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CASE REPORT
Year : 2020  |  Volume : 38  |  Issue : 2  |  Page : 190-199
 

Oral submucous fibrosis in children and adolescents: Analysis of 36 cases


1 Department of Oral Medicine and Maxillofacial Radiology, K. M. Shah Dental College and Hospital, Sumandeep Vidyapeeth Deemed University, Vadodara, Gujarat, India
2 Harvard Medical school, Harvard University; Division of Oral Medicine and Dentistry, Brigham and Women's Hospital, Boston, Massachusetts, USA
3 Director, Ajinkya D Y Patil University, Pune, Maharashtra, India
4 Department of Neuroscience, University of Illinois at Chicago, Illinois, USA
5 Department of Oral Pathology, B. P. Koirala Institute of Health Sciences, Dharan, Nepal
6 Department of Pedodontics and Preventive Dentistry, Maulana Azad Institute of Dental Sciences, New Delhi, India

Date of Submission14-Apr-2020
Date of Acceptance03-Jun-2020
Date of Web Publication28-Jun-2020

Correspondence Address:
Dr. Naman R Rao
Harvard Medical School, Harvard University, Boston, Massachusetts
USA
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JISPPD.JISPPD_173_20

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   Abstract 


Oral submucous fibrosis (OSMF) is a chronic debilitating irreversible oral potentially malignant disorder affecting any part of the oral cavity. It is usually seen in adults but rarely noticed in children and adolescents. Since the paucity of the cases, there exists a gap of knowledge in the causative habits, root reasons of habit initiation, age of habit initiation, and the common clinical representation of this disorder. The current article aims to bridge this gap by presenting unusual 36 cases of children and adolescents reported at the tertiary care hospital of Vadodara, Gujarat, India, with specific areca nut chewing habit and distinct features of OSMF. Furthermore, the present case series is the first of its kind in the scientific literature with a high number of OSMF cases in children and adolescents.


Keywords: Adolescent, arecanut, children, gutkha, oral potential malignant disorders, oral submucous fibrosis


How to cite this article:
More CB, Rao NR, Hegde R, Brahmbhatt RM, Shrestha A, Kumar G. Oral submucous fibrosis in children and adolescents: Analysis of 36 cases. J Indian Soc Pedod Prev Dent 2020;38:190-9

How to cite this URL:
More CB, Rao NR, Hegde R, Brahmbhatt RM, Shrestha A, Kumar G. Oral submucous fibrosis in children and adolescents: Analysis of 36 cases. J Indian Soc Pedod Prev Dent [serial online] 2020 [cited 2020 Jul 7];38:190-9. Available from: http://www.jisppd.com/text.asp?2020/38/2/190/288219





   Introduction Top


Oral submucous fibrosis (OSMF) is a potentially malignant disorder described by Schwartz J in 1952 as “Atropica idiopathica mucosae oris” and later defined (based on pathology) in 1966 by Pindborg.[1],[2] Recently, based on its clinical behavior, a new definition was proposed that described OSMF as “a debilitating, progressive, irreversible collagen metabolic disorder induced by chronic chewing of areca nut and its commercial preparations; affecting the oral mucosa and occasionally the pharynx and esophagus; leading to mucosal stiffness and functional morbidity; and has a potential risk of malignant transformation.”[3]

Despite having a multifactorial etiopathogenesis, areca nut chewing in any formulation is considered as the chief causative agent.[4] Other contributors/associated risk factors include chewing or consumption of smokeless tobacco, toxic levels of copper in foodstuffs and masticatories, high intake of chilies, vitamin deficiencies, malnutrition resulting in low levels of serum proteins, anemia, and immunological and/or genetic predisposition.[5],[6],[7] This disorder is more commonly reported in South and South-East Asian countries with an increased predilection in male adults.[8] Due to the varying geographical locations, sample size, and study methodology, the prevalence of OSMF shows a broader range that falls between 0.1% and 30%.[9] The recent data also show an increased incidence of OSMF in Western countries, considering the immigrant population and surge in the availability of processed areca nut products.[10]

