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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 8  |  Issue : 3  |  Page : 203-207

An unusual case of molar incisor hypomineralization with a rare combination of fusion, hypodontia, and ankyloglossia in 11-year-old male


Department of Pedodontics and Preventive Dentistry, Dr. R Ahmed Dental College and Hospital, Kolkata, West Bengal, India

Date of Submission19-May-2021
Date of Decision18-Jun-2021
Date of Acceptance20-Jun-2021
Date of Web Publication23-Aug-2021

Correspondence Address:
Supreet Shirolkar
Department of Pedodontics and Preventive Dentistry, Dr. R. Ahmed Dental College and Hospital, Kolkata-700014, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdrr.jdrr_91_21

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  Abstract 


Molar-incisor hypomineralization (MIH) is a developmentally acquired dental defect that directly affects the enamel and dentin of one to four first molars, frequently associated with affected incisors. Due to alteration in matrix production, crystal arrangement and formation, enamel matrix secretion a compromised enamel structure may be seen. This condition can also be called a idiopathic enamel hypomineralization. In the article, we report a case of 11-year-old male presenting with typical features of MIH affecting all four permanent molars in combination with fusion of mandibular right permanent central and lateral incisor, hypodontia of mandibular left central incisor and ankyloglossia, and a possible management of the clinical condition.

Keywords: Amelogenesis, fusion, hypodontia, Kotlow's, stainless steel crown, tongue-tie


How to cite this article:
Gayen K, Shirolkar S, Biswas R, Chatterjee AN, Sarkar S. An unusual case of molar incisor hypomineralization with a rare combination of fusion, hypodontia, and ankyloglossia in 11-year-old male. J Dent Res Rev 2021;8:203-7

How to cite this URL:
Gayen K, Shirolkar S, Biswas R, Chatterjee AN, Sarkar S. An unusual case of molar incisor hypomineralization with a rare combination of fusion, hypodontia, and ankyloglossia in 11-year-old male. J Dent Res Rev [serial online] 2021 [cited 2021 Nov 27];8:203-7. Available from: https://www.jdrr.org/text.asp?2021/8/3/203/324423




  Introduction Top


Although the prevalence of caries has been greatly declined over the past decades in both primary and permanent teeth, a major concern remains for the practicing dentists where first permanent molars (FPMs) get affected soon after their eruption in the oral cavity due to some developmental defects.[1] The term molar-incisor hypomineralization (MIH) was suggested to this painful condition by Weerheijm et al. and was defined as “hypomineralization of systemic origin of one to four permanent first molars frequently associated with affected incisors.”[2] Further conjecture was raised that if the permanent cuspids get mineralized at the same period as FPMs and permanent incisors, the tip of the permanent cuspids might show similar defects.[2] The clinical representation of MIH is yellow or brownish opacities and usually, there is loss of tooth structure under masticatory forces due to the enamel being soft, porous, and brittle in nature.[3] Due to this, the teeth become highly susceptible to caries development at an earlier age. In some cases, there is an unique representation where MIH is represented with other systemic diseases or other dental anomalies also.

Ankyloglossia or tongue-tie is a congenital anomaly which can be clinically represented with short lingual frenum and limited tongue movement known as partial ankyloglossia or there can be complete ankyloglossia where the ventral surface of the tongue gets fused with the floor of the mouth and it is rare.[4] The presentation of tongue-tie can be with or without any syndrome, the incidence of which is 10%.[5] Hypodontia is a condition referring absence of one or more teeth and if it is seen in the anterior region it can seriously affect a child both physically and mentally.[6]

Fusion is a developmental anomaly of the tooth where two originally separate tooth buds unite before the crown is yet to be mineralized forming a single large tooth.[7] Fusion and gemination are two different developmental anomalies with almost similar clinical presentation of “double teeth” and that is why often makes it difficult in differentiation. Gemination can be explained as an attempt to form a supernumerary tooth.

Most human congenital malformations and dental defects are caused by mutations in developmental regulatory genes.[8]

This case report describes an atypical representation of MIH along with the presence of ankyloglossia, hypodontia, and fusion in mandibular anterior permanent teeth.


  Case Report Top


A 11-year-old male patient visited to the Department of Pedodontics and Preventive dentistry with the complaint of multiple broken teeth in the posterior region of both the jaws. Patient also claimed of having sensitivity in the lower posterior teeth of both sides while eating. Mother of the child gave a history of premature birth and child being affected with recurrent fevers, cold, and cough at the early ages of life which can be contributing factors. However, no documentation was present with the parents about those illnesses like which medications he used to take.

