International Journal of Science and Healthcare Research Vol.5; Issue: 2; April-June 2020 Website: ijshr.com Case Report ISSN: 2455-7587

Chronic Desquamative : A Case Report

Kousain Sehar1, Nadia Irshad2, Navneet Kour1, Mir Tabish Syeed3, Burhan Altaf Misgar4, Manju Verma2, Hilal Ahmad Hela5

1MDS Dept of and Implantology, BRS Dental College and Hospital Sultanpur Panchkula. 2MDS Dept of Paedontics and Preventive Dentistry, BRS Dental College and Hospital Sultanpur Panchkula. 3MDS Dept of Paedontics and Preventive Dentistry, Swami Devi Dayal Hospital and Dental College, Barwala, Panchkula. 4MDS Dept of Paedontics and Preventive Dentistry, Guru Nanak Dev Dental College and Research Institute, Sunam, Punjab. 5MDS Dept. of Prosthodontics, Govt Dental College and General Hospital, Srinagar.

Corresponding Author: Navneet Kour

ABSTRACT causes for desquamative gingivitis. It may occur due to aging, abnormal response to Desquamative gingivitis is a type of gingival bacterial plaque, allergy, idiopathic, chronic keratinization disorder, characterized by chronic infections, autoimmune diseases or ulceration, desquamative erosions and idiopathic1 edematous erythema of the free and attached Patients may be asymptomatic, gingiva. Desquamative gingivitis has been known to be refractory in treatment and only a however, when symptomatic, their few cases have been reported. Various etiologic complaint ranges from mild burning factors are present for the appearance of such sensation to an intense pain. Approximately . Despite of considering etiology, 50% of desquamative gingivitis cases are treatment is often provided by systemic or localized to gingiva, although involvement topical corticosteroids. Since the cause of this of the gingiva and intraoral/extraoral sites condition is still unclear, symptomatic therapy are not uncommon. remains the mainstay of treatment. Here, Initially, the cause of this condition presenting the case of a patient who had was not clear with a variety of possibilities moderate periodontitis with chronic that had been suggested. Etiologic factors desquamative gingivitis. have been considered which includes

Keywords: Corticosteroids, Desquamative, estrogen deficiency, hypothyroidism and nutritional deficiencies. However, Foss, Erythema, Periodontitis 3 Glickman and Smulow considered the INTRODUCTION disease to be primarily a degenerative Although first recognized and process. In 1953 Glickman, speculated that reported in 1894 by Tomes and Tomes, the chronic desquamative gingivitis may term chronic desquamative gingivitis was represent a manifestation of several disease coined in 1932 by Prinz1 to describe a processes. While he favored an endocrine peculiar condition characterized by intense etiology, he thought that the condition might erythema, desquamation, and ulceration of be a variant of chronic mucous membrane the free and attached gingiva.1 It is seen or bullous . mainly in adults, especially women, Because most of the cases were diagnosed although rare cases have been observed in in women in the fourth- fifth decades of life children2. Oral lichen planus, mucous (but desquamative gingivitis may occur as membrane pemphigoid and early as puberty or as late as the seventh or vulgaris have emerged as most common eighth decades), a hormonal derangement was suspected. However, in 1960 McCarthy

