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Table of Contents
Year : 2021  |  Volume : 23  |  Issue : 4  |  Page : 138-143

Otological manifestations in oral submucous fibrosis

1 Department of Otorhinolaryngology and Head and Neck Surgery, IMS and SUM Hospital, Siksha “O” Anusandhan University, Bhubaneswar, Odisha, India
2 Department of Oral Pathology and Microbiology, IDS and SUM Hospital, Siksha “O” Anusandhan University, Bhubaneswar, Odisha, India

Date of Submission13-Sep-2021
Date of Decision04-Oct-2021
Date of Acceptance09-Oct-2021
Date of Web Publication10-Dec-2021

Correspondence Address:
Prof. Santosh Kumar Swain
Department of Otorhinolaryngology and Head and Neck Surgery, IMS and SUM Hospital, Siksha “O” Anusandhan University, K8, Kalinga Nagar, Bhubaneswar - 751 003, Odisha
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/sjoh.sjoh_39_21

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Oral submucous fibrosis (OSMF) is a chronic and progressive disease where fibrous bands formed in the oral mucosa, resulting in severe restriction of movements of the jaw and also tongue. OSMF is one of the commonly seen potentially malignant disorders which can cause severe morbidity. It is prevalent in the Indian subcontinent. The exact etiology of OSMF is not known. Chronic irritation of the oral mucosa is thought to be the underlying etiology for OSMF. The common irritants are betel nut, tobacco, and chilies. The pathological changes not only affect mucosa and submucosa of the oral cavity and pharynx but also affect deeper parts with the involvement of underlying muscles. Atrophic and degenerative changes in the tubal and paratubal muscles of the eustachian tube, leading to eustachian tube dysfunctions. The eustachian tube dysfunction in OSMF results in different otological symptoms such as hearing impairment, otalgia, and fullness in the ear. Dysfunction of the eustachian tube in OSMF may be due to fibrosis in the palatal muscles, resulting in a conductive type of hearing loss. While treating OSMF, otological manifestations such as eustachian tube dysfunctions and hearing impairment have to be kept in mind and vice versa because managing hearing disability without taking care of OSMF will not be successful. All literature related to otological manifestations in OSMF were identified through Scopus, Google Scholar, Medline, and Pub Med and analyzed individually. This review article discusses the etiopathology, epidemiology, clinical manifestations, and management of the OSMF with otological manifestations.

Keywords: Eustachian tube, hearing impairment, oral submucous fibrosis, otological manifestations

How to cite this article:
Swain SK, Debta P. Otological manifestations in oral submucous fibrosis. Saudi J Otorhinolaryngol Head Neck Surg 2021;23:138-43

How to cite this URL:
Swain SK, Debta P. Otological manifestations in oral submucous fibrosis. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2021 [cited 2022 Oct 4];23:138-43. Available from: https://www.sjohns.org/text.asp?2021/23/4/138/332094

  Introduction Top

Oral submucous fibrosis (OSMF) is a chronic insidious disease, involving any part of the oral cavity and sometimes the pharynx.[1] It is usually recognized as a collagen disease with great similarity to morphea or localized scleroderma.[1] In OSMF, sometimes vesicles formation occurs in the mucosal lining of the oral cavity which results associated with a juxtaepithelial inflammatory reaction followed by fibroelastic changes at the lamina propria along with epithelial atrophy, resulting in trismus and inability to eat food materials.[2] The exact cause for OSMF is still obscure, but several factors such as tobacco, chewing betel nut, smoking, and consuming chillies are contributory.[3] OSMF causes progressive fibrosis in the mucus membrane of the oral cavity, mainly buccal mucosa, lip mucosa, soft palate, and anterior pillars. Rarely, it affects the mucosal lining of the pharyngeal wall or vocal folds but can affect the  Eustachian tube More Details.[4] The involvement of muscles of the soft palate may affect the eustachian tube and result in otological manifestations such as eustachian tube dysfunctions and hearing impairment.[5] There is a paucity of information or studies related to OSMF with otological manifestations in the medical literature. This review article discusses the details of epidemiology, etiopathology, clinical manifestations, otological symptoms, diagnosis, and treatment of OSMF with otological manifestations.

  Methods of Literature Search Top

Multiple systematic methods were used to find current research publications on orals submucous fibrosis causing otological manifestations or hearing loss. We started by searching the Scopus, Pub Med, Medline, and Google Scholar databases online. A search strategy using PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analysis) guidelines was developed. This search strategy recognized the abstracts of published articles, while other research articles were discovered manually from the citations. Randomized controlled studies, observational studies, comparative studies, case series, and case reports were evaluated for eligibility. There were total numbers of articles 72 (30 case reports; 34 cases series; and 8 original articles). This paper focuses only on otological manifestations in patients with OSMF. This paper examines the epidemiology, etiopathogenesis, clinical manifestations, diagnosis, and treatment of OSMF causing otological manifestations. This analysis provides a foundation for future prospective trials in OSMF and otological manifestations. It will also serve as a catalyst for additional study into OSMF causing otological manifestations and allowing early detection and treatment.

