Lata Goyal, MDS; Shivani Bansal, MD, DNB, MNAMS; Arshad Ernakulum, MDS; and Manjit Kaur Rana, MD, DNB, MNAMS
Plasma cell gingivitis and cheilitis are diagnostic challenges for dental practitioners. To the best of the authors' knowledge, concomitant plasma cell gingivitis and plasma cell cheilitis in the same patient has not been reported in the dental literature, although these conditions have been reported separately (on lips and gingiva) several times. Underreporting of these coexisting clinical conditions may be attributed to a lack of awareness of both conditions and underperformance of comprehensive histopathological examinations. This article describes a case of plasma cell gingivitis with plasma cell cheilitis in a 63-year-old female patient. Histopathology and immunohistochemistry evaluations confirmed the diagnosis. After being informed about her condition and educated about possible allergens, which could be present in certain toothpastes, foods, and other foreign substances, the patient responded well to the stoppage of the use of suspected allergens. Identification and elimination of causative allergens is considered the accepted treatment for these conditions.
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Plasma cell gingivitis and plasma cell cheilitis are benign, inflammatory conditions that are found infrequently on the gingiva and commisures of the lips. Their estimated prevalence is unknown. In these conditions, the subepithelial connective tissue is infiltrated with plasma cells.1 Plasma cell infiltration has been reported in various body parts with mucosal coverings, including the lips, tongue, larynx, epiglottis, vulva, conjunctiva, and nasal aperture.1 The exact cause of such plasma cell infiltration is unknown, although it may be due to allergen exposure causing local hypersensitivity reactions. These allergens may be present in toothpaste, chewing gum, food, and/or other foreign substances.2 Flavoring agents like cinnamon and cinnamaldehyde used in toothpaste or chewing gum, for example, act as allergens and have demonstrated increased risk of oral symptomatology associated with their consumption.
Besides allergens as a possible cause, these conditions can also be of benign neoplastic origin, or the cause may be unknown.3 Plasma cell gingivitis associated with cheilitis is considered an infrequent condition. A thorough literature search shows case reports of the former condition, but none of them mentioned concomitant plasma cell cheilitis, although it is possible that cheilitis went unrecognized in these reports. Hence, this article reports a rare case of plasma cell gingivitis associated with cheilitis.
A 63-year-old female patient presented to the AIIMS Department of Dentistry in Bathinda, India, complaining of a burning sensation on her lips and gums for the past 5 years. She was using topical corticosteroids to treat the condition with no relief. The patient was referred from the Department of Dermatology, where she was evaluated for a reddish discoloration of the lower lip. Her medical history was unremarkable. On extraoral examination, erythematous plaque with a glazed surface was present on the lower lip. Intraorally, generalized gingival erythema was present (Figure 1). Gingival stippling was absent and gingival tissues easily bled on probing. Calculus deposits were present on the lingual of the lower anterior region. The patient was diagnosed to have generalized stage IV grade B periodontitis4 and generalized gingival recession with mean gingival recession depth of 3.38 mm. The rest of the examination was normal.
Routine blood investigations, including hemogram, liver function, and kidney function tests, were within normal range, and panoramic radiography demonstrated no noted pathology. Incisional biopsies were performed at the affected gingiva site (in the region of teeth Nos. 13, 27) and lower lip site (middle part), including marginal gingiva and attached gingiva. The biopsies were placed in 10% formalin in a sterile container and sent for histopathological examination.
On histopathological examination of the specimen taken from the gingiva, intact stratified squamous lining epithelium was present. The subepithelial tissues exhibited diffuse and marked infiltration by predominantly mature plasma cells and lymphomononuclear cells. No atypia or necrosis was noted in the tissue samples. The above-mentioned features were indicative of plasma cell gingivitis (Figure 2). In the specimen taken from the middle part of the lower lip, the dermis showed dense infiltration of chronic inflammatory cells comprised of predominantly plasma cells admixed with a lesser number of lymphocytes suggestive of plasma cell cheilitis (Figure 2). To confirm the diagnosis, an immunohistochemical examination was conducted, which showed both lip and gingiva positivity for kappa (κ) and lambda (λ) immunoglobulin light chain expression. Therefore, the final diagnosis of plasma cell gingivitis and plasma cell cheilitis was confirmed (Figure 3).
