Extranodal Rosai-Dorfman Disease Associated With Increased Numbers of Immunoglobulin G4 Plasma Cells Involving the Colon: Case Report With Literature Review

2013 ◽  
Vol 137 (7) ◽  
pp. 999-1004 ◽  
Author(s):  
Daniel B. Wimmer ◽  
Jae Y. Ro ◽  
Annisa Lewis ◽  
Mary R. Schwartz ◽  
Richard Caplan ◽  
...  
Keyword(s):  
Case Report ◽  
Plasma Cells ◽  
Granulomatous Inflammation ◽  
Acid Reflux ◽  
Right Hemicolectomy ◽  
Rare Presentation ◽  
Immunoglobulin G4 ◽  
First Case ◽  
Multiple Biopsies ◽  
Rosai Dorfman Disease

A 49-year-old woman presented with fever, weight loss, night sweats, hematochezia, and acid reflux symptoms. Two large, firm cecal lesions were seen at colonoscopy, but multiple biopsies were inconclusive. The patient underwent a right hemicolectomy for a clinical diagnosis of colon cancer. Noncaseating granulomatous inflammation with background lymphocytes, plasma cells, and histiocytes exhibiting emperipolesis were identified. With these histologic features and immunoreactivity for S-100 protein and CD68, a diagnosis of Rosai-Dorfman disease was rendered. Other areas had storiform fibrosis admixed with numerous immunoglobulin G4 (IgG4)–positive plasma cells. Although a few preliminary reports have noted an increased number of IgG4-positive plasma cells in Rosai-Dorfman disease, the relationship between these 2 conditions is unclear. To our knowledge, this is the first case report of a possible association of colonic Rosai-Dorfman disease with an increased number of IgG4-positive plasma cells. Reviews of colonic Rosai-Dorfman disease and IgG4-related sclerosis are presented to heighten awareness of this rare presentation.

scholarly journals Case report of Mikulicz`s disease: A modern concept of an old entity

2016 ◽  
Vol 73 (4) ◽  
pp. 393-396 ◽  
Author(s):  
Ksenija Bozic ◽  
Branislava Glisic ◽  
Olga Radic-Tasic ◽  
Bojana Knezevic
Keyword(s):  
Case Report ◽  
Plasma Cells ◽  
Clinical Presentation ◽  
Elevated Serum ◽  
Lymphoproliferative Disease ◽  
Modern Concept ◽  
Immunoglobulin G4 ◽  
Important Differential Diagnosis ◽  
First Case ◽  
Storiform Fibrosis

Introduction. Modern knowlegde defines Mikulicz?s disease as a part of immunoglobulin G4-related disease. The main feature is the presence of lymphoplasmacytic infiltrates, immunoglobulin G4 plasma cells positivity, distinctive storiform fibrosis and moderate eosinophilia. Case report. A 59-years old male presented with a mild keratoconjuctivitis sicca and enlarged lacrimal and salivary glands during the last two years. Althought clinical presentation of the patient was typical, earlier testing did not pinpoint Mikulicz ?s disease. By typical clinical presentation, elevated serum immunoglobulin G4 level and histopathological finding of lacrimal glands tissue we diagnosed Mikulicz?s disease successfully treated with corticosteroid therapy. Conclusion. We reported the first case of IgG4-related Mikulicz?s disease in Serbia. Our report highlights IgG4-related Mikulicz` s disease as an important differential diagnosis with Sj?gren`s syndrome and lymphoproliferative disease in rheumatological practice.

scholarly journals Endometritis and Cystic Endometrial Hyperplasia in a Goat

2005 ◽  
Vol 17 (4) ◽  
pp. 393-395 ◽  
Author(s):  
Zaher A. Radi
Keyword(s):  
Case Report ◽  
Caesarean Section ◽  
Endometrial Hyperplasia ◽  
Plasma Cells ◽  
Histopathologic Examination ◽  
Boer Goat ◽  
First Case ◽  
Cellular Debris ◽  
Superficial Epithelium ◽  
Cystic Masses

Histologic examination was performed on uterine biopsy samples of irregular cystic masses noted during caesarean section of a 2-year-old female Boer goat. Histopathologic examination revealed multifocal erosions of the superficial epithelium and multifocal infiltration of the endometrium by widely scattered viable and degenerate neutrophils, lymphocytes, and plasma cells admixed with mild amounts of cellular debris and hemorrhage. The endometrium was markedly expanded by many irregular cystic and hyperplastic glands. This is the first case report of endometritis and cystic endometrial hyperplasia in a goat in North America.

Cutaneous Rosai-Dorfman Disease With Increased Number of Eosinophils: Coincidence or Histologic Variant?

