scholarly journals Immunoglobulin G4-related periodontitis: case report and review of the literature

2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Jinmei Zhang ◽  
Lei Zhao ◽  
Jieyu Zhou ◽  
Wei Dong ◽  
Yafei Wu

Abstract Background Immunoglobulin G4-related disease (IgG4-RD) is a chronic inflammatory systemic disease of unknown etiology that can affect one or multiple organs. The disease can mimic many infectious and inflammatory diseases, mainly causing organ enlargement or hyperplasia. Its diagnosis primarily relies on clinical, serologic, and histological features (lymphoplasmacytic infiltrates, storiform fibrosis, and obliterative phlebitis of IgG4 + plasma cells). Here, we report a rare case of IgG4-related periodontitis, and review the relevant literatures. Case presentation A 38-year-old Chinese man visited the Department of Periodontics with gingival enlargement, loose teeth, and tooth loss. The patient had very poor oral hygiene and a large amount of calculus. Gingivae were edematous with deep periodontal pockets and attachment loss. Panoramic radiography showed alveolar bone loss. Serologic examination showed that IgG was 23.70 g/L and IgG4 concentration was 2.800 g/L. There was significant lymphoplasmacytic infiltration, a storiform pattern of fibrosis, and mitotic figures with hematoxylin and eosin staining; immunohistochemical staining showed 10 scattered IgG4-positive plasma cells in a high-power field. The patient was diagnosed as IgG4-related periodontitis. He received a course of corticosteroids with periodontal therapy, and the enlargement was significantly improved without recurrence. Conclusion IgG4-RD in the oral and maxillofacial region mainly involves salivary glands, but this rare case was characterized by enlarged gingivae. The differential diagnosis of IgG4-RD should be based on the clinical features and serologic (IgG4) and histopathological examinations. Corticosteroid therapy is effective for most IgG4-RD patients. Taken together, we hope this case report and the literature review can help dentists to improve their understanding of the IgG4-RD.

2018 ◽  
Vol 2018 ◽  
pp. 1-5
Author(s):  
Pragya Shrestha ◽  
Brian Le ◽  
Brent Wagner ◽  
William Pompella ◽  
Paras Karmacharya

IgG4-related sclerosing cholangitis (IgG4-SC) is one of the most common extra-pancreatic manifestation of IgG4-related disease (IgG4-RD) and is clinically distinct from primary sclerosing cholangitis (PSC). IgG4-RD is an increasingly recognized immune-mediated fibroinflammatory systemic disease, mostly affecting middle-aged and older male populations that can affect multiple organs. The presence of extra-biliary clinical manifestations of IgG4-RD, such as parotid and lacrimal swelling, lymphadenopathy, autoimmune pancreatitis, and retroperitoneal fibrosis, if present could provide important clues to diagnosis. High serum IgG4 levels, characteristic radiological (e.g., sausage-shaped pancreas or periaortitis) or biopsy findings (high percentage of IgG4+ plasma cells, lymphoplasmacytic infiltrate, storiform fibrosis, or obliterative phlebitis) in the setting of these features is diagnostic of this disease process. However, isolated IgG4-SC might be a diagnostic challenge, and the distinction is important as management of this disorder is vastly different from other causes of cholangitis such as PSC. Systemic corticosteroid therapy is the mainstay of therapy.


2021 ◽  
pp. 71-74
Author(s):  
Vilma Alejandra Umanzor Bonilla ◽  
Claudette Arambú ◽  
Hugo Romero ◽  
Juan Jose Guifarro

Plasma cell gingivitis is a benign lesion of unknown etiology characterized by massive infiltration of plasma cells into the connective tissue of the gingiva. Clinically it presents as a gingival enlargement with erythema and some areas with the presence of desquamation, it is usually asymptomatic, but on some occasions the patient may present pain and gingival bleeding. Diagnosis requires clinical-pathological correlation. Based on the foregoing, we present a case report of a 25-year-old female patient diagnosed with plasma cell gingivitis with idiopathic etiology based on the clinical and histopathological study.


