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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 4  |  Issue : 3  |  Page : 69-71

Riga–Fede disease in association with natal teeth


1 Department of Pediatric and Preventive Dentistry, AME's Dental College and Hospital, Raichur, Karnataka, India
2 Department of Pediatric and Preventive Dentistry, Faculty of Dental Sciences, M S Ramaiah University of Applied Sciences, Bengaluru, Karnataka, India

Date of Web Publication11-Jan-2018

Correspondence Address:
Sujatha Paranna
Department of Pediatric and Preventive Dentistry, AME's Dental College and Hospital, Benjanagera Road, Raichur - 584 102, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdrr.jdrr_62_17

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  Abstract 

The purpose of this paper is to report a case of a 16-day-old infant with two natal teeth in mandibular anterior region associated with ulceration on the tip and ventral surface of the tongue. Clinical presentation revealed two natal teeth with Grade II mobility in the mandibular anterior region and Riga–Fede Disease. The treatment planned was to extract both the natal teeth followed by curettage of the extraction socket to prevent aspiration, swallowing of teeth, and for early resolution of ulcerative lesion. Satisfactory healing of ulcerative lesion was observed at 10 days follow-up visit.

Keywords: Aspiration, feeding, natal teeth, Riga–Fede


How to cite this article:
Paranna S, Kamath P. Riga–Fede disease in association with natal teeth. J Dent Res Rev 2017;4:69-71

How to cite this URL:
Paranna S, Kamath P. Riga–Fede disease in association with natal teeth. J Dent Res Rev [serial online] 2017 [cited 2021 Dec 4];4:69-71. Available from: https://www.jdrr.org/text.asp?2017/4/3/69/223056


  Introduction Top


 Riga-Fede disease More Details which was first described by Antonio Riga in 1881 and subsequently with histological examinations by Francesco Fede in 1890, is a rare pediatric condition in which chronic lingual ulceration results from repetitive trauma.[1] The presence of teeth at birth may lead to the development of Riga-Fede Disease that may appear as traumatic ulcers which may be commonly located on the ventral surface of the tongue or lip and the mother's breast.[2]

Several terms have been used in the literature to designate teeth that erupt earlier than scheduled time, such as congenital teeth, fetal teeth, predeciduous teeth, and dentitia praecox.[3] According to the definition presented by Massler and Savara, taking only the time of eruption as reference, natal teeth are those observable in the oral cavity at birth and neonatal teeth are those that erupt during the first 30 days of life.[4]

The purpose of this article is to report a case of a 16 days old infant with two natal teeth in mandibular anterior region associated with Riga–Fede disease and its management.


  Case Report Top


A 16-day-old female infant accompanied by mother was referred to the Department of Pediatric and Preventive Dentistry, Faculty of Dental Sciences, M. S. Ramaiah University of Applied Sciences, Bengaluru, with a chief complaint of teeth in the lower front region of jaw and difficulty in feeding. The intraoral examination revealed two natal teeth with Grade II mobility in the mandibular anterior region and presence of ulceration on the ventral surface of the tongue [Figure 1]. Inspection of the ulcer revealed a circular lesion of four mm in diameter on the tip and ventral surface of the tongue with a white fibrous plaque on the surface. The ulcer was painful on palpation. The case was diagnosed as precocious Riga–Fede Disease. It was decided to manage the case with the extraction of natal teeth due to mobility of the teeth which could otherwise lead to complications such as swallowing or aspiration during nursing and prevent the resolution of the ulcerated lesion. Parental consent was obtained, and the extraction of natal teeth was done under topical anesthesia [Figure 2] followed by curettage of the extraction socket. The extracted teeth were yellowish white, opaque without root. The ulcer was resolved, and improvement in suckling was observed at 10 day-follow-up visit.
Figure 1: Two natal teeth and ulceration on tongue

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Figure 2: Extracted natal teeth

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  Discussion Top


The presence of natal and neonatal teeth is definitely a disturbance of biological chronology whose etiology is still unknown.[5] It has been related to several factors, such as superficial position of the tooth germ, infection or malnutrition, febrile states, eruption accelerated by febrile incidents or hormonal stimulation, hereditary transmission of a dominant, autosomal gene, osteoblastic activity inside the germ is related to the remodeling phenomenon, and hypovitaminosis.[6] In the present case, the cause for the natal teeth could be superficial position of the tooth germ.