2011 ◽  
Vol 135 (12) ◽  
pp. 1597-1600 ◽  
Author(s):  
John J Cangelosi ◽  
Victor G Prieto ◽  
Doina Ivan
Keyword(s):  
Differential Diagnosis ◽  
Pituitary Gland ◽  
Plasma Cells ◽  
S100 Protein ◽  
Clinical Context ◽  
Cutaneous Involvement ◽  
Eosinophilic Infiltrate ◽  
First Case ◽  
Rosai Dorfman Disease ◽  
Skin Biopsies

Rosai-Dorfman disease (RDD) is characterized histologically by a dense histiocytic infiltrate with emperipolesis and associated lymphocytes, plasma cells, and neutrophils. Eosinophils are not commonly associated. We report a patient with initial thymus and pituitary gland involvement by RDD, who later developed papules on the groin and axilla. Skin biopsies showed admixed histiocytic infiltrates (lymphocytes, neutrophils, and plasma cells) without emperipolesis. A prominent eosinophilic infiltrate was also observed, a feature not, to our knowledge, previously reported. Immunohistochemistry revealed positivity for CD68 (most cells) and S100 protein (scattered cells) and was negative for anti-CD1a. The diagnosis of RDD was established in the clinical context after comparison with the thymic and pituitary lesions (similar histologic features, albeit with fewer eosinophils, and immunohistochemical profiles). We present the first case, to our knowledge, of multicentric RDD with cutaneous involvement and associated prominent eosinophilic infiltrate. Thus, RDD should be included in the differential diagnosis of mononuclear infiltrates containing eosinophils.

scholarly journals Subacute Thyroiditis After SARS-CoV-2 Infection Presenting With Pyrexia of Unknown Origin

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A957-A957
Author(s):  
Santosh Singh
Keyword(s):  
Case Report ◽  
Case Reports ◽  
Unknown Origin ◽  
Subacute Thyroiditis ◽  
Pathophysiological Mechanism ◽  
Rare Presentation ◽  
Pyrexia Of Unknown Origin ◽  
Upper Limit ◽  
First Case ◽  
Heart Rhythm Disorders

Abstract Subacute thyroiditis(SAT) is associated with viral(destructive) or post-viral(inflammatory) origin.[1]The most common clinical characteristics of SAT are female sex preponderance, anterior neck pain and fever.[2,3] Heart rhythm disorders and silent cervical forms have been described in SAT associated with SARS-CoV-2 infection,which occurs 16 to 36 days after resolution of COVID-19.[1,3] Symptomatic improvement occurs in a few days after initiation of therapy with sterods or NSAID.[3] Pyrexia of Unknown Origin (PUO) is a very rare presentation of SAT.[2] Hereby, a case of SAT, presenting with painful neck swelling and persistent fever (5weeks duration), two weeks after resolution of COVID-19, is being discussed. The index patient was a 50-year old obese,normotensive, diabetic (10 years duration, HbA1c-6.6% on SU, metformin, sitagliptin and dapagliflozin) male. TSH was suppressed (0.02 mIU/L), FT4 (3.06 ng/dl, upper limit of normal-1.48ng/dl) and FT3 (3.9 pg/ml, upper limit of normal-3.71 pg/ml) were elevated. Total T4 and T3 were normal. HS-CRP was markedly elevated. IL-6 and TBG were not estimated. Cervical USG revealed diffuse hypoechogenecity of thyroid gland and thyromegaly. There was reduced uptake in thyroid scan (technetium). The patient became afebrile after 4 days of initiation of 30 mg prednisolone (tapered by 10 mg every 5 days). The initial tachycardia reverted to sinus rhythm with marked reduction of neck tenderness. This case highlights certain considerations for SAT associated with SARS-CoV-2 infection. Firstly, it can present with PUO (first case report). Secondly, it may be associated with normal total T4 and T3 which can happen due to reduced TBG consequent upon increased IL-6.[4] References: 1. Caron Philippe: Thyroid disorders and SARS-CoV-2 infection: From pathophysiological mechanism to patient management. Ann Endocrinol (Paris), 2020, Sept. 2. Fever of Unknown Origin as a Sole Presentation of Subacute Throiditis in an Elderly Patient. A Case Report with Literature Review. Raj R, Yada S, Jacob A et al: Hindawi Case Reports in Endocrinology, 2018, Article ID 5041724 3. Brancatella A, Ricci D, Latrofa F et al: Is subacute thyroidits an underestimated manifestation of SARS-CoV-2 infection? Insights from a case serie. J Clin Endocrinol Metab, 2020, Aug 11, dgaa537. 4. Bartaleno L, Brogioni S, Grasso L and Martino E: Increased serum interleukin-6 concentration in patients with subacute thyroiditis:relationship with concomitant changes in serum T4-binding globulin concentration. Journal of Endocrinological Investigation, 1993, 16, 213-218.

scholarly journals A rare presentation of genital self-mutilation in a patient of schizophrenia

2017 ◽  
Vol 4 (3) ◽  
pp. 1131
Author(s):  
Md. Omar Tabrez ◽  
Md. Tabrez Aziz
Keyword(s):  
Case Report ◽  
Clinical Evaluation ◽  
Local Anaesthesia ◽  
Clinical Entity ◽  
Surgical Removal ◽  
Rare Presentation ◽  
Self Mutilation ◽  
First Case ◽  
Psychiatric State ◽  
Type Of Injury