After informed consent was obtained from the patient, treatment began with nonsurgical therapy comprising thorough scaling and root planing, as pocket depths were less than 4 mm. The patient was advised to maintain proper oral hygiene. She was also advised to stop the use of products with suspected allergens that can cause a hypersensitivity reaction. As stated earlier, these may include cosmetic products, chewing gums, and nonessential additives used in food. The patient was also instructed to change her herbal toothpaste, which she used regularly for oral hygiene, since its ingredients might also act as a causative factor in plasma cell gingivitis and plasma cell cheilitis.2
The patient stringently adhered to these instructions and demonstrated complete resolution of symptoms of both gingiva and lips within 3weeks (Figure 4).
In 1986, White et al grouped these conditions-plasma cell gingivitis and plasma cell cheilitis-under plasma-cell orofacial mucositis (PCM).1,5 This group encompasses the inflammatory condition that has plasma cells in histopathology occurring at the oral mucosa and upper aerodigestive tract. Plasma cell gingivitis and plasma cell cheilitis can be considered benign PCM subtypes.6 The defining characteristic of both conditions is the presence of plasma cell infiltrates in the connective tissue.7 Based on etiology, Gargiulo et al further categorized plasma cell gingivitis into three categories: that which is caused by allergens, that which is of neoplastic origin, and that which is of an unknown cause.8
Identifying the allergen or hypersensitivity-causing agent is one of the main factors in diagnosing and managing this condition. Clinical, histopathologic, and hematologic screening should be done to rule out any other pathology. Plasma cell gingivitis usually presents as inflammation or enlargement in the anterior gingiva; clinically, its classic feature is a non-ulcerated, fire-engine red appearance of the attached gingiva. In the present case, increased enlargement of the facial gingiva was present, especially in mandibular anterior regions. Plasma cell cheilitis typically presents with erythematous plaque, erosion, fissuring, bleeding, and crusting on lip mucosa.
As far as the authors know, plasma cell gingivitis has been reported with cheilitis but not specifically with plasma cell cheilitis. Prasanna et al reported a case of plasma cell gingivitis along with cheilitis in which lip lesions subsided following gingival therapy.9 Chauhan et al reported a case of plasma cell gingivitis and cheilitis in which lip lesions subsided following the gingivectomy procedure.3 A case by Janam et al showed plasma cell gingivitis and upper lip swelling, in which the lip swelling subsided after gingivectomy and oral prophylaxis.10 In the present case, the lip lesion subsided following the remission of lesions on the gingiva, indicating that both occurred due to hypersensitivity similar to contact dermatitis, as reported by Abhishek et al.11 It should be noted that in all of these previous reports, histopathological examination revealed no presence of plasma cells in lip lesions.
Regarding diagnosis and treatment, the treatment of both conditions is challenging. Plasma cell gingivitis is a nonspecific entity, and clinical features resemble several clinical conditions like oral clinical lichen planus, gingival enlargement, erythematous gingiva, granuloma, and mucous membrane pemphigoid.12-14 Medical and clinical history, clinical presentation, and histopathological examination are of utmost importance in identifying and managing this condition.15
Management of plasma cell gingivitis and plasma cell cheilitis involves both medical and surgical methods. Besides elimination of allergens, medical management primarily includes the use of topical corticosteroids, intra-lesional steroid application, systemic steroids, and antibiotics. Tissue destruction using cryotherapy, electrocoagulation, ablative lasers, surgical excision, radiation therapy, and photodynamic therapy have also been used.
In the present case, the patient was advised to stop the use of suspected allergens and comply with follow-up evaluations.16 Within 3 weeks, the patient was completely relieved of symptoms, which may indicate that elimination of allergens is an important treatment modality that should be considered before prescribing anything. Moreover, preventive therapies such as ozone, photobiomodulation, and probiotics may be appropriate future avenues of investigation in order to understand their effects on plasma cell gingivitis.17-19
The concomitant presentation of plasma cell gingivitis and plasma cell cheilitis is a diagnostic challenge for clinicians because of the clinical resemblance to other entities. This condition is believed to be a nonspecific, inflammatory response to an unknown exogenous agent. Careful history, histopathological, and immunohistochemistry examinations are mandatory for a confirmatory diagnosis. To achieve complete resolution, identification of the suspected allergen is essential. In the present case, the patient was educated about this condition and all the possible allergens that could cause it and advised to refrain from them, which led to resolution of her situation.
Associate Professor, Periodontology Division, Department of Dentistry, All India Institute of Medical Sciences (AIIMS), Bathinda, Punjab, India
Assistant Professor, Department of Dermatology, All India Institute of Medical Sciences (AIIMS), Bathinda, Punjab, India
Senior Resident, Department of Dentistry, All India Institute of Medical Sciences (AIIMS), Bathinda, Punjab, India
Additional Professor, Department of Pathology, All India Institute of Medical Sciences (AIIMS), Bathinda, Punjab, India
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