Case Report
An Uncommon Case of Plasma Cell Mucositis of the Tongue in a Young Man
Alessandro Antonelli ,1Fiorella Averta,1Federica Diodati,1Danila Muraca,1 Ylenia Brancaccio,1Chiara Mignogna,2and Amerigo Giudice 1
1School of Dentistry, Department of Health Sciences,“Magna Graecia” University of Catanzaro, Italy
2Interdepartmental Service Center,“Magna Graecia” University of Catanzaro, Italy Correspondence should be addressed to Alessandro Antonelli; antonellicz@gmail.com Received 3 December 2019; Accepted 5 February 2020; Published 19 February 2020 Academic Editor: Evanthia Chrysomali
Copyright © 2020 Alessandro Antonelli et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Plasma cell mucositis (PCM) is an unusual plasma cell proliferative disorder of the upper aerodigestive tract. It is a rare disease, and its etiology is not yet known with variable clinical features. Symptoms include dysphagia, oral pain, and swelling. We described a case of PCM involving the tongue of a 14-year-old man. In thefirst place, several diagnostic hypotheses were proposed, most of them discarded for incompatibility with blood and laboratory tests. This disease rarely manifests itself on the tongue, especially in young patients with no comorbidities. The management of PCM is mainly aimed at reducing the symptoms, and in our report, the treatment involved the use of systemic prednisone with an improvement of the quality of life. At 1-year follow-up, there was no recurrence of the disease. Many therapeutic treatments are able to stabilize but not able to induce a complete remission. PCM is considered an uncommon benign disorder with a favorable prognosis and should be considered in the differential diagnosis with other inflammatory or neoplastic conditions.
1. Introduction
Plasma cell mucositis (PCM) is an unusual plasma cell prolif- erative disorder of the upper aerodigestive tract [1]. In the past, this pathology has been reported under various names.
The name indicated the anatomical structure involved together with the component of plasma cells, such as idio- pathic plasmacytosis of the gingiva, plasma cell vulvitis, oral papillary plasmacytosis, or mucous membrane plasmacytosis of the upper aerodigestive tract [2]. It is a rare disease, and its etiology is not yet known; it is considered a benign condition of adults, and there are no correlations in literature with the development of plasma cell neoplasm. Clinical features are an intensely erythematous mucosa with papillomatous, cob- blestone, nodular, or velvety surface changes. Symptoms include dysphagia, oral pain, sore throat, and pharyngitis [2]. PCM can be treated with corticosteroids, administrated topically, systemically, or intralesionally. Usually, the treat- ment is not resolutive; however, the main outcome is the
improvement of the symptoms. Generally, PCM patients have a previous history of an autoimmune or immunologi- cally mediated disease like Sjögren syndrome or possible autoimmune hepatitis; however, these are not present in all cases and no single disease is consistently associated.
2. Case Report
We described a case of PCM involving the tongue of a 14-year-old man. The patient was referred to the Oral Pathology Unit at the Faculty of Dentistry, Magna Graecia University of Catanzaro, in August 2019. The referring prac- titioner suspected squamous cell carcinoma. His past medical history was not relevant, and the patient was a nonsmoker and nonalcohol user. The patient had previously used a noc- turnal bite to control bruxism. He reported a burning sensa- tion in his mouth, local dysgeusia, and pain on the tip and on the right lingual border for 1 year. The patient also referred that he was unable to eat or drink any hot or spicy foods
https://doi.org/10.1155/2020/3429632
due to burning and pain on his tongue. He stated persistent hoarseness, sore throat, and difficulty sleeping due to contin- uous oral pain.