Clinically, signs and symptoms of this disorder include pain, burning sensation, and ulceration.[11],[12] Progressive restriction of mouth opening, mucosal blanching, tongue depapillation, and loss of pigmentation are other classical features.[13] Dysphonia and hearing impairment are also reported in the advanced cases of OSMF.[14] Since the clinical presentation varies based on the stage of the disorder, various treatment modalities are proposed to date.[15],[16]

This disorder known to be occurring in the middle-aged population has now begun to get noticed in the children and adolescent population.[17] Since there exist few numbers of children and adolescent cases reported in the literature, there exists a gap of evidence-based literature and knowledge in the commonly adapted habits, root reasons of habit initiation, age of habit initiation, and the common clinical representation of this disorder. The current article aims to bridge this gap by presenting 36 cases of children and adolescent patients of OSMF reported at the tertiary care hospital in Vadodara, Gujarat. Furthermore, the present case series is the first of its kind in the scientific literature with a high number of OSMF cases in children and adolescents.


   Cases Presentation Top


All the cases presented in this article come from the Department of Oral Medicine and Maxillofacial Radiology, K. M. Shah Dental College and Hospital, Sumandeep Vidyapeeth, Vadodara, Gujarat state, India [Table 1] and [Figure 1]. The patients reported in this article were of age ≤ 17 years and were clinically examined by the residents/specialist attendings as per the department's standard operating protocol (SOP). A full descriptive history of the patient was taken that included demographics, types of habit, age of habit initiation, frequency of habit, and reasons for habit initiation. Confirmatory diagnosis of the disease was made by the attending based on the clinically observed symptoms of the patients. Dr. More et al. classification system of OSMF was used for the clinical diagnosis of the patients.[18] No biopsies were taken. Palliative treatment for OSMF including habit cessation and dietary modification was rendered to these patients. Regular referrals for other treatment needs were made available as per the regular hospital protocol. Per the departmental SOP, permission to publish clinical images for educational purposes was obtained from the patient's parents/guardian. Permission to use those clinical images from the archive of the Oral Medicine and Maxillofacial Radiology (OMR) was approved by the head of the department.
Table 1: Individual case presentation of 36 patients

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Figure 1: The case series showing the involvement of different parts of the oral mucosa. (a) (Blanching of the soft palate), (b) (presence of blanching and thin fibrous bands in the left buccal mucosa), (c) (presence of blanching and thin fibrous bands in the right buccal mucosa) shows oral submucous fibrosis signs in a 15-year-old male patient with a history of gutkha chewing. (d) (Isolated involvement of lower labial mucosa) shows oral submucous fibrosis signs in a 12-year-old female patient with a history of flavored areca nut chewing

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Analytical methods

To construe all these 36 cases, a descriptive statistics of patient demographics, types of habit, age of habit initiation, frequency of habit, reasons for habit initiation, clinical presentation, and diagnosis was reported.


   Results Top


Baseline characteristics

Of the 36 diagnosed patients of OSMF, there were 75% (n = 27) male and 25% (n = 9) female patients with the majority of patients belonging to the age bar of 12–14 years. Eighty-one percent (n = 29) of all were diagnosed with Stage I and rest with Stage II (19%, n = 7) [Table 2].
Table 2: Baseline characteristics

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Types of habit

Of all habits, chewing/consumption of the flavored areca nut (n = 18, 50%) was noted with majority of the patients, followed by pan masala (n = 5, 14%), mawa (n = 4, 11%), betel leaf quid with areca nut excluding tobacco (n = 4, 11%), baked areca nut products (n = 3, 8%), and gutkha (n = 2, 6%) [Table 3].
Table 3: Types of habit

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Age of habit initiation

Majority of the patients initiated these habits at the age of 4–5 years (n = 12, 33%), followed by 6–7 years (n = 9, 25%), 8–9 years (n = 8, 22%), and 10–11 years (n = 7, 19%) [Table 4].
Table 4: Age of habit initiation

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Reasons for habit initiation

