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Cheilitis Glandularis Treated with Intralesional Steroids: a Rare Case Report and Review 1Rezwana Begum Mohammed , 2G Shirisha Rani, 3M Pavana Sandya, 4S Jyotsna

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Cheilitis Glandularis Treated with Intralesional Steroids: a Rare Case Report and Review 1Rezwana Begum Mohammed , 2G Shirisha Rani, 3M Pavana Sandya, 4S Jyotsna OMPJ Rezwana Begum Mohammed et al 10.5005/jp-journals-10037-1025 CASE REPORT Cheilitis Glandularis Treated with Intralesional Steroids: A Rare Case Report and Review 1Rezwana Begum Mohammed , 2G Shirisha Rani, 3M Pavana Sandya, 4S Jyotsna ABSTRACT CG was fi rst used by Volkmann in 1870 to describe a disorder Cheilitis glandularis (CG) is a rare disorder characterized by that presented as a chronic, suppurative infl ammation of the swelling of the lip with hyperplasia of the labial salivary glands. lower lip, characterized by swelling of the mucous glands CG is most commonly seen in the lower lip, in middle-aged to and associated with mucopurulent discharge through dilated older Caucasian men; however, rarely reported in Asians. The ductal openings.3 CG is a rare condition most commonly exact cause of CG is unknown, but various agents have been attributed like smoking, chronic irritation, poor oral hygiene, seen in the lower lip of adult male with little predilection allergy, bacterial infections, syphilis, chronic exposure to sunlight to female and children,4 in middle aged to older Caucasian and wind, compromised immune system, and genetic trans- men;5 Japanese and Asian cases are very rare. In the litera- mission. In this paper, we report a rare case of CG of the super- fi cial suppurative type in a 43-year-old Indian female affecting ture, three types of CG have been described, the simple, lower lip which was diagnosed based on clinical and histo- superfi cial suppurative and deep suppurative.6 However, it is pathological fi ndings and was treated with intralesional steroid now believed that these represent three stages in progression injections. Even though the incidence and occurrence of CG of a single disease entity.7 Clinically, CG may resemble many in clinical practice is rare, it presents a diagnostic challenge to dentists because the etiologic factors are uncommon with other conditions. Differential diagnoses includes multiple variations in clinical presentation. Early diagnosis with defi - mucocele, chronic sialadenitis of the minor salivary glands, nitive treatment and frequent monitoring should be carried out sialolithiasis of minor salivary gland, factitious cheilitis, to prevent further complications. actinic cheilitis, cheilitis granulomatosa, angioedema, and Keywords: Cheilitis glandularis, Suppurative, Minor salivary benign and malignant salivary gland tumors.2,8 glands, Intralesional steroids. Histologically, CG is a nonspecifi c chronic infl amma- How to cite this article: Mohammed RB, Rani GS, Sandya MP, Jyotsna S. Cheilitis Glandularis Treated with Intralesional Steroids: tory lesion characterized by localized dense accumulation A Rare Case Report and Review. Oral Maxillofac Pathol J 2014; of infl ammatory cells and infl ammation of mucous glands 5(2):510-513. associated with glandular distension and ductal dilatation Source of support: Nil with notable loss of acinar structure.9 Roda and Yacobi and Confl ict of interest: None Brown consider duct ectasia and infl ammation of minor glands and adjacent tissue to be the essential microscopic 10, 11 INTRODUCTION features of CG. Various treatment modalities have been described for Cheilitis glandularis (CG) is a rare chronic infl ammatory CG based on clinical presentation, diagnostic information disease affecting the minor labial salivary glands and charac- obtained from histopathologic analysis and the identifi - 1,2 terized clinically by edema and focal ulceration. The term cation of likely etiologic factors responsible for the disease. It includes administration of antihistamines, antimicrobials, 1-3Assistant Professor, 4Postgraduate Student topical or intralesional steroids12,13 and topical immuno- 1 Department of Oral Medicine and Radiology, Gitam Dental suppressants. In cases with severe dysplasia, surgical stripping, College and Hospital, Visakhapatnam, Andhra Pradesh, India vermilionectomy, cryosurgery, laser surgery, or topical 2 Department of Oral Medicine and Radiology, Meghna Institute chemotherapy with 5-fl uorouracil found to be the effective of Dental Sciences, Nizamabad, Andhra Pradesh, India treatment modalities.4,12 3Department of Public Health Dentistry, Mamata Dental College and Hospital, Khammam, Andhra Pradesh, India Herein we report a rare case of CG of superfi cial suppu- rative type in a 43-year-old Indian female, successfully treated 4Department of Oral Medicine and Radiology, Panineeya Institute of Dental Sciences, Hyderabad, Andhra Pradesh, India with intralesional steroid injections. Corresponding Author: Rezwana Begum Mohammed Assistant