  Epidemiology Top

OSMF was first described by Schwartz in 1952 among five East African women of Indian origin.[6] This was followed by the first documentation in India in 1953 in quick succession.[7] The prevalence of OSMF is between 0.03% and 3.2% in India.[7],[8] A clinical condition resembling OSMF was documented as early as 600 BC by Sushruta, and it was named VIDARI with features of progressive narrowing of the oral cavity, depigmentation of the oral mucosa, and pain on taking food.[9] OSMF is predominantly found in Southeast Asia and the Indian subcontinent with few cases documented from South Africa, Greece, and the United Kingdom.[10] The increasing prevalence of OSMF is due to the upsurge of popularity of commercially made areca nut and tobacco preparations such as gutkha, pan masla, mawa, and flavored supari.[11] The Worldwide estimation of OSMF from 1996 was about 2.5 million people.[12] However, the study showed that more than 5 million people were suffering from OSMF in 2002.[13] In addition, up to 20% of the world's population are consuming betel nut in some form, so the prevalence of OSMF is higher than that documented in the literature.[14] The malignant transformation rate in OSMF is about 4.5%–7.6%.[15] The premalignant nature of OSMF was first documented by Paymaster, who described the occurrence of squamous cell carcinoma in one-third of patients with OSMF.[15],[16]

One study on OSMF with hearing loss reported that hearing loss was normal in 67%, mild hearing loss in 22%, and moderate mixed hearing loss in 11% with no statistical significance between grades of OSMF and hearing loss.[17]

  Etiopathology Top

The etiology for OSMF is considered to be multifactorial.[18] There are severe factors such as betel nut alkaloids, capsaicin, autoimmunity, hypersensitivity, genetic predisposition, chronic Vitamin B-complex deficiency, and iron deficiency result in OSMF.[19] Many epidemiological studies which included case series reports, large cross-sectional surveys, case–control studies, cohort, and intervention studies have shown the areca nut as the major causative agent for OSMF.[20] Areca nut chewing is a common practice and still a popular habit in the Indian subcontinent. Areca nut mainly consists of arecoline and tannin. These chemicals interfere with the molecular process of deposition or degradation of collagen which causes progression of fibrosis submucosally in the oral cavity and pharynx. During fibrogenesis, transforming growth factor β is responsible for increased production of collagen and reduced collagen degradation in OSMF.[21] The overexpression of connective tissue growth factor has been associated with fibrosis in OSMF.[22] OSMF may affect the nasopharyngeal orifice of the eustachian tube and paratubal muscles, so result in abnormal functions of the eustachian tube. There are several researchers have described the pathological changes in the soft palate. Once the soft palate muscles such as tensor palatine and levator veli palatine are affected by OSMF along with the involvement of them cause dysfunction of the eustachian tube along with the involvement of soft palate muscles such as tensor palatine and levator veli palatine and any involvement of other muscles such as paratubal muscles and accessory muscles of the eustachian tube. Fibrosis results in narrowing of the eustachian tube, which causes in the retracted tympanic membrane and otitis media with effusion. The eustachian tube dysfunction can cause chronic otitis media which manifests hearing impairment in patients with OSMF.[23]

  Eustachian Tube Dysfunction in Oral Submucous Fibrosis Top

Physiologically, the closing and opening of the eustachian tube are important for middle ear function. Normal opening of the eustachian tube usually equalizes the atmospheric pressure in the middle ear cleft.[24] Closing of the eustachian tube protects the middle ear cleft from unwanted pressure fluctuations and loud sounds. Dysfunction of the eustachian tube may cause pathological changes in the middle ear cleft which result in different otological manifestations.[25] The eustachian tube connects the nasopharynx to the middle ear. Tensor veli palatine and levator veli palatine muscles are two important muscles that are attached to the soft palate and eustachian tube.[26] OSMF causes ultrastructural changes in the muscle fibers of the palate and causes severe degenerative changes in a high proportion of OSMF patients.[25] Defective patency of the eustachian tube due to the involvement of parapalatal muscle fibrosis, tensor veli palatine, and levator veli palatine muscles which result in discomfort in ear and mild-to-moderate conductive hearing loss in OSMF patients.[27]

