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Thanking you
With sincere regards
Dr. Rajendra Kumar Ghritlaharey, M.S., M. Ch., FAIS
Associate Professor,
Department of Paediatric Surgery, Gandhi Medical College & Associated
Kamla Nehru & Hamidia Hospitals Bhopal, Madhya Pradesh 462 001 (India)
E-mail: drrajendrak1@rediffmail.com
On May 11,2011




Dr. Shankar P.R.

"On looking back through my Gmail archives after being requested by the journal to write a short editorial about my experiences of publishing with the Journal of Clinical and Diagnostic Research (JCDR), I came across an e-mail from Dr. Hemant Jain, Editor, in March 2007, which introduced the new electronic journal. The main features of the journal which were outlined in the e-mail were extensive author support, cash rewards, the peer review process, and other salient features of the journal.
Over a span of over four years, we (I and my colleagues) have published around 25 articles in the journal. In this editorial, I plan to briefly discuss my experiences of publishing with JCDR and the strengths of the journal and to finally address the areas for improvement.
My experiences of publishing with JCDR: Overall, my experiences of publishing withJCDR have been positive. The best point about the journal is that it responds to queries from the author. This may seem to be simple and not too much to ask for, but unfortunately, many journals in the subcontinent and from many developing countries do not respond or they respond with a long delay to the queries from the authors 1. The reasons could be many, including lack of optimal secretarial and other support. Another problem with many journals is the slowness of the review process. Editorial processing and peer review can take anywhere between a year to two years with some journals. Also, some journals do not keep the contributors informed about the progress of the review process. Due to the long review process, the articles can lose their relevance and topicality. A major benefit with JCDR is the timeliness and promptness of its response. In Dr Jain's e-mail which was sent to me in 2007, before the introduction of the Pre-publishing system, he had stated that he had received my submission and that he would get back to me within seven days and he did!
Most of the manuscripts are published within 3 to 4 months of their submission if they are found to be suitable after the review process. JCDR is published bimonthly and the accepted articles were usually published in the next issue. Recently, due to the increased volume of the submissions, the review process has become slower and it ?? Section can take from 4 to 6 months for the articles to be reviewed. The journal has an extensive author support system and it has recently introduced a paid expedited review process. The journal also mentions the average time for processing the manuscript under different submission systems - regular submission and expedited review.
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Dr. P. Ravi Shankar
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On April 2011
Anuradha

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Dr. Anuradha
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On Jan 2020

Important Notice

Case report
Year : 2021 | Month : August | Volume : 15 | Issue : 8 | Page : ZD01 - ZD04 Full Version

Management of Histologically Proven Oral Submucous Fibrosis with Intralesional Steroids and Hyaluronidase- A Report of Two Cases


Published: August 1, 2021 | DOI: https://doi.org/10.7860/JCDR/2021/48909.15202
R Preethi, S Aravind Warrier, CV Divyambika

1. Postgraduate Student, Department of Oral Medicine and Radiology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India. 2. Professor and Head, Department of Oral Medicine and Radiology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India. 3. Associate Professor, Department of Oral Medicine and Radiology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India.

Correspondence Address :
Dr. S Aravind Warrier,
Professor and Head, Department of Oral Medicine and Radiology, Sri Ramachandra
Institute of Higher Education and Research, Porur, Chennai-600116, Tamil Nadu, India.
E-mail: dentalwarrier@gmail.com

Abstract

Oral Submucous Fibrosis (OSMF) is a chronic debilitating disease characterised by progressive fibrosis. The disease is predominantly seen in South East Asia and mainly attributed to areca nut chewing habit. The classical presentation includes generalised blanching with palpable fibrotic bands in the oral mucosa. Patients experience severe burning sensation, trismus and difficulty in gustatory functions, thus resulting in compromised nutritional status. The OSMF has been grouped under Oral Potentially Malignant Disorders (OPMD) and has high rates of malignant transformation. Although randomised controlled trials on different treatment modalities have been conducted, currently there is no effective medical management. This paper highlights two cases of OSMF presenting with severe burning sensation and trismus, who underwent incisional biopsy for histopathological confirmation and to rule out dysplasia. Both the patients were treated with dexamethasone and hyaluronidase, biweekly for four weeks. Post-treatment there was significant improvement in the clinical symptoms, thus providing the patients with better quality of life.