2021 ◽  
Vol 12 ◽  
pp. 557
Author(s):  
Julie Mayeku ◽  
Jeremy Deisch ◽  
Miguel Angel Lopez-Gonzalez

Background: Immunoglobulin G4-related disease (IgG4-RD) is a rare systemic disease of unknown etiology. It is characterized by tissue infiltration caused by IgG4 plasma cells and sclerosing inflammation of various body organs. At present, there are very few reported cases of IgG4-RD invasion of cavernous sinus and the orbit. Case Description: A 56-year-old female with a history of rheumatoid arthritis was presented with a gradual onset of right oculomotor, abducens, and trigeminal nerve deficits. Four weeks after the onset of symptoms, the patient developed gradual visual deficit. Following this, a trial of steroids was administered to the patient. However, the treatment did not work as expected and patient’s condition worsened. She progressed on to suffer complete visual loss in the right eye. Extensive work-up conducted on her turned out to be nondiagnostic. After this, the patient was referred to us for our evaluation. Neuroimaging revealed a right-sided cavernous sinus and orbital apex lesion. Given the lack of diagnosis and response to steroid treatment, we recommended surgical intervention and performed a modified pterional and pretemporal approach with extradural anterior clinoidectomy and transcavernous approach. We performed a lesion biopsy and cavernous sinus decompression, which helped in the partial recovery of visual function. The pathology report was consistent with IgG4-RD. Conclusion: IgG4-RD is a rare disease that occurs even less in combination with cavernous sinus and orbit invasion. The rarity of the disease and the diverse presentation of symptoms have sometimes caused delayed diagnosis and intervention. Patients who failed to respond to conservative management and patients in the fibrotic stage of the disease without other organ involvement may benefit from surgical intervention if amenable. Early suspicion, diagnosis, and intervention can facilitate better prognosis.


2009 ◽  
Vol 133 (8) ◽  
pp. 1251-1255 ◽  
Author(s):  
Manisha Mehta ◽  
Frederick Jakobiec ◽  
Aaron Fay

Abstract Progressive sclerosing orbital pseudotumors are a subset of usually primary and localized idiopathic fibroinflammatory disorders. We report on a 66-year-old man who developed this condition along the facial tissue planes with extension into the orbit and preferential involvement of the periorbital membrane. Fibrocollagenous tissue with scattered lymphoid aggregates without follicle formation dominated the process. There was a light dispersion of B and T lymphocytes and histiocytes in the stroma. Immunoglobulin G4 (IgG4)–positive plasma cells (>35 per high-power field) were identified mostly in the lymphoid clusters, as has been discovered in similar IgG4–related fibrosclerosing conditions of other nonorbital sites. No associated systemic disease emerged during a 20-year clinical course. Previously reported orbital cases of IgG4-positive disease have all involved the lacrimal gland, usually bilaterally, and more closely resembled hypercellular reactive lymphoid hyperplasias with moderate interlobular fibrosis, rather than representing an essentially sclerosing process from the beginning.


2017 ◽  
Vol 07 (02) ◽  
pp. 164-167
Author(s):  
Hanish Bansal ◽  
Ashwani Chaudhary ◽  
Dipesh Batra ◽  
Rohit Jindal

AbstractWe report a very rare case of a 63-year-old man with penile carcinoma who developed brain metastasis. Brain metastasis from penile carcinoma is very rare, and only six cases have been reported so far as per our literature review. In view of controlled primary tumor and absence of systemic disease, tumor was excised and the patient remained clinically controlled till 3 months after follow-up.