Among all, maximum number of the patients reported initiation of habit due to influential surrounding of parents/siblings/teachers/relatives who are involved in chewing various forms of areca nut products (n = 18, 50%), followed by peer pressure (n = 17, 47%), attractive packaging (n = 8, 22%), attractive advertisement of the areca nut-related products (n = 8, 22%), to avoid pressure of studies (n = 5, 14%), influenced by film actors consuming various forms of areca nut (n = 5, 14%), to look mature (n = 3, 8%), to distract from domestic violence of parents (n = 2, 6%), and to increase social interaction (n = 3, 8%) [Table 5].
Table 5: Reasons for habit initiation

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Methods and amount of areca nut and its by-product consumption

On observing the packet size and frequency of areca nut and its by-product (ANBP) consumption, majority of the patients reported consumption of small packets (n = 13, 36%), followed by large packets (n = 12, 33%), ball (n = 4, 11%), fold (n = 4, 11%), and whole seed (n = 3, 8%). Among patients consuming small packet, 19% reported consumption of 4–6 packets/day, followed by 11% (1–3 packets/day) and 6% (7–9 packets/day). Among patients consuming large packet, 22% reported consumption of 4–6 packets/day, followed by 8% (7–9 packets/day) and 6% (1–3 packets/day). Among patients consuming ball, 6% reported consumption of 1–3 and 4–6 balls/day. Among patients consuming fold, 8% reported consumption of 4–6 folds/day and 3% with 1–3 folds/day. Among patients consuming whole seed, 8% reported consumption of 1–3 seeds/day [Table 6].
Table 6: Methods and amount of areca nut and its by.product consumption

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Clinical signs and symptoms

Per the clinical reporting, majority of the patients complained of burning sensation of the mouth (n = 24, 67%) and loss of taste sensation (n = 9, 25%), followed by other complaints. Per the clinical observations, most of the patients showed blanching of the buccal mucosa (n = 27, 75%) and thin palpable fibrous bands on both sides of the buccal mucosa (n = 22, 61%), followed by other signs [Table 7].
Table 7: Clinical signs and symptoms

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   Discussion Top


With a global prevalence of 5 million patients, OSMF is an increasing oral potentially malignant disorder (OPMD) most commonly noted in the South and South-East Asian countries.[9],[19],[20] Since the inception of this disorder, innumerable studies showcasing different aspects of demographics and diagnostic and treatment modalities have been published in the literature. These literatures have shown an alarming trend in demographics, especially in the age group per se. Earlier, this disorder, being more prevalent among middle-aged, older age patients (above 40 years), and adult patients (>20–<40 years),[19],[20],[21],[22],[23],[24] has lately and unfortunately started affecting adolescents and teenagers (below 20 years).[25],[26],[27] Despite the increased reported incidence in the adolescent population, there still exists a paucity of data that provide causative habits, root reasons for habit initiation, age of habit initiation, and common clinical representation of this disorder in the children and adolescent population. The current article is therefore presented to bridge this gap.

Most of the case reports presented to date have mentioned OSMF to be rare among the adolescent. However, Ali et al. in their study had mentioned the incidence of 7.4% of OSMF among school-going males.[26] Clinicopathological study conducted by Cai et al. among 647 cases of OSMF among the population of China showed the prevalence to be 2.37% in the age group of 10–19 years, whereas it was 35.2% in a study conducted by Hazarey et al. in 1000 cases of OSMF from Central India.[20],[27] In the present article among 36 cases of OSMF, majority of cases were males, which can be correlated with a higher prevalence of areca nut chewing habit in school-going males in the study conducted by Oakley et al.[24] The result was in contrast to review paper published by Jain and Taneja where the incidence was higher in females.[17]

Among adolescents, the prevalence of OSMF has been observed with a marked variation. Per our data, the age for OSMF ranges from 9 to 17 years with the highest prevalence within the age range of 12–14 years, whereas the study conducted by Basagoudanavar et al. observed the range of 16–18 years.[28] Jain and Taneja in their study mentioned the minimum age for OSMF presentation to be 2.5 years to a maximum of 10 years.[17] A study conducted by Babu et al. mentioned the prevalent age to be 14–19 years.[29]

The clinical presentation of OSMF in adolescent groups remained similar to those with adults and older age including the classical feature of reduced mouth opening, burning sensation, blanching of the oral mucosa, restricted tongue movement, the palpable fibrotic band in the buccal mucosa, retromolar area, and soft palate.[27] Per our data, we observed burning sensation and blanching of the buccal mucosa as the most common symptom and sign, respectively, being clinical Stages I and II the most prevalent.

