131J INDIAN SOC PEDOD PREVENT DENT | Apr - Jun 2009 | Issue 2 | Vol 27 |

CASE REPORT

Dentin dysplasia type I with pyogenic granuloma in a 12-year-old girl

Nirmala SVSG, Sivakumar N, Usha KDepartment of Pedodontics and Preventive Dentistry, Narayana Dental College and Hospital, Nellore, Andhra Pradesh-524002, India

Correspondence:Correspondence: Dr. N. Sivakumar, Department of Pedodontics and Preventive Dentistry, Narayana Dental College and Hospital, Nellore, Andhra Pradesh - 524 002, India. E-mail: dentist4kids@gmail.com

Abstract

This paper presents a case of dentin dysplasia, a rarely reported disorder that is also associated with pyogenic granuloma in a 12-year-old girl. The case presented as excessively mobile teeth that appeared radiographically as rootless teeth and also as a soft tissue lesion in the right maxillary anterior region.

Key words

Dentin dysplasia, rootless teeth, pulp obliteration, thistle tube teeth

DOI: 10.4103/0970-4388.55343

Introduction

Dentin dysplasia is a rare disturbance of dentin formation, characterized by normal enamel formation, but atypical dentin with abnormal pulpal morphology that is inherited as an autosomal dominant trait.[1] It is classified into type I (Radicular dysplasia) and type II (coronal dysplasia) which affects both the dentition.

Type I is characterized by the presence of primary and permanent teeth with normal appearance of crown, but no or rudimentary root development, incomplete or total obliteration of the pulp chamber and periapical radiolucent areas or cysts. Teeth exhibits extreme mobility and exfoliate prematurely because of short roots. Type II is characterized by primary teeth with complete pulpal obliteration and yellow, brown, or bluish grey opalescent appearance similar to that seen in hereditary opalescent dentin.[2] Permanent teeth have a normal appearance or a slight amber coloration; the roots are normal in size and shape with a ‘thistle-tube’-shaped pulp chamber and with pulp stones[3,4] and the absence of periapical radiolucencies.

The purpose of this case report is to present a case of

dentin dysplasia type I, which is also associated with pyogenic granuloma in a 12-year-old girl.

Case Report

A 12-year-old girl presented to the Department of Pedodontics and Preventive Dentistry, Narayana Dental College and Hospital, Nellore, Andhra Pradesh, India complaining mobile teeth and a swelling in the upper right front jaw since 2 years. The extra oral examination revealed that there were no other abnormalities [Figure 1]. The intraoral examination revealed a soft tissue lesion that was lobulated, pedunculated, ulcerated, soft to hard in consistency, and pale pinkish red in color present in relation to right permanent lateral incisor [Figure 2]. It was 1 x 1cm in size, situated at the facial aspect of gingiva and extending between central and lateral incisors which was painless and bleeding easily [Figure 3]. The patient had a history of the presence of lesion since 2 years that was started as a small nodule and gradually increased up to the present size. The teeth present in the oral cavity were

17 16 15 14 53 12 11 21 22 24 25 26 27

47 46 45 44 42 41 31 32 33 34 35 36 37

The patient was having class III malocclusion with spacing between maxillary and mandibular anteriors. There was grade I mobility of 17, 16, 12, 11, 21, 22, 26, 27, 31, 32, 33, 36, 37, 41, 42, 46, and 47 and grade

132 J INDIAN SOC PEDOD PREVENT DENT | Apr - Jun 2009 | Issue 2 | Vol 27 |

II mobility with 14, 15, 24, 25, 34, 35, 44, and 45. All the teeth were normal in shape and size. Under development of maxilla was observed. Oral hygiene was

very poor with great amount of plaque and calculus.

The panoramic radiograph revealed features of dentine

Figure 1: Facial profi le of the patient Figure 2: Frontal view of the oral cavity showing pyogenic granuloma in relation to the right maxillary lateral incisor

Figure 3: Mirror view of the maxilla showing the presence of pyogenic granuloma extending between right maxillary central and lateral incisors

Figure 4: The panoramic radiograph revealed the absence of roots in relation to 14, 15, 17, 24, 25, 27, 33, 34, 37, 44, 45, 47, partial root formation in relation to 11, 12, 21, 22, and two-third of root formation in relation to 16, 26, 36, 46, 31, 32, 41, 42

Nirmala et al.: Rootless teeth with Pyogenic granuloma

Figure 5: Post treatment photograph Figure 6: The microscopic picture of pyogenic granuloma

133J INDIAN SOC PEDOD PREVENT DENT | Apr - Jun 2009 | Issue 2 | Vol 27 |

dysplasia type I with normal appearance of crown but no root development in relation to 14, 15, 17, 24, 25, 27, 33, 34, 37, 44, 45, 47, slight root formation in relation to 11, 12, 21, 22, and two-third root formation in relation to 16, 26, 36, 46, 31, 32, 41, 42. Both the maxillary canines and mandibular right canine were horizontally impacted. Pulp chambers were obliterated and bone below the teeth showed well-defined margins, and periapical radiolucencies were not seen [Figure 4].

