|Year : 2012 | Volume
| Issue : 1 | Page : 115-119
Plasma cell gingivitis associated with cheilitis: A diagnostic dilemma!
Presanthila Janam, Bindu R Nayar, Remya Mohan, A Suchitra
Department of Periodontics, Government Dental College, Trivandrum, Kerala, India
|Date of Submission||15-Jun-2010|
|Date of Acceptance||09-Nov-2011|
|Date of Web Publication||3-Apr-2012|
Department of Periodontics, Government Dental College, Trivandrum, Kerala
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Plasma cell gingivitis is a rare condition characterized by diffuse and massive infiltration of plasma cells into the sub-epithelial connective tissue. Clinically, it appears as a diffuse reddening and edematous swelling of the gingiva with a sharp demarcation along the mucogingival border. Though considered as a hypersensitive reaction to an allergen, the etiology of this bizarre condition is still not properly understood. Here, we present an interesting case of plasma cell gingivitis associated with an enlarged and fissured upper lip, which is quite a rarity. The condition was diagnosed based on clinical and histopathologic findings and treated by gingivectomy. The associated cheilitis has dramatically reduced after treatment of the gingival lesion.
Keywords: Cheilitis, gingivectomy, hypersensitivity, plasma cell gingivitis
|How to cite this article:|
Janam P, Nayar BR, Mohan R, Suchitra A. Plasma cell gingivitis associated with cheilitis: A diagnostic dilemma!. J Indian Soc Periodontol 2012;16:115-9
|How to cite this URL:|
Janam P, Nayar BR, Mohan R, Suchitra A. Plasma cell gingivitis associated with cheilitis: A diagnostic dilemma!. J Indian Soc Periodontol [serial online] 2012 [cited 2016 May 5];16:115-9. Available from: http://www.jisponline.com/text.asp?2012/16/1/115/94618
| Introduction|| |
Plasma cell gingivitis is a rare benign condition of the gingiva characterized by sharply demarcated erythematous and edematous gingivitis often extending to the mucogingival junction. , Other terminologies previously used for this unusual type of conditioned enlargement were atypical gingivitis,  allergic gingivitis,  plasmacytosis of the gingiva,  plasma cell-gingivostomatitis  and idiopathic gingivostomatitis. A localized lesion called plasma cell granuloma has also been reported . Plasma cell gingivitis is considered a hypersensitive reaction to some antigen, often to chewing gum, toothpaste, and other foreign substances. ,,,,, The allergens identified were mostly cinnamonaldehyde and cinnamon, used as flavoring agents in chewing gums and dentifrices. ,, Clinically, the gingiva appears red, friable and bleeds easily. Histopathologically, a dense and massive infiltration of mature plasma cells in the lamina propria is noted.  Gingival ulceration is rare. 
Lesions may mimic that of acute leukemia and histologically imitate multiple myeloma and extramedullary plasmacytoma. Hence, the diagnosis requires hematological screening in addition to clinical and histopathological examinations. Plasma cell gingivitis is purely benign, and the detection and elimination of the exposure to the antigenic agent will bring about the remission of the condition. However, the allergen in most cases is unknown, despite extensive allergy testing. Plasma cell gingivitis associated with cheilitis is a very rare condition, and only a few cases have been reported in the literature. Kerr et al. in 1971 gave an excellent report on patients presented with a triad of plasma cell gingivitis, cheilitis and glossitis due to allergic reactions to chewing gum additives.  We are reporting a case of plasma cell gingivitis associated with an enlarged upper lip in an otherwise healthy patient, and not related to any known allergen.
| Case Report|| |
A 15 year old female patient presented to the Department of Periodontics, Government Dental College, Trivandrum, with the chief complaint of a swollen upper lip and enlarged gums [Figure 1] and [Figure 2]. The patient first noticed a mild reddish discoloration involving her upper lip around 5 years back, which progressively increased in size without any apparent discomfort. Gingival changes started at around the same time, the gingiva appearing fiery red in color and friable with an edematous consistency and granular surface texture. There was no associated pain; however, bleeding on brushing was noted. The patient's medical history was unremarkable except for a case of mild dust allergy. She also reported about the occasional appearance of an isolated red patch on her face on excessive exposure to sunlight.
|Figure 1: Extraoral appearance of an enlarged and fissured upper lip in the patient of the study|
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|Figure 2: Characteristic appearance of the gingival enlargement in the patient of the study|
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Excision of the gingival lesion of the upper arch was done 4 years back; however, it gradually reappeared after 1 year. Patient couldn't associate the recurrence of symptoms with the use of any specific food, drug, cosmetic items or dentifrice. She was previously treated for the lip lesion by the physician with a course of systemic and topical steroids for 1 month without any perceivable improvement of the condition, and hence the treatment was stopped as per medical advice.