Early exposure of areca nut is mostly attributed to surrounding influences that include parents, relatives, friends, film actors, and other significant having the habit of ANBP consumption. Stress, domestic violence, avoidance of hunger, craving, aid to concentration, pleasure, taste, look mature, attractive packaging, and refresh breath are also some documented drivers that lead to exposure of areca nut in early age life.[25] Per our observation, the influence of parents, relatives, friends, and teachers was the predominant cause for exposer of areca nut and its product among adolescents.

Studies have suggested that the severity of OSMF increases with an increase in the frequency of the habit.[22],[30] In the present article, most of the cases had a frequency of 4–6 times/day irrespective of packet size or form of areca nut and its product. The habit of chewing areca nut is the prime etiological agent for causing OSMF. All the cases in the present article showed the habit of ANBP consumption, among which flavored areca nut was observed to be predominant.

The increasing incidence of OSMF in the younger population is a big challenge not only locally but also from the global disease burden standpoint. There exists a limited available resource depicting the information regarding OSMF in the adolescent/teenage population. Hence, this scientific information could be very contributory to further perform an observational study to understand the depth of this disorder in the younger age group population.

The strengths of this article include its original hospital-based data from one of the reputed academic tertiary care hospitals of Vadodara, Gujarat, providing a large cohort of OSMF children and adolescent cases. The present article had several limitations that must be considered. Some of these limitations include those inherent to the case reporting methods including missing records of follow-up visits. Since the presented data characterize the rural cohort of Vadodara, Gujarat, the findings can differ among the regions of Gujarat, states as well as other countries. Although, one of the largest reported cohorts to date, the data remain limited to a descriptive representation due to a small sample size.


   Conclusion Top


OSMF, an uncertain OPMD with the devastating outcome, has now begun to get noticed in the children and adolescents. Since it is known to have no obvious treatment modalities, it is rather important to develop preventive strategies. The data presented in the article increase concern related to the ANBP abuse among the children and adolescent groups. This needs to be urgently addressed by the national and local government bodies. There is a strong need to understand the drivers of habit initiation among the younger population. Age and culturally appropriate control and preventive strategies might be helpful for this group. Development of early educational resources at primary education level, special intoxication, and rehabilitation centers should perhaps be developed for this population in the region of Vadodara, Gujarat. Of course, further observational studies would significantly improve and provide evidence to fight this dreadful disorder.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

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Schwartz J. Atrophia Idiopathica Tropica Mucosa Oris. 11th International Dental Congress. London; 1952.  Back to cited text no. 1
    
2.
Pindborg JJ. Oral submucous fibrosis as a precancerous condition. J Dent Res 1966;45:546-53.  Back to cited text no. 2
    
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More CB, Rao NR. Proposed clinical definition for oral submucous fibrosis. J Oral Biol Craniofac Res 2019;9:311-4.  Back to cited text no. 3
    
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More C, Shilu K, Gavli N, Rao NR. Etiopathogenesis and clinical manifestations of oral submucous fibrosis, a potentially malignant disorder: An update. Int J Curr Res 2018;10:71816-20.  Back to cited text no. 4
    
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Shih YH, Wang TH, Shieh TM, Tseng YH. Oral submucous fibrosis: A review on etiopathogenesis, diagnosis, and therapy. Int J Mol Sci 2019;20:2940.  Back to cited text no. 5
    
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More C, Rao NR, More S, Johnson NW. Reasons for initiation of areca nut and related products in patients with oral submucous fibrosis within an endemic area in Gujarat, India. Substance Misuse 2020. [doi: 10.1080/10826084.2019.1660678].  Back to cited text no. 6
    
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More C, Peter R, Nishma G, Chen Y, Rao N. Association of Candida species with oral submucous fibrosis and oral leukoplakia: A case control study. Ann Clin Lab Res 2018;6:248.  Back to cited text no. 7
    
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Cox SC, Walker DM. Oral submucous fibrosis. A review. Aust Dent J 1996;41:294-9.  Back to cited text no. 8
    
