Letters to the Editors

Plasma cell , an underestimated condition in patients with Sjögren’s syndrome?

Sir, We would like to share with the medical and scientific community this case that de- Fig. 1. A- Initial clinical view of the gum. The patient suffered from a serious carious disease and a moderate scribes a plasma cell gingivitis in a woman generalized periodontitis with redness and oedema of the gum, and generalized dryness (mouth, eyes). The clinical with primary Sjögren’s syndrome (pSS). characteristics of the gum are coherent with the importance of the dental lesions, the bad control of plaque and oral A 61-year-old woman was referred to hos- dryness. B - Clinical view after treatment of carious disease and periodontitis. After tooth extraction, treatment of pital dental department for comprehensive dental caries, temporary oral rehabilitation and periodontal treatment, the remain red and swollen at the level of care. Her medical history revealed a IgG the marginal gum and the papillae. Spontaneous bleeding is visible around the right maxillary canine. The aspect of the gums is not coherent with a controlled oral hygiene and a stabilized . In parallel, a diagnosis of kappa (MM) in complete pSS was made. C- Clinical view after local treatment of the gingiva with a topical corticosteroid for 3 months to remission after intensive chemotherapy fol- treat plasma cell gingivitis. Note that clinical signs of inflammation of the gums persist, indicating a specific form lowed by autologous stem cell transplanta- of gingiva in a patient with pSS. tion 8 years before, a hypothyroïdism treat- ed by L-thyroxine and several depressive ponent, and prescribed a topical corticoid Please address correspondence to: episodes. She complains from dry mouth, treatment (40 mg of prednisolone mouth- Prof. Marjolaine Gosset, dry eyes, and sore and bleeding gums when wash twice a day during three months) (6). Université de Paris, UR2496, brushing for the past 2.5 years. A severe F-92120 Montrouge, France. No improvement was observed reinforcing E-mail: [email protected] carious disease and moderate generalized the idea of a non-allergic etiology (Fig. 1C). © Copyright Clinical and periodontitis (stade II grade B) associated Plasma cells gingivitis is a rare disease that with hyposalivation were diagnosed (Fig. Experimental Rheumatology 2021. could be linked to type IV hypersensitivities Competing interests: none declared. 1A). A diagnosis workup to search for pSS with allergenic (4) or idiopathic (5) origin. revealed objective ocular dryness without Interestingly, it has also been described in References keratitis, positive AAN antibodies (high patients with auto-immune disease, i.e., se- titers=1/2560, with no specificity), and mi- 1. SHIBOSKI CH, SHIBOSKI SC, SEROR R et al.: ronegative rheumatoid arthritis (6) and coe- 2016 American College of Rheumatology/European nor salivary gland revealed a focal liac disease (7). pSS is a rare auto-immune League Against Rheumatism Classification Criteria (Focus score =1.4) confirming disease that is characterized by lymphocytic for Primary Sjögren’s Syndrome: A Consensus and the diagnosis of pSS according to the classi- infiltration of the exocrine glands leading Data-Driven Methodology Involving Three Interna- Arthritis Rheumatol fication of the 2016 ACR/EULAR (1). The to their destruction and dry syndrome. Gin- tional Patient Cohorts. 2017; patient did not present other immunologi- 69: 35‑45. giva inflammation is current inpatients with 2. LIKAR-MANOOKIN K, STEWART C, AL-HASHIMI cal abnormalities (normal gammaglobulin pSS because of the absence of humidifica- I et al.: Prevalence of oral lesions of autoimmune and complement level, no cryoglobulin), or tion, lubrication, clearance, buffering ca- etiology in patients with primary Sjögren’s syn- systemic manifestations. pacity, and local immunity (8). Herein, the drome. Oral Dis 2013; 19: 598‑603. The clean-up treatment with tooth extrac- local role of a lack of saliva could not be 3. CALONJE JE, BRENN T, LAZAR AJ, BILLINGS SD: McKee’s Pathology of the Skin, 2 Volume Set E- tion, caries treatment and a periodontal excluded. On the other hand, the gum could treatment (oral hygiene instruction and Book. In: 5th Revised edition. Elsevier Health Sci- also be a place of inflammation of the pSS ences; 2019, 436‑7. scaling and root planning), lead to clinical autoimmune process. Interestingly, Likar- 4. SERIO FG, SIEGEL MA, SLADE BE: Plasma Cell improvement. However, gums remain lo- Manookin et al., reported in a multi-center Gingivitis of Unusual Origin. A Case Report. J Peri- cally red and swollen at 6 weeks and the retrospective cohort study on 155 pSS pa- odontol 1991; 62: 390‑3. patient still complained about soreness and tients that 12% had lesions of 5. ARDUINO PG, D’AIUTO F, CAVALLITO C et al.: Professional Oral Hygiene as a Therapeutic Option bleeding on brushing (Fig. 1B). As oral mu- auto-immune etiology, mainly cosal lesions in patients with pSS had been for Pediatric Patients With Plasma Cell Gingivitis: and aphtous , and rarely chronic Preliminary Results of a Prospective Case Series. J described in the literature (2), a gingiva ulcerative stomatitis, but no case of plasma Periodontol 2011; 82: 1670‑5. biopsy was performed. It revealed a highly cells gingivitis (2). Therefore, in case of 6. TIMMS MS, SLOAN P: Association of supraglottic vascularised inflammatory cellular infiltrate refractory gingivitis in a patient with pSS, and gingival idiopathic plasmacytosis. Oral Surg Oral Med Oral Pathol made of polytypic plasma cells both kappa plasma cell gingivitis might be search since 1991; 71: 451‑3. and lambda. IgG4 staining revealed no evi- 7. SMITH ME, CRIGHTON AJ, CHISHOLM DM, the treatment of this entity differ from that MOUNTAIN RE: Plasma cell mucositis: a review dence of significant IgG4 plasma cell infil- of classical pSS involvement. and case report. J Oral Pathol Med 1999; 28: 183‑6. tration. Together, these features were con- 8. NAPEÑAS JJ, ROULEAU TS: Oral Complications sistent with a diagnosis of plasma cells gin- of Sjögren’s Syndrome. Oral Maxillofac Surg Clin 1 givitis (3). Due to the patient medical histo- L. Genet , DMD North Am 2014; 26: 55‑62. M. Jalladaud1,2, DMD ry, a relapse of MM or flare of autoimmune 3,4 disease were investigated. MM was still in R. Seror , MD, PhD, Prof M. Gosset1,2,5, DMD, PhD, Prof complete remission and the polytypic na- 1 ture of the plasma cell infiltrate excluded a Service de Médecine Bucco-Dentaire, AP-HP, Hôpital Charles Foix, Ivry/Seine; possible link between MM and this gingivi- 2Université de Paris, Faculté de Santé tis. Moreover, no systemic flare of pSS was Odontologie, Montrouge; observed. We investigated allergenic fac- 3Department of Rheumatology, AP-HP, tors in the diet (red pepper, chewing-gum) Hôpital Bicêtre, Le Kremlin Bicêtre, and cosmetic (herbal toothpaste) but we and Université Paris-Sud 11; 4IMVA (Immunology of Viral Infections and didn’t find any. Nevertheless, we decided to Autoimmune Diseases), INSERM U1012, change the patient’s toothpaste to one that Le Kremlin Bicêtre; did not contain sodium lauryl sulphate, a 5Université de Paris, URP2496, very common and highly allergenic com- Montrouge, France.

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