Neonatal Peripheral Ossifying Fibroma 55 CASE REPORT Available At

CASE REPORT available at www.jstage.jst.go.jp/browse/islsm

940 nm Diode Laser assisted excision of Peripheral Ossifying Fibroma in a neonate.

Nitesh Tewari 1, Vijay Prakash Mathur 1, Asit Mridha 2, Kalpana Bansal 1, Divesh Sardana 1

1: Pedodontics & Preventive Dentistry, Centre for Dental Education and Research, All India Institute of Medical Sciences 2: Department of Pathology, All India Institute of Medical Sciences

Background: Peripheral ossifying fibroma associated with neonatal tooth extraction is a rare, benign reactive lesion, but its nature and location often scares the patient & parents for possibility of neoplasm. A high recurrence rate makes its histopathological examination and long term follow up important. Case Report: A 2 months old boy presented with enlarging soft tissue growth on the anterior mandibular ridge. The history revealed extraction of two neonatal teeth at 2 weeks of age. Lesion was excised using 940 nm diode laser and histopathological examination revealed hypercellularity and prominent dystrophic calcification, confirming it to be Peripheral Ossifying Fibroma. There was no recurrence after 18 months follow up. Conclusion: Paediatric dentists should be aware of possible outcomes of natal and neonatal teeth extraction and histopathological features of soft tissue lesions in neonates and infants. This report also highlights that 940 nm diode laser can be safely used for minor oral soft tissue surgeries in neonates and infants.

Key words: peripheral ossifying fibroma • neonatal teeth • laser excisional biopsy

Introduction sent a group of lesions with overlapping clinical and histologic features. 1, 2) Peripheral ossifying fibroma Human gingiva and periodontium is subjected to con- (POF) is a non neoplastic, reactive lesion seen exclu- stant irritation through materia alba, plaque, calculus, sively in gingiva. 2, 3) Clinically it appears as red to irregular tooth surfaces, faulty restorations, inadequate pink, solitary, smooth/lobulated, pedunculated or ses- removable or fixed prosthesis and trauma. Eversole sile, nodular mass of 0.2-3 cm in size, emerging from and Rovin (1972) 1) emphasized that similar etiologic interdental papilla. 1, 2) Most common site of occur- irritants lead to varied “Reactive lesions” of gingiva. In rence has been maxillary anterior region with a higher recent times, Buchner et al (2010)2 described Localised female susceptibility and Caucasian predilection. 2, 3) hyperplastic reactive lesions (LHRLs) of gingiva as- 1) Natal teeth are present since birth while neonatal focal fibrous hyperplasia (FFH), 2) pyogenic granulo- teeth erupt within first month. Their prevalence has ma (PG), 3) peripheral ossifying fibroma (POF) and 4) been recorded as 1:800 to 1:3000 in different popula- peripheral giant cell granuloma (PGCG). These repre- tions with natal teeth being three times more common than neo natal teeth. 4, 5) Severe infections secondary Addressee for Correspondence: to natal and neonatal teeth have also been described in Nitesh Tewari past. 5) Extraction is recommended in many cases Division of Pedodontics & Preventive Dentistry 6th Floor, Centre for Dental Education & Research though conservative means like smoothening of edges All India Institute of Medical Sciences can be attempted for treating the associated sub lingual New Delhi, India-110029 Email: [email protected] Received date: July 27th, 2016 Phone: +91-9839311058 Accepted date: September 27th, 2016 ©2017 JMLL, Tokyo, Japan Laser Therapy 26.1: 53-57 53 CASE REPORT available at www.jstage.jst.go.jp/browse/islsm traumatic ulcers. 4 ,5) LHRLs of gingiva in neonates and discomfort in breast feeding. A consultation with pae- within first 10 months of life are very rare with only 5 diatrician was sought and they were referred to a gen- cases reported till date (Table I). 6-10) POF associated eral dentist who extracted two mobile teeth (as with neonatal tooth has been even rarer with only one described by parents) under local anesthesia. Healing case in literature. 7) A case of peripheral ossifying was uneventful till mother got alarmed by presence of fibroma secondary to neonatal tooth extraction, treated a mass on lower jaw at six weeks of age. They consult- by Diode laser assisted excision, has been presented in ed the dentist and were advised that it will regress on this report. its own. However, the lesion kept growing and they were referred to this tertiary care centre. Case Report Clinical examination revealed a healthy male child with no extra oral swelling or deformity. Intra A two month old boy was referred to the outpatient oral examination showed a midline, pink, nodular, department from a paediatrician with a soft tissue pedunculated, non tender soft tissue growth of 2.5 cm enlargement on the anterior mandibular ridge that was x 1 cm x 1 cm with smooth intact surface, present on increasing in size. Parents reported uncomplicated the anterior mandibular alveolar mucosa (Figure 1). A antenatal history, normal vaginal delivery after full term clinical diagnosis of epulis was made and excisional and a birth weight of 3.5 Kilograms. He was the first biopsy under local anesthesia was planned. A diode born and only child of healthy parents with unremark- laser (Biolase, USA- 940 nm, power settings of 2 Watt, able post natal medical episodes. continuous mode and a tip of 300 microns diameter) Presence of neonatal teeth was discovered by assisted excisional biopsy was performed under topical parents in second week after birth when mother felt local anesthesia with child held in parent’s lap and