  Clinical Manifestations Top

OSMF is a well-established potentially malignant disorder of the oral cavity with the characteristic of fibrosis of the mucosal lining at the upper digestive tract involving the oral cavity, oropharynx, and sometimes the upper part of the esophagus.[28] OSMF is seen at any age group but most commonly in adolescents and adults, particularly between 16 and 35 years.[29] OSMF patients often complain of soreness in the mouth, intolerance to spicy/chilies foods, burning sensation in the oral cavity, ulceration in the oral cavity mucosa, and unable to protrude the tongue outside and also unable to open the mouth completely.[30] On examination of the oral cavity, there is blanching of the oral mucosa and palpable fibrous bands are seen [Figure 1]. Blanching of the lining of the mucosa in the oral cavity has been recognized as an early sign of OSMF. Blanching is persistent as a white marble-like appearance of the oral mucosa.[31] There may be inflammatory erythematous areas and ulcerations found in the oral mucosa. Dryness in the oral cavity may be found with the absence of salivary pooling in the floor of the mouth. OSMF causes difficulty in speech and swallowing throat pain and ears and also causes hearing impairment.[32] In the advanced form of OSMF, fibrous bands are seen in buccal mucosa running in the vertical direction. Faucial pillars and palate are first involved followed by gradual impairment of tongue movement and difficulty in opening of the mouth.
Figure 1: Blanching of the oral mucosa and fibrous bands in oral cavity of OSMF patient

Click here to view

Based on the clinical manifestations, OSMF is graded into Grade I, II, and III.[32] Early OSMF (Grade I) shows clinical features of burning sensation to hot and spicy food, blanching, palpable fibrosis in buccal mucosa and faucial pillars, soft palate, and pterygomandibular raphae. The mouth opening in Stage I OSMF is about 25–35 mm. In moderate OSMF (Grade II), patients present with burning sensation to hot and spicy food, palpable fibrosis in the buccal mucosa, pterygomandibular raphae, faucial pillars, soft palate, and the floor of mouth and tongue. Patients of Grade II OSMF also present with restricted tongue movement, loss of flexibility of buccal mucosa, and mouth opening is approximately 15 mm to 25 mm. In severe types of OSMF (Grade III), patients present with a burning sensation in the absence of any stimuli or irritants and severe fibrosis in the entire oral cavity. In Garde III OSMF, there is a severe restriction of tongue movements, severe loss of flexibility of buccal mucosa, and circular band around lips and mouth. There is fibrosis of the soft palate and shrunken uvula. Patients with a severe variety of OSMF show difficulty in speech and nasal voice with restricted mouth opening <15 mm.

  Otological Manifestations Top

In advanced cases of OSMF, patients present with nasal twang due to fibrosis at the nasopharynx and hearing impairment because of stenosis of the eustachian tube opening at the nasopharynx.[33] Involvement of eustachian tube opening at the nasopharynx can occur in OSMF[Figure 2]. The eustachian tube dysfunction results in otalgia and hearing loss. One study clinically evaluated the function of the eustachian tube with the help of pure tone audiometry and found significant hearing loss among patients with OSMF.[34] In one more study, there was a significant correlation found between the degree of palatal muscles fibrosis and hearing impairment.[32] It is well known that the aging process is associated with hearing loss.[35] However, the eustachian dysfunction in OSMF at a younger age was found with hearing impairment.[36] There is an increased chance of OSMF at a younger age because of more use of areca quid and gutkha. OSMF with eustachian dysfunction and hearing impairment in younger age group likely is to affect the quality of life.
Figure 2: Blanching of the mucosal lining in the eustachian opening at the nasopharynx of OSMF patient (yellow arrow)

Click here to view

  Diagnosis Top

In OSMF patients with eustachian dysfunction, the patient may get middle ear pathology where pure tone audiometry often shows the conductive type of hearing loss in the affected side of the eustachian tube. The patients often show mild-to-moderate conductive type of hearing loss. Based on air conduction (AC)-bone conduction (BC) gaP values, the grading of hearing is usually quantified into different categories such as 0–25 decibel – normal hearing; 26–40 decibel – mild deafness; 41–55 decibel – moderate deafness; 56–70 decibel – moderate-to-severe hearing loss; 71–90 decibel – severe hearing loss, and more than 90 decibel suggests profound hearing loss.[37] Tympanometry test may reveal Type B and C types of tympanograms in case of eustachian tube dysfunction by OSMF.[5]