On clinical evaluation which was carried out on clean wet teeth, extensive enamel breakdown was seen in all four FPMs and also irregular brown opacities was there in all of them. All the FPMs were found to be grossly affected with caries. Demarcated opacities were seen in all maxillary and mandibular permanent incisors ranging from white-creamy to yellow-brown defects which had distinct borders with the adjacent sound enamel along with maxillary and mandibular permanent canine which showed some linear defects [Figure 1]. As patient was having full permanent dentition, evaluation of the deciduous dentition could not be done but as per the history given by his mother, the milk teeth were also affected in similar pattern in both anterior and posterior region.
Figure 1: Preoperative intraoral photographs

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Along with this, on intraoral examination presence of ankyloglossia was also found causing limited tongue movement [Figure 2]. According to Kotlow's assessment, this ankyloglossia was classified as class II [Table 1]. The appearance of the tongue when it was lifted was “v” shaped [Figure 3]. However, it was not affecting the normal function such as speech of the child.
Figure 2: Ankyloglossia or tongue-tie

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Figure 3: Picture showing limited tongue movement due to ankyloglossia

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Table 1: Kotlow's classification

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On further evaluation a fused tooth was found in the mandibular anterior region which was a fusion of mandibular right permanent central and lateral incisor and mandibular left central incisor was also missing in the lower arch which denotes the count of whole teeth presence in lower arch was <2 from normal which signifies the presence of hypodontia [Figure 4].
Figure 4: Orthopantomograph showing presence of fused tooth

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No other medical issues were found or told by the parents. As per the parents history, the child was a prematured delivered baby. No other family members were affected with such kind of abnormalities as dictated by the parents. Marriage between the parents was also not consanguineous.

In the present case, parents of the patient had not sought any consultation for their child's tongue-tie because they felt that no treatment was essential as his daily activities were not affected and after addressing them about the problems related to this, they were not ready for any surgical intervention at this phase of time. As their parent's concern was only about the sensitivity and pain in all four FPM teeth so no intervention was done to tongue-tie.

Esthetic restoration was done in all permanent anterior teeth [Figure 5]. Caries removal was done in all permanent first molar teeth and after doing the needful procedures they were provided full coverage restoration with preformed stainless steel crowns [Figure 6].
Figure 5: Postoperative intraoral photograph

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Figure 6: Full coverage restoration with preformed stainless steel crowns

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  Discussion Top


Although at present time the etiology of MIH still remains unclear, the various speculations that are made in the literature about the etiology behind MIH are environmental conditions, prenatal, perinatal, and postnatal complications such as maternal diseases such as viral infections, hypertension, diabetes, and medication, oxygen starvation of the child during birth combined with a low birth weight, frequent childhood diseases (respiratory infection, asthma, bronchitis, and otitis), or the use of antibiotics, calcium, and phosphate metabolic disorders.[9],[10],[11] During amelogenesis (enamel formation) if any disturbance occurs during the later stage of mineralization or maturation qualitative defect of enamel occurs known as hypomineralization. MIH denotes such a qualitative defect where on clinical presentation there is a clear demarcation present between sound and affected enamel.[3],[12] In 2013, Fagrell et al. estimated that the mineralization of teeth gets disturbed at around the first 6–7 months of age.[13] Permanent first molars and incisors usually get affected because their mineralization times usually overlap. In this present case, canines were also found to be affected. Previously in a study, 25.7% of MIH-affected individuals of age 14 years showed MIH-like defects in canines.[13] Another study found 22.8% of MIH-affected individuals of age 16 years had MIH such as defects in canines.[14],[15] In this case, the history of premature birth of the child was only present which possibly can be the causative factor.

Management of MIH cases can be highly problematic due to teeth being highly sensitive and highly susceptible to caries development. Successful management of MIH-affected children can be difficult due to less cooperation from young children, difficulty in achieving anaesthesia, and frequent marginal breakdown of restorations. In the present case, all first molars were restored with full coverage restoration and composite restorations were done in all incisors.[6]

Fusion is one of those anomalies or anatomic variations of the tooth which causes major difficulties for diagnosis and treatment. The etiology behind this anomaly is still unclear but some contributing factors which were reported are some physical force or pressure/trauma and also genetic predisposition also considered.[7] In this case, the fusion between the two tooth buds occurred at both crown and root level (enamel and dentin) generating a single tooth during odontogenesis. Due to such malformation, aesthetic and also maintenance of oral hygiene get compromised. There are various treatment protocols for fused teeth as illustrated by numerous case reports such as intraoral or extraoral hemisection of the tooth with intentional dental replantation.[16] In this present case, patient was not willing to get any sorts of treatment so no such intervention was done and only esthetic modification was done for the hypoplastic teeth.

Ankyloglossia is a Greek word that is derived from two words, agkylos (tied) and glossa (tongue).[17] Tongue movements are associated with normal orofacial functions such as sucking, chewing, swallowing, speech and articulation which can get affected due to limited tongue movement in ankyloglossia. There has been an association of aberrations in T-bx22 genes with abnormality in the development of the palate (cleft palate) and tongue (ankyloglossia).[18] It can be associated with syndromes such as Optiz syndrome, Beckwitz-Wiedemann syndrome, Orofacial-digital syndrome, cleft palate or can have nonsyndromic presentation. The presence of tongue-tie may interfere with speech articulation and less sweeping action of the tongue may result in caries of teeth so early surgical intervention is indicated. The available surgical interventions are frenotomy, frenectomy, revision by electrocautery or monopolar diathermy, and/or revision of the frenum by laser.[19]

Hypodontia or the absence of one or few more teeth usually require various treatment approaches such as orthodontic, combined orthodontic prosthetic, and implant-related prosthesis. However in this case, as there was no spacing due to the absence of one tooth so no requirement of any intervention was there.