Table I: Showing the review of salient features of reported cases of LHRLs in neonates and infants. Author & Age/ Sex Region & Final History, Clinical Course & Treatment, Follow up Year Diagnosis Pre surgical clinical features & Recurrence Yip WK 7 days Maxillary Right - Present since birth Surgical excision at 7th & Yeow Female Posterior-Deciduous - No history of natal tooth day under local anes- CS 1973 first molar region - Soft pedunculated swelling 1.5 cm x 1 cm thesia. Peripheral Ossifying 5 months No recur- Fibroma rence Muench 6 days Mandibular Anterior - Natal tooth attached to a loose mass. Surgical excision at et al 1992 Male region. Pyogenic - Extraction of natal tooth < 6 days. 16th day under local Granuloma - Rapid enlargement after extraction. anesthesia. - 0.6 cm x 0.4 cm x 0.4 cm soft pedunculated mass. 1 month No recurrence

Kohli et al 2 hours Mandibular Anterior - 2 cm x 1.2 cm x 0.6 cm soft fluid pink fluctuant Surgical excision at the 1998 Female Region Peripheral mass since birth. age of 4 weeks under Ossifying Fibroma - Neonatal tooth at 2 weeks. local anesthesia. - Extraction at 2 weeks. 2 weeks No recurrence - Growth of mass (0.8 cm x 0.4 cm x 0.4 cm) over extraction site on 3rd week. Singh et al 4.5 Mandibular Anterior - Natal tooth Surgical excision with 2004 months Region Reactive - Soft growth covering the tooth at 2 months. natal tooth at 4.5 Male Fibrous Hyperplasia - Soft pedunculated 0.5 cm x 1.5 cm mass months under local anesthesia 1 month No recurrence Sethi et al 8 weeks Mandibular Anterior - Natal tooth - Growth around natal tooth at 6-7 Surgical excision with 2015 Female Region Reactive weeks which was increasing in size. Natal tooth at 8 weeks Fibrous Hyperplasia - Smooth, pink 0.5 cm x 1.3 cm pedunculated mass under local anesthesia. with embedded natal tooth. 1 month No recurrence

