INTRODUCTION
In 1952, Dr. Zoon described a dermatological condition which he termed as “plasma cell infiltrate” which had unique characteristic, presented histologically as a dense infiltration of plasma cells in the subepithelial connective tissue.[123] Gingiva is a rare site for its presentation, making it a clinical dilemma. This benign inflammatory condition of the gingiva has got various synonyms such as plasma cell gingivitis (PCG), idiopathic gingivostomatitis, plasma cell gingivostomatitis,[4] atypical gingivostomatitis,[5] allergic gingivostomatitis,[6] and plasmacytosis of the gingiva.[7] PCG can be classified on the basis of its etiology into three categories: PCG due to allergens, due to neoplastic origin, and due to unknown cause.[8] The most common causative agents are the allergens known to cause hypersensitivity reaction in the tissue. Many such allergens are cinnamon, cinnamaldehyde, red pepper, chewing gums, mint, certain components of toothpaste,[910] and khat leaves.[11] PCG clinical presented as reddish and friable gingiva which easily bleeds on provocation. On examination, PCG shows the pathognomic feature of sharply demarcated erythematous and edematous gingiva which may extend to the mucogingival junction. This lesion clinically mimics acute leukemia whereas histologically imitates extramedullary plasmacytoma and multiple myeloma. Thus, it is necessary to examine the case of PCG – clinically, histologically as well as hematologically.[12] This case report elaborates on the diagnosis and management of a rare case of PCG which reported to the department of periodontology.
CASE REPORT
A 29 year old female patient reported to the Department of Periodontology with a chief complaint of bleeding from the gums on the slightest provocation which is bright red for 11 months. She was also complaining of poor esthetics due to redness and swollen gums in upper and lower front tooth region. She noticed that the size of the gums increased gradually to the present size over a period of time. On further elicitation, there was no history of trauma and medical history was nonrelevant. Personal history and family history were noncontributory. General physical examination did not show any signs or symptoms suggestive of any systemic illness.
The intraoral examination revealed that gingiva was bright fiery red, highly inflamed, including marginal, papillary, and attached gingiva extending from 13 to 23 in the maxillary arch and 33 to 43 in the mandibular arch [Figure 1]. The gingiva was edematous which showed profuse bleeding on slight or gentle probing and loss of stippling and contour was noticed. The gingiva on the posterior teeth was relatively less edematous for both upper and lower arches. Minimal plaque deposition was noticed around affected teeth in maxillary and mandibular anterior sextants. Orthopantomograph examination revealed minimal to no bone loss. Blood investigations included total leukocyte count, differential leukocyte count, hemoglobin, platelet count, blood glucose levels, blood coagulation test such as PT, aPTT, INR, IgE levels, IgG levels, and IgG subclass 4 levels. All were reported within normal limits. Urine examinations were within normal limits, showing no pathological changes. Blood estradiol and progesterone levels were within normal limits (luteal phase).
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Figure 1: Arrow showing preoperative, bright fiery red, highly inflamed, edematous gingiva including marginal, papillary, and attached gingiva extending from 13 to 23 in the maxillary arch and 33 to 43 in the mandibular arch
Based upon the history and clinical findings, the patient was provisionally diagnosed as a case of inflammatory gingival overgrowth. A biopsy was taken from the affected attached gingiva, i.e., 22 and 32 regions. The tissue sample was transported in 10% formalin for histopathological examination. The patient was recalled after 1 week, and healing of the biopsy site was checked.
Phase I therapy was carried out after ensuring the healing of the biopsy region. The patient was educated and motivated for maintaining proper oral hygiene. The patient was on maintenance.
Histopathological examination comprising of hematoxylin and eosin-stained sections under ×10 magnification showed stratified squamous lining epithelium exhibiting marked acanthosis with minimal keratinization along with anastomosing rete pegs. The subepithelial tissue was edematous and showed lymphomononuclear inflammatory infiltrate. Higher magnification (×40) of sections revealed predominant plasma cell infiltrate having the characteristic eccentrically placed nucleus [Figure 2]. Immunohistochemistry (IHC) with CD38 [Figure 3] and CD138 [Figure 4] was positive and plasma cells did not show any light chain restriction with immunohistochemical staining for kappa and lambda chains [Figures 5 and 6]. Sections did not show any abnormal or malignant cells. The patient was also investigated for IgG subclass 4, which came negative [Figure 7]. Based on the histopathological examination and IHC study, the final diagnosis of “PCG” was given [Figure 8].