Natal teeth presents with complications such as discomfort during suckling, irritation, and trauma to infant's tongue, sublingual ulceration (Riga-Fede disease), laceration of the mother's breast, and risk of aspiration if mobility exists.[7],[8] Prolonged gingival irritation may increase the incidence of gingival fibrous dysplasia. As the feeding of the child is affected during the early infancy and associated with difficulty in feeding, the growth and development of the child are affected as the only source of nutrition during infancy is milk, particularly from the mother.[9] Breastfeeding occurs not only as a response to hunger but also as a mechanism of decreasing a child's stress and discomfort, breastfed children are more cooperative and less likely to drop out of school, therefore, would be expected to play an important role in the child's psychosocial development.[10] This is consistent with Peruvian mother's description of breastfeeding as a means of providing the child with comfort, love, security, and communication.[11]

In 1997, Hebling classified natal teeth into four types based on the clinical appearance:[12]

  1. Shell-shaped crown, poorly fixed to the alveolus by gingival tissue, and absence of a root
  2. Solid crown, poorly fixed to the alveolus by gingival tissue, and little or no root
  3. Eruption of the incisal margin of the crown through the gingival tissues
  4. Edema of gingival tissue with an unerupted but palpable tooth.


According to this classification, the extracted teeth in the present case were of Type 2 which had solid crown, poorly fixed to the alveolus by gingival tissue without root.

Riga-Fede disease is a rare clinicopathological entity with unknown etiology although a relation with chronic trauma due to the teeth has been proposed by many authors.[13],[14],[15] Narang et al. described the constant traction due to tongue-tie as another cause in addition to the trauma caused by the teeth.[16] Tang et al. proposed that the trauma is only a contributing factor in the development of Riga-Fede disease that could lead to viral and toxic agents penetrating in the submucosa and into the traumatic area causing inflammatory reaction and tissue loss.[13],[17] The Riga-Fede disease corresponds to a lesion on the mucosa of the tongue resulting from trauma by the primary teeth during tongue forward and backward movements.[18] Hence, in the present case, the possible etiology for Riga–Fede disease is the presence of natal teeth. Clinical presentation, laboratory data, biopsy, and follow-up can help in the differential diagnosis. In fact, ulceration of the tongue may also be due to other causes, including bacterial or mycotic infections, allergy and immunologic diseases, tumors (traumatic neuroma, granular cell myoblastoma, lymphoma, lymphangioma, salivary gland tumors, and metastatic tumors),[19] genetic disorders,[20] primary syphilis, tubercolosis, and agranulocytosis.[13]

Domingues-Cruz et al.[21] proposed a new classification of Riga–Fede disease, dividing the disease into precocious and late. The “precocious Riga–Fede Disease” is associated with natal-neonatal teeth, which appear in the first 6 months of the life and has no correlation with neurological disorders; the “late Riga–Fede Disease” typically appears after 6–8 months of life, with the first dentition, and may be related to neurological disorders. According to this classification, the present case is precocious Riga–Fede Disease.

Treatment of Riga-Fede disease has varied from excision of the lesion, to smoothening of the sharp incisal edges or rounding of the sharp edges of the tooth with composite increments, the later being the chosen treatment where only mild-to-moderate ulceration occurs.[22] In the present case, the ulceration of tongue was large, and natal teeth were mobile; hence, the treatment chosen was extraction of natal teeth over a more conservative approach.

Removal of natal teeth is indicated when they are poorly developed, interfere with feeding, highly mobile, and associated with soft-tissue growth. Prophylactic administration of Vitamin K (0.5–1.0 mg, intramuscular) is advocated because of the risk of hemorrhage as the commensal flora of the intestine might not have been established until the child is 10 days old, and since Vitamin K is essential for the production of prothrombin in the liver.[23] In this case, as the infant was older than 10 days delaying management or administration of Vitamin K was not considered.


  Conclusion Top


The presence of natal teeth creates anxiety in the child and parents as well as difficulty in feeding and rarely the Riga–Fede Disease which is an ulceration on the ventral surface of tongue. Early diagnosis helps in early resolution of ulcerative lesion and aids in resuming normal feeding. Treatment of natal teeth should be carefully decided based on factors such as morphology and mobility of natal teeth as well as the presence of other complicating factors such as Riga–Fede Disease.

