Oral Submucous Fibrosis Secondary to Buccal Mucosal Graft for Urethroplasty

Abstract

Oral submucous fibrosis is a collagen disorder commonly seen in the population with areca nut chewing habit, which is more prevalent in the Indian subcontinent. Various studies have suggested a multifactorial origin with a high incidence of the disease in association with consumption of the areca nut. However, it has never reported being secondary to buccal mucosal graft procedures. We are presenting a case of submucous fibrosis secondary to buccal mucosal graft for urethroplasty.

Background

Oral submucous fibrosis (OSMF) is an insidious disease affecting the oral cavity, pharynx, and upper digestive tract. It is an established precancerous condition [1]. Its aetiology is linked directly to betel nut usage, common to the Indian subcontinent and southeast Asia [2]. Though autoimmune and genetic etiologies are proposed, betel nut usage is the primary cause of oral submucous fibrosis. Although mucosal fibrosis is a common complication after harvesting buccal mucosal graft for urethroplasty, it is least described in the literature as a cause of OSMF. We are presenting a case of OSMF secondary to buccal mucosal graft for urethroplasty.

Case Presentation

A 55-year-old male patient presented to our Out-Patient-Department, with a chief complaint of progressive trismus and inability to chew. He noted that his mouth opening decreased over the last two years without oral burning, irritation, and any vesicle formation. He denied any deleterious habits of betel nut and tobacco chewing. The patient had a history of urethroplasty using a buccal mucosal graft harvested from his right cheek for urethral stricture two years ago. As the donor site (right buccal mucosa) healing progressed, his mouth opening reduced progressively due to fibrosis.

On examination, the patient was well-nourished without any remarkable extraoral changes. Maximal interincisal opening (MIO) was 16 mm (Fig. 1). Both maxillary and mandibular vestibules were extremely tight on the right side, with soft tissue bands running through both vestibules. The unattached gingiva in both vestibules appeared pale and indurated. (Fig. 2) On further examination, the buccal mucosa was firm leathery and indurated on the right side, with numerous soft-tissue bands running vertically. Left buccal mucosa and oropharynx appeared near normal.

Fig. 1.

Restricted mouth opening with maximum interincisal opening of 16 mm

Fig. 2.

Blanched right buccal mucosa and retromolar trigone

A preliminary diagnosis of surgical site fibrosis was thought of. However, the presence of well-formed fibrous bands running across the full length of the buccal mucosa, obliteration of the vestibules and progressive decrease in the mouth opening led us to the final diagnosis of oral submucous fibrosis.

The patient was started on treatment of submucous fibrosis as per out institutional protocol wherein we inject intralesional triamcinolone acetate on weekly basis of 5 weeks, following which if the mouth opening persists to be decreased, we treat the patient surgically by intraoral band release with interposition with bilateral nasolabial flap.

Discussion

Oral Submucous fibrosis is an insidious, chronic disease that may affect any part of the oral cavity and sometimes the pharynx, leading to stiffness of the oral mucosa and causing trismus [1–3] This disease is most frequently found in India and is not uncommon in Southeast Asia. Betel nut chewing appears to be the main factor correlating with this disease, but other uncommon cause may also contribute to the development of this disease as in our case.

The use of autologous free buccal mucosal grafts has been described for the reconstruction of urethral strictures [4–6] The buccal mucosa has properties that make it the graft of choice in many circumstances. Oral mucosa and penile skin are the most commonly used [6] Since the first reported use of buccal mucosa in urethroplasty for adults, by El-Kasaby et al. [7], increasingly many urologists are using it. Indeed, since 1995, cheek mucosa has been used more often than penile skin [8] Buccal mucosa gives excellent results because it has a high capillary density, is resilient and easily harvested [9] Unfortunately, few reports on oral complications after cheek mucosal harvesting and cite only minor complications.

Restriction of mouth opening seems to be a common problem and, in the first week, was seen in 10 of 12 patients in the study of Tolstunov et al. [9], and in half the patients (n = 35) in the study by Dublin et al. [10] In this case, the patient did not follow on regular intervals and presented two years after buccal mucosal graft harvest as trismus progressed. The patient had not done any mouth opening physiotherapy after the surgery.

The literature contains few references to the successful treatment of OSMF. Various treatments to improve mouth opening have been attempted, including surgical excision of the fibrotic bands, but have been reported as generally unsatisfactory [11, 12] Yen was the first to succeed in coveting the buccal defect with a split-thickness skin graft in treating a case of OSMF [13] After that, various autologous grafts, including buccal fat pad, full-thickness skin graft, abdominal fat graft and different types of nasolabial flaps, have been tried. Khanna & Andrade reported the new surgical technique of covering the buccal defects with a palatal island flap in combination with temporalis myotomy and coronoidectomy [14] Early and intensive postoperative mouth-opening exercises are very important to achieve adequate mouth opening after any surgical procedure for the treatment of OSMF.

Conclusion

• All patients with trismus and submucous fibrosis may not have betel nut chewing habit.

• Past surgical history is also important for the diagnosis of a disease.

• Aggressive mouth opening physiotherapy is essential for patients undergoing buccal mucosal graft harvesting.

Declarations

Conflict of interest

None of the authors has any financial or personal interest associated with this article. No funding has been received by any author in relation to this study.

Ethical approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed consent

A written informed consent was obtained from all individual participants included in the study.