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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 19  |  Issue : 3  |  Page : 177-180

Oral submucous fibrosis: A rare case in a child and its management


1 Department of Paediatric & Preventive Dentistry, Faculty of Medicine, A.M.U., Uttar Pradesh, India
2 Department of Pediatric and Preventive Dentistry, Dr Ziauddin Ahmad Dental College and Hospital, Faculty of Medicine, Aligarh Muslim University, Aligarh, Uttar Pradesh, India

Date of Submission17-Apr-2022
Date of Decision15-May-2022
Date of Acceptance19-May-2022
Date of Web Publication14-Sep-2022

Correspondence Address:
Mohammad Kamran Khan
BDS, MDS (AMU, Aligarh), Hamdard Nagar.A, Civil Line, Aligarh, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/tdj.tdj_11_22

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  Abstract 


Oral submucous fibrosis (OSMF) is a potentially malignant condition mainly found in people of Indian subcontinent. Although, mainly adults affected with OSMF have been seen in literature, but its incidence is increasing in pediatric population in recent years. The increased trend of habit of chewing the areca nut in children has been associated with this condition. There is paucity of published literature regarding OSMF in pediatric age group. Hence, the current article presents a case report of OSMF in an 8.5-year-old boy having chronic habit of chewing flavored supari (areca-nut) five to seven packets per day in the last 3 years. Patient was managed by counseling, medicinal treatment and oral physiotherapy. Follow-up evaluation of patient showed the significant reduction in signs and symptoms of OSMF.

Keywords: areca nut, betel nut, child, flavored supari, oral premalignant condition, oral submucous fibrosis, pediatric oral submucous fibrosis, restricted mouth opening


How to cite this article:
Khan MK, Jindal MK. Oral submucous fibrosis: A rare case in a child and its management. Tanta Dent J 2022;19:177-80

How to cite this URL:
Khan MK, Jindal MK. Oral submucous fibrosis: A rare case in a child and its management. Tanta Dent J [serial online] 2022 [cited 2023 Jan 30];19:177-80. Available from: http://www.tmj.eg.net/text.asp?2022/19/3/177/356077




  Introduction Top


Oral submucous fibrosis (OSMF) is a potentially malignant condition mainly found in Indian subcontinent with a prevalence rate of 0.5% and mainly affects females in a ratio of 3:1 [1].

OSMF is a chronic disorder characterized by progressive fibrosis of the mucosa of oral cavity, oropharynx, and esophagus, which results in difficulty in eating spicy foods, burning sensation of the mouth, xerostomia, reduction in mouth opening, restricted tongue movements and, also causes dysphagia and impairment of phonation [2]. OSMF has 7–13% malignant transformation rate [3].

OSMF has an uncertain etiology [1]. Various etiological factors for OSMF have been reported in literature like areca nut chewing habit, capsaicin in chili, autoimmunity, hormones, nutritional deficiencies (minerals and vitamins), and genetic factors (HLA 10, DR3, DR7) [3]. However, habit of areca nut chewing in any form is believed as the chief causative agent for OSMF as it contains great amount of copper that accelerates the lysyl oxidase activity resulting in fibrosis of mucosa [4].

Although, majority of the adults of age group 20–40 years affected with OSMF have been seen in literature [5], but in pediatric population with OSMF in India, the incidence rate of 0.2–1.2% has been reported [6]. This pathological condition has started occurring relatively more frequently in children in recent years and the possible reason is the increased habit of areca nut chewing [1].

Still there is a paucity of literature about OSMF in pediatric age group. The current article presents a case report of OSMF in an 8 year-old healthy boy.

This case-report has been written as per the CARE checklist case report guidelines.


  Case presentation Top


An 8.5-year-old boy from a village reported to the Outpatient Department of Pediatric Dentistry with chief complaints of reduced mouth opening and intolerance to spicy foods for last 8 months. Patient's parents revealed the history of habit of chewing the flavored areca nut (supari) five to seven packets per day for the past 3 years. Medical history was not significant. Personal and family history revealed that his friends and elder family members also had a habit of chewing areca nut. Patient was from low socioeconomic group.

General examination revealed normal health condition [Figure 1]. On extra-oral examination, mouth opening was restricted with inter-incisal distance of 1.8 cm [Figure 2]. On intra-oral examination, buccal mucosa and soft palate was pale, opaque and blanched [Figure 3]a, [Figure 3]b, [Figure 3]c. Restricted tongue movements on protrusion were present (Fig. 3d). The buccal mucosa was inelastic, with the presence of palpable vertical fibrous bands. Poor oral hygiene was present with high plaque and calculus score with generalized stains on surfaces of mixed dentition.
Figure 1: Photograph showing normal facial features.

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Figure 2: Photograph showing the restricted mouth opening measured as 18 mm.