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.
Eley KA, Watt-Smith PA, Watt-Smith SR. Deformity of the tongue in an infant: Riga-Fede disease. Paediatr Child Health 2010;15:581-2.  Back to cited text no. 1
    
2.
Campos-Muñoz L, Quesada-Cortés A, Corral-De la Calle M, Arranz-Sánchez D, Gonzalez-Beato MJ, De Lucas R, et al. Tongue ulcer in a child: Riga-Fede disease. J Eur Acad Dermatol Venereol 2006;20:1357-9.  Back to cited text no. 2
    
3.
Zhu J, King D. Natal and neonatal teeth. ASDC J Dent Child 1995;62:123-8.  Back to cited text no. 3
    
4.
Massler M, Savara BS. Natal and neonatal teeth; a review of 24 cases reported in the literature. J Pediatr 1950;36:349-59.  Back to cited text no. 4
    
5.
Bigeard L, Hemmerle J, Sommermater JI. Clinical and ultrastructural study of the natal tooth: Enamel and dentin assessments. ASDC J Dent Child 1996;63:23-31.  Back to cited text no. 5
    
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7.
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8.
Spouge JD, Feasby WH. Erupted teeth in the newborn. Oral Surg Oral Med Oral Pathol 1966;22:198-208.  Back to cited text no. 8
    
9.
Nirmala SV, Prabhu RV, Veluru S, Tharay N, Kolli NK, Hemant KJ. Natal teeth – A case report with decision support system. Pediatr Neonatal Care 2015;2:00073.  Back to cited text no. 9
    
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Lawrence RA, Lawrence RM. Breastfeeding: A Guide for the Medical Profession. 5th ed. St. Louis, MO: Mosby; 1999.  Back to cited text no. 10
    
11.
Marquis GS, Díaz J, Bartolini R, Creed de Kanashiro H, Rasmussen KM. Recognizing the reversible nature of child-feeding decisions: Breastfeeding, weaning, and relactation patterns in a shanty town community of Lima, Peru. Soc Sci Med 1998;47:645-56.  Back to cited text no. 11
    
12.
Hebling J, Zuanon AC, Vianna DR. Dente natal – A case of natal teeth. Odontol Clín 1997;7:37-40.  Back to cited text no. 12
    
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Ceyhan AM, Yildirim M, Basak PY, Akkaya VB, Ayata A. Traumatic lingual ulcer in a child: Riga-Fede disease. Clin Exp Dermatol 2009;34:186-8.  Back to cited text no. 13
    
14.
Hegde RJ. Sublingual traumatic ulceration due to neonatal teeth (Riga-Fede disease). J Indian Soc Pedod Prev Dent 2005;23:51-2.  Back to cited text no. 14
[PUBMED]  [Full text]  
15.
Goho C. Neonatal sublingual traumatic ulceration (Riga-Fede disease): Reports of cases. ASDC J Dent Child 1996;63:362-4.  Back to cited text no. 15
    
16.
Narang T, De D, Kanwar AJ. Riga-Fede disease: Trauma due to teeth or tongue tie? J Eur Acad Dermatol Venereol 2008;22:395-6.  Back to cited text no. 16
    
17.
Tang TT, Glicklich M, Hodach AE, Oechler HW, McCreadie SR. Ulcerative eosinophilic granuloma of the tongue. A light- and electron-microscopic study. Am J Clin Pathol 1981;75:420-5.  Back to cited text no. 17
    
18.
Mohan RP, Verma S, Gill N, Singh U. Riga-Fede disease (Cardarelli's aphthae): A report of nine cases. S Afr J Child Health 2014;8:72-4.  Back to cited text no. 18
    
19.
Joseph BK, BairavaSundaram D. Oral traumatic granuloma: Report of a case and review of literature. Dent Traumatol 2010;26:94-7.  Back to cited text no. 19
    
20.
Baroni A, Capristo C, Rossiello L, Faccenda F, Satriano RA. Lingual traumatic ulceration (Riga-Fede disease). Int J Dermatol 2006;45:1096-7.  Back to cited text no. 20
    
21.
Domingues-Cruz J, Herrera A, Fernandez-Crehuet P, Garcia-Bravo B, Camacho F. Riga-Fede disease associated with postanoxic encephalopathy and trisomy 21: A proposed classification. Pediatr Dermatol 2007;24:663-5.  Back to cited text no. 21
    
22.
Slayton RL. Treatment alternatives for sublingual traumatic ulceration (Riga-Fede disease). Pediatr Dent 2000;22:413-4.  Back to cited text no. 22
    
23.
Rao RS, Mathad SV. Natal teeth: Case report and review of literature. J Oral Maxillofac Pathol 2009;13:41-6.  Back to cited text no. 23
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