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SHORT COMMUNICATION  
Year : 2014  |  Volume : 25  |  Issue : 6  |  Page : 832-834
Dentin dysplasia type 1d: A rare case


1 Department of Oral Pathology and Microbiology, Institute of Dental sciences and Hospital, SOA University Bhubaneswar, Odisha, India
2 Department of Conservative Dentistry and Endodontics, Institute of Dental sciences and Hospital, SOA University Bhubaneswar, Odisha, India

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Date of Submission10-May-2013
Date of Decision31-Mar-2014
Date of Acceptance14-Jan-2015
Date of Web Publication02-Mar-2015
 

   Abstract 

Dentin dysplasia is a rare hereditary disturbance of dentin formation characterized by a defective dentin development with clinically normal-appearing crowns, severe hypermobility of teeth and spontaneous dental abscesses or cysts. Radiographic analysis shows obliteration of all pulp chambers by pulp stones, short, blunted and malformed or absent roots, peri-apical radiolucencies of noncarious teeth. We present a case of dentin dysplasia type 1d in a 19-year-old boy along with the clinical, radiographic findings of this condition and treatment. There are still many inconclusive issues in the diagnosis and management of patients with dentin dysplasia. The diagnostic features of this rare disturbance will remain incompletely defined until additional cases have been described.

Keywords: Dentin dysplasia type 1d, hereditary, pulp stone

How to cite this article:
Sahoo SR, Aggarwal S. Dentin dysplasia type 1d: A rare case. Indian J Dent Res 2014;25:832-4

How to cite this URL:
Sahoo SR, Aggarwal S. Dentin dysplasia type 1d: A rare case. Indian J Dent Res [serial online] 2014 [cited 2019 Oct 23];25:832-4. Available from: http://www.ijdr.in/text.asp?2014/25/6/832/152217
Pulp stones are foci of calcification in the dental pulp. Etiological factors that have been implicated in the formation of pulp stones include pulp degeneration, inductive interactions between epithelium and pulp tissue, age, circulatory disturbances in pulp, orthodontic tooth movements, idiopathic factors, and genetic predisposition. [1]

Dentinal dysplasia (DD), so named by Rushton, is an autosomal dominant hereditary condition affecting dentin formation that is rarely encountered in dental practice. DD is divided into two basic types, that is type 1 and type 2. Type 1 is further subdivided into four subtypes; 1a, 1b, 1c and 1d. Type 1d is characterized by normal root formation, which sometimes may be bulbous in the coronal 3 rd and within the pulp canal "a stone" may be found. [2]

This case report presents a child with generalized pulp stones in the dentition, which is suggestive of a case of dentin dysplasia type 1d - a rare entity, which manifested itself as coronal bulging of roots of some teeth and shortened roots of maxillary premolars.


   Case report Top


A 19-year-old male patient reported to the Department of Conservative Dentistry and Endodontics with the chief complaint of broken upper front right tooth since 6 months [Figure 1]. He gave a history of trauma to the tooth due to fall from a bicycle. On clinical examination, Ellis Class II fracture was found in tooth 11. Two teeth 11 and 21 were tender on percussion. All other teeth were of normal size, morphology, and color. The extraoral examination showed no abnormalities. Pulp vitality test was negative for both teeth 11 and 21. Periapical radiograph was taken, which showed unusual finding that is pulp stones in all anterior teeth visible on that intraoral periapical (IOPA) film with bulging of the root corresponding to the pulp stones [Figure 2]. Above and below the pulp stones there was little constriction and remaining root was normal. IOPAs of posterior teeth showed the similar findings [Figure 3]. A panoramic radiograph was taken, revealing a dentition with multiple pulp stones, shortened roots of premolars and retained deciduous left second molar [Figure 4]. Medical history given was noncontributory to the diagnosis. To see the hereditary pattern panoramic radiograph of the patient's sibling was taken, which showed no characteristic features, but there was congenitally missing maxillary second premolar and retained 65 [Figure 5]. Root canal treatment of 11 and 21 was initiated. After gaining access to root canal, K-file number 6 was inserted into the canal. With the tactile sensation, it was found that pulp stones were free as they were moving with the file inside the canal. Due to bulging of the coronal third of the root canal corresponding to the pulp stones; removal of pulp stone was difficult and time-consuming. Coronal constriction was removed by cutting radicular dentin with the help of Gates-Glidden drill No. 2 and 3. Pulp stones were held with the file toward the orifice, which was then grabbed with twizzer and then removed. With the repetitive use of irrigating solutions, root canal treatment was done successfully [Figure 6].
Figure 1: Ellis class II fracture in 11

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Figure 2: Preoperative radiograph – pulp stones in 11, 21, 22 with bulging of roots in the cervical third of the root

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Figure 3: Intraoral periapicals of posterior teeth of the patient. (a) Maxillary right posterior teeth. (b) Maxillary left posterior teeth. (c) Mandibular right posterior teeth. (d) Mandibular left posterior teeth