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Figure 3: Intra-oral photograph showing pallor, opaque, blanching of (a) soft palate, facial pillars; buccal mucosa (b) right side (c) left side; (d) restricted tongue movement.

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Orthopantomogram [Figure 4] showed normal radiographic findings. Complete blood count test was advised to assess the hematological status of the patient and was found normal.
Figure 4: Orthopantomogram radiograph showing the normal radiographic features.

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Based on the patient's history and examination findings, a clinical diagnosis of OSMF in stage-II (Pindborg's classification of stages of OSMF based on its clinical features) [7] was established.

Differential diagnosis included the OSMF, localized Scleroderma, Cranio-facial syndrome microsomia, TMJ ankylosis. But the chewing habit of areca nut and typical sign and symptoms confirmed the provisional diagnosis of OSMF.

Patient's parents were informed and explained about the diagnosis of OSMF. Treatment plan was made as per the patient's clinical condition and his sign and symptoms. The informed consent was taken from the patient's parents prior to commencement of the proposed treatment plan.

Counseling, medicinal treatment and oral physiotherapy exercises constituted the treatment plan for managing the present patient.

First, a counseling of patient and parents was done regarding the potentially malignant condition premalignant condition of OSMF and deleterious effects and consequences of habit of chewing areca nut. Patient and parents were instructed to discontinue the habit. Diet counseling was given emphasizing on having healthy nutritious meals, but spicy hot food was advised to avoid. Physiotherapy oral exercises using the wooden ice-cream stick and blowing the balloons several times a day were advised to increase the mouth opening. Warm saline rinses were also advised. Medicinal treatment included multivitamin capsule (Vitamin-B complex with vitamin-A,-C and -E) once a day for 1 month. Oral prophylaxis with scaling was done. Oral hygiene instructions were given to the patient. Patient was recalled after 1 month for follow-up.

On follow-up visit after 1 month, patient was evaluated. A remarkable improvement was noticed in patient condition. Parents revealed that patient had discontinued habit of chewing eating areca nut and relieved from burning sensation on eating spicy foods. On clinical examination, mouth opening was significantly improved by 3 mm (inter-incisal distance = 21 mm) [Figure 5]a. Opacity, paleness and blanching of oral mucosa were found slightly reduced [Figure 5]b. Patient was advised to continue multivitamins for another 2 weeks. Physiotherapy exercises and warm-saline rinses were advised to continue. Healthy nutritious diet has been advised. Patient has been kept for long term follow-up at 3 months interval.
Figure 5: Follow-up photograph showing (a) increased mouth opening to 21 mm; (b) reduction in paleness, opacity and blanching of buccal mucosa.

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  Discussion Top


OSMF is a chronic condition with obscure etiology, however areca nut chewing habit is strongly associated as causative factor, which results in functional morbidity and have accentuated risk of malignancy [8]. Areca nut is rich in alkaloids, nitrosamines, and tannins [1]. Arecoline is the major alkaloid in areca nut which is carcinogenic [1]. The contents of areca nut have genotoxic effects on oral mucosal fibroblast which induces its proliferation and collagen synthesis [1]. Harvey et al. [9] reported in their research that alkaloids of betel-nut increase collagen synthesis by 170% by induced fibroblasts in OSMF. The progressive fibrosis of lamina propria of mucosal epithelium causes inelasticity and stiffing of oral mucosa and hence, leads to reduction in mouth opening [1].

Histological features of OSMF include absence of rete ridges, atrophic epithelium, hyalinization and homogenization of collagen in the underlying lamina propria. Blood vessels and cellular elements are also decreased [1],[8].

In old literature, OSMF has been observed in adults, but in recent years this condition is being reported in children also [10]. However, still there is a paucity of literature regarding OSMF in pediatric age group. Hence, this present article will add another new case report of OSMF in a pediatric age group in the literature.

The consumption of using areca and its production nut have increased [8]. The colorful attractive packets containing scented and flavored areca nut attract school-aged children and adolescents. The easier availability around schools is making children more addicted towards consuming areca nut [1],[11]. The possibility of addiction and deleterious consequences of areca-nut chewing habit are increased when an individual is indulged in habit at younger age [12]. Mostly parents and children are unaware about the ill-effects of habit of areca nut chewing [1]. Majority of children belonging to low socio-economic status have been reported with OSMF [1]. In the present case report also, the patient was belonging to low socioeconomic group.

Definitive cure of OSMF is still not known [8]. Hence, prevention is believed to be the best measure to decline the incidence of OSMF in children [8]. Prevention can be accomplished by educating and motivating children, parents, and teachers about the OSMF, its association with harmful habit of areca nut consumption and its malignancy transformation potential. Management of OSMF starts with cessation of habit of areca nut chewing. The early lesions of OSMF have a good prognosis along with improvement in its symptoms, only if this habit is stopped earlier [8].