Extraoral examination was unremarkable, while intraoral examination revealed the presence of an ulcer and intense erythema of the tongue (Figures 1 and 2) and gingival edema and erythema (Figure 3); however, the other parts of the oral mucosa was clinically normal. In thefirst place, several diag- nostic hypotheses were proposed, most of them discarded for incompatibility with full blood count, serum B12 and folate, urea and electrolytes, liver function tests, glucose, anti- nuclear antibody, and syphilis IgG that were normal or neg- ative. The patient underwent an incisional biopsy under local anesthesia. The specimen was stored in a tube containing for- malin 10% and sent to a laboratory for histopathological analysis. Microscopically, a large area of ulceration of the coating epithelium subtended by dense plasma cell infiltrate was observed (Figures 4 and 5). CD20 and CD3 showed focal positivity for B and T cells, respectively. The plasma cell
infiltrate was positive for CD138 (Figures 6 and 7) and showed kappa light chain restriction (Figures 8 and 9) with a kappa/- lambda ratio of 20 : 1 (Figures 10 and 11). The final histo- pathological diagnosis was “plasmacytosis of the mucous membranes with restriction for the kappa chains.” Initial pharmacotherapy with prednisone began with 50 mg/day for two weeks, and the patient referred an improvement of his conditions (reduction of pain and swelling). He repeated this treatment one month later, and he experienced a partial Figure 1: Ulceration on the tip of the tongue.
Figure 2: Lateral view of the tongue.
Figure 3: Gingival edema and erythema.
Figure 4: Medium-power hematoxylin and eosine-stained biopsy sample from the tongue mucosa showing a dense polyclonal plasmacytic inflammatory infiltrate throughout the connective tissue.
Figure 5: Histological examination of a biopsy taken from the tongue showed a denuded surface epithelium with the superficial and mid dermis demonstrating a dense infiltration of plasma cells.
Figure 6: CD138. A high-power view shows a monomorphic population of mature plasma cells (×10).
regression of the lesions. At 1-year follow-up, there was no regression of the disease and no recurrence of the lesions (Figure 12).
3. Discussion
Plasma cell mucositis (PCM) is an idiopathic disorder histo- logically characterized by dense infiltrates of lymphocytes and plasma cells in submucosa. Usually, the lesions are mainly observed on the mucosal surfaces, vulva and penis, gingiva, lips, tongue, buccal mucosa, epiglottis, and larynx.
A review of the English-language literature was per- formed. The keywords “plasma cell mucositis” and “oral”
were entered in the searchfields of PubMed. 103 results were identified. Another research was performed including the keywords“plasmacytosis” and “oral.” 40 results were identi- fied. The research was conducted by considering the articles published until November 2019. Excluded were cases that were not about the oral cavity, not in English, not human study, and not compatible with the PCM diagnosis or report lacking immunohistochemical analysis. According to the exclusion criteria, we selected 26 studies [1–26] that included 45 cases of PCM. We analysed patients’ age, gender, symp- toms, lesion location, treatment, and follow-up. The princi- pal features and data pertaining the selected cases are compiled in Table 1. Age data were available in all selected studies, and the average age was 56:06 ± 16:33 years. The youngest patient was 13 years old [17] and the oldest 83 years old [20]. All studies included in our review reported the gen- der of the subjects. The analysis of the data collected Figure 7: CD138. A high-power view shows a monomorphic
population of mature plasma cells (×20).
Figure 8: Plasma cells showing kappa light chain restriction (×10).
Figure 9: Plasma cells showing kappa light chain restriction (×20).
Figure 10: Plasma cells showing lambda light chain restriction (×10).
Figure 11: Plasma cells showing lambda light chain restriction (×20).
Figure 12: Follow-up at 1 year.