On clinical examination, the patient was systemically healthy without any apparent skin lesions. Her lips were incompetent; and, irregular verrucoid plaque lesions were noted on the inner aspect of both lips. The upper lip appeared edematous and dry with exaggerated fissures [Figure 1]. Intra oral examination showed generalized enlargement of marginal and attached gingivae [Figure 2]. Abundant plaque deposits were also present. Panoramic radiograph couldn't detect any bony pathology, and there were no changes in the lamina dura, periodontal ligament space and alveolar bone crest [Figure 3]. Investigations included routine blood examinations, peripheral smear and biochemical screening, and all were found to be within normal limits. Ultrasonography of upper lip could not spot any focal lesions or abnormalities. However, an incisional biopsy revealed a chronic inflammatory lesion. Bone marrow aspiration cytology and bone trephine showed normal patterns. A provisional diagnosis of generalized chronic inflammatory gingival enlargement was established, and treatment was planned. Differential diagnoses included plasma cell gingivitis and solitary plasmacytoma for the gingival lesion; and, cheilitis granulomatosa, dermatitis venenata, plasma cell cheilitis and actinic cheilitis for the lip lesion.
|Figure 3: Pre treatment panoramic radiographic view of the patient in the study|
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Non surgical therapy included thorough oral prophylaxis, oral hygiene instructions and an antihistamine mouthwash. The patient was instructed to avoid possible allergens like chewing gums, cosmetics and food additives. A strict elimination diet was advised and a change of the regular toothpaste was tried. Herbal toothpastes were not prescribed since reports have shown them as an etiologic factor in the initiation of these types of lesions.  Though all these modalities were followed stringently, there was no true regression of the disease. Hence, a surgical approach was planned for the gingival lesion. External bevel gingivectomy was done in all six sextants and the specimens were sent for histopathological examination [Figure 4]a-f. Coe-pak was placed and post operative antibiotics and analgesics were prescribed.
|Figure 4: Surgical views of maxillary and mandibular anterior sextants of the patient in the study. (a) Pre surgical view; (b) After gingivectomy; (c) Excised tissue; (d) Pre surgical view; (e) After gingivectomy; (f) Excised tissue|
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Histological examination showed hyperplastic parakeratinized stratified squamous epithelium proliferating into the connective tissue in an arcading pattern. The connective tissue was densely collagenous with focal dense collections of chronic inflammatory cells, chiefly plasma cells. There was increased vascularity with dilated and engorged blood vessels [Figure 5]. The histologic appearance was compatible with plasma cell gingivitis.
|Figure 5: Histopathological appearance (hematoxylin and eosin staining) showing infi ltrate of plasma cells in a dense collagenous stroma. (a) Low power view (×10); (b) High power view (×40)|
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After 2 weeks of excision, the surgical site was healing and the lesion showed remission; however, the gingiva appeared reddish and atrophic even after one month [Figure 6] and [Figure 7]. Most interestingly, there was a dramatic reduction in the lip enlargement without any additional therapeutic procedures [Figure 8] and [Figure 9]. It remained in the same condition for the next 8 months, while the gingival lesions showed mild recurrence [Figure 10] and [Figure 11]. Now the patient is under regular follow up.
|Figure 9: An evident reduction in the lip size post treatment compared to the appearance before the treatment in the patient of the study|
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|Figure 10: Recurrence of lesion after 8 months correlating with local irritants in the patient of the study|
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| Discussion|| |
Plasma cell gingivitis is a rare condition characterized by a massive infiltration of plasma cells into the sub-epithelial gingival tissue. Clinically, the illness presents as a diffuse enlargement with oedematous swelling of the gingiva in the maxillary and mandibular anterior segments. Although the exact mechanism behind this condition is not known, the presence of plasma cells suggests an allergic origin. However, most of the cases seen are without any known or identifiable causes. Gargiulo, Timms et al. in 1995 classified Plasma cell gingivitis into 3 types  - caused by an allergen, neoplastic nature and of unknown origin. The case report which we depicted here also is of unknown etiology, and hence belongs to type 3. Plasma cell gingivitis usually occurs in the anterior gingiva, most frequently in the maxilla. Even in our case, the patient showed pronounced enlargement of the facial gingiva in the anterior maxillary and mandibular regions.