International Journal of Science and Healthcare Research (www.ijshr.com) 289 Vol.5; Issue: 2; April-June 2020 Kousain Sehar et.al. Chronic desquamative gingivitis: a case report and colleagues suggested that desquamative over 95% of the dermatologic cases. gingivitis was not a specific disease entity, However, other mucocutaneous but a gingival response associated with a autoimmune conditions such as lupus variety of conditions. This concept was erythematosus, , further supported by numerous , pemphigus immunopathologic studies. They also vulgaris, linear immunoglobulin A (IgA), proposed a classification of desquamative and chronic ulcerative can gingivitis based on etiologic considerations: clinically manifest as desquamative Dermatoses, Hormonal influences, gingivitis. Abnormal responses to irritation, Chronic Other conditions considered in the infections and Idiopathic causes. Among differential diagnosis of desquamative dermatoses manifest as desquamative gingivitis are chronic bacterial, fungal, and gingivitis, the authors included benign viral infections, as well as reactions to mucous membrane pemphigoid (now often medications, , and chewing called cicatricial pemphigoid), pemphigus gum. Although less common, Crohn's and lichen planus. Some reports have disease, , some , and suggested that psoriasis also may manifest even factitious lesions have also been as desquamative gingival lesions. reported to clinically present as The diseases that can cause desquamative gingivitis. desquamative gingivitis can be classified Therefore it is of paramount into 2 categories: immunological importance to ascertain the identity of (autoimmune or auto-immune like) and disease responsible for desquamative idiopathic. Immunological diseases causing gingivitis to establish an appropriate desquamative gingivitis include erosive therapeutic approach and management. To lichen planus, lichenoid mucositis, benign achieve this goal, the clinical examination mucous membrane pemphigoid, pemphigus has to be coupled with a thorough history, vulgaris, paraneoplastic pemphigus, linear and routine histologic and IgA disease, and bullous pemphigoid. immunofluorescent studies. It should be Idiopathic lesions are probably not mentioned, however, that despite this autoimmune mediated, and may be caused diagnostic approach, the cause of by chronic bacterial, fungal, and viral desquamative gingivitis cannot be infections, or other factors capable of elucidated in up to one third of the cases. causing chronic irritation and inflammation. The current classification system for CASE REPORT periodontal diseases and conditions includes A 51-year-old female patient non plaque induced gingival disorders and reported to the Department of gingival manifestations of systemic Periodontology, BRS Dental College & conditions. The mucocutaneous disorders Hospital, Panchkula, having a chief e.g., lichen planus, pemphigoid, pemphigus complaint of burning sensation in lower vulgaris, , lupus posterior gingiva since 9 months which erythematosus, drug induced lesions and aggravates on having spicy & hot others are listed in this subgroup together beverages. On examination, red, linear, with allergic reactions to dental materials, diffused patches were noticed on the buccal foods, and other substances for topical marginal gingiva with involvement of application. attached gingival showing signs of The advent of clinical and laboratory desquamation. (Fig 1) Limitation of oral parameters revealed that approximately 75% function and speech difficulties due to pain of desquamative gingivitis cases have a was also reported by the patient. Non- dermatologic genesis. Cicatricial significant dental and medical history was pemphigoid and lichen planus account for given was given by the patient. Patient had a

International Journal of Science and Healthcare Research (www.ijshr.com) 290 Vol.5; Issue: 2; April-June 2020 Kousain Sehar et.al. Chronic desquamative gingivitis: a case report habit of tobacco chewing, 3-4 times in a day did not show any improvement clinically since last 10 years but she had quitted her with topical steroids application in the habit 3 yrs back. She has also given a follow up visits (Figure:3) history of change in tooth paste a year back which was continued by her for 6 months and again she shifted to her regular tooth paste. Due to the discontinuation of the tooth-paste, the appeared desquamated didn’t healed. Exact etiology for the appearance of desquamated lesions in this patient was not identified. After obtaining written consent, an incisional gingival biopsy was taken from mandibular posterior region for the histopathological examination. The hematoxylin & eosin (H Figure:1 Desquamative gingivitis in mandibular posterior & E) stained tissue showed clefts at the sub- region epithelium with underlying connective tissue showing bundles of collagen fibers arranged haphazardly along with fibroblast & small foci of inflammatory cells. (Fig.2). epithelium clefts were suggestive of desquamative lesions. Thorough oral prophylaxis (scaling & polishing) was done. Patient was advised to rinse her mouth with 0.2% , enforced to do modified bass technique for brushing her teeth & also was asked to maintain good . Topical application of steroid Figure:2 H & E stained tissue for histopathological (0.1% Triamcinolone acetonide) twice in a examination day for three weeks was prescribed. Lesions