Table1:ReviewofPCM. Study/yearofpublicationAge∗GenderSymptomsLocationofthelesionsTreatmentFollow-up Poswilloetal.[7]/1967
37FRedness,swellingGingivaResection,oralhygiene programmeAWD,2years 39FRedness,hypertrophyGingivaResectionNotavailable 21FHyperplasticredlesion, hypertrophy,erythemaMaxillarylabialvestibule, gingivaResection,oralhygiene programmeNotavailable Whiteetal.[3]/198647FDysphonia,sorethroatLips,mouth,tongue, supraglotticlarynxResection,prednisoneAWD,9years(f/ureportedby Ferreiroetal.[1]) Timmsetal.[8]/198870FHoarseness,cough,stridorGingiva,supraglottic,hard palateCO2laser,prednisolone, topicalsteroidsprayAWD,1year TimmsandSloan[9]/199132FHoarseness,sorethroatGingiva,falsecord, mucobuccalSystemicandtopical steroidsMarginalimprovement,nof/u epiglottis Ferreiroetal.[1]/1994
60FDysphonia,dysphagiaSupraglotticlarynxCO2laser,prednisone, antibiotics,beclomethasone (Vanceril)sprayAWD,1year 41MDysphonia,stridorSupraglottic,glotticlarynx, nose,pharynxAntibioticsAWD,7years;tracheostomy 62FDysphonia,stridor,dry eyes,dysphagiaSupraglottic,glotticlarynx, tracheaNotavailableAWD,16years;tracheostomy 54MDysphoniaSupraglottic,glotticlarynxResectionAWD,1year 40MDysphonia,soremouthLips,tongue,palate, pharynx,supraglottic& glotticlarynx
Prednisone,isotretinoin (Accutane),CO2laserAWD,15years;sleepapnea 67MSoremouthLips,mouth,tongue,palatePrednisoneAWD,3years 61MUnknownLipsthroughlarynxNotavailableNotavailable 56MUnknownNose,palate,pharynxResectionNotavailable VandeKerkhofand VanBaar[26]/199580FPain,soreness,erythema, swellingLipsBetamethasone dipropionatePartialsymptomaticrelief,f/unot available 57FSorethroatGingiva,supraglotticlarynxTopicalsteroidsGingivalimprovement,nof/u Khanetal.[5]/199769MHoarseness,dysphagia, hemoptysis
Leftfaucialpillar, hypopharynx,epiglottis, larynx
Beclomethasonespray, CorsodylmouthwashNorecurrence,20monthsf/u Noorily[25]/199767MInduration,erythema, crustingLowerlipResection,primaryclosureNotavailable Smithetal.[4]/199959MSwelling,hoarseness,sore throat,erythemaSoftpalate,gingiva, oropharynx,nasopharynxNoneAsymptomatic,6monthsf/u
Table1:Continued. Study/yearofpublicationAge∗GenderSymptomsLocationofthelesionsTreatmentFollow-up Kauretal.[24]/200147MSwelling,erythema, inflammationUpperlipTriamcinoloneacetonide injectionsNorecurrence,3monthsf/u BhartiandSmith[10]/200342FPain,dysphagiaBuccalmucosa,palateTopicalandsystemic antifungals,corticosteroidsPartialsymptomaticrelief,no regressionofthedisease,nof/u Heinemannetal.[23]/200661FPain,ulcerations,erythema, erosionsTongue,lips,buccal mucosa,vulvaePrednisolone,cyclosporinCompleteremission,6monthsf/u Solomonetal.[2]/200860FPain,sorethroat,erosions, swelling,erythemaGingiva,lips,ventral tongue,hardpalatePrednisoneDiseaseremission,f/unotavailable Senoletal.[11]/200846MInflammation,maceration, pruritic,eczema Oralcommissures, gingivobuccalmucosa,toe- webs,groins,preputium, perineum,umbilicus
PrednisoloneNorecurrence,1yearf/u Najarianetal.[12]/200856MPain,impetigo,bleedingLowerlipTopicalandsystemic glucocorticosteroid, cryotherapyNorecurrence,6monthsf/u Pepperetal.[13]/200977MAtrophy,hyperkeratosis, erosion,painCommissure,lips,cheekTacrolimus,methotrexate, betamethasonemouthwash, CO2laser,radiotherapy
