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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 27  |  Issue : 1  |  Page : 105-111

Oral submucous fibrosis in children: Report of three cases and review


1 Department of Oral Medicine and Radiology, Saraswati Dhanwantari Dental College and Hospital, Parbhani, Maharashtra, India
2 Department of Oral Medicine and Radiology, Government Dental College and Hospital, Hyderabad, Telangana, India
3 Department of Maxillofacial Pathology and Microbiology, Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre, Hyderabad, Telangana, India
4 Department of Oral Medicine and Radiology, Lenora Institute of Dental Sciences, Rajahmundry, Andhra Pradesh, India

Date of Submission30-Dec-2014
Date of Acceptance14-Sep-2015
Date of Web Publication12-Oct-2015

Correspondence Address:
Tulasi Lakshmi Duggirala
Department of Oral Medicine and Radiology, Saraswati Dhanwantari Dental College and Hospital, Parbhani, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.167127

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   Abstract 

Oral submucous fibrosis (OSMF) is a chronic, insidious, generalized, and debilitating condition of the oral mucosa predominantly encountered in South-East Asian countries. Oral submucous fibrosis is etiologically linked to the consumption of the areca nut in flavored formulations or as an ingredient in the betel quid chewed by the communities in these countries. The sweet supari, in their multicolored attractive pouches, is the most common chewed product in children. It is considered a harmless mouth freshener and therefore is consumed in larger amount and is kept in the mouth for a longer time and swallowed by the ignorant children. Factors involved in the consumption of sweet supari are levels of awareness, household environment, peer pressure, low cost, easy availability, etc. Here, we report three cases of OSMF in a 9-year-old girl, 13-year-old boy and a 15-year-old girl and literature review.

Keywords: Areca nut, children, oral submucous fibrosis, OSMF, sweet supari


How to cite this article:
Duggirala TL, Marthala M, Gannepalli A, Podduturi SR. Oral submucous fibrosis in children: Report of three cases and review. J Indian Acad Oral Med Radiol 2015;27:105-11

How to cite this URL:
Duggirala TL, Marthala M, Gannepalli A, Podduturi SR. Oral submucous fibrosis in children: Report of three cases and review. J Indian Acad Oral Med Radiol [serial online] 2015 [cited 2020 May 25];27:105-11. Available from: http://www.jiaomr.in/text.asp?2015/27/1/105/167127


   Introduction Top


Oral submucous fibrosis (OSMF) is a chronic debilitating and a well-recognized potentially malignant condition of the oral and oropharyngeal mucosa with initial inflammation followed by progressive fibrosis of the underlying connective tissues. The morbidity and mortality rates are associated with significant masticatory dysfunction and oral discomfort as well as an increased risk of developing squamous cell carcinoma. This condition was first described in ancient Indian medical manuscripts by Sushruta in around 600 B.C. describing it as "Vidari", the symptoms of which resemble symptomatology of OSMF. [1] It was first described in the modern literature by Schwartz in 1952. [2] Joshi first described the condition in India and suggested the name oral submucous fibrosis. [3]

The strongest evidence regarding the etiology of OSMF associates it with the habit of areca nut chewing and is prevalent in the population of South Asian, South-East Asian and among South Asian immigrants in Western countries. [4] The prevalence of OSMF in India varies between 0.03% and 3.2% according to various epidemiological studies. [5],[6] Although OSMF is generally considered an adult disease, it has been reported among children from communities with betel and areca nut chewing habits. In recent years marked increase in the occurrence of OSMF was observed in many parts of India like Bihar, Madhya Pradesh, Gujarat and Maharashtra. The younger generation is suffering more due to areca nut products in different multicolored attractive pouches. [7] The epidemiological studies conducted on the prevalence of OSMF in children are few. Several small surveys conducted in schools and colleges in several states of India have reported that 13-50% of students chew areca nut in its different forms. [8] Very few cases of OSMF have been reported in children in the literature. Here we are presenting three cases of OSMF in children; in a 9-year-old girl, 13-year-old boy and a 15-year-old girl.