Keywords

Areca nut, Malignant transformation, Oral potentially malignant disorder

Case Report

Case 1

A 41-year-old male patient reported to the Outpatient Department of Oral Medicine and Radiology with a chief complaint of limited mouth opening since four months. On eliciting the personal history, the patient gave history of pan chewing for past four years, 10 times a day, and had habit of keeping it in left buccal mucosa for 15 minutes before spitting it out. The patient gave a Visual Analogue Scale (VAS) score of seven for burning sensation. On examination, there was evidence of generalised blanching (Table/Fig 1)a,b, with restricted tongue movements and mouth opening of 30 mm (Table/Fig 2)a,b. On palpation, the buccal mucosa was leathery in texture with evident palpable fibrotic bands bilaterally on the buccal mucosa. A provisional diagnosis of Oral Submucous Fibrosis (OSMF) (Stage II-based on Khanna and Andrade Functional staging (1) was given. Informed consent was obtained from the patient prior to investigative work up. The haematological findings were within normal limits with haemoglobin at 14.4 gm/dL and incisional biopsy was done on the left buccal mucosa. The histopathology report revealed the presence of atrophic stratified squamous epithelium, with underlying connective tissue showing hyalinisation, fibrosis, with few inflammatory cells and absence of dysplasia, correlating with histopathological Grade III based on histopathological grading of OSMF by Pindborg and Sirsat (Table/Fig 3) (2).

Case 2

A 40-year-old male patient reported to the Outpatient Department of Oral Medicine and Radiology with a chief complaint of burning sensation to spicy foods and limited mouth opening since one year. Personal history revealed that the patient had pan chewing habit for two years, two times a day, and had habit of keeping it in left and right buccal mucosa for five minutes before spitting. The patient gave a VAS score of eight for burning sensation. On examination there was evidence of generalised blanching of oral mucosa with marble like appearance (Table/Fig 4)a-d. The mouth opening measured as interincisal distance using metallic scale and divider was 30 mm (Table/Fig 5)a,b. Tongue movements were restricted and there was evidence of bud shaped uvula (Table/Fig 6). On palpation, bilateral fibrous bands were evident bilaterally on the buccal mucosa. A provisional diagnosis of OSMF (Stage II-based on Khanna and Andrade functional staging (1) was given. Patient’s consent was obtained before subjecting to investigations. The haematological parameters were within normal limits with haemoglobin levels at 12.3 gm/dL and incisional biopsy was performed in relation to right buccal mucosa. The histopathology report revealed the presence of orthokeratotic atrophic stratified squamous epithelium, with underlying connective tissue showing hyalinisation, fibrosis, with few inflammatory cells and absence of dysplasia, correlating with histopathological Grade III based on histopathological grading of OSMF by Pindborg and Sirsat (Table/Fig 7) (2). The differential diagnosis for both the cases, included scleroderma, iron deficiency anaemia and generalised fibromatoses (3), however they were ruled out based on the habit history and classical clinical manifestations of OSMF.

Treatment: Both the patients were counselled to stop pan chewing habit. The treatment included topical application of 0.1% triamcinolone acetonide twice daily, vitamin B complex tablet, and intralesional injection with 1.5 ml of dexamethasone (4 mg/mL) and hyaluronidase (1500 IU); with 0.5 ml lignocaine HCL, biweekly for four weeks on right and left buccal mucosa in multiple sites, based on James L et al., (4). Patient was advised mouth opening exercises for 2-3 times a day.

