens. Each author reviewed the paper for content and contributed to the writing of the manuscript. Allauthors approved the finalreport.

Competing interests

The authors declare that they have no competing interests.

Received: 25 May 2011 Accepted: 28 October 2011 Published: 28 October 2011

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

Cite this article as: Bargale and Kiran: Non-syndromic occurrence of true generalized microdontia with mandibular mesiodens - a rare case. Head & Face Medicine 2011 7:19.

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