   Case Reports Top


Case-1

A 9-year-old girl reported to the Department of Oral medicine and Radiology with the chief complaint of restricted mouth opening and severe burning sensation on eating spicy food. History of present illness showed chewing sweet supari 9-10 times a day since 3 years. Burning sensation on taking spicy foods was noticed since 1 year with increase in severity and progressive restriction of the mouth opening since 6 months. Her history revealed that she belonged to lower socio-economic status and was a cowherd. She used to chew supari (a sweetened form of areca nut) continuously while looking after the cows and was not aware of the deleterious effects. She got into the habit with other children due to its sweet taste. On examination, inter-incisal distance (IID) was 1.4 cm [Figure 1]. Intraoral examination showed pale, opaque, blanched and fibrotic labial mucosa, buccal mucosa, palate and floor of the mouth [Figure 2]. Gingiva was pale with loss of stippling. Generalized areas of pigmentation were seen with marble-like appearance. Tongue was pale, stiff, atrophic and depapillated with restricted movement [Figure 3]. On palpation, lips and cheeks were rigid, and difficult to retract with decreased resiliency. Fibrous bands were appreciated around the entire rima oris and vertical bands in the buccal mucosa which were thick in the posterior region towards retro-molar area. Palate was fibrotic with shrunken uvula. Oral hygiene was poor with stains and calculus.
Figure 1: Case-1: 9-year-old girl with interincisal distance of 1.4 cm

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Figure 2: Case-1: Blanching and opaque oral mucosa

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Figure 3: Case-1: Pale, stiff, atrophic and de-papillated tongue with restricted movement

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Routine hematological investigations showed hemoglobin 9.3 gm%. The patient was counseled and was advised to stop the habit of chewing supari. Oral supplements of vitamin A chewable tablets (25,000 IU) OD, Evion (Vitamin E) 400-mg capsules OD, zinc acetate syrup 5 ml bid for 3 months and oral ferrous fumarate tablets 200 mg daily for 1 month were advised along with mouth-opening exercises. The patient was advised regular checkups and was recalled after 15 days, but the patient failed to report for further follow up.

Case-2

A 13-year-old boy who was a laborer reported to the Department with a chief complaint of progressive inability to open the mouth since 1 year and severe burning sensation on taking spicy foods since 2 years. History revealed habit of chewing pan 2-3 times a day since 4 years and gutka and supari once or twice in a week. He used to chew pan/gutka for several minutes and retain it in the mouth. Family history showed parents had a habit of chewing pan for several years and peer pressure from fellow laborers encouraged him with the habit of areca nut in its different forms i.e., gutka and supari.

He was moderately built with normal general condition. On examination IID was 2.2 cm [Figure 4]. Soft tissue examination revealed blanched, opaque appearance of buccal mucosa and soft palate, tongue showed depapillation with mild blanching and movement was mildly restricted [Figure 5]. On palpation mucosa was leathery in consistency and vertical fibrous bands were appreciated in the posterior buccal mucosa near retromolar region. Hard tissue examination revealed moderate tobacco stains and calculus.
Figure 4: Case-2: 13-year-old boy with interincisal distance of 2.2 cm

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Figure 5: Case-2: Blanching of the oral mucosa

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Routine hematological investigations showed no abnormality. The patient along with his parents were counseled to immediately stop the usage of areca nut in its different forms and was advised oral prophylaxis. Oral supplements of vitamin A chewable tablets (25,000 IU) OD, Evion (Vitamin E) 400-mg capsules OD, zinc acetate syrup 5 ml bid for 3 months were prescribed. Intralesional injections of corticosteroids - injection dexamethasone 4 mg with lignocaine 2% was given weekly for 2 months in the regions with palpable fibrous bands. Each time, 2 ml of the solution was deposited around the specific region on both sides. There was a remarkable improvement in the burning sensation of the mouth and moderate improvement in the mouth opening with IID 2.5 cm. Patient was advised warm saline rinses and mouth-opening exercises.

Case-3

A 15-year-old girl reported to the Department with a chief complaint of recurrent oral ulcers since 6 months and burning sensation in the mouth particularly when eating hot and spicy foods since 1 year. She also complained of restricted mouth opening since 4 months. She reported that the ulcers were recurrent and occurred in different areas each time. History revealed chewing of sweet supari for several minutes for 6-7 times in a day since 4 years. She used to chew supari and swallow it while engaged in household work. She was unaware of the deleterious effects of areca nut chewing and thought it to be a natural product which aid digestion.

On examination IID was 2.8 cm [Figure 6]. On inspection mucosa was pale, opaque, blanched with few erythematous areas. Fibrous bands were prominent in the retro molar region with fibrosis of pterygomandibular raphe [Figure 7]. Erytematous areas were observed at the junction of hard and soft palate [Figure 8]. The faucial pillars appeared short, thick and hard on palpation. Uvula was shrunken and bud like. There was depapillation of tongue with loss of gustatory sensation.
Figure 6: Case-3: 15-year-old girl with interincisal distance of 2.8 cm

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Figure 7: Case-3: Blanching of the oral mucosa with prominent fi brosis of the pterygomandibular raphe and shrunken, bud-like uvula (arrow)

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Figure 8: Case-3: Erythematous area (arrow) at the junction of hard and soft palate on the right side