Plaque induced gingivitis would normally involve the marginal gingiva alone and not the entire width of attached gingiva. In the present case, there was an inflammation of marginal and attached gingiva which was not responding to local therapy, and hence inconsistent with a plaque related etiology. Biopsy had helped to rule out oral granulomatous lesions. Blood picture and bone marrow tests cleared doubts about hematologic malignancies. Vickers et al. suggested candida albicans as an etiological factor; however, an absence of fungal hyphae under microscopic examination excluded any such infection in this case.
Differential diagnoses for the lip lesion included cheilitis granulomatosa, dermatitis venenata, actinic cheilitis and plasma cell cheilitis. Plasma cell cheilitis is a very rare and benign inflammatory mucosal condition characterized by a dense plasma cell infiltrate within the mucosa. The classic clinical appearance is a flat-to-slightly raised, eroded area, usually on the lower lip of an elderly patient.  Plasma cell cheilitis was ruled out here because of a lack of erosion or crusting and also as there was enlargement of the upper lip alone. Granulomatous cheilitis was ruled out by biopsy. Actinic cheilitis was excluded since this usually involves the external aspect of lips. Dermatitis venenata is too in question since the lesion can be due to a contact allergy to an antigen which is shared by the gingival lesion.
Kerr et al. reported occurrence of plasma cell gingivitis, cheilitis and glossitis in patients, and identified the culprit as the cinnamonaldehyde in chewing gums. The condition regressed completely on discontinuity of the use of these gums.  Silverman and Lozada described plasma-cell gingivostomatitis as a syndrome, consisting of gingivitis, cheilitis, and glossitis.  In the above case, when the clinical and histological findings are correlated, the case can be diagnosed as a part of plasma cell mucositis. Mucous membrane plasmacytosis of the upper aerodigestive tract is a rare, benign disorder in which the mucous membranes are infiltrated by plasma cells.  Cases have been reported about plasma-cell infiltrates on the buccal mucosa, palate, nasal aperture, gingiva, lips, tongue, epiglottis, larynx, and other orificial surfaces. In 1986, White et al. grouped all such lesions under the name plasma-cell orificial mucositis because all the cases reported had clinical and histologic findings that were indistinguishable from one another. Management of plasma cell mucositis involves both medical and surgical approaches. Although several treatment modalities have been tried, including corticosteroids (topical, intralesional, and systemic), antibiotics, destruction of the tissue (liquid nitrogen, Carbon dioxide laser and electrocoagulation), excision of the tissue, and radiation therapy, no treatment clearly stands out as consistently effective.
The patient in this case report had not responded to topical or systemic corticosteroids and hence was managed surgically. The cheilitis which was very evident previously has regressed spontaneously after gingivectomy, and has shown no recurrence till date.
| Conclusions|| |
Plasma cell gingivitis is a diagnosis of exclusion, distinguished primarily by the histologic finding of a marked submucosal plasma-cell infiltrate, after conditions such as infection and plasmacytoma have been eliminated. Careful history taking, biopsy and hematological examinations are mandatory to exclude leukemia and other local manifestations of systemic diseases. Though recurrences are common, no studies till date report a progression of this condition to a malignancy of any type. The condition is believed to be a nonspecific inflammatory response, in the form of a plasma-cell infiltrate, to an unknown exogenous agent. However attempts to induce plasma-cell infiltrations on mucosal and nonmucosal surfaces by allergic and irritant stimuli were not successful. Roman hypothesized that plasma-cell gingivitis may be associated with low levels of serum IgA and secretory IgA, which allows localized, repetitive, subclinical infections that could lead to the plasma-cell infiltrate.
In this case, the gingivitis and cheilitis suggest a contact allergy to some antigenic agent which is yet to be found out. The patient should be regularly followed up to assess oral hygiene maintenance as well as identification of a possible allergen to avoid recurrences. During the quest for a true allergen, repeated surgical excisions may be required for the gingival enlargement to facilitate plaque control. Plasma cell gingivitis should be included in the differential diagnosis of nonspecific enlargements of the gingiva with characteristic clinical and histological appearance, which cannot be attributed to any other disease entity. This condition, without any doubt, presents as a diagnostic dilemma and therapeutic challenge to the specialist.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]