2015 ◽  
Vol 8 (3) ◽  
pp. 124-126
Author(s):  
Ankita Joshi ◽  
Chinmay Sundarray ◽  
Krishna Arpita Sahoo

ABSTRACT Paranasal sinus anatomical anomalies with unknown etiology are common. Paranasal sinus-related diseases are associated with so high rate of morbidities, it becomes essential to identify the structure and pathophysiology of the paranasal sinuses. Computed tomography (CT) is a valuable tool in displaying its anatomy, any anatomic variations and diseases. As paranasal sinus development is a complex and long-lasting process, there are great structural variations between individuals. Maxillary and/or frontal sinus aplasia or hypoplasia are more common than the other types. Several degrees and combinations of aplasias and hypoplasias have been reported; however, there is only two case of total paranasal sinus aplasia in the literature. Here, we present the third case of total paranasal sinus aplasia, and the first ever case reported from India. How to cite this article Joshi A, Sundarray C, Sahoo KA. Bilateral Total Aplasia of Paranasal Sinuses: A Rare Case Report. Clin Rhinol An Int J 2015;8(3):124-126.


2021 ◽  
Vol 126 (1) ◽  
Author(s):  
Helya Hashemi ◽  
Andreas Thor ◽  
Erik Hellbacher ◽  
Marie Carlson ◽  
Miklós Gulyás ◽  
...  

Immunoglobulin G4-related disease (IgG4-RD) is an immune-mediated fibroinflammatory condition that can affect multiple organs. IgG4-RD may show a variety of initial symptoms. In the oral mucosa, lesions present as inflammatory fibrosis with a large number of IgG4-positive plasma cells. Evaluating treatment is a well-known problem in IgG4-RD due to the absence of an established assessment system. There are difficulties in defining the severity of the disease, which is why treatment is primarily based on its clinical manifestations. We present a case report of localized IgG4-RD with ulcerative and proliferative manifestations on the tongue, which clinically mimicked oral squamous cell carcinoma. A tumor-like lesion on the tongue can indicate something else other than the malignant or reactive changes commonly found in the oral mucosa. Multiple differential diagnoses of these atypical oral lesions, including localized IgG4-RD, should be considered.


2018 ◽  
Vol 2018 ◽  
pp. 1-4
Author(s):  
C. A. Simões ◽  
M. R. Tavares ◽  
N. M. M. Andrade ◽  
T. M. Uehara ◽  
R. A. Dedivitis ◽  
...  

Riedel’s thyroiditis (RT) represents one type of IgG4-related thyroid disease (IgG4RTD) and the diagnosis involves quantitative immunohistochemistry showing dense lymphoplasmacellular inflammatory infiltrate consisting of IgG4-positive plasma cells with storiform fibrosis and obliterative phlebitis. We report a case of RT with progressive enlargement of the anterior neck, severe dysphagia, odynophagia, and dyspnea. The patient underwent surgical decompression of the airway, protection tracheotomy, and gastrostomy for nutritional intake 6 months after first symptoms. Complete resolution occurred after surgical treatment combined with prednisolone. Immunostaining revealed IgG4-positive plasma cells 12/HPF (high-power field) and the IgG4/IgG ratio 25%, values that were disproportionate to the intensity of the patient’s symptoms. As to this case and the few cases described and analyzed in the literature, our impression is that there is no relation between the intensity of symptoms in RT with the total number of IgG4-positive plasma cells and the IgG4/IgG ratio, but more studies are needed.


2016 ◽  
Vol 30 (3) ◽  
pp. 412-418
Author(s):  
Luis Rafael Moscote-Salazar ◽  
Hernando Raphael Alvis-Miranda ◽  
Willem Guillermo Calderon-Miranda ◽  
Zenen Antonio Carmona Meza ◽  
Nidia Escobar Hernandez ◽  
...  

Abstract Solitary plasmacytoma and extramedullary plasmocytoma are tumors of malignant character composed of plasma cells, with a mean age of onset at 60 years. They can appear anywhere where the reticuloendothelial system is present. Usually these tumors lead to the development of multiple myeloma in a period of time ranging from 3 to 5 years. We present a rare case handled in our neurosurgery service associated with an unusually long period of evolution.


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