Indian Journal of Dental ResearchIndian Journal of Dental ResearchIndian Journal of Dental Research
HOME | ABOUT US | EDITORIAL BOARD | AHEAD OF PRINT | CURRENT ISSUE | ARCHIVES | INSTRUCTIONS | SUBSCRIBE | ADVERTISE | CONTACT
Indian Journal of Dental Research   Login   |  Users online: 508

Home Bookmark this page Print this page Email this page Small font sizeDefault font size Increase font size         

 


 
Table of Contents   
CASE REPORT  
Year : 2019  |  Volume : 30  |  Issue : 3  |  Page : 472-477
Recurrent oral submucous fibrosis with nil mouth opening surgical management and reconstruction with bilateral nasolabial flap: A case report and review of literature


International Medical Center, Jeddah, Saudi Arabia; Department of Oral and Maxillofacial Surgery, SRM Kattankulathur Dental College and Hospital, Chennai, Tamil Nadu, India

Click here for correspondence address and email

Date of Web Publication9-Aug-2019
 

   Abstract 

Oral submucous fibrosis [OSF] is a premalignant condition characterized by inflammation and progressive fibrosis of submucosal tissue, resulting in trismus. It is associated with chewing of areca nut in betel quid. Mortality rate is significant because it transforms into oral squamous cell carcinoma at a rate of 2.3%–7.6%. The aim of this article is to share our experience in managing a case of recurrent oral submucous fibrosis with nil mouth opening by surgical excision, coronoidotomy, and reconstruction of buccal defect using bilateral inferiorly based nasolabial flap, followed by active oral physiotherapy. The patient had reached an acceptable mouth opening with no further recurrence. The patient was observed closely for any malignant transformation. Surgical excision of bands and coronoidotomy followed by reconstruction with nasolabial flaps and active physiotherapy in the postoperative period remains a good option for recurrent and advanced cases of OSF with acceptable functional and cosmetic results.

Keywords: Nasolabial flaps, nil mouth opening, recurrent oral submucous fibrosis, squamous cell carcinoma, surgical excision

How to cite this article:
Ravikumar KK, Khan U N, Priyadarshini, Ramakrishnan K, Nachiappan S. Recurrent oral submucous fibrosis with nil mouth opening surgical management and reconstruction with bilateral nasolabial flap: A case report and review of literature. Indian J Dent Res 2019;30:472-7

How to cite this URL:
Ravikumar KK, Khan U N, Priyadarshini, Ramakrishnan K, Nachiappan S. Recurrent oral submucous fibrosis with nil mouth opening surgical management and reconstruction with bilateral nasolabial flap: A case report and review of literature. Indian J Dent Res [serial online] 2019 [cited 2019 Oct 16];30:472-7. Available from: http://www.ijdr.in/text.asp?2019/30/3/472/264117

   Introduction Top


Oral submucous fibrosis [OSF] is a premalignant condition characterized by inflammation and progressive fibrosis of submucosal tissue, resulting in trismus. It is associated with chewing of areca nut in betel quid and is very common in Southeast Asia but has started to spread to Europe and North America due to its widespread use. Mortality rate is significant because it transforms into oral squamous cell carcinoma at a rate of 2.3%–7.6%.[1] In ancient medicine, Susruta described a condition similar to OSF as “vidari,” under the umbrella of mouth and throat diseases.[2] In 1952, Schwartz [3] described a condition of the oral mucosa as “atrophia idiopathica mucosa oris,” with the term OSF coined by Joshi in 1953.[4] The initial presentation of OSF is inflammation. Inflammation is followed by hypovascularity and fibrosis, visible as blanching of the oral mucosa with a marble-like appearance. Blanching may be localized, diffuse, or reticular. In some cases, small vesicles may develop that rupture and form erosions.[5] OSF does not disappear after cessation of the habit but remains permanent.[6],[7]

The severity of trismus can be graded by measuring the interincisor opening. The mouth opening is categorized into stage I (>3 cm), stage II (2–3 cm), and stage III (<2 cm).[8],[9],[10]

Clinical features of advanced OSF include the loss of puffed-out appearance of cheeks when a patient blows a whistle.

Fibrosis of tongue and mouth impairs tongue movement and leads to depapillation and blanching of mucosa. Fibrosis may also affect the soft palate and uvula, whereas gingival involvement is relatively uncommon. Sometimes, the blockage of  Eustachian tube More Detailss impairs hearing, and oesophageal fibrosis causes problems in swallowing.