Onsetofsquamouscellcarcinoma, partialresolutionofmucosal plasmacytosis Puvanendranetal.[22]/201274MUlceration,erythemaUvula,hypopharyngealwallResectionLesionresolved,6monthsf/u Guptaetal.[21]/201472M
Difficultyinswallowing, soreness,burningsensation, sorethroat,ulcerations, swelling,bleedinggums, erosions Buccalmucosa,palate, tongue,pharynx,gingiva Topicalcorticosteroids (betamethasone1mg, triamcinoloneacetonidegel 0.1%),prednisolone,topical antifungals
Partialsymptomaticrelief,no regressionofthedisease,nof/u MadhavarajanandTighe [15]/201563MPain,ulcerationCommissure,buccal mucosa,gingivaNotreatmentSpontaneousresolving,6monthsf/u Cottometal.[20]/2015
54FErythemaSoftpalateNotavailableNotavailable 51MErythema,ulceration, erosionsBorderoftongueNotavailableNotavailable 50MInflammationGingivaNotavailableNotavailable 67MPain,bleeding,erosionsGingivaNotavailableNotavailable 83FErosionsGingivaNotavailableNotavailable 65MInflammationGingiva,hardpalateNotavailableNotavailable 72MInflammationGingivaNotavailableNotavailable
Table1:Continued. Study/yearofpublicationAge∗GenderSymptomsLocationofthelesionsTreatmentFollow-up Galvinetal.[14]/2016
68FErythema,ulcerationAlveolarridges,palate, buccalmucosa,gingiva Oralfluconazole, chlorhexidinemouthwash, betamethasonecreamand tablets
AWD,7years 61FSoremouth,ulceration, erythema,edemaGingiva,buccalmucosaPrednisolone,adalimumabNorecurrence,18months 69MOropharynxcongestionGingiva,dorsumand bordersofthetongue,soft palate,uvula
Mycophenolatemofetil, prednisolonePartialsymptomaticrelief,no regressionofthedisease,nof/u Trehanetal.[19]/201639MUlceration,dysphagia, erosionsLips,buccalmucosa, gingivaPrednisoloneGoodresponse,f/unotavailable Arunetal.[17]/201713MSwelling,bleeding, erythema,Upperlip,gingivaTriamcinoloneacetonide injections,Norecurrence,1yearf/u Liuetal.[18]/201718FSwelling,bleeding,erosions, painLips Methylprednisolone, dapsone, hydroxychloroquine, antibiotics
Symptomsdecreasing,6monthsf/u Gasparroetal.[6]/201978FPain,erythema,ulcerations, erosionsBuccalmucosaInjectableplatelet-rich fibrin(i-PRF)Nocompletehealing,inflammation reduction,6monthsf/u Shanahanetal.[16]/201962FDrymouth,ulceration, swelling,dysphagia, hoarsenessSoftpalate,buccalmucosaPrednisolone,dapsone, mycophenolatemofetilNorecurrenceofsymptoms, 1yearf/u AWD:alivewithdisease;F:female;f/u:follow-up;M:male.∗Ageatpresentation(years).
suggested a male predilection with a male-to-female ratio of 1.25 : 1 (45 cases: 25 M, 20 F). A graph of the age and gender distribution is reported in Figure 13. Rarely the lesions are isolated, and in 45 cases reviewed, the oral cavity zones most affected by the lesions were the gingiva (20 cases), the lips (14 cases), the upper aerodigestive tract (13 cases), the palate (12 cases), the buccal mucosa (11 cases), and the tongue (8 cases). In our review, the symptoms most present are typical of inflammation with a particular interest of the oropharynx; the main symptoms are erythema (13 cases), pain (10 cases), swelling (9 cases), sore throat (7 cases), dys- phonia, and dysphagia (6 cases). The analysis of the treatment evidenced 24 cases (53.3%) treated with corticosteroids in different forms (systemic, topical, and injections), and in 10 cases of these, there was no recurrence of the disease.
