Oral submucous fibrosis in a youngpatient M. J. Yazdanpanah, M. Banihashemi, F. Pezeshkpoor, S. Famili, P. Layegh, M. Katebi, and H. Hamidi Summary - A seventeen-year-old male presented with a restriction in the oral cavity. The oral mucosa was white and he could not protrude his tongue. For 4 years he had habitually held a powdery material containing betel nut in the oral vestibule for several minutes a day. A biopsy of the labial mucosa was carried out and a diagnosis of oral submucous fibrosis was confirmed. This is a premalignant condition. Oral submucous fibrosis is very rare in young patients. Case report This case report relates to a seventeen-year-old male referred to Ghaem Hospital in Mashhad, Iran, with progressive inability to open his mouth. This condition had manifested itself 2 years earlier and was progressive. On examination, the inter-incisal opening was approximately 20 mm. The oral mucosa was blanched in appearance and he could not protrude his tongue (Fig. 1). Questioning revealed that he was in habit of holding a powdery material (containing betel nut, catechu, tobacco, lime, spices, and flavors) in the oral vestibule for several minutes a day starting 4 years earlier, that his symptoms started 2 years later, and that they were progressive. A biopsy of the labial mucosa was carried out and a histological diagnosis of oral submucous fibrosis was made (Figs. 2, 3). The patient was otherwise normal upon physical examination. Laboratory investigations such as complete blood count and liver and kidney function tests were within normal ranges. Fig. 1. Blanched appearance of mucosa. KEY WORDS oral submucous fibrosis, betel nut Fig. 2. Microscopic view of histological section of OSMF lesions; chronic inflammatory reaction and deep hypovascular fibrosis under squamous epithelium with focal atrophy to hyperplasia, H&E staining, X100. Fig. 3. Microscopic view of OSMF lesion; A: Hypovascular hyalinized collagenous fibrosis in superior to inferior dermis, B: Note entrapment of striated muscle fascicles by deep and dense fibrosis, H&E staining, X400. Discussion Oral submucous fibrosis (OSMF) is a chronic disease with chronic inflammation and fibrosis of submucous tissues, causing restriction of the mouth opening (1). Areca nut chewing has a significant role in its etiology (2). Cases have been reported worldwide wherever Asians migrate, but it occurs most commonly in India and southeast Asia (3, 4). The majority of OSMF cases belong to the 20- to 40-year-old age group and there is a male-to-female ratio of 1.8 to 2:1 (5—7). Most OSMF cases occur in lower socioeconomic groups (8). In the pathogenesis of OSMF, it is suggested that a multifactorial mechanism is at work, including areca nut chewing, the ingestion of spicy red pepper, nutritional deficiency including vitamins and trace elements, hypersensitivity to various dietary constituents, and genetic and immunological susceptibility (9—13). References - Histopathology findings are the mainstay of diagnosis at present. The principal features of OSMF are less vascularized collagenous submucosa with a range of atrophy in the neighboring striated muscle fibers, mild to moderate chronic inflammation, and epithelial changes consisting of atrophy and a variable degree of dysplasia (14—17). The important pathological feature of OSMF is submucosal accumulation of collagen leading to epithelial atrophy (18, 19). It has been discovered that exposure of buccal mucosal fibroblasts to alkaloids may cause aggregation of collagen (20). Oral submucous fibrosis is a chronic condition of the oral mucosa and oropharynx with the potential for malignant transformation. Squamous cell carcinoma may occur in 7.6% of cases (9). An interesting point in our patient is his relatively young age. Proper preventive measures such as public education must be taken to reduce this serious disease. 