The aim of this article is to share our experience in managing a case of recurrent oral submucous fibrosis with nil/zero mouth opening by surgical excision and reconstruction of buccal defect using bilateral inferiorly-based nasolabial flap, followed by active oral physiotherapy in a 66-year-old male patient. Small effort has been made in the present report aiming to endure adequate, functional mouth opening and to detect any developing malignant change at its earliest.


   Case Report Top


A 66-year-old male patient reported to department of oral and maxillofacial surgery in our Dental College and Hospital with progressive inability to open his mouth for past three months which had resulted in his current stage of nil/zero mouth opening. He could only liquids with great difficulty and had experienced weight loss.

His past medical history revealed surgery for OSF, followed by reconstruction with skin graft five years ago. No other medical comorbidity was present.

He had been a betel nut chewer for more than 20 years and claimed to have reduced its frequency since his first surgery but was noncompliant.

On general examination, he was a moderately built patient with complete trismus and nil mouth opening [Figure 1]. Tight bands were palpable at the commissures and no further clinical examination was possible due to severe trismus.
Figure 1: Preoperative mouth opening

Click here to view


Panaromic film revealed attrition of dentition with multiple non-restorable teeth and no other significant dental or bony pathology [Figure 2]. An MRI scan of cheek and neck revealed fibrosis of cheek mucosa with no other significant lesions suggestive of malignancy [Figure 3].
Figure 2: Panoramic film

Click here to view
Figure 3: MRI images

Click here to view


Based on clinical and radiological examination, a clinical diagnosis of recurrent oral submucous fibrosis was made. The patient was reviewed with findings and based on the present condition surgical intervention was the only viable option to improve his mouth opening and his feeding status. The patient was planned for surgical release of fibrous bands, bilateral coronoidotomy, extraction of non-restorable teeth and reconstruction with bilateral inferiorly based nasolabial flap for which he had a written informed consent.

Surgical procedure

Anaesthesia was achieved through fiberoptic nasal endotrachreal intubation. Diathermy was used with caution, starting with the release of bands from commissure, extending posteriorly, taking care not to damage the Stenson's duct by staying below the level of the occlusal plane bilaterally. With combined blunt and sharp dissection and application of brisement force, the mouth opening improved intraoperatively to facilitate bilateral coronoidotomy [Figure 4]. Following the excision of fibrous bands and gradual application of force with Fergusson mouthgag mouth opening improved to 28 mm [Figure 5]. Dissection of inferior fibres of temporalis and deep fibres of masseter was undertaken with biopsy specimen from cheek for histopathology which later came back negative for malignancy.
Figure 4: Intraoperative-coronoidectomy

Click here to view
Figure 5: Intraoperative mouth opening

Click here to view


Bilateral inferior-based nasolabial flap was raised and an orocutaneous tunnel was created in the cheek [Figure 6] and [Figure 7]. The flap was rotated orally extending from the retromolar region to the commissure and achieving closure bilaterally [Figure 8] and [Figure 9].
Figure 6: Bilateral inferiorly based nasolabial flap outline

Click here to view
Figure 7: Bilateral inferiorly based nasolabial flap

Click here to view
Figure 8: Immediate postoperative left buccal mucosa

Click here to view
Figure 9: Immediate postoperative right buccal mucosa

Click here to view


The patient was extubated uneventfully with Ryles tube in place for RT feeds. Oral physiotherapy was initiated 48 hours post-surgery using Heister's mouth gag and the patient was motivated to actively continue the same to maintain the achieved mouth opening [Figure 10] and [Figure 11]. The flap division was done 21 days later and the oro-cutaneous tunnel was closed. The patient is maintaining interincisal opening of 28 mm after five years with no further recurrence or deterioration in mouth opening [Figure 12], [Figure 13], [Figure 14], [Figure 15], [Figure 16].
Figure 10: Immeditate postoperative mouth opening frontal view

Click here to view
Figure 11: Immediate postoperative mouth opening lateral view

Click here to view
Figure 12: Left buccal mucosa five years follow-up

Click here to view
Figure 13: Right buccal mucosa five years follow-up

Click here to view
Figure 14: Mouth opening five years follow-up frontal view

Click here to view
Figure 15: Five years follow-up left lateral

Click here to view
Figure 16: Five years follow-up right lateral

Click here to view



   Discussion Top


The hallmark of diagnosing OSF is clinical and histological. Clinically, one or more of the following symptoms should be present:

  • Blanching of oral mucosa defined as a persistent, white, marble-like appearance of the oral mucosa, which may be localized, diffuse or reticular
  • Tough, leathery texture of the mucosa
  • Palpable, whitish, fibrous bands.


This should be accompanied by histopathological investigations. OSF is characterized by epithelial atrophy with loss of rete ridges and hyalinization of the lamina propria and the underlying muscle.