Figure:3 Appearance of desquamative gingival lesions in mandibular region after steroid therapy

DISCUSSION of any given therapeutic approach depends Desquamative gingivitis is only a on the re-inforcement of an appropriate final clinical term that describes a peculiar diagnosis. The following represents a clinical picture. This term is not a diagnosis systematic approach to elucidate the disease per se and once it is rendered, a series of triggering desquamative gingivitis. laboratory procedures should be used to Differential diagnosis of multiple oral ulcers arrive to a final diagnosis. Thus the success may include aphthous ulcers major, erosive

International Journal of Science and Healthcare Research (www.ijshr.com) 291 Vol.5; Issue: 2; April-June 2020 Kousain Sehar et.al. Chronic desquamative gingivitis: a case report lichen planus, mucous membrane acute herpetic gingivostomatitis, and pemphigoid, , acute erythema multiforme.4 necrotizing ulcerative gingivitis, allergies,

Figure:4 Representing identification of gingival lesions

Desquamative gingivitis is entirely a based on expert opinion rather than common disorder in which the gingiva is empirical evidence. Several treatment desquamated. Chronic soreness is methods have been reported (Carrozzo & commonly seen and intake of spicy foods Gandolfo, 1999; Chan et al., 2002; Endo et may further worsen the condition. al., 2008b; Fatahzadeh et al., 2006; Erythematous gingiva with loss of , Kirtschig et al., 2003; Lamey et al., 1992; extends apically from the gingival margins Motta et al., 2009; Nisengard, 1996; to the alveolar mucosa is frequent. Severity Nisengard & Levine, 1995). Treatments are may range from mild, almost insignificant available in the form of systemic or topical small patches to widespread erythema with steroids, antimetabolites glazed appearance. Occasionally, such (cyclophosphamide, azathioprine, lesions may occur in the absence of mycophenolate mofetil, methotrexate), bacterial plaque1. Sometimes contact antibiotics (), and dapsone allergic reactions to various oral hygiene intravenous immunoglobulins, products have also been reported in plasmapheresis, and Low level laser therapy desquamative gingivitis patients2. But oral which causes pain relief and accelerates mucosa is less commonly prone to contact regeneration of damaged tissues.. Treatment allergic reactions, when compared to skin, of desquamative gingivitis requires though the latter is exposed to a wide elimination or control of local irritants. variety of antigenic stimuli4. The therapeutic Rough restorations, illfitting dentures, approaches to desquamative gingivits are traumatic oral hygiene procedures, and