This case presentation is unique with so many dental abnormalities such as MIH, fusion and absence of tooth along with tongue-tie. Nonsyndromic representation with so many abnormalities is very rare to be found.


  Conclusion Top


To the best of our knowledge, the present case report is the first case that acknowledges MIH to be presented with all the other dental abnormalities - fusion, hypodontia and along with ankyloglossia. It also elicits the various contemporary treatment options available to all the abnormalities - hypoplastic enamel defects of MIH, fused teeth, tongue–tie, and proper selection of the treatment plan so that early intervention can be done to manage the aesthetic, functional problems and in result to improve the quality of life of a patient.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Zameer M, Peeran SA, Basheer SN, Peeran SW, Birajdar SB, Alzahrani FM, et al. Molar incisor hypomineralization (MIH) in a child with congenital chronic intestinal pseudoobstruction (CIPO). Case Rep Dent 2020;2020:8894657.  Back to cited text no. 1
    
2.
Weerheijm KL, Jälevik B, Alaluusua S. Molar-incisor hypomineralisation. Caries Res 2001;35:390-1.  Back to cited text no. 2
    
3.
Weerheijm KL. Molar incisor hypomineralization (MIH): Clinical presentation, aetiology and management. Dent Update 2004;31:9-12.  Back to cited text no. 3
    
4.
Horton CE, Crawford HH, Adamson JE, Ashbell TS. Tongue-tie. Cleft palate J 1969;6:8-23.  Back to cited text no. 4
    
5.
Segal LM, Stephenson R, Dawes M, Feldman P. Prevalence, diagnosis, and treatment of ankyloglossia: Methodologic review. Can Fam Physician 2007;53:1027-33.  Back to cited text no. 5
    
6.
Valle AL, Lorenzoni FC, Martins LM, Valle CV, Henriques JF, Almeida AL, et al. A multidisciplinary approach for the management of hypodontia: Case report. J Appl Oral Sci 2011;19:544-8.  Back to cited text no. 6
    
7.
Ozden B, Gunduz K, Ozer S, Oz A, Otan Ozden F. The multidisciplinary management of a fused maxillary central incisor with a talon cusp. Aust Dent J 2012;57:98-102.  Back to cited text no. 7
    
8.
Thesleff I. Genetic basis of tooth development and dental defects. Acta Odontol Scand 2000;58:191-4.  Back to cited text no. 8
    
9.
Koch G, Hallonsten AL, Ludvigsson N, Hansson BO, Holst A, Ullbro C. Epidemiologic study of idiopathic enamel hypomineralization in permanent teeth of Swedish children. Community Dent Oral Epidemiol 1987;15:279-85.  Back to cited text no. 9
    
10.
Johnson D, Kreji C, Hack M, Fanaroff A. Distribution of enamel defects and the association with respiratory distress in very low birth weight infants. J Dent Res 1984;63:59-64.  Back to cited text no. 10
    
11.
Beentjes VE, Weerheijm KL, Groen HJ. Factors involved in the aetiology of molar-incisor hypomineralisation (MIH). Eur J Paediatr Dent 2002;3:9-13.  Back to cited text no. 11
    
12.
Allazzam SM, Alaki SM, El Meligy OA. Molar incisor hypomineralization, prevalence, and etiology. Int J Dent 2014;2014:234508.  Back to cited text no. 12
    
13.
Fagrell TG, Salmon P, Melin L, Norén JG. Onset of molar incisor hypomineralization (MIH). Swed Dent J 2013;37:61-70.  Back to cited text no. 13
    
14.
Kevrekidou A, Kosma I, Kotsanos I, Arapostathis KN, Kotsanos N. Enamel opacities in all other than molar incisor hypomineralisation index teeth of adolescents. Int J Paediatr Dent 2021;31:270-7.  Back to cited text no. 14
    
15.
Schmalfuss A, Stenhagen KR, Tveit AB, Crossner CG, Espelid I. Canines are affected in 16-year-olds with molar-incisor hypomineralisation (MIH): An epidemiological study based on the Tromsø study: “Fit Futures”. Eur Arch Paediatr Dent 2016;17:107-13.  Back to cited text no. 15
    
16.
Olivan-Rosas G, López-Jiménez J, Giménez-Prats MJ, Piqueras-Hernández M. Considerations and differences in the treatment of a fused tooth. Med Oral 2004;9:224-8.  Back to cited text no. 16
    
17.
Kaushik M, Masih U, Rathore K, Mahay P. A rare case report of nonsyndromic hypodontia with ankyloglossia. Int J Adv Res (Indore) 2019;7:292-7.  Back to cited text no. 17
    
18.
Chandrashekar L, Kashinath KR, Suhas S. Labial ankyloglossia associated with oligodontia: A case report. J Dent (Tehran) 2014;11:481-4.  Back to cited text no. 18
    
19.
Klockars T. Familial ankyloglossia (tongue-tie). Int J Pediatr Otorhinolaryngol 2007;71:1321-4.  Back to cited text no. 19
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
 
 
    Tables

  [Table 1]



 

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