Home | About Us | Editorial Board | Current Issue | Archives | Search | Instructions | Subscription | Feedback | e-Alerts | Login 
Journal of Indian Society of Pedodontics and Preventive Dentistry Official publication of Indian Society of Pedodontics and Preventive Dentistry
 Users Online: 1655  
 
  Print this page Email this page   Small font sizeDefault font sizeIncrease font size


 
  Table of Contents    
CASE REPORT
Year : 2019  |  Volume : 37  |  Issue : 1  |  Page : 107-109
 

Neonatal molar in a child with Langerhan cell histiocytosis


Department of Pedodontics and Preventive Dentistry, UCMS and GTB Hospital, New Delhi, India

Date of Web Publication25-Feb-2019

Correspondence Address:
Dr. Sunil Kumar
Department of Pedodontics and Preventive Dentistry, UCMS and GTB Hospital, New Delhi - 110 095
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JISPPD.JISPPD_194_18

Rights and Permissions

 

   Abstract 


Teeth which erupt in the 1st month of postnatal life are known as “neonatal tooth.” The incidence of these teeth ranges from 1:2000 to 1:3500 live births. Natal teeth are more common in mandibular central incisor region, followed by maxillary incisor region and mandibular canine region. The neonatal or natal teeth in the maxillary molar region are a rare occurrence. This article represents a rare case of the neonatal tooth with Langerhans cell histiocytosis.


Keywords: Histiocytosis, Langerhans cell, neonatal teeth


How to cite this article:
Kalra N, Tyagi R, Khatri A, Kumar S, MM A, Khandelwal D. Neonatal molar in a child with Langerhan cell histiocytosis. J Indian Soc Pedod Prev Dent 2019;37:107-9

How to cite this URL:
Kalra N, Tyagi R, Khatri A, Kumar S, MM A, Khandelwal D. Neonatal molar in a child with Langerhan cell histiocytosis. J Indian Soc Pedod Prev Dent [serial online] 2019 [cited 2019 Oct 19];37:107-9. Available from: http://www.jisppd.com/text.asp?2019/37/1/107/252851





   Introduction Top


Tooth eruption at about 6 months of age is a milestone both in terms of functional and physiological changes in a child's life and emotional terms for parents.[1] Several terms have been used in the literature to designate teeth that erupt before the normal time, such as congenital teeth, fatal teeth, predeciduous teeth, and dentitia precox.[2],[3]

Massler and Savara defined these teeth as natal and neonatal teeth. Natal teeth are those teeth that erupt in the oral cavity at the time of birth and neonatal teeth are those that erupt during the first 30 days of life. The exact etiology of natal and neonatal teeth is unknown.[4]

These teeth are more common in the mandibular arch than maxillary arch and most commonly found in the incisor region than in the canine and molar regions. Various investigators have shown that 85% of these teeth erupt in the mandibular incisor region followed by 11% in the maxillary incisor region, 3% in the mandibular canine region, and 1% in the maxillary canine and molar regions.[4],[5]

Langerhans cell histiocytosis (LCH) was formerly known as histiocytosis X and refers to a group of conditions characteristics by the uncontrolled stimulation and proliferation of a normal antigen-processing cell.[6],[7] It is currently regarded as a myeloid neoplasm, with a remarkably broad clinical spectrum, ranging from isolated skin or bone lesions to a disseminated disease that can involve nearly any organ.[8] A neonatal tooth in the molar region with LCH is a rare finding. This article presents a case of a neonatal maxillary molar with LCH in a 2½-month-old male child.


   Case Report Top


A 2½-month-old male child was brought to the Department of Pedodontics and Preventive Dentistry, UCMS and GTB Hospital, New Delhi, with a chief complaint of an unusual tooth in the upper left back gum pad region. A consent was taken from the guardian before performing the intraoral examination.

On intraoral examination, an unusual neonatal tooth was found in the maxillary arch (in the left deciduous 1st molar region) [Figure 1]. The tooth was not producing any discomfort on feeding; no associated ulcer was found on the opposing arch and tooth was not mobile. On soft-tissue examination, the complete gum pad was reddish, with small petechiae. Medical history of the child revealed that the child was admitted to the Department of Paediatrics with the chief complaint of vesicles all over the body, loose stool, cough, fever, and abdominal distress for several days [Figure 2]. Through proper examination and investigation with the help of skin and bone biopsy, it was diagnosed with a case of LCH.
Figure 1: Neonatal molar

Click here to view
Figure 2: Small petechiae were present on the abdomen

Click here to view


As the tooth was not producing any discomfort to the child or mother, we left the tooth without any intervention and advised parents to be on regular follow-up.


   Discussion Top


LCH is a heterogeneous disease, characterized by the accumulation of dendritic cells with features like epidermal Langerhans cells in various organs. Any organ or system of the human body can be affected, but more frequently involved are the skeleton (80%), the skin (33%), and the pituitary (25%). Other organs involved are the liver, spleen, hematopoietic system and the lungs (15% each), lymph nodes (5%–10%), and the central nervous system, excluding the pituitary (2%–4%).[8]

In our case, the lesion was present on the complete upper alveolar ridge and gingiva along with the neonatal molar on the left side, which is a unique finding and could be taken as reference for further researches. The incidence of neonatal teeth in the maxillary arch in the molar region is only 1%.[9] Natal teeth may be associated with syndromes such as Ellis–Van Creveld, Jackson–Lawler, Hallermann–Streiff, and steatocystoma multiplex with natal teeth.[10]