At the end of four weeks, case 1 gave a VAS score of 0 (pre-treatment VAS score-7) and increase in mouth opening as interincisal distance improved to 35 mm (an increase by 5 mm) (Table/Fig 8)a,b; case 2 gave a VAS score of 0 (pre-treatment VAS score-8) and interincisal distance improved to 37 mm (an increase by 7 mm) (Table/Fig 8)c,d. Both the patients gave a subjective improvement in the tongue movements and the elasticity of the right and left buccal mucosa (measured using divider and scale) improved in both the patients, which was performed based on Reddy V et al., (5). The patients were asked to continue mouth opening exercises for another six weeks, and the patients were kept under follow-up.

Discussion

Oral Submucous Fibrosis (OSMF) is a chronic debilitating disease seen in betel nut chewers, characterised by progressive fibrosis. The clinical features include burning sensation, trismus, blanching, palpable fibrotic bands and altered gustatory function. The burning sensation seen in OSMF is mainly attributed to the atrophy of the epithelium, which results in reduction of the distance of intra-epithelial nerve endings from the surface (6). The areca alkaloids in the betel nut-namely arecoline, arecaidine, guvacine and guvacoline play major role in the pathogenesis of OSMF. Arecoline stimulates fibroblastic proliferation, collagen synthesis and decreases collagen breakdown. Areca nut chewing causes continuous local irritation of the soft tissues of oral cavity, leading to injury related chronic inflammation, oxidative stress and cytokine production. Oxidative stress and subsequent Reactive Oxygen Species (ROS) generation induces cell proliferation and apoptosis; these events lead to preneoplastic changes and subsequently to oral malignancy (7),(8).

Various OPMD include oral leukoplakia, erythroplakia, erythroleukoplakia, OSMF, palatal lesions in reverse smokers, oral lichen planus, oral lichenoid reactions, graft versus host Disease (GvHD), oral lupus erythematosus, actinic chelitis of lower lip and some hereditary conditions, such as dyskeratosis congenita and epidemolysis bullosa. Most of these conditions tend to be asymptomatic in early stage and hence early identification is of utmost importance for better prognosis and prevents malignant transformation (9). High rates of malignant transformation has been reported in OSMF amongst OPMD, with malignant transformation rate of 7-13% (7),(8). The different treatment strategies include local injections of steroids (dexamethasone or betamethsone), placental extracts, hyaluronidase, lycopene, pentoxyphylline, IFN-gamma, colchicine, Vitamin A and other anti-oxidants. However, it has been seen that various treatment modalities have limited efficacy with currently no single effective treatment available (10). Previous studies indicate that the anti-inflammatory effect of steroids along with breakdown of inter-cementing substance by hyaluronidase has proven symptomatic relief in OSMF (4),(11). The current report highlights two cases of histologically proven cases of OSMF treated with dexamethasone and hyaluronidase resulting in substantial symptomatic relief at four weeks post treatment.

Steroids namely dexamethasone, betamethasone, hydrocortisone, triamcinolone which have been employed in the management of OSMF, have proven anti-inflammatory action. Steroids further reduce the fibroblastic proliferation and the collagen deposition (11). The symptomatic relief resulting in reducing the burning sensation and increasing the mouth opening in both the cases could be attributed to the potent anti-inflammatory and antifibrotic effect of steroids. Hyaluronidase has the potency to break the inter-cementing substance (hyaluronic acid); the combination with steroid has provided better results in symptomatic improvement among OSMF patients (12). Despite steroids being conventional treatment regimen for different clinical stages of OSMF, a study by Borle RM et al., showed increased fibrosis with repeated steroid injections and has favoured conservative approach for OSMF in the form of topical application of betamethasone, chewable vitamin A tablets and iron supplements (13). An unusual case of bilateral buccal space abscess and a case of central serous chorioretinopathy were observed after intralesional steroid injections in OSMF patients (14). Hence, it is mandatory to consider the possibility of such rare side effects during the entire course of treatment and a thorough evaluation of medical history is necessary before the initiation of treatment. Some of the contraindications include hypersensitivity to steroids or hyaluronidase, medically compromised patients including uncontrolled diabetes, hypertension and pregnant women (15),(16).