All patients with OSMF and ulcerations in the oral cavity should undergo a biopsy to confirm the diagnosis and also correlate the clinical and histopathological findings. Incisional biopsy should be taken from retromolar and buccal mucosal areas. Additional biopsies can be collected from places where the mucosa has changed clinically, indicating atypia or malignant transformation. Biopsy from fibrotic or ulcerative lesions of the oral cavity of OSMF is done to rule out malignancy or dysplastic changes in the oral cavity. Histopathologically, the OSMF is characterized by marked epithelial atrophy, lack of rete pegs, and subepithelial hyalinization with abnormal excessive collagen deposition, resulting in abundant fibrosis and degeneration of muscles.[9] Epithelium may reveal dysplastic changes signifying the high-risk malignant transformation in the oral cavity and pharyngeal mucosa.[9],[38]

  Treatment Top

OSMF is a disease of insidious origin and is not amenable to reverse at any stage of the progression of the disease, either spontaneously or with cessation of the habit.[9] OSMF is a potentially malignant disorder that needs close monitoring and follows up for different morbidities of this clinical entity. This clinical condition remains either stationary or becomes severe, leaving a person handicapped, both physically and psychologically.[9] Depending based on the stage of OSMF, several management therapies are present which include medical and surgical approaches. There is no definitive treatment available for curing the OSMF other than several treatment options. No single medication or drug has effectively reversed the initiation and development of the OSMF.

The medical treatment is usually symptomatic and often aimed to improve the mouth opening and hearing impairment in OSMF patients. Intralesional injection of steroids/betamethasone, placentrex, and hyaluronidase may help to relieve the restricted mouth opening and burn sensation in the mouth.[39] These treatment options also help to stop the spreading of the diseases to other parts of the oral cavity and oropharynx. Intralesional steroids work by inhibiting the function of soluble factors released by sensitized lymphocytes after they have been activated by certain antigens or triggering stimuli, resulting in OSMF. By preventing inflammatory reactions, steroids prevent fibrosis by reducing fibroblastic proliferation and deposition of collagen.[39] In OSMF, the symptomatic relief by intralesional steroids could be due to anti-inflammatory actions which help in decreasing the juxtaepithelial inflammation.[40] Although uncommon, long-term intralesional injection of steroids has adverse effects such as osteoporosis, myopathies, peptic ulcer, or central serous chorioretinopathy.[41] Hence, in a situation where adequate mouth opening is achievable by the use of intralesional injection of hyaluronidase alone the use of steroids should be avoided to reduce the adverse effects. Placentrex is an aqueous extract of the human placenta which contains nucleotides, enzymes, vitamins, amino acids, and steroids.[42] The action of placentrex is usually biogenic stimulation, and it stimulates the pituitary and adrenal cortex and regulates the metabolism of tissues. It also increases the vascularity of the tissues.[42] Hyaluronidase causes breakage and dissolution of the fibrous band in the oral cavity and so relief from the OSMF.[43] Hyaluronidase plays an important role in relieving the patients with restricted mouth opening and also blockage of the eustachian tube. Aloe vera is an emollient and a protein that contains several amino acids known as wound healing hormones.[44] Surgical treatment is indicated in OSMF patients with severe trismus, and biopsy can be done to rule out any dysplastic/malignant lesions in the oral cavity. Release and excision of the fibrotic bands in OSMF are important surgical steps to prevent further complications and are often considered a challenging task for surgeons.

Although different surgical approaches are available, numerous morbid outcomes such as increased fibrotic changes are found following the postsurgical period. Simple excision of the fibrous bands in the oral cavity, excision of the fibrous bands with myotomy with or without coronoidectomy, covering of the raw area with skin grafts, collagen membrane, fresh amnions, a buccal pad of fat, local flaps, or vascularized free flaps, followed by postoperative jaw physiotherapy with antioxidants and appropriate nutrition and regular follow-ups are needed to ensure maintaining mouth opening and early detection of malignant transformation. The laser can be used for the excision of fibrous bands in patients with OSMF.[45] Laser is a reliable, reproducible technique for preventing postoperative morbidity.[45] Balloon eustachian tuboplasty is a new technique for the treatment of eustachian tube dysfunction.[36] This technique is helpful in the case of OSMF with eustachian tube dysfunction. The use of lasers should be encouraged in general practice for the treatment of OSMF.

  Conclusion Top

There is a significant association between OSMF and eustachian tube function. OSMF may result in eustachian tube dysfunction and hearing impairment. Hence, while managing OSMF, eustachian tube dysfunction and hearing disability have to be kept in mind and vice versa, as treating otological manifestations without taking care of OSMF will not be successful. Further studies are needed to elucidate the association between the eustachian tube dysfunctions with the staging of the OSMF and manifestations of hearing loss. Early diagnosis, treatment, and appropriate patient counseling help to improve the condition drastically. Because of the lack of curative treatment of OSMF and the precancerous nature of this disease, it is often essential to follow-up with the patients on regular basis along with available treatment for reducing the fibrosis bands in the oral cavity and pharynx.

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