Oral manifestations may be the earliest sign of this disorder. The incidence of oral lesions is as frequent as 77%. Gingivitis with hyperplasia, bleeding, recession and necrosis, mucosal ulcerations and destruction of the alveolar bone and periodontal support, impaired healing, and abnormal primary dentition mobility (floating teeth) are common findings in pediatric patients. Furthermore, lytic bone lesions in the jaw are predominant (5%–10% of cases), primarily in the posterior region (distal to the canines) and ramus of the mandible, occasionally accompanied by facial pain and swelling, and, in severe cases, pathological jaw fractures; they may disturb the tooth germ development and cause enamel hypoplasia of the permanent teeth.[11]

Previous observational studies have shown that head and neck are the most common anatomical areas where LCH bone lesions are present, with an incidence rate of 65%–90%, including the skull (particularly the calvarium, temporal bones, and the sella turcica), mandible, or maxilla.[11]

Artzi et al. mentioned that LCH patients with head-and-neck lesions manifested characteristic signs and symptoms of the periodontal disease. Thus, the evidence of destructive periodontitis, abnormal mobility, or tooth loss as the initial child's complaints during the primary dentition stage may be indicative of a systemic condition, such as LCH; for purposes of confirming the diagnosis using immunofluorescence tests, it is strongly recommended that the dentist collect enough gingival biopsy material, with or without tooth extraction.[12]

According to the Histiocyte Society, the three points of confidence for a precise diagnosis of LCH in children are as follows: (1) clinical and radiographic oral characteristics, for example, displaced teeth and developing permanent tooth follicles with altered eruption patterns or dentoalveolar growth; (2) two or more positive stains for adenosine triphosphatase, S-100 protein, alpha-D-mannosidase, or presence of peanut lectin; and (3) detection of Birbeck granules and CD 1a antigen (T6) on the surface of Langerhans cells. Once the diagnosis is confirmed, the treatment team must perform skeletal and visceral system assessments to verify the presence of other lesions in the rest of the patient's body.[11]

Treatment depends on the severity and progression of the clinical signs and symptoms. Treatment methods for LCH include observation for spontaneous regression, surgical curettage, radiation, topical injection of steroids, and systemic steroid therapy. Surgical curettage and topical injection of steroids are usually the preferred treatments for solitary jaw lesions. In young children, radiation therapy to the jaw may result in damage to the developing teeth and bone and is thus not the first choice. Steroid therapy is usually used when multiple organs are involved or when organ dysfunction is present.[13]

This case illustrates the contribution of the dental team to the functional rehabilitation of the patient. The children with teeth at the time of birth should be thoroughly examined and proper treatment should be provided at the appropriate age. The parents should be counseled properly in these cases.

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.
Zhu J, King D. Natal and neonatal teeth. ASDC J Dent Child 1995;62:123-8.  Back to cited text no. 1
    
2.
Bodenhoff J. Natal and neonatal teeth. J Odontal Tidskr 1959;67:645-95.  Back to cited text no. 2
    
3.
Mayhall JT. Natal and neonatal teeth among the Tlinget Indian. J Dent Res 1967;46:748-9.  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.
Bodenhoff J. Natal and neonatal teeth. Odon T 1959;67:645-95.  Back to cited text no. 5
    
6.
Stein SL, Paller AS, Haut PR, Mancini AJ. Langerhans cell histiocytosis presenting in the neonatal period: A retrospective case series. Arch Pediatr Adolesc Med 2001;155:778-83.  Back to cited text no. 6
    
7.
Batra P, Roychoudhary A, Prakash H. Langerhans cell histiocytosis – A case report. J Indian Soc Pedod Prev Dent 2004;22:168-70.  Back to cited text no. 7
[PUBMED]    
8.
Haupt R, Minkov M, Astigarraga I, Schäfer E, Nanduri V, Jubran R, et al. Langerhans cell histiocytosis (LCH): Guidelines for diagnosis, clinical work-up, and treatment for patients till the age of 18 years. Pediatr Blood Cancer 2013;60:175-84.  Back to cited text no. 8
    
9.
Sarkar S, Sarkar S. Unusual neonatal tooth in maxillary 1st molar region: A case report. J Indian Soc Pedod Prev Dent 2007;25 Suppl 1:S41-2.  Back to cited text no. 9
    
10.
Kumar A, Grewal H, Verma M. Posterior neonatal teeth. J Indian Soc Pedod Prev Dent 2011;29:68-70.  Back to cited text no. 10
[PUBMED]  [Full text]  
11.
Ramos-Gutiérrez E, Alejo-González F, Ruiz-Rodríguez S, Garrocho-Rangel JA, Pozos-Guillén A. Langerhans cell histiocytosis: Current concepts in dentistry and case report. J Clin Exp Dent 2016;8:e102-8.  Back to cited text no. 11
    
12.
Artzi Z, Grosky M, Raviv M. Periodontal manifestations of adult onset of histiocytosis X. J Periodontol 1989;60:57-66.  Back to cited text no. 12
    
13.
Mitomi T, Tomizawa M, Noda T. Tooth development included in the multifocal jaw lesions of Langerhans cell histiocytosis. Int J Paediatr Dent 2005;15:123-6.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2]



 

Top
Print this article  Email this article
 

    

 
  Search
 
  
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Article in PDF (707 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case Report
   Discussion
    References
    Article Figures

 Article Access Statistics
    Viewed849    
    Printed23    
    Emailed0    
    PDF Downloaded165    
    Comments [Add]    

Recommend this journal


Contact us | Sitemap | Advertise | What's New | Copyright and Disclaimer 
  2005 - Journal of Indian Society of Pedodontics and Preventive Dentistry | Published by Wolters Kluwer - Medknow 
Online since 1st May '05