Treatments for OSF are mainly symptomatic because the aetiology of the disease is not conclusive and it is a progressive disease. Conservative treatment includes vitamin, iron supplements, intralesional injections of hyaluronidase, placental extracts and steroids. Submucosal injections of various drugs may produce temporary symptomatic relief but can lead to aggravated fibrosis, pronounced trismus, and increased morbidity from mechanical injury secondary to insertion of needle and chemical irritation from the drugs.[11]

Surgical excision of fibrous bands and propping the mouth open to allow secondary epithelialisation causes rebound fibrosis during healing with variable success rates. Split thickness skin grafting has high recurrence from contracture which we also encountered in our patient as it was a recurrent case and the survival of full thickness skin graft is questionable.[12]

The use of island palatal flaps based on greater palatine artery has limited involvement of donor site by fibrosis, limited reach of the flap and need for extraction of maxillary second molars to cover the defect without tension.[12] The limitations of bilateral tongue flap includes severe dysplasia, disarticulation, risk of postoperative aspiration, limited donor tissue for coverage of defect, uncontrolled tongue movements leading to lack of stability and dehiscence and involvement of tongue in 38% of cases, which precludes its use for reconstruction.[12]

Buccal fat pad (BFP) is simple due to easy access and could be used to cover defect after excision of fibrous bands. The limitations of BFP are atrophy of the fat in chronic and recurrent disease as in our case and also its anterior reach beyond the cuspids. Any raw area heals by secondary intention and leading to secondary fibrosis and relapse.[13]

The radial forearm free flap can be used to replace oral mucosa, and to decrease the trismus associated with resections. However, bilateral radial forearm free flaps are not ideal for defects less than 6–7 cm in diameter and 40% of patients require secondary debulking procedures and the facilities for free tissue transfer are not universally available.[14] Contemporary oral reconstructive options include use of AlloDerm and tissue engineered mucosal grafts.

The nasolabial flap based on superiorly or inferiorly on the subcutaneous blood supply from the transverse, facial, and angular vessels can be used for buccal mucosal defect reconstruction. The rich collateral blood supply of the cheek is derived from the massetric, buccal, infraorbital and transverse facial arteries.[15] A simultaneous ligation of facial artery in unilateral neck dissection does not appear to be a contraindication for ipsilateral inferiorly or superiorly based nasolabial flap, due to the collateral circulation.[16],[17] There is an abundant dermosubdermal plexus supplying the whole area which exhibits a good degree of axiality ensuring good perfusion to the distal aspect of the flap which contributes to the flaps reliability. In one study an ipsilateral second nasolabial flap was used for reconstruction of oral SCC proving the robust nature of the flap.[18]

As our case was a recurrent case of OSF with additional scarring and fibrosis as a result from the first surgery where skin graft was used for closing the defect, the functional outcome of the inferiorly based nasolabial flap was satisfactory. The patient was able to open his mouth from nil mouth opening to near normal at five years follow-up with no further fibrosis. The patient was able to maintain the initially achieved maximum interincisal opening. The nasolabial flap provided local accessibility in close proximity to the buccal mucosal defect. The donor site could be closed primarily with a well camouflaged scar in the nasolabial fold and no morbidity [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14]. Postoperation, the trismus was relieved to a great extent with the patient able to chew solid food.

However there are certain limitations to nasolabial flaps which include and are not restricted to the following:

  1. A second surgery is needed to close the buccal tunnel which is usually a minor intervention. In certain cases, it can be overcome by deepithelialization of the part in the region of buccal tunnel to facilitate primary closure
  2. The width of the flap depends on the laxity of skin in the nasolabial region and is considered more in older age group than in younger age
  3. Risk of orocuraneous fistula, hematoma, infection and hypertrophic scarring particularly, if the donor site is closed under tension
  4. The other drawback we encountered as in other studies was the intraoral hair growth and the patient had his hair trimmed in his follow-up sessions which subsequently reduced after one year with the mucolization of the grafted tissue.[19]


Our conclusion from the case report is that random pattern of nasolabial flaps could be considered a good option for such recurrent cases of OSF requiring a second surgery and reconstruction of the fibrotomy buccal mucosal defects. It has yielded good functional and cosmetic results with minimal complication. The second surgical intervention not only relieved the trismus but also facilitated future oral examination for early detection of recurrence and malignant transformation.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Canniff JP, Harvey W. The aetiology of oral submucous fibrosis: The stimulation of collagen synthesis by extracts of areca nut. Int J Oral Surg 1981;10:163-7.  Back to cited text no. 1
    