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Rao NR, Villa A, More CB, Jayasinghe RD, Kerr AR, Johnson NW. Oral submucous fibrosis: A contemporary narrative review with a proposed inter-professional approach for an early diagnosis and clinical management. J Otolaryngol Head Neck Surg 2020;49:3.  Back to cited text no. 9
    
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Wahi PN, Kapur VL, Luthra UK, Srivastava MC. Submucous fibrosis of the oral cavity. 1. Clinical features. Bull World Health Organ 1966;35:789-92.  Back to cited text no. 11
    
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More C, Pawar R, Rao N, Shah P, Gavli N. Oral ulcer: An overview with an emphasis on differential diagnosis. Int J Oral Health Sci Adv 2015;3:1-13.  Back to cited text no. 12
    
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More C, Shah P, Rao N, Pawar R. Oral submucous fibrosis: An overview with evidence-based management. Int J Oral Health Sci Adv 2015;3:40-9.  Back to cited text no. 13
    
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Devi P, Singh I, Setru R, Tyagi K, Singh D, Thiyam B. Evaluation of hearing deficit in patients with oral submucous fibrosis. J Oral Sci 2015;57:109-13.  Back to cited text no. 14
    
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[PUBMED]  [Full text]  
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Jain A, Taneja S. Oral submucous fibrosis in pediatric patients: A systematic review and protocol for management. Int J Surg Oncol 2019;2019:3497136.  Back to cited text no. 17
    
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More CB, Das S, Patel H, Adalja C, Kamatchi V, Venkatesh R. Proposed clinical classification for oral submucous fibrosis. Oral Oncol 2012;48:200-2.  Back to cited text no. 18
    
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Jayanthi V, Probert CS, Sher KS, Mayberry JF. Oral submucosal fibrosis--a preventable disease. Gut 1992;33:4-6.  Back to cited text no. 19
    
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Hazarey VK, Erlewad DM, Mundhe KA, Ughade SN. Oral submucous fibrosis: Study of 1000 cases from central India. J Oral Pathol Med 2007;36:12-7.  Back to cited text no. 20
    
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Goyal S, Ahmed J. O21. Oral submucous fibrosis – A clinico-histopathological comparative study in population of Southern Rajasthan, India. Oral Oncol 2011;47:S35.  Back to cited text no. 22
    
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Oakley E, Demaine L, Warnakulasuriya S. Areca (betel) nut chewing habit among high-school children in the Commonwealth of the Northern Mariana Islands (Micronesia). Bull World Health Organ 2005;83:656-60.  Back to cited text no. 24
    
25.
Joseph N, Nagaraj K, Shashidhar Kotian M. Areca nut and tobacco use among school children in a village in South India – A cross sectional study. Australas Med J 2010;1:299-303.  Back to cited text no. 25
    
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Ali SM, Qureshi R, Jamal S. Prevalence of oral submucous fibrosis and use of tobacco and related products amongst school going males. Pak Oral Dent J 2013;31:384-7.  Back to cited text no. 26
    
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Cai X, Yao Z, Liu G, Cui L, Li H, Huang J. Oral submucous fibrosis: A clinicopathological study of 674 cases in China. J Oral Pathol Med 2019;48:321-5.  Back to cited text no. 27
    
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Basagoudanavar H, Shrikrishna AC. Jyothi ABSTRACT. Prevalence of areca nut eating habits and its association with oral submucous fibrosis in preuniversity college- going adolescents of Raichur in Karnataka, India: A prospective cross-sectional survey. Int J Head Neck Surg 2016;7:197-203.  Back to cited text no. 28
    
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Ara SA, Arora V, Zakaullah S, Raheel SA, Rampure P, Ashraf S. Correlation of habits and clinical findings with histopathological diagnosis in oral submucosal fibrosis patients. Asian Pac J Cancer Prev 2013;14:7075-80.  Back to cited text no. 30
    


    Figures

  [Figure 1]
 
 
    Tables

  [Table 1], [Table 2], [Table 3], [Table 4], [Table 5], [Table 6], [Table 7]



 

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    Abstract
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