In a report of three cases, intralesional injection of steroids along with hyaluronidase resulted in a significant relief of symptoms (17). Study done by Tilekaratne WM et al., employed intralesional injections of 40 mg of prednisolone on 116 patients in histologically proven cases of OSMF with mouth opening less than 30 mm. The study showed that around 60% of cases had 5 mm or <5 mm improvement of mouth opening at one year follow-up from the first dose, after being treated at monthly intervals for a period of six months. The improvement in mouth opening after corticosteroid administration has been correlated with the anti-inflammatory and upregulation of immune mediated fibrolytic pathways (18). It has been observed in a study that intralesional triamcinolone acetonide (40 mg/mL; 1 mL) showed superior clinical improvement compared with intralesional placental extract (placental extract 2 mL) after 10 weeks of therapy (19). The current case report highlights good clinical response of reduced burning sensation and improvement in mouth opening after four weeks post-treatment with intralesional dexamethasone and hyaluronidase. With currently no effective medical management for OSMF, steroids along with hyaluronidase play a promising role in symptomatic relief for patients thus reducing the morbidity associated with the disease.

Conclusion

Currently, management of OSMF with respect to alleviation of burning sensation and trismus is challenging. Intralesional dexamethasone with hyaluronidase aids in symptomatic relief and improvement in mouth opening, resulting in better nutritional intake. Oral physiotherapy during the treatment and adequate follow-up of such patients helps in monitoring them for any malignant transformation, thus enhancing the quality of life of OSMF patients.

References

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Daga D, Singh RK, Pal US, Gurung T, Gangwar S. Efficacy of oral colchicine with intralesional hyaluronidase or triamcinolone acetonide in the Grade II oral submucous fibrosis. National Journal of Maxillofacial Surgery. 2017;8(1):50-54. [crossref] [PubMed]
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Kar IB, Sethi AK. A rare ocular complication following treatment of oral submucous fibrosis with steroids. National Journal of Maxillofacial Surgery. 2011;2(1):93. [crossref] [PubMed]
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Nagaraj T, Okade D, Biswas A, Sahu P, Saxena S. Intralesional injections in oral submucous fibrosis-A series of case reports. Journal of Medicine, Radiology, Pathology and Surgery. 2018;5(5):23-26. [crossref]
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Tilakaratne WM, Ekanayaka RP, Herath M, Jayasinghe RD, Sitheeque M, Amarasinghe H. Intralesional corticosteroids as a treatment for restricted mouth opening in oral submucous fibrosis. Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology. 2016;122(2):224-31. [crossref] [PubMed]
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Shinde CV, Saawarn N, Kohli S, Khare P, Singh A, Sagar KM. Comparative efficacy of intralesional placental extract and intralesional triamcinolone acetonide in the management of OSMF. Journal of Indian Academy of Oral Medicine and Radiology. 2019;31(4):328.

DOI and Others

10.7860/JCDR/2021/48909.15202

Date of Submission: Feb 08, 2021
Date of Peer Review: May 10, 2021
Date of Acceptance: Jun 02, 2021
Date of Publishing: Aug 01, 2021

AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. Yes

PLAGIARISM CHECKING METHODS:
• Plagiarism X-checker: Feb 09, 2021
• Manual Googling: May 25, 2021
• iThenticate Software: Jul 05, 2021 (6%)

ETYMOLOGY: Author Origin

JCDR is now Monthly and more widely Indexed .
  • Emerging Sources Citation Index (Web of Science, thomsonreuters)
  • Index Copernicus ICV 2017: 134.54
  • Academic Search Complete Database
  • Directory of Open Access Journals (DOAJ)
  • Embase
  • EBSCOhost
  • Google Scholar
  • HINARI Access to Research in Health Programme
  • Indian Science Abstracts (ISA)
  • Journal seek Database
  • Google
  • Popline (reproductive health literature)
  • www.omnimedicalsearch.com