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Routine hematological investigations showed hemoglobin 9.1%. A punch biopsy from the erythematous areas on posterior buccal mucosa was taken and sent for histopathological examination. Microscopic picture with H&E stain showed epithelium with absence of rete ridges and underlying connective tissue showed hyalinization and dense collagen bundles with moderate number of chronic inflammatory cells [Figure 9]. The diagnosis of OSMF was made on clinical basis which was confirmed by histopathological examination.
Figure 9: Case-3: H&E histomicrograph showing epithelium with juxta-epithelial hyalinization and dense collagen bundles

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The patient was advised to stop the habit of chewing supari and prescribed vitamin A chewable tablets 25,000 IU OD, Evion (Vitamin E) 400-mg capsules OD, zinc acetate syrup 5 ml bid for 3 months and oral ferrous fumarate tablets 200 mg daily for 1 month and Rexidine M forte gel for local application on the erythematous areas for temporary relief. Submucous (intralesional) injections of corticosteroids - injection dexamethasone 4 mg with lignocaine 2% was given weekly for 2 months in the regions with palpable fibrous bands. The patient was also advised warm saline rinses and mouth-opening exercises. Reduction of burning sensation, erythematous areas and slight improvement in the mouth opening with IID 3 cm were noted.


   Discussion Top


Oral submucous fibrosis is a well-recognized potentially malignant condition of the oral cavity associated with betel quid (BQ), areca nut and smokeless tobacco-chewing habits. Epidemiological data and intervention studies suggest that areca nut is the main predisposing factor for OSMF. [9] Other etiological factors suggested are chillies, lime, tobacco, nutritional deficiencies such as iron and zinc, immunological disorders, and collagen disorders. [10]

The pathogenesis of the disease is not well established, but is believed to be multifactorial. Various mechanisms suggested include:

  1. Clonal selection of fibroblasts with a high amount of collagen production during long-term exposure to areca nut,
  2. Stabilization of collagen structure by catechin and tannins from BQ,
  3. Production of stable collagen (type I) by OSMF fibroblasts,
  4. Increase in collagen cross-linking by upregulation of lysyl oxidase,
  5. Deficient collagen phagocytosis, and
  6. Micronutrient and vitamin deficiencies. [11]


The molecular events in the causation of OSMF takes place through collagen production pathway and collagen degradation pathway. Synthesis of collagen is influenced by variety of mediators including growth factors, hormones, cytokines and lymphokines. Transforming growth factor-beta (TGF-β) plays a major role, it causes deposition of extracellular matrix (ECM) by increasing the synthesis of matrix proteins such as collagen and decreasing its degradation by stimulating various inhibitory mechanisms. OSMF is regarded as a collagen metabolic disorder with an overall increased collagen production and decreased collagen degradation resulting in increased collagen deposition in the oral tissues, and fibrosis due to alkaloid exposure. [12]

There are studies published in literature which reported areca nut addiction among school children in various countries such as Taiwan, Pakistan, London, Micronesia and India. [13],[14],[15],[16],[17] Very few studies were conducted on the prevalence of OSMF in the pediatric population. A recent study conducted in Karachi on OSMF found its incidence to be 6.6% among school children. [18] A high school survey reported a prevalence of OSMF to be 8.8%. [16] There are around 16 case reports of OSMF in children and adolescents in the literature [Table 1]. [16],[19],[20],[21],[22],[23],[24],[25],[26],[27],[28],[29],[30],[31],[32],[33] The age group in the pediatric population ranged from 4 to 15 years. Hayes reported first case of OSMF in a 4-year-old girl. [19] No sex predilection is reported in children but few studies reported areca nut chewing to be more prevalent in boys. [14],[17],[34]
Table 1: Summary of reported cases of OSMF in children and their chewing habits

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Areca nut is the main etiological factor for OSMF and all the three cases reported in this study have the habit of chewing areca nut in its different forms - pan, sweet supari and gutka. Sweetened versions of betel nut are sold to children as sweet supari, gua, mawa or mistee pan. [33] All the three cases in our study chewed sweet supari (sweetened version of areca nut) which has been reported to be more common in children from previous studies. [19],[20],[21],[22],[27],[29],[31],[33] Because of its sweet and pleasant taste, it is considered as a harmless mouth freshener and therefore consumed in larger amount and kept in the mouth for a longer time by the ignorant children. [35]

Gutka is a dry, relatively nonperishable commercial preparation containing areca nut, slaked lime, catechu, condiments and powdered tobacco. The same mixture without tobacco is called pan masala. Both gutka and pan masala come in attractive foil packets (sachets) and tins which can be stored and carried conveniently. Aggressive advertising, targeted at the middle class and adolescents since the early 1980s, has enhanced the sales of these products. [36] Dhariwal et al. and Chakraborty et al. both reported gutka chewing in 10-year-old boys and both the cases were influenced by peers. [28],[30] Similarly the 13-year-old boy in our study reported gutka chewing which was encouraged by his fellow laborers. Shah et al., Shirzaii M, Dhariwal et al. reported family history of chewing areca nut in their reports but only 14-year-old boy had acquired the habit of paan from parents. [22],[29],[30] Family environment plays a major role in acquiring habit and also cultural habits predispose to the condition. Factors involved in consumption of various forms of areca nut are levels of awareness, household environment, economic status, peer pressure, low cost, easy availability, etc. [31] The three cases in our series were not aware of the harmful effects of areca nut products and were of low socio-economic status.