Plasma cell mucositis (PCM) is classified as idiopathic and rarely manifests itself on the tongue, especially in young patients with no comorbidities. Despite the fact that plasma cell mucositis often involves the oral and genital mucosa [3], there was no genital involvement in this circumstance.
Since it is a very rare pathology, in the diagnosis of PCM, it is important to exclude other pathologies with similar patho- logical features, such as erosive lichen planus, mucous mem- brane pemphigoid, sarcoidosis, allergic gingivostomatitis, extramedullary plasmacytoma, rhinoscleroma, pemphigus, erythroplasia, squamous cell carcinoma, and fungal infec- tions [4, 5]. Lichen planus is a chronic-relapsing inflamma- tory disease, with cutaneous and mucous involvement. It can be distinguished in oral lichen planus (OLP) and oral lichenoid drug reactions (OLDRs). The lesions may cause pain and, generally, appear as white patches with erosive aspects. A typical OLP model is identified with bilateral and symmetrical lesions; moreover, a hyperkeratosis is observed in the lower epidermis and in the upper dermis [27]. Pemphi-
goid of the mucous membranes affects mainly older individ- uals, as well as the plasma cell mucositis. Unlike the PCM, it presents itself with erosions to the mucous membranes of variable gravity that are mainly located in the oral cavity [5]. Sarcoidosis is a rare acquired systemic granulomatous disease. The respiratory system is more interested, but in a smaller number of cases, there is the involvement of the oral and perioral mucosa. Oral lesions can be solitary or multiple;
they appear as a well-delimited and occasionally ulcerated red swelling [28]. Allergic gingivostomatitis is the result of an immunologic injury reaction; it is a complex disease which can have more than one etiologic factor [29]. Extrame- dullary plasmacytoma is a tumor that rarely affects the head and neck, especially the upper airways, and the lesions are generally polypoidal in appearance [5]. Primary Non- Hodgkin’s Lymphoma (NHL) can rarely arise from lym- phoid tissue of the tongue and give manifestations that mimic PCM [30].
Several times, PCM lesions can enter into differential diagnosis with squamous cell carcinoma (SCC) [4–22]; in lit- erature, there are cases in which a differential diagnosis was made with oral carcinoma and there is a case report where a SCC arose from a mucosal plasmacytosis [13]. Therefore, a correct diagnosis and adequate management of the disease are essential.
The management of PCM is mainly aimed at reducing the symptoms; in fact, the patient treatment involved the use of systemic prednisone 50 mg/day to improve his quality of life.
However, in literature, there are some reported cases where the use of PRF injections is associated with cortisone therapy [6]. The benefit of PRF is to allow a continuous release of growth factors, inducing neoangiogenesis and fibroblast activations. Therefore, PRF creates an optimal scaf- fold for the tissue healing process, with the advantage of low cost and easy to prepare [31].
Although many therapeutic treatments are able to sta- bilize the disease, they are not able to induce a complete remission. The condition is generally of long-standing duration and the disease has a significant impact on the patient’s quality of life. The disease treatment is mainly tar- geted to the management of symptoms. PCM is considered an uncommon benign disorder with a favorable prognosis.
The distinctions between the conditions that are present in the oral cavity with the histologicfinding of a dense submu- cosal plasma cell infiltrate are not well documented in oral pathology literature.
4. Conclusion
It is important that PCM is recognized in the dental commu- nity, because diagnosis is dependent on clinical pathologic correlation. Only close communication between specialists in several disciplines prevented inappropriate treatment of a patient with an extremely rare condition. Nevertheless, it is very important to differentiate the PCM disease from other neoplastic conditions in order to achieve a better clinical management of the patients, so it is necessary to investigate this disease in depth.
12
9
6
3
0 0-20 21-40 41-60
Age (years) >60 Male
Female
Figure 13: Distribution of age and sex in the 45 cases of PCM of the oral cavity reviewed.
Conflicts of Interest
The authors declare that they have no conflicts of interest.
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