54 N Tewari et al CASE REPORT available at www.jstage.jst.go.jp/browse/islsm head supported by a trained nurse. (Figure 2a & b) histopathology studies from various parts of the world Excised tissue was sent for histopathological examina- have reported its prevalence rate to be 1-3 % among all tion as per the protocol. The procedure was complete- gingival biopsies and 16-40 % of studied LHRLs. 2, 3, 11) ly blood less and child was discharged without any medications, after basic infant oral health counselling. The histopathology revealed pseudo-epithelioma- tous hyperplasia of stratified squamous epithelium with parakeratinization. Sub epithelial connective tissue revealed a highly cellular mass of proliferating fibrob- lasts and collagenous stroma with prominent dystroph- ic calcifications. On the basis of distinctive histopatho- logical features, the lesion was diagnosed as peripheral ossifying fibroma. (Figure 3 a-d) The child was fol- lowed up after 24 hrs, 1 week, 1 month, 6 months and 18 months. The clinical findings exhibited healed ante- rior mandibular ridge and no recurrence. Most recent follow up (18 months) showed erupted primary maxil- lary central and lateral incisors and mandibular lateral incisors. (Figure 4 a-d)

Discussion Figure 3: (a&b) Haematoxylin and eosin stained sec- tions showing marked pseudoepitheliomatous Peripheral ossifying fibroma is benign reactive lesion but hyperplasia of stratified squamous epithelium its nature and location often scares the patient & parents with a calcified area in the subepithelial con- for possibility of a neoplasm. 11) Retrospective nective tissue (a, 40x; b, 40x). (c&d) The lesion comprised of hyalinised fibrocollagenous tissue with dystrophic calcifi- cation and was surrounded by a cellular mass of proliferating fibroblastic cells (c, 100x; d, 200x).

Figure 1: Showing pink, lobulated and sessile soft tis- sue growth of 2.5 cm x 1 cm x 1 cm present at the midline of anterior part of mandible.

Figure 4: (a) 1 week follow up showing healed anteri- or mandibular ridge. (b) 12 months follow up showing missing primary mandibular central incisors and erupting primary mandibular lateral incisors. (c,d) 18 months follow up showing erupted primary maxillary central and lateral incisors, Figure 2: (a) Diode laser assisted excisional biopsy mandibular lateral incisors, missing under topical local anaesthesia. mandibular central incisors and no recur- (b) Excised soft tissue lesion. rence of the lesion.

Neonatal peripheral ossifying fibroma 55 CASE REPORT available at www.jstage.jst.go.jp/browse/islsm