Professor, Department of Oral Medicine and CASE REPORT Radiology, Gitam Dental College and Hospital, Rushikonda Visakhapatnam-530045, Andhra Pradesh, India, Phone: A 45-year-old Indian female reported with burning sensation 07382406226, Fax: 08912790033, e-mail: rizwana6603@ and ulceration of lower lip since 1 year. She had history of gmail.com trauma to lower lip region 2 years back which later healed 510 OMPJ Cheilitis Glandularis Treated with Intralesional Steroids: A Rare Case Report and Review with pigmentation. After a year, she developed a small ulcer (Figs 1 to 3). Right submandibular lymph nodes were pal- in the middle of lower lip and it spread to the corner of mouth pable but asymptomatic. Oral mucosa was not affected. on both sides. She also noticed swelling of lip with frequent On the basis of above clinical fi ndings, it was provisio- alteration in size. Even on slight touch, there was bleeding nally diagnosed as CG affecting lower lip. Differential diag- from lip region. During night times, lips stick together and nosis included was actinic cheilitis, cheilitis granulomatosa early morning she had severe pain and bleeding with dry, and syphilitic ulcer. Routine blood investigations performed cracked lower lip. She consulted general physician for the were within normal limits except for elevated erythrocyte same problem but the lesion did not subside on using the sedimentation rate. C-reactive protein levels, serum glucose, medication prescribed. She was not allergic to any drugs. hepatic and renal parameters were normal. The treponema She also revealed that she was a house wife with no frequent pallidum hemagglutination test, the HIV-screening test sun exposure. was nonreactive. Incisional biopsy was done to confi rm On clinical examination, lips were dry, incompetent the diagnosis. and lower lip presented with well-defi ned solitary ulce- Histopathology revealed nonspecific inflammatory rated lesion above vermillion border of size 5 × 1 cm with infi ltration with lymphocytes, plasma cells and neutrophils. bloody crustations. Lower lip was infl amed and swollen. The labial glands were hyperplastic with focal infi ltration On palpation, lower lip was edematous, soft in consistency of lymphocytes and plasma cells with ductal dilatation and with exudates over the lesion and severe bleeding from mucin pooled areas (Fig. 4). Treatment consisted of adminis- the cracks even on slight touch with moderate tenderness tration of intralesional steroid injections of dexamethasone Fig. 1: Extraoral photograph showing ulcerated lower lip Fig. 2: Infl amed, swollen, edematous lower lip with exudates over the lesion Fig. 3: Severe bleeding from the lower lip even on slight touch Fig. 4: Photomicrograph showing nonspecifi c infl ammatory infi ltration with lymphocytes, plasma cells and neutrophils. The labial glands were hyperplastic with focal infi ltration of lymphocytes and plasma cells with ductal dilatation and mucin pooled areas (H & E, ×10) Oral and Maxillofacial Pathology Journal, July-December 2014;5(2):510-513 511 Rezwana Begum Mohammed et al 2 ml weekly twice for 1 month along with analgesics and tracts.6,7 The latter two types have the highest association topical antimicrobial metronidazole gel. The lesion resolved with dysplasia and carcinoma. spontaneously within 3 months and recurrence was not Differential diagnosis may include actinic cheilitis, noticed during 3 years of regular follow-up (Figs 5 and 6). atopic dermatitis, angioedema, exfoliative cheilitis, chronic sialadenitis of the minor salivary glands, factitious cheilitis, DISCUSSION and cheilitis granulomatosa.17 Reported cases of CG were associated with other clinical conditions. Stoopler et al8 Cheilitis glandularis is a rare disorder characterized by reported a papillary cystadenoma like ductal growth pattern swelling of the lip with hyperplasia of the labial salivary in one patient with CG. Musa et al2 reported a case with a glands. Sutton stated that the characteristic lip swelling was attributable to a congenital adenomatous enlargement more generalized presentation of CG, termed as suppurative 18 of the labial salivary glands.14 Swerlick and Cooper stated stomatitis glandularis. Reichart et al reported a retention that there was no evidence to support the assertion that sub- cyst in the upper lip of an elderly patient with the ‘simplex’ 1 mucosal salivary gland acinar hyperplasia is either responsible form of CG. Leao et al reported a case with CG which was for or a consistent feature of established CG.15 later discovered to have undiagnosed HIV infection. Butt 19 The etiology of CG is still unknown, but it has been et al reported a case of CG that progressed to squamous cell 20 suggested to be an autosomal dominant hereditary disease carcinoma in an HIV-infected patient. Carrington and Horn for which smoking, chronic irritation, poor oral hygiene, reported a case of CG related to actinic damage following allergy, bacterial infections, syphilis,
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