Get Permission Ghoshal and Singh: A rare case report of natal tooth, neonatal tooth (dentitia praecox) and fibrous hyperplasia (Riga fede disease) of same newborn patient


Introduction

Natal tooth of a child is defined as a tooth that is already present at birth, which differs from Neonatal tooth which usually used to emerge in during the first 30 days after birth. Natal teeth are more frequent than neonatal teeth with an approximate ratio of 3:1. Neonatal and natal teeth are considered as an anomaly which has a fundamental importance from the point of view of a dental surgeon as well a pediatrician as patients with dentitia praecox face numerous problems like pain in suckling breast milk and refusal to feed which may lead to nutritional deficit, dehydration and growth retardation. Sometimes from continuous mechanical friction from natal or neonatal teeth there is a deformity or mutilation of tongue is observed.1 Riga Fede disease (RFD) is an uncommon benign mucosal ulceration usually takes place on the ventral surface of tongue because of the repetitive trauma due to forward and backward motion of the tongue over the edges of mandibular anterior incisors. There may be complete cessation of suckling reflex due to the severe painful condition.2 Various terms are there to describe the lesion, like Riga’s disease, Riga-Fede’s disease, sublingual ulcer, sublingual granuloma, traumatic sublingual ulceration, eosinophilic granuloma, traumatic eosinophilic ulceration of the tongue and oral mucosa, sublingual fibrogranuloma, sublingual growth in infants and traumatic atrophic glossitis.3

Clinical Case Presentation

21 days old male child reported to the clinic with the chief complaint of a large ulcerative lesion on the ventral surface of the tongue which was causing pain and bleeding. The baby was crying during breast feeding and refusing to take milk or water since 48 hours. His mother gave the history of Natal tooth (present at the time of birth) and eruption of neonatal teeth (on the 16th day of birth). The consultant pediatrician advocated extracting the teeth under the supervision of pedodontist but out of apprehension and as there was no problem arrived at that time she escaped the essential procedure. Since 3 days she observed a big whitish ulcerative lesion in the ventral surface of the tongue of her baby with complete denial of food.

On clinical examination, two teeth like structures in the mandibular anterior region corresponding to 71 (natal tooth) and 81(partially erupted neonatal tooth) were detected. No mobility was observed. There was a white granular necrotic plaque measuring about 8 mm X 5 mm which elicited pain on palpation. It was extended from the anterior border of the tongue to the lingual frenum. Base on clinical finding findings and history the child was diagnosed with RFD. The family history is negative for developmental disorders and congenital syndromes.

Figure 1

Preoperative natal and neonatal (partially erupted)

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Figure 2

Preoperative riga fede disesase

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Treatment

Since the lesion was extremely painful and complete denial of food was really serious as it would lead to dehydration and nutritional deficiency which might require hospitalization of the infant. So extraction of the teeth was the ultimate choice to avoid unfavorable situations.

With parental consent, the both teeth were extracted with pediatric tooth forceps followed by the gentle curettage of sockets were carried out after proper application of surface anesthetics and labial infiltration of 2% Lignocaine hydrochloride solution under the medical supervision of consultant pediatrician and pediatric nurse.

Figure 3

Extracted natal tooth (left) and neonatal tooth (right)

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The extracted teeth showed a normal morphology, hypomineralised, 3.5x 7 mm (left natal tooth), 2.5x4.5 mm (right neonatal tooth) were in dimension and whitish opaque coronal structure and presence of root formation. The child was prescribed metronidzole suspension 7.5 Ml/kg wt every 6 hrs for 7 days and topical solutions like sucralfate, amlexanox were advocated with topical anesthetic gel (Benzocaine 20% w/w) for symptomatic relief. The suckling reflex of the child was replenished within 5 days and after 2 weeks follow up there was complete cricatrization of RFD.

Figure 4

Post operative- Complete remission of RFD

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Figure 5

4 months after follow up

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Discussion

It is found that there is an inherited tendency to developing natal teeth with up to 60% of cases reporting a positive family history with an autosomal dominant pattern (meaning about half the children of an affected individual are affected). There are some syndromes where natal/neonatal tooth is thought to be a recognized feature.4

Figure 6

Some of the syndromes relevant to natal/neonatal tooth5

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Management of natal/neonatal teeth is decided by a lot factors including tooth prognosis, development of root, chance of aspiration, degree of mobility, interference in breastfeeding, difficulty in suckling reflex, risk of hemorrhage which should be taken into consideration.