A peak of incidence is seen in second and third decade these lesions in future. of life with substantial fall after that. Prevalence in 0-10 Approximately 90% of natal and neonatal teeth years age group has been reported as 1-2% by most are prematurely erupted deciduous teeth, appearing as authors. 2) POF has a multitude of differential diagnoses delicate, mobile and shell like structures. 4) This was ranging from other LRHLs to hemangioma and traumatic also seen in present case with absence of primary fibroma. 2, 3) A high recurrence rate of 16-20% and mandibular central incisors at 20 months of age. Most ambiguous clinical picture necessitates a histopathologi- often they are situated on a mound of soft tissue and cal examination after their excision and a long term fol- associated with difficulty in breast feeding and other low up. 2, 3, 11) consequences as neonatal sublingual traumatic ulcer POFs are believed to be derived from irritation (NSTU) or riga-fede disease. 5) Their movement can induced hyper-reactivity of progenitor cells of peri- lead to irritation and microtrauma in gingival tissues, odontal ligament. 1) Immunohistochemical examina- periodontal ligament progenitors and periosteum tions have further strengthened this theory. 12, 13) resulting in LRHLs and POFs. 6, 7) Similar etiopathogen- Earlier belief that pyogenic granulomas mature into esis was reported by Singh et al (2004)8 and Sethi et al POF has not been fully negated, though their de novo (2015) 10). Pyogenic Granuloma after extraction of natal development on interdental papilla has gathered more tooth was reported by Muench et al (1992) 6) and POF evidence. 13) Even in cases, where they were reported with neonatal extraction was described by Kohli et al in edentulous areas (Trasad et al, 2011) 14) their deriva- (1998) 7). A case of congenital POF without any natal tion due to chronic irritation from affected teeth and or neonatal tooth was reported by Yip & Yeow (1973) possibility of extraction as causative factors have been 9). After reviewing the handful of reported cases of proposed. LHRLs in neonates and infants, we have presented Although smaller lesions are usually not associat- their salient features in Table I. The irritation and trau- ed with any ulceration, pain or displacement of adja- ma caused by extraction at 2nd week in the present cent teeth and alveolar bone, giant POFs (5-8 cms) 15) case could have attributed to development of POF in and multicentric POFs (multiple sites) 16) with adverse anterior mandible, though its simultaneous occurrence symptoms have been previously reported. and progression with neonatal tooth could not be Radiographic, tomographic and magnetic resonance ruled out on the basis of available records. imaging in larger lesions have also demonstrated null Reactive lesions of neonates and infants are scary to varying degree of erosion in alveolar bone and pres- for the parents and require surgical excision mostly ence of mineralized component in matrix. 17) under general anaesthesia, with in medical considera- The classical histopathological features include tions. 11) dense fibrocellular proliferation with random focal American Academy of Pediatric Dentistry adopted deposits of calcified material varying from ovoid-irreg- the policy on use of lasers in 2013 which recognized ular dystrophic/metaplastic calcification to laminated, “Lasers as an alternative and complementary method of concentric deposits resembling Liesegang ring. Another providing soft and hard tissue dental procedures for pattern of osseous lamellae and trabeculae with cir- infants, children, adolescents, and persons with special cumferential osteoid has also been observed. The health care needs”, with prime importance given on degree of mineralisation has been regarded as a part of Laser safety at all times. 30) Its benefits in soft tissue maturation of POF and its distinctive hypercellularity as procedures include precision, hemostasis, less thermal a histopathologic marker. 1, 2) necrosis than electrosurgery, rapid wound healing, no Immunohistochemical study of POF by Shumway suturing, reduced post-operative discomfort and lesser et al (2013) questioned the overlapping nature of need for analgesics. Most of the procedures can be LHRLs and proved POFs to be benign (low prolifera- done with topical local anesthesia leading to better tive index- Ki67) with myofibroblastic (reactivity to patient acceptance and reduced procedural time. vimentin & SMA) or fibroblastic (reactivity to Further, the bactericidal properties of lasers on tissues fibronectin ED-A) in nature. 12) Elanagai et al (2015) require less prescribing of antibiotics post-operatively. 19) explored the role of osteopontin in mineralisation of The choice of Diode laser in present case was LRHLs. 13) Proliferation index using nucleolar organis- based on its deeper penetration and proven safety in ing regions (AgNOR technique) and proliferating cellu- soft tissue surgeries. Surgical excision of a 2.5 cm x 1 cm lar nuclear antigen (PCNA) have been used to describe x 1 cm mass in a 2 month old child by conventional the nature of these lesions. 18) Similar applications of scalpel & blade would have been definitely more inva- technology will definitely help us in understanding sive requiring hemostasis, suturing and post-operative

56 N Tewari et al CASE REPORT available at www.jstage.jst.go.jp/browse/islsm medications. Excision of LRHLs using Nd:YAG, 810 nm Conclusion Diode and CO2 Lasers have also been successfully per- formed in past without recurrence. 20) The 940 nm POF and LRHLs of neonates are rare but possible out- diode laser is further associated with coefficient of come of extraction of natal and neonatal teeth requir- absorption in both water and haemoglobin with lesser ing a long term follow ups and histopathological depth of penetration as compared to 810 nm diode examination. The paediatric dentists also need to be laser. This property is significant in reducing the surgi- aware of classical features of these lesions and can cal time and effect on deeper layers, making it very safely use 940 nm diode laser in minor soft tissue effective for use in neonates and infants. surgery in neonates.

References

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