Family history revealed that there was no such abnormality among the other family members. Maternal history revealed no history of medication or illness during pregnancy and the patient was born through normal delivery. Medical history revealed that the patient was having dermatological lesions on her soles that were diagnosed as juvenile dermatitis and she was under medication. Blood and radiographic investigations were done and revealed that there was no systemic involvement. Depending upon clinical and radiographic examinations, the patient was diagnosed with dentin dysplasia type I. Based upon clinical examination, the soft tissue lesion was diagnosed provisionally as pyogenic granuloma. An excisional biopsy of the lesion was done under local anesthesia, and a biopsy was sent for histopathological investigation. Thorough oral prophylaxis with ultrasonic scalers and extraction of over-retained primary canine was done prior to the excisional biopsy. The patient was recalled after a week and was observed with an excellent improvement in oral hygiene. [Figure 5].

Microscopic examination showed fibro cellular, edematous connective tissue with intense chronic (plasma cells) inflammatory cell infiltration in large aggregates and many capillaries (some dilated and large), separated by fibro vascular connective tissue septae coursing in various directions. Ulcerated areas showed intense chronic inflammatory changes with many canalizing blood vessels with an overlying thick fibrinopurulent membrane with intense acute inflammatory cell infiltration suggestive of pyogenic granuloma [Figure 6].

Discussion

Dentine dysplasia is rarely seen in children, the occurrence is 1 in 100,000. The abnormal root morphology is postulated secondary to the abnormal differentiation or function of the ectomesenchymally derived odontoblast.[7] The first case was reported by

Ballschmiede in 1920 with spontaneous exfoliation of multiple teeth in seven children of one family and he called this phenomenon as rootless teeth.[5] Later in 1939, Rushton described it as dentin dysplasia.[6]

Recently, another case was reported by Depprich et al. in 2007, a 17-year-old girl with excessive mobility of teeth with periapical cysts for which extraction of all teeth and cystectomy were done followed by surgical implants.[2] Treatment of a child with dentin dysplasia requires a multidisciplinary approach and thorough knowledge of behavioral management of the pediatric patient. Treatment should be aimed at maintaining the teeth as long as possible. Oral rehabilitation of teeth in case of early exfoliation can be done.

Another clinical entity noted in this present case was diagnosed as pyogenic granuloma which was a response of tissues to minor trauma or to a non-specific infection which provides a pathway for the invasion of non-specific types of organisms. 75% of pyogenic granulomas shows predilection for gingiva. Gingival irritation and inflammation result from poor oral hygiene may be a precipitating factor. It can develop at any age but more common in children and young adults. Females are more commonly affected because of vascular effects of female hormones. Some of them resolve without any treatment or undergo fibrous maturation and resemble a fibroma. Treatment is conservative surgical excision. Occasionally, recurrence is seen and re-excission is necessary.

Conclusion

Treatment for children with dentin dysplasia type I mainly aims at preventive care. As a result of shortened roots, early loss from periodontitis is frequent. So, meticulous oral hygiene must be established and maintained for the retention of teeth to help children to have natural teeth as long as possible. Oral rehabilitation of prematurely exfoliated teeth is the treatment of choice in future.

References

1. Shafer W. G, Hine MK, Levy BM. Developmental Disturbances of Oral and Paraoral Structures: In A Text Book Of Oral Pathology; 4th Ed, Elsevier Science, Saunders Co: Philadelphia; 2003. p. 2-85.

2. Depprich RA, Ommerborn MA, Handschel JG, Naujoks CD, Meyer U, Kübler NR. Dentin dysplasia type I: A challenge for treatment with dental implants. Head Face Med 2007;3:31.

Nirmala et al.: Rootless teeth with Pyogenic granuloma

134 J INDIAN SOC PEDOD PREVENT DENT | Apr - Jun 2009 | Issue 2 | Vol 27 |

3. 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.

4. Ommerborn M, Raab W. Allgemeinerkrankungen und Schäden der Zahnhartsubstanzen. Prophylaxe Impuls 2005;9:66-73.

5. Steidler NE, Radden BG, Reade PC. Dentinal dysplasia: a clinicopathological study of eight cases and review of the literature. Br J Oral Maxillofac Surg 1984;22:274-84.

6. Rushton MA. A case of Dentinal dysplasia. Guys Hosp Rep 1939;89:369-73.

7. Melnick M, Levin LS, Brady J. Dentin dysplasia type I: a scanning electron microscopic analysis of the primary dentition. Oral Surg Oral Med Oral Pathol 1980;50:335-40.

Source of Support: Nil, Confl ict of Interest: Nil

Nirmala et al.: Rootless teeth with Pyogenic granuloma