Genital self-mutilation (GSM) is an uncommon self-inflicted injury. GSM is not a single clinical entity, and it can occur in any psychiatric state or condition. The instruments that are used for self-mutilation can vary, and treatment of these injuries requires a detailed clinical evaluation and multi-disciplinary approach. We report here a case report of a 28-year-old psychotic man who came to our emergency with complaint of self-hammered nails in his both scrotum with fear of not able to reproduce child after marriage. He underwent surgical removal of nail under local anaesthesia and was promptly reviewed by the psychiatrists who managed appropriately. To our knowledge this is the first case reported in literature and the rarest type of injury presented as a bilateral scrotal self-mutilation as should be managed wisely.

scholarly journals A Rare Presentation of Cavernous Hemangioma of Both Inferior Turbinates

2017 ◽  
Vol 25 (2) ◽  
pp. 111-114
Author(s):  
KN Salimath ◽  
N Ramakrishnan ◽  
JR Galagali
Keyword(s):  
Case Report ◽  
Nasal Cavity ◽  
Histological Examination ◽  
Nasal Obstruction ◽  
Cavernous Hemangioma ◽  
Inferior Turbinate ◽  
Mesenchymal Tumors ◽  
Rare Presentation ◽  
First Case ◽  
Inferior Aspect

The nasal cavity presents with various types of neoplasms, including epithelial and mesenchymal tumors. Cavernous hemangioma of nasal cavity is quite rare. It usually presents as a unilateral mass arising from mucosa of nasal cavity. Case Report                                       We describe here, a case of 25-year-old serving soldier, who was referred to our hospital with a mass in both nasal cavities with bilateral nasal obstruction. On examination, the mass originated from the inferior aspect of inferior turbinate of both sides. Subsequently on histological examination after complete endoscopic excision revealed that the mass was a cavernous hemangioma arising from both inferior turbinates. Discussion To our knowledge, this is the first case of cavernous hemangioma arising from both inferior turbinate reported in literature.

scholarly journals Localized IgG4-related disease manifested on the tongue: a case report

2021 ◽  
Vol 126 (1) ◽  
Author(s):  
Helya Hashemi ◽  
Andreas Thor ◽  
Erik Hellbacher ◽  
Marie Carlson ◽  
Miklós Gulyás ◽  
...  
Keyword(s):  
Case Report ◽  
Oral Mucosa ◽  
Plasma Cells ◽  
Clinical Manifestations ◽  
Assessment System ◽  
Immunoglobulin G4 ◽  
Related Disease ◽  
Multiple Organs ◽  
Igg4 Related Disease ◽  
Initial Symptoms

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.

A Solitary Rosai-Dorfman Disease of The Talus: The First Case Report of Bisphosphonate Therapy with Imaging Follow-up

2021 ◽  
Vol 104 (12) ◽  
pp. 1988-1991
Keyword(s):  
Case Report ◽  
Clinical Symptoms ◽  
Bisphosphonate Therapy ◽  
Histological Feature ◽  
Ankle Pain ◽  
First Report ◽  
Osseous Lesion ◽  
First Case ◽  
Rosai Dorfman Disease

Rosai-Dorfman disease (RDD) manifesting as a solitary osseous lesion especially of talus bone is rare. The authors reported a 31-year-old Thai man who had chronic left ankle pain and the biopsy of his talar lesion demonstrated emperipolesis, the typical histological feature of RDD. He was treated with curettage and adjuvant bisphosphonate and appeared to show improvement in clinical symptoms and radiological evidence. To the authors’ knowledge, this is the first report of an intraosseous RDD lesion treated with bisphosphonate with imaging follow-up. Keywords: Rosai-Dorfman disease; Talus; Solitary; Radiology; Bisphosphonate

scholarly journals Ewing's Sarcoma of the Zygomatic Arch Presenting in a 69-Year Old: An Unusual Case Report

2011 ◽  
Vol 2011 ◽  
pp. 1-4 ◽  
Author(s):  
Joanna C. Mennie ◽  
Robin Reid ◽  
Fiona Cowie ◽  
Omar Hilmi
Keyword(s):  
Case Report ◽  
World Literature ◽  
Ewing’S Sarcoma ◽  
Ewing's Sarcoma ◽  
Surgical Excision ◽  
Zygomatic Arch ◽  
Rare Presentation ◽  
Children And Adolescent ◽  
First Case ◽  
Neuroectodermal Origin

Objective. We report a rare case of Ewing's sarcoma of the zygomatic arch presenting in a 69-year-old patient.Method. Case report and a review of the world literature on Ewing's sarcoma incidence and management.Results. Ewing's sarcoma is a malignant round cell tumour of neuroectodermal origin that typically presents in the pelvis and long bones of children and adolescent boys. This report is the first to document the presentation of ewing's sarcoma of the zygomatic arch in a 69-year-old lady. Our patient underwent surgical excision and radiotherapy and at 4-year followup has no signs of recurrence or metastasis.Conclusion. To our knowledge this is the first case report to document Ewing's sarcoma of this location in a 69-year-old patient. This case report highlights the importance of diagnostic investigations in Ewing's sarcoma and discusses the management issues that this rare presentation raises.

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