Figure 7

Hebling Classification of Natal/ Neonatal tooth (1997)6

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When the conservative measures fail to resolve the lesion and when the child is dehydrated or malnourished, natal/neonatal impedes in breastfeeding, mobility of tooth imposes the risk of aspiration, extraction of those teeth is necessary.7 The extraction should be delayed up to 10 days or more until and unless the optimum level of viamint K is not present in newborn blood. During the period normal commensal flora is established in intestine which is mandatory to produce 2, 7,9,10 in liver. If the procedure is essential before the period, the need of vitamin K should be consulted with pediatrician if the newborn is medicated with Vitamin injection at the time of birth. Vitamin K (0.5-1mg) is generally administered intramuscularly after birth to prevent the chances of developing hemorrhagic diseases.8 Once the extraction is carried out, dental papilla cells with Hertwigs epithelium root sheath (HERS) should be enucleated with curette to remove any chance to development of root from the tissues if left in the socket.

RFD starts as an ulcerated lesion with prominent raised borders. With repetitive traumata from irregular edges of newly erupted teeth the lesion leads to an enlarged fibrotic mass which appears like an ulcerative granuloma accompanied with superficial necrosis. Histopathologically, the lesion exhibits granulation tissue with inflammatory infiltration of mast cells, macrophge lymphocytes and eosinophils.9

RFD is generally classified as ‘Early’ and ‘Late’ lesions (before 6 months of age) depending on the time of occurrence. Early RFD is relevant to neonatal or natal tooth which is characterized by hypoplastic enamel, partially developed root and resultant early mobility. Late lesions are related to primary dentition which may be associated with neurological and developmental disorders like familial dysautonomia, Down syndrome, microcephaly, congenial autonomic dysfunction and cerebral palsy.10, 11

In our case report, the child presented with both natal and neonatal tooth resulted in RFD. No family history of neurological or developmental anomalies were related. The natal teeth exhibited 1 degree mobility where as partially erupted neonatal tooth showed no mobility. Because of the presence of RFD, patient’s refusal to breast milk and denial of feeding was the major concern where the extraction under neonatal care was the only option left. But after 2 weeks follow up the lesion resolved and at 4 months follow up, the gum pad was found completely healthy and imprint of erupting lower central incisors.

Conclusion

The definite etiology behind the natal or neonatal teeth is still unknown. The conservative approach like smoothening of the edges or rounding of the sharp edges by composite increments are preferred in those situations where the baby is asymptomatic because invasive procedure of newborns is always challenging to the dentist and extraction may have some disadvantages like dehydration, loss of space, arch collapse delayed eruption of deciduous tooth. But when the baby is unable to breastfeed or devoid of any nutrition because of RFD, or when the teeth are poorly implanted which poses a risk of aspiration, extraction is the only way out to avoid further complication. So, early diagnosis is mandatory for better prognosis that allows the early resolution of ulcerative lesion and restore the feeding of the newborn.

Source of Funding

None.

Conflict of Interest

None.

References

1 

BK Joseph DB Sundaram Oral traumatic granuloma: report of a case and review of literatureDent Traumatol2010261947

2 

SC Choi JH Park YC Choi GT Kim Sublingual traumatic ulceration (a Riga-Fede disease): report of two casesDent Traumatol2009254850

3 

A Guzman G Mendoza Dientes natales y enfermedad de Riga-FedeDermatol Pediatr Lat2005321527

4 

EHVD Meij TW DeVries HF Eggink JG DeVisscher Traumatic lingual ulceration in a newborn: Riga-Fede diseaseItal J Pediatr20123820

5 

JD Farsi MM Ahmed Natal and neonatal teethSaudi Med J201435499503

6 

J Hebling ACC Zuanon DR Vianna Dente Natal- A case of natal teethOdontol Clin197573740

7 

RL Slayton Treatment alternatives for sublingual traumatic ulcerationPediatr Dent20002254134

8 

M Costacurta P Maturo R Docimo Riga-Fede disease and neonatal teethOral Implantol (Rome)2012512630

9 

V Khandelwal UA Nayak PA Nayak Y Bafna Management of an infant having natal teethBMJ Case Rep20132013bcr2013010049

10 

N Sharma S Chander S Soni S Singh MG Chodhary Riga-fede disease due to neonatal tooth: A case reportInt J Oral Maxillofac Pathol201232434

11 

PSP Kumar KS Dhull RS Dhull S Panda S Yadav MD Indira Riga Fede Syndrome: A review of literature and report of three casesInt J Oral Maxillofac Pathol201342404



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Article History

Received : 08-12-2023

Accepted : 29-12-2023


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Article DOI

https://doi.org/ 10.18231/j.idjsr.2023.037


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