2.
Gupta SC, Yadav YC. “Misi” an aetiological factor in oral submucous fibrosis. Ind J Otolaryngol 1978;30:5-6.  Back to cited text no. 2
    
3.
Schwartz J. Atrophia idiopathica mucosa oris; Demonstrated at the 11th International Dental Congress. London; 1952.  Back to cited text no. 3
    
4.
Joshi SG. Fibrosis of the palate and pillars. Ind J Otolaryngol 1953;4:1.  Back to cited text no. 4
    
5.
Haider SM, Merchant AT, Fikree FF, Rahbar MH. Clinical and functional staging of oral submucous fibrosis. Br J Oral Maxillofac Surg 2000;38:12-5.  Back to cited text no. 5
    
6.
IARC Working Group on the Evaluation of Carcinogenic Risks to Humans. Betel-quid and areca-nut chewing and some areca-nut derived nitrosamines. IARC Monogr Eval Carcinog Risks Hum 2004;85:1-334.  Back to cited text no. 6
    
7.
Al-Rmalli SW, Jenkins RO, Haris PI. Betel quid chewing elevates human exposure to arsenic, cadmium and lead. J Hazard Mater 2011;190:69-74.  Back to cited text no. 7
    
8.
Pandya S, Chaudhary AK, Singh M, Singh M, Mehrotra R. Correlation of histopathological diagnosis with habits and clinical findings in oral submucous fibrosis. Head Neck Oncol 2009;1:10.  Back to cited text no. 8
    
9.
Ceena DE, Bastian TS, Ashok L, Annigeri RG. Comparative study of clinicofunctional staging of oral submucous fibrosis with qualitative analysis of collagen fibers under polarizing microscopy. Indian J Dent Res 2009;20:271-6.  Back to cited text no. 9
[PUBMED]  [Full text]  
10.
Patil S, Maheshwari S. Proposed new grading of oral submucous fibrosis based on cheek flexibility. J Clin Exp Dent 2014;6:e255-8.  Back to cited text no. 10
    
11.
Borle RM, Borle SR. Management of oral submucous fibrosis: A conservative approach. J Oral Maxillofac Surg 1991;49:788-91.  Back to cited text no. 11
    
12.
Khanna JN, Andrade NN. Oral submucous fibrosis: A new concept in surgical management. Report of 100 cases. Int J Oral Maxillofac Surg 1995;24:433-9.  Back to cited text no. 12
    
13.
Yeh CJ. Application of the buccal fat pad to the surgical treatment of oral submucous fibrosis. Int J Oral Maxillofac Surg 1996;25:130-3.  Back to cited text no. 13
    
14.
Wei FC, Chang YM, Kildal M, Tsang WS, Chen HC. Bilateral small radial forearm flaps for the reconstruction of buccal mucosa after surgical release of submucosal fibrosis: A new, reliable approach. Plast Reconstr Surg 2001;107:1679-83.  Back to cited text no. 14
    
15.
Herbert DC, Harrison RG. Nasolabial subcutaneous pedicle flaps. Br J Plast Surg 1975;28:85-9.  Back to cited text no. 15
    
16.
Napolitano M, Mast BA. The nasolabial flap revisited as an adjunct to floor-of-mouth reconstruction. Ann Plast Surg 2001;46:265-8.  Back to cited text no. 16
    
17.
Lazaridis N. Unilateral subcutaneous pedicled nasolabial island flap for anterior mouth floor reconstruction. J Oral Maxillofac Surg 2003;61:182-90.  Back to cited text no. 17
    
18.
James K, Stassen LF. Ipsilateral second nasolabial flap reconstruction for oral squamous cell carcinoma. Br J Oral Maxillofac Surg 2013;51:269-70.  Back to cited text no. 18
    
19.
Lambade P, Meshram V, Thorat P, Dawane P, Thorat A, Rajkhor D. Efficacy of nasolabial flap in reconstruction of fibrotomy defects in surgical management of oral submucous fibrosis: A prospective study. Oral Maxillofac Surg 2016;20:45-50.  Back to cited text no. 19
    

Top
Correspondence Address:
Dr. Kamal Kanthan Ravikumar
International Medical Center, Post Box 2172, Jeddah 21451

Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.IJDR_423_17

Rights and Permissions


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14], [Figure 15], [Figure 16]



 

Top
 
 
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Email Alert *
    Add to My List *
* Registration required (free)  
 


    Abstract
   Introduction
   Case Report
   Discussion
    References
    Article Figures

 Article Access Statistics
    Viewed201    
    Printed1    
    Emailed0    
    PDF Downloaded31    
    Comments [Add]    

Recommend this journal