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.

Cutaneous Rosai–Dorfman disease: a challenging diagnosis

2021 ◽  
Vol 14 (2) ◽  
pp. e239244
Author(s):  
Rita Sampaio ◽  
Leandro Silva ◽  
Goreti Catorze ◽  
Isabel Viana
Keyword(s):  
Weight Loss ◽  
Differential Diagnosis ◽  
Physical Examination ◽  
Plasma Cells ◽  
Oral Prednisolone ◽  
Inflammatory Cells ◽  
Rosai Dorfman Disease ◽  
History Of ◽  
Nodular Lesions ◽  
Proliferative Disease

Rosai–Dorfman disease is a rare benign histiocytic proliferative disease of unknown cause that, in exceptional cases, presents with lesions confined to the skin. Clinically variable types of lesions such as papules, nodules and plaques have been reported. We present a case of a 27-year-old woman with a 1-year history of erythematous papular and nodular lesions on the malar and right axillary regions, previously misdiagnosed as acne. She reported no fever, malaise or weight loss, while physical examination and laboratory workup were normal. Bacteriological and mycobacteriological cultures were negative. Histopathological findings showed dense infiltration of inflammatory cells involving the entire dermis, consisting of large macrophages with emperipolesis, S100 and CD68 positive, neutrophils, eosinophils, lymphocytes and plasma cells. The patient was treated with oral prednisolone without improvement. Dapsone was subsequently initiated with favourable clinical response. The present article aimed to emphasise the clinical and histological differential diagnosis and share the treatment experience.

scholarly journals Asymptomatic flow of Rosai-Dorfman disease

2014 ◽  
Vol 71 (8) ◽  
pp. 780-783 ◽  
Author(s):  
Dragan Petrovic ◽  
Dragan Mihailovic ◽  
Sladjana Petrovic ◽  
Nikola Zivkovic ◽  
Zaklina Mijovic ◽  
...  
Keyword(s):  
Plasma Cells ◽  
S100 Protein ◽  
Unknown Etiology ◽  
Neoplastic Disease ◽  
Diagnostic Dilemma ◽  
Regional Lymph Nodes ◽  
Parotid Region ◽  
Massive Lymphadenopathy ◽  
Rosai Dorfman Disease ◽  
Abundant Cytoplasm

Introduction. Sinus histiocytosis with massive lymphadenopathy is a rare benign self