Gingival plasmacytoid granuloma is a rare disease. In this paper, we report an unusual case of plasmacytoid granuloma that affected multiple teeth in different areas within a relatively short period of time, resulting in periodontal destruction and tooth loss.
Clinical information
1. Medical history report: The patient, male, 30 years old, sought treatment with the main complaint of “left upper gingival swelling for more than one month”, and had bleeding from the left upper gingiva with no obvious cause one month ago, which was stopped by self-compression. She was admitted with the diagnosis of “left upper gingival mass”. He was in good general condition and his vital signs were stable.
Specialized examination: a 2.5*1.5*1.5 cm swelling on the corresponding buccal gingiva, involving the gingival papilla, soft, reddish in color, bleeding easily, with clear borders, and the corresponding tooth IIIo loosening. It was suspected to be a “malignant mass”. The associated teeth were extracted and a partial enlarged resection including some normal gingiva and alveolar bone was performed to the depth of the left maxillary sinus floor, preserving the intact sinus mucosal wall. The trabecular cavity was filled with iodoform gauze. The pathological diagnosis: (consistent with) left maxillary gingival plasma cell granuloma. 10 days later the gauze strips were removed and the patient was discharged.
Postoperatively, a “granuloma-like” mass appeared on the buccal gingival papilla in January 765 and was loosened in 5II. The mass was surgically removed and periodontally scraped. Pathology consultation, results as before. One week after surgery, the mass recurred and was again excised, 65 teeth were extracted and scraping was expanded, and the patient was cured two weeks later. Laboratory tests: bone marrow aspiration and cellular immunoassay were normal, and no Benzo’s protein was seen in the urine.
Three months after the initial surgery, 56 “granulomatous lesions” at the tip of the gingival papilla, small in extent, 56 no loosening, resection of the lesions, expanded scraping and postoperative hormonal therapy. 3 weeks later, the medication was discontinued and no local recurrence was observed, but 5 distal mesial alveolar bone resorption and 5 distal mesial root surfaces were exposed. Pathology was again consulted and a diagnosis of plasmacytoid granuloma was made. Local low-dose radiotherapy was given to promote healing. 1 month later, no recurrence of gingival mass was seen, but 5 IIIo was loosened and extracted.
Two weeks after the completion of radiotherapy, the patient complained of discomfort in the right upper posterior teeth, and examination revealed 78 gingival papillae apically red, soft, and bleeding easily; the teeth were not loosened and were immediately excised, thoroughly scraped subgingivally, and treated with Chinese medicine. According to the dialectical treatment of Chinese medicine, the mechanism of the disease is heat and toxicity, blood stagnation, phlegm in the local gingiva. Treatment: Benefit Qi and nourish Yin, clear heat and detoxification, activate blood stasis, soften hardness and disperse live. The formula uses Xianfang Livelihood Drink, Wu Wei Disinfection Drink and Si Jun Zi Tang plus or minus. After two months of treatment, the disease was completely improved, and no recurrence was seen in the six-month follow-up.
2. Pathology and immunohistochemistry.
Microscopic examination: mucosal squamous epithelium was broken, and wheel-like collagen fibers and a large number of new capillaries were seen in the submucosal tissue, between which a large number of plasma cells were infiltrated, and plasma cells were differentiated and mature, with more neutrophils, small lymphocytes and scattered eosinophils and histiocytes infiltration.
Immunohistochemistry: plasma cells and most lymphocytes CD20(+), CD79(+), κ(+), λ(+), IgG(+), IgA(+), plasma cells CD138(-), CD38(+), Ki-67(+), few lymphocytes CD3(+), CD45RO(+), all cells CD56( -).
DISCUSSION
Plasmacytoid granuloma (PCG) is an idiopathic inflammatory pseudotumor of unknown etiology with aggressive clinical and imaging changes, characterized by benign proliferation of inflammatory cells dominated by mature plasma cells containing Russel vesicles, lesions sometimes also including lymphocytes p mast cells p eosinophils and large fat-filled monocytes with a stroma of granulation tissue, which may contain fibroblastic tissue in between whorls123.
IPT consists of inflammatory cells such as plasma cells, leukocytes, histiocytes, and polymorphonuclear cells mixed with collagen fibers. If plasma cells predominate, it is called plasmacytoid granuloma. However, if histiocytes predominate, it is called histiocytomatosis, xanthogranulomatous inflammatory pseudotumor, or inflammatory myofibroblastoma.
PCG is most commonly found in the lungs, in roughly equal numbers in men and women, with an average age of 30 years. However, PCG can occur in many parts of the body, including the central nervous system p orbital p paranasal sinuses p esophagus p trachea, and oral cavity. In the oral maxillofacial region, PCG is seen and reported in the tongue, maxillary sinus, posterior molar pad, and gingiva. Gingival PCG is a less frequent disease and has not been reported in the Chinese biomedical literature search from 1979 to the present. mediline search, 12 relevant articles between 1966 and May 2007, mostly in the form of case reports.
In the case reported here, “rapid progression” refers not only to the short time between the onset of symptoms and the identification of the disease, but also to the occurrence of multiple tooth loss in different gingival sites within a relatively short period of time, and no similar reports have been found. Based on the close observation of the onset process, the following clinical manifestations are summarized.
(1) Age of onset: 30 years old, consistent with the epidemiological characteristics of PCG, appearing in young adulthood.