Diagnosis of OSMF is usually based on the clinical signs and symptoms, which do not differ much in children and adults. They include burning sensation of the mouth, particularly during consumption of spicy foods and is often accompanied by the formation of vesicles and ulcerations. Gradually, patients develop stiffening of the mucosa, with a dramatic reduction in mouth opening and with difficulty in swallowing and speaking. The mucosa appears blanched and opaque with the appearance of fibrotic bands that can be easily palpated. The bands usually involve the buccal mucosa, soft palate, posterior pharynx, lips and tongue. [37] All the three cases showed stiffening of the oral mucosa and palpable fibrous bands with restricted tongue movement and reduced mouth opening. A recent study reported OSMF in five pre-school children aged 2-3 years by considering depigmentation of the oral mucosa as the earliest possible manifestation. [26]

Histologically, OSMF shows the characteristic features of severely atrophic epithelium with complete loss of rete ridges. Varying degrees of epithelial atypia may be present. The underlying lamina propria exhibits severe hyalinization, with homogenization of collagen. Cellular elements and blood vessels are greatly reduced. [38] Biopsy of the lesion is rarely performed due to the observation that such investigation leads to further fibrous scar development and worsening of the symptoms. [22],[29]

There is no known cure for OSMF. Therefore, the initial treatment starts with the cessation of the chewing habit. If this is achieved, the early lesions have a good prognosis and might regress with improvement in symptoms. Several conservative approaches have been tried with little success such as oral antioxidants, micronutrients and minerals, turmeric, pentoxyfilline, interferon gamma, submucosal steroid injections of dexamethasone, hyaluronic acid (hyalase), chymotrypsin, placental extract (placentrix), milk from immunized cows, intralesional injections of stem cells, physiotherapy, regular mouth-opening exercises and local heat therapy. Surgical care is indicated in patients with severe trismus, dysplasia or neoplasia. Surgical modalities include myotomy, coronoidectomy and simple excision of fibrous bands, mucosal or non-vascularized split thickness grafts. They have been ineffective in few cases and also have often exacerbated the condition, with added scar tissue. [39],[40]

In our case series, the first case failed to report for follow up. The second and third cases responded to initial treatment. Both the subjects stopped chewing areca nut and were given vitamin A tablets, vitamin E capsules, zinc acetate syrup for 3 months and intralesional injections of dexamethasone along with mouth opening exercises and warm saline rinses. Both the cases showed remarkable improvement in the burning sensation and mouth opening. Reduction of ulcers was noted in the third case. Vitamin A is an essential nutrient needed for normal functioning of the visual systems, growth and development, maintenance of epithelial integrity and immune functions. [41] Vitamin E is an antioxidant. Its main actions are free radical scavenging, maintenance of membrane integrity, immune function, inhibition of cancer cell growth/differentiation, cytotoxicity, inhibits mutagenicity and nitrosamine formation, inhibition of DNA and RNA, protein synthesis in cancer cells. [42] Zinc is an essential component of a large number of enzymes participating in the synthesis of carbohydrates, lipids, proteins, nucleic acids and also plays a central role in humoral and cellular immune system. Moreover zinc is the antagonist of copper and prevents its role in enzyme-induced collagen production. [43] Dexamethasone opposes the action of soluble factors released by sensitized lymphocytes following activation by specific antigens. It prevents inflammatory reaction and fibrosis by decreasing fibroblastic proliferation and depositing of collagen. [44]


   Conclusion Top


The case series shows even children and young adults are prone for OSMF and the main predisposing factor involved is the habit of areca nut chewing in its different forms. The chewed product was sweet supari, areca nut in its sweetened and flavored form and is thought as a mouth freshener by many. There is no definitive cure and the only option is prevention which can be achieved by avoiding the habit of chewing areca nut products especially in children. It is the responsibility of the parents, teachers, dentists, pediatricians, other health care professionals and the general population to spread awareness of the potential risk factors involved in causing morbidity and mortality of this new but avoidable menace in children and protect them.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9]
 
 
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