The treatment approaches have been used for OSMF are the micronutrients, minerals, oral antioxidants, pentoxifylline, turmeric, interferon-gamma, submucosal steroid injections of dexamethasone, hyaluronic acid (hyalase), placental extract (placentrix), chymotrypsin, intralesional injections of stem cells, oral physiotherapy, regular mouth-opening exercises and local heat therapy [1],[8]. In patients with severe OSMF, surgical care is indicated such as, myotomy, simple excision of fibrous bands, coronoidectomy and mucosal or nonvascularized split thickness grafts [8]. Management of OSMF requires combination of counseling, medicinal and physiotherapy exercises because of its multifactorial etiology [8],[10]. In present case report, patient was managed with combination of counseling, cessation of habit, multivitamin capsules (vitamin B-complex, vitamin A, C and E, antioxidants) for 1 months, along with oral physiotherapy exercises and warm-saline rinses which resulted in reduction of burning sensation and increased mouth opening satisfactorily in initial 1 month follow-up evaluation.

Biopsy is usually not advised because it results in further formation of fibrous scar which may worsen the condition of OSMF [9],[13]. In this case report also, biopsy was not suggested as the patient presented the characteristic features of OSMF along with positive history of areca nut chewing and normal radiographic findings confirmed the clinical diagnosis of OSMF.

Adequate nutrition to children in growing age is influenced due to morbidity of OSMF. Growth and quality of life of growing children may be affected if such premalignant condition is not prevented and treated on right time. Prevention and early management are crucial to reduce the prevalence of OSMF in children.


  Conclusion Top


Incidence of OSMF in children can be prevented by conducting the dental awareness camps in schools and in societies to educate the children, parents and teachers about the OSMF and the adverse effects of consuming areca-nut. Early intervention of such premalignant condition is pivotal for treating the morbidity and for preventing the malignant transformation of OSMF.

Acknowledgements

Authors' contributions: Mohammad K. Khan was involved in clinical examination, diagnosis, preparing a treatment plan and clinical management and follow-up evaluation of the patient. He prepared this manuscript. Mahendra K. Jindal was involved in clinical examination, diagnosis, preparing a treatment plan and clinical management.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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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; 3497136:1–6.  Back to cited text no. 1
    
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Rajendran R. Oral submucous fibrosis-etiology, pathogenesis and future research. Bull World Health Organ 1994; 72:985–996.  Back to cited text no. 2
    
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Tilakaratne WM, Klinikowski MF, Saku T, Peters TJ, Warnakulasuriya S. Oral submucous fibrosis: review on aetiology and pathogenesis. Oral Oncol 2006; 42:561–568.  Back to cited text no. 3
    
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Bari S, Metgud R, Vyas Z, Tak A. An update on studies on etiological factors, disease progression, and malignant transformation in oral submucous fibrosis. J Canc Res Therapeut 2017; 13:399–405.  Back to cited text no. 4
    
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Pindborg JJ, Sirsat SM. Oral submucous fibrosis. Oral Surg Oral Med Oral Pathol 1966; 22:764–779.  Back to cited text no. 5
    
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Jayanthi V, Probert CSJ, Sher KS, Mayberry JF. Oral submucous fibrosis: a preventable disease. Gut 1992; 33:4–6.  Back to cited text no. 6
    
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Gupta H, Grover N, Tyagi N, Misra A. Classification systems in oral submucous fibrosis patients: a review. TMU J Dent 2018; 2:13–19.  Back to cited text no. 7
    
8.
Kariya P, Khobragade V, Sura S, Singh S. No age predilection for a disease like OSMF. A case report of 5-year-old child. J Oral Biol Craniofac Res 2020; 10:153–157.  Back to cited text no. 8
    
9.
Harvey W, Scutt A, Meghji S, Canniff JP. Stimulation of human buccal mucosa fibroblasts in vitro by betel-nut alkaloids. Arch Oral Biol 1986; 31:45–49.  Back to cited text no. 9
    
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Rai A, Jain A, Arora A, Motiwale T. Oral submucous fibrosis in children: an alarming condition and challenges in management. BMJ Case Rep 2021; 14:e238718.  Back to cited text no. 10
    
11.
Auluck A, Hislop G, Poh C, Zhang L, Rosin MP. Areca nut and betel quid chewing among South Asian immigrants to Western countries and its implications for oral cancer screening. Rural Remote Health 2009; 9:1118–1127.  Back to cited text no. 11
    
12.
Warnakulasuriya S, Trivedy C, Peters TJ. Areca nut use: an independent risk factor for oral cancer. BMJ 2002; 324:799–800.  Back to cited text no. 12
    
13.
Canniff JP, Harvey W, Harris M. Oral submucous fibrosis: its pathogenesis and management. Br Dent J 1986; 160:429–434.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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