(2) The swelling and discomfort of the associated teeth appear first, followed by the formation of “granuloma” at the gingival papillae. 5-7 days later, the “granuloma” spreads to the entire gingival papillae of the associated teeth, manifesting as red, edematous gingiva with a shiny surface and easy bleeding.
(3) The early stage is vertical resorption of the periodontium, followed by destruction of adjacent alveolar bone and loosening of teeth within a short period of time, resembling the biological behavior of a “malignant tumor”.
(4) The lesions are mostly found in the bicuspids and adjacent molar areas.
(5) The onset of each lesion is 1-2 months apart.
(6) X-ray: from vertical resorption of the periodontium to the destruction of the alveolar bone process.
(7) Laboratory tests: routine blood, bone marrow aspiration, and immune function tests are within normal limits, and periapical protein (-). Because of the low incidence, there is no unified understanding of the disease and a lack of statistical basis.
The pathogenesis of PCG is still debated. It has been reported that pulmonary PCG often has a history of infection and some believe that it is caused by bacterial infection based on the fact that PCG can be relieved with antibiotics,and that Coxiella Burniti and Pseudomonas Psittici bacteria have been identified in IPT. However, there is not much evidence to support bacterial infection; many consider it to be related to immune dysfunction in the body.
Thami et al. considered plasma cell perioral mucositis (PCOM) associated with PCG of the gingiva as a reactive polyclonal plasma cell disease occurring in many sites of PCOM, including the tongue, palate, and epiglottis. Plasma cells and IgA are the main mucosal defense system of the organism, and the oral mucosal system is often stimulated by various external factors; gummies, menthol, dental powders and some xenogenic substances can mediate the infiltration of plasma cells.
On the other hand, the altered host immune state can also produce reactive polyclonal mucosal plasma cell infiltration against the normal organismal environment, leading to the development of PCG.
Because of the extremely low incidence of plasma cell granuloma and its “malignancy-like” biological behavior, it is often misdiagnosed clinically, and its repeated occurrence at different sites often makes diagnosis difficult. It needs to be differentiated from the following diseases.
(1) granuloma gingivalis: clinically it is very similar to PCG, but it is often found to have obvious local irritants such as tartar or poor restorations, develops slowly, and has less impact on the alveolar bone and periodontium. Pathologically, granulation tissue and inflammatory infiltrates are predominant, but plasma cells are less frequent.
(2) Plasmacytoma: PCG can also be confused with leukocyte proliferative disease, and plasmacytoma can also occur in the gingival region, although it is rare. Plasmacytoma is a tumor and precancerous lesion that also presents similarly to PCG, often as a well-defined swelling or the formation of a papillary nodule, approximately one to several centimeters in diameter. Laboratory tests may reveal elevated serum electrophoretic gammaglobulin. Pathology microscopically reveals an almost entirely plasma cell composition without connective tissue mesenchyme, with abnormal, atypical plasma cells and a few schwannomatous images.
Immunohistochemical analysis of plasmacytoma has monoclonal plasma cells, whereas plasmacytoid granuloma is a nonneoplastic proliferation of inflammatory cells with an inflammatory lesion in nature, with positive expression of both κ and λ chains in the cytoplasm, and plasma cells are polyclonal, whereby the two can be distinguished.
(3) Multiple myeloma: Also known as plasmacytoid sarcoma, it often occurs in older individuals with an age of onset up to 70 years, and very rarely occurs before the age of 40. The first symptom of extramedullary myeloma is local pain and a soft tissue mass with clear circumference. Laboratory tests mostly show progressive anemia, increased plasma globulin, inverted white sphere ratio, urine finding Benzo’s protein, and bone marrow aspiration finding tumorigenic plasma cells can confirm the diagnosis
(4) Histiocytosis:Pathologically some PCG should be differentiated from histiocytosis X. The latter tissue contains a large number of eosinophils with certain structural features of the cytoplasm, and some PCG contain eosinophils, but the number is usually small enough to diagnose histiocytosis X.
Most of the plasmacytoid granulomas are cured after complete surgical excision, but some cases have recurred. Hormonal and radiotherapy have also been reported to be effective in controlling the development of PCG. The gingival plasmacytoid granuloma reported here developed rapidly and regularly affected three of the four quadrants of the oral cavity in the bicuspid and first molar regions of the gingiva, causing multiple tooth loss in a short period of time. The clinical presentation has a tendency to be recurrent compared to general PCG and multifocal compared to recurrent PCG, rather than multiple occurrences at the same site.
This not only causes great psychological stress to the patient, but also makes the treatment of this disease very difficult. The initial treatment proved that surgical excision could cure the local lesion, and the subsequent surgical excision of the lesion, postoperative hormone and radiation therapy were effective in controlling the rapid development of PCG, but failed to prevent the occurrence of PCG in other gingival sites. In the follow-up treatment, on the one hand, the lesions were detected early and controlled by surgical excision and subgingival scraping. On the other hand, TCM treatment was used to regulate the organism status.
After regular oral checkups and Chinese herbal treatment, the patient’s condition did not develop further. It has been proved that Chinese medicine adds a new way to the treatment of PCG.
In conclusion, gingival plasmacytoid granuloma is a rare “tumor-like” inflammatory lesion, and the rapidly progressive multifocal PCG may be a specific type with an unclear etiology. Definitive diagnosis depends on pathological diagnosis and immunohistochemistry, and in addition to surgical excision, hormonal therapy and radiotherapy, herbal medicine is also an effective treatment.