1. WHO. Meeting report. Control of oral cancer in developing countries. WHO Bull. 1984;62:617. 2. Babu S, Bhat RV, Kumar PU, et al. A comparative clinico-pathological study of oral submucous fibrosis in habitual chewers of panmasala and betel quid. Clin Toxicol. 1996;34:317-22. 3. Tang JG, Jian XF, Gao ML, et al. Epidemiological survey of oral submucous fibrosis in Xiangtan city, Hunan Province, China. Community Dent Oral Epidemiol. 1997;25:177-80. 4. Shah B, Lewis MA, Bedi R. Oral submucous fibrosis in an 11-year-old Bangladeshi girl living in the United Kingdom. Br Dent J. 2001;191:130-2. 5. Sirsat SM, Khanolkar VR. Submucous fibrosis of the palate in diet-preconditioned Wistar rats: Induction by local painting of capsaicin-an optical and electron microscopic study. Arch Pathol. 1960;70:171-9. 6. Shah N, Sharma PP. Role of chewing and smoking habits in the etiology of oral submucous fibrosis: A case control study. J Oral Pathol Med. 1998;27:475-9. 7. Wahi PN, Kapoor VL, Luthra UK, et al. Submucous fibrosis of the oral cavity: 2. Studies on epidemiology. Bull World Health Organ. 1966;35:793-9. 8. Ramanathan K. OSMF-An alternative hypothesis as to its causes. Med J Malaysia. 1981;36:243-5. 9. Aziz SR. Oral submucous fibrosis: an unusual disease. J N J Dent Assoc. 1997;68:17-19. 10. Liao PH, Lee TL, Yang LC, et al. Adenomatous polyposis coli gene mutation and decreased wild-type p53 protein expression in oral submucous fibrosis: a preliminary investigation. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2001;92:202-7. 11. van Wyk CW, Grobler-Rabie AF, Martell RW, et al. HLA-antigens in oral submucous fibrosis. J Oral Pathol Med. 1994;23:23-7. 12. Paul RR, Chatterjee J, Das AK, et al. Altered elemental profile as indicator of homeostatic imbalance in pathogenesis of oral submucous fibrosis. Biol Trace Elem Res. 2002;87:45-56. 13. Paul RR, Chatterjee J, Das AK, et al. Zinc and iron as bioindicators of precancerous nature of oral submucous fibrosis. Biol Trace Elem Res. 1996;54:213-30. 14. Rosai J. Rosai and Ackerman's surgical pathology. 9th ed., vol. 1. Edinburgh: Mosbey; 2004. 15. Neville BW, Damm DD, Allen CM, et al. Oral & maxillofacial pathology. 2nd ed. Philadelphia: W. B. Saunders Company; 2002. 16. Canniff JP, Harvey W, Harris M. Oral submucous fibrosis: its pathogenesis and management. Br Dent J. 1986;160:429-34. 17. Pindborg JJ, Sirsat SM. Oral submucous fibrosis. Oral Surg Oral Med Oral Pathol. 1966;22:764-79. 18. Hardie J. Oral submucous fibrosis. A review with case reports. J Can Dent Assoc. 1987;53:389-93. 19. Huang IY, Shieh TY. Collagen content and types in oral submucous fibrosis. Gaoxiong Yi Xue Ke Xue Za Zhi. 1989;5:162-71. 20. Harvey W, Scutt A, Meghji S, et al. Stimulation of human buccal mucosa fibroblasts in vitro by betel-nut alkaloids. Arch Oral Biol. 1986;31:45-9. autors' Mohammad Javad Yazdanpanah, MD, Associate Professor of Dermatology, addresses Department of Dermatology, Ghaem Hospital, Mashhad University of Medical Sciences, Mashhad, Iran, corresponding author, Tel.: +98 511 8012861, +98 511 8409612, E-mail: yazdanpanahmj@mums.ac.ir Mahnaz Banihashemi, MD, Associate Professor of Dermatology, same address Fakhrozaman Pezeshkpoor, MD, Associate Professor of Dermatology, same address Soroor Famili, MD, Associate Professor of Dermatology, same address Pouran Layegh, MD, Associate Professor of Dermatology, same address Mehrdad Katebi, MD, Assistant Professor of Pathology, same address Hamid Hamidi, MD, Resident of Dermatology, same address