International Journal of Science and Healthcare Research (www.ijshr.com) 292 Vol.5; Issue: 2; April-June 2020 Kousain Sehar et.al. Chronic desquamative gingivitis: a case report dysfunctional oral habits should be then submitted for microscopic corrected5. In most of the cases, such lesions examination. Buffered formalin (10%) are successfully managed with topical should be used to fix the tissue for corticosteroids combined with effective conventional hematoxylin and eosin (H&E) plaque control6,7. Plaque accumulation itself evaluation. Michell's buffer (ammonium acts as a stimulus for worsening the sulfate buffer, pH 7.0) is used as transport condition of desquamative gingivitis, but the solution for immunofluorescent assessment.. plaque itself does not cause such lesions. However, there are notable exceptions such Patient with desquamative gingivitis are as in lichen planus and subacute lupus unable to maintain a proper oral hygiene erythematosus where only the lesional tissue due to unbearable pain. Therefore, their oral will exhibit the corresponding immunologic hygiene is likely to be ineffective, making it markers. difficult to treat this condition. Lack of Microscopic Examination: correct oral hygiene and the accumulation of Approximately 5-μm sections of plaque may increase the long-term risk for formalin-fixed, paraffin embedded, tissue plaque-induced periodontal diseases along stained with conventional H&E are obtained with desquamative gingivitis.8 for light microscopy examination. Clinical Examination: Immunofluorescence: Recognition of pattern of For direct immunofluorescence, distribution of the lesions (i.e., focal or unfixed frozen sections are incubated with a multifocal, with or without confinement to variety of fluorescein-labeled, anti-human the gingival tissues) provides leading serum (anti-IgG, anti-IgA, anti-IgM, anti- information to begin the formulation of a fibrin and anti-C3). With indirect differential diagnosis. A clinical maneuver immunofluorescence, unfixed frozen such as Nikolsky's sign offers insight into sections of oral or esophageal mucosa from the plausibility for the presence of any an animal such as a monkey are first vesiculobullous lesions. incubated with the patient's serum to allow Biopsy: attachment of any serum antibodies to the Given the extent and number of mucosal tissue. The tissue is then incubated lesions that may be present in a given with fluorescein-labeled antihuman serum individual, an incisional biopsy is the best (anti-IgG, anti-IgA, anti-IgM, anti-fibrin, alternative to begin the microscopic and and anti-C3). Immunofluorescence tests are immunological evaluation. An important positive if a fluorescent signal is observed consideration is selection of the biopsy site. either in the epithelium, its associated Once the tissue is excised from the oral basement membrane or in the underlying cavity, the specimen can be bisected and connective tissue.

Figure:5 Direct/indirect immunofluorescence

International Journal of Science and Healthcare Research (www.ijshr.com) 293 Vol.5; Issue: 2; April-June 2020 Kousain Sehar et.al. Chronic desquamative gingivitis: a case report

CONCLUSION 5. Fatahzadeh, M., Radfar L., and Sirois, D.A., The management of desquamative Dental care of patients with autoimmune gingivitis is challenging because lesions vesiculobullous diseases: case reports and reoccur after it goes into remission. literature review, Quintessence International, 37(10), 2006 Nov-Dec, 77-87. REFERENCES 6. Endo H., Rees T D, Kuyama K, Matsue M, 1. Shamimul Hasan, Desquamative gingivitis - and Yamamoto H, Successful treatment A clinical sign in mucous membrane using occlusive steroid therapy in patients pemphigoid: Report of a case and review of with erosive lichen planus: a report on 2 literature, Jornal of Pharmacy & Bioallied cases, Quintessence International, 39(4), Sciences, 6(2), 2014, 122–126. 20008 April, 162-172. 2. Hiroyasu Endo and Terry D. Rees, 7. Guiglia R, Di Liberto C, Pizzo G, Picone L, Diagnosis and Management of Lo Muzio L and Gallo P D, A combined Desquamative Gingivitis, Gingival Diseases treatment regimen for et al. gingivitis in – Their Aetiology, Prevention and patients with oral lichen planus, Journal of Treatment, www.intecopen.com, 171-188. Oral Pathology & Medicine, 36(2), 2007 3. Glikman I, Smulow JB. Chronic February, 110-116. desquamative gingivitis: Its nature and 8. Pradeep A.R, Manojkumar S.T, and Arjun treatment. J Periodontol 1964;35:397-405 R, Pemphigus vulgaris with significant 4. Baljeet Singh, Alka Sharma, and Avnika periodontal findings: a case report, Jornal of Garg, Herbal oral gel induced contact the California Dental Association, 38(5), stomatitis along with desquamative 2010 May, 343-346

gingivitis due to a coloring agent, Journal of Indian Society of Periodontology, 19(5), How to cite this article: Sehar K, Irshad N, Kour 2015, 569–572. N et.al. Chronic desquamative gingivitis: a case report. International Journal of Science & Healthcare Research. 2020; 5(2): 289-294.

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International Journal of Science and Healthcare Research (www.ijshr.com) 294 Vol.5; Issue: 2; April-June 2020