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Figure 4: Orthopantomogram showed pulp stones in multiple teeth of the patient with shortened roots of maxillary premolars and retained 75

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Figure 5: Orthopantomogram of patient's sibling showed no characteristic features but there was congenitally missing maxillary second premolars and retained 65

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Figure 6: Postobturation radiographs

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   Discussion Top


Dentine dysplasia is an autosomal dominant trait that resembles opalescent dentin. It is a rare anomaly of unknown etiology that affects approximately one patient in every 100,000. [3]

In 1920, Balchsmiede first reported eight cases as "rootless teeth." Later, Rushton in 1939 described it as "DD," since disturbance of dentine development was recognized such as highly atypical dentine, sometimes with pulpal obliteration and defective root formations. Years later, Stafne and Gibilisco reported three cases with similar radiographic findings, but with no microscopic descriptions.

Two main types of DD have been described. Type 1, also called radicular dentin dysplasia, primarily affects the root portions of both primary and permanent teeth and produces teeth with shortened and periapical radiolucencies. [4] Type 2, also called coronal pulp dentin dysplasia, is characterized by large pulp chambers containing denticles in permanent teeth and total obliteration of the pulp chambers in primary teeth. [5] According to O Carroll et al. [2],[6] the permanent teeth in DD type 1 can have any one of the four fairly distinct radiographic appearances:

  • Complete obliteration of pulp chambers and no root development with many periapical radiolucent areas (DD1a)
  • Horizontal, crescent-shaped, radiolucent pulpal remnants and few millimeters of root development with many periapical radiolucent areas (DD1b)
  • Two horizontal, crescent-shaped, radiolucent lines and significant but incomplete root development, with or without periapical radiolucent areas (DD1c)
  • Visible pulp chambers and oval pulp stones in the coronal third of the canal, with bulging of the root around the stone, and few, if any, periapical radiolucent areas (DD1d).
In DD1d, the pulp chamber is usually not obliterated and normal root formation occurs. This is the least severe form of DD. In these cases, the pulp around the stones is healthy. In other cases, the denticle is continuous with dentinal walls.

In literature case reports exist where generalized pulp stones are found in the dentitions of individuals with various systemic or genetic disease conditions like tumoral calcinosis, DD type 2, dentinogenesis imperfecta, Saethre-Chotzen syndrome, elfin facies syndrome, familial expansile osteolysis, ehlers danlos syndrome type I, osteogenesis imperfecta type I, otodental syndrome, van der Woude syndrome, hypercalcemia, gout and renal lithiasis, coronary atherosclerosis. Unusual cases of idiopathic generalized pulp stone formation have also been reported. Other suggested correlations to pulp stones have been plethoric as opposed to anemic personalities, metabolic imbalance or dysfunction and orthodontic treatment and traumatic occlusion. [7]

In our case, findings did not support the association of systemic disorders. Hereditary pattern was also not established from the panoramic radiograph of patient's sibling. Findings in patients were same as that of dentin dysplasia type 1d, that is bulging of the coronal third of the roots with pulp stones, shortened roots of premolars, some retained deciduous teeth. It would have been better if panoramic radiographs of patient's parents were available, but because of some reasons we could not acquire them.

Until date, to our knowledge only one case of DD type 1d has been reported by Comer and Gound where the authors had established hereditary pattern of the same. [8]



 
   References Top

1.
Hill TJ. Pathology of the dental pulp. J Am Dent Assoc 1934;21:820-8.  Back to cited text no. 1
    
2.
O Carroll MK, Duncan WK, Perkins TM. Dentin dysplasia: Review of the literature and a proposed subclassification based on radiographic findings. Oral Surg Oral Med Oral Pathol 1991;72:119-25.  Back to cited text no. 2
    
3.
Kim JW, Simmer JP. Hereditary dentin defects. J Dent Res 2007;86:392-9.  Back to cited text no. 3
    
4.
Witkop CJ Jr. Hereditary defects of dentin. Dent Clin North Am 1975;19:25-45.  Back to cited text no. 4
    
5.
Shields ED, Bixler D, el-Kafrawy AM. A proposed classification for heritable human dentine defects with a description of a new entity. Arch Oral Biol 1973;18:543-53.  Back to cited text no. 5
    
6.
Neville BW, Damm DD, Allen CM, Bouquot JE, editors. Abnormalities of teeth. In: Oral and Maxillofacial Pathology. 2 nd ed. Philadelphia: W.B. Saunders Company; 2002. p. 96-9.  Back to cited text no. 6
    
7.
Nayak M, Kumar J, Prasad LK. A radiographic correlation between systemic disorders and pulp stones. Indian J Dent Res 2010;21:369-73.  Back to cited text no. 7
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8.
Comer TL, Gound TG. Hereditary pattern for dentinal dysplasia type Id: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:51-3.  Back to cited text no. 8
    

Top
Correspondence Address:
Sonia Aggarwal
Department of Conservative Dentistry and Endodontics, Institute of Dental sciences and Hospital, SOA University Bhubaneswar, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.152217

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    Figures

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



 

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