Figure 2: Histopathological examination sections under ×40. Arrow showing predominant plasma cell infiltrates having the characteristic eccentrically placed nucleus
Figure 3: Photomicrograph showing immunohistochemistry with CD38
Figure 4: Photomicrograph showing immunohistochemistry with CD138
Figure 5: Photomicrograph showing positive immunohistochemistry with CD38. Plasma cells do not show any light chain restriction with immunohistochemical staining for kappa and lambda chains
Figure 6: Photomicrograph showing positive immunohistochemistry with CD138 and plasma cells do not show any light chain restriction with immunohistochemical staining for kappa and lambda chains
Figure 7: Serum IgG immunoglobulin subclass 4 test – negative
Figure 8: Final diagnosis based on histopathological examination and immunohistochemistry study – plasma cell gingivitis
After the diagnosis was confirmed, the patient was advised to use the topical application of steroids (0.1% Triamcinolone acetonide – TESS®, thrice daily for 4 weeks and tapered gradually) and antihistaminic (tablet Levocetirizine 10 mg twice daily for 14 days). Clinically, there was an excellent resolution of inflammation. The patient was advised to maintain strict oral hygiene and was recalled for review for 2 years. At present, the patient is asymptomatic and on regular recalls [Figure 9].
Figure 9: Arrow showing condition of gingiva – 2-year postoperatively
DISCUSSION
PCG is a rare condition which histologically presents as a massive infiltration of plasma cells in the subepithelial gingival tissue. On clinical examination, the condition presents as an erythematous diffused gingival enlargement with edema in the marginal and complete width of the attached gingival of the maxillary and mandibular anterior tooth region. It is considered to be a type of hypersensitivity reaction to an allergen, but the exact cause of the plasma cell infiltration is not known. The occurrence of PCG is commonly in the maxillary anterior region. In 1995, PCG was classified by Sollecito et al. into three types on the basis of their etiology: [8] Type 1 – caused by an allergen, Type 2 – neoplastic nature, and Type 3 – unknown origin. According to the above-mentioned classification, this case report discusses a Type 3 PGC. This case report also presents pronounced erythematous enlargement of the maxillary and mandibular anterior region covering almost complete width of the attached gingiva till the mucogingival junction whereas plaque-induced gingivitis would only involve the marginal gingiva and not the entire width of attached gingiva. In the present case, gingiva responded to local therapy, i.e., scaling and root planing but did not resolve the complete inflammation where in case of plaque-induced gingivitis, it would have resolved completely and hence inconsistent with a plaque-related etiology.
Biopsy of the lesion which is confirmatory to the diagnosis helped to rule out oral granulomatous lesions and other red lesions.[13] Blood pictures showed the absence of any infections and hematologic malignancies. A study done by Vickers and Hudson also suggested candida albicans to be one of the etiological factors; however, microscopic examination of the biopsy showed an absence of fungal hyphae.[14] Román et al. 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.[3]
Plasma cell lesions of head-and-neck possess great diagnostic and therapeutic challenge. Plasma cell lesions of head and neck mainly categorized into four types: multiple myeloma, solitary myeloma (solitary plasmacytoma of bone), soft-tissue myeloma (extramedullary plasmacytoma), and plasma cell granuloma. The first two multiple myeloma and solitary myeloma are tumors of bone while the soft tissue myeloma and PCG are lesions of the soft tissue origin. It is very important to differentiate the last two soft-tissue lesions, as the soft-tissue myeloma (plasmacytoma) may represent early stages of multiple myeloma also.[15]
IHC examination of the biopsy with markers such as CD38 and CD138 with kappa and lamba gives confirmatory results toward PCG. This disease can be differentiated from various vesiculobullous lesion such as pemphigus, which is a desquamative lesion, auto-immune disease such as lichen planus which has histopathological signs such as saw-shaped rete pegs, acanthosis, supra basilar split, which are absent in PCG. Leukemia can be differentiated using hematological examination and plasma cell infiltrates in the histopathological examination of PGC differentiates it from multiple myeloma and plasmacytoma.[16]
The clinical, histologic, and immunological findings were suggestive of case of PCG. The case was managed with a formulated treatment plan which comprised of Phase I therapy: nonsurgical periodontal therapy, scaling, and root planing for the removal of the plaque followed by Phase IV for a re-evaluation of plaque and calculus and also reinforce oral hygiene practice which is very important for patients with such conditions.
Definite treatment regimen consists the use of topical application of steroids (0.1% Triamcinolone acetonide – TESS®, thrice daily for 4 weeks, which was tapered gradually over a period of 2 months) and antihistaminic (tablet Levocetirizine 10 mg twice daily for 14 days). The patient was recalled after every week for assessment. Post 4 weeks complete resolution of the signs and symptoms were seen. 24-month postoperation showed stable result and the patient is kept under follow-up and periodic recall is being carried out.
CONCLUSION
PCG 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, histological, and hematological examinations are mandatory to exclude leukemia and other local manifestations of systemic diseases. Although 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.
PCG 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. In this case report, diagnosis of Type-3 PGC was made with the help of immunohistopathological examination ruling out other probabilities and managed using topical steroid and antihistaminic resulting in asymptomatic condition and resolution of all the signs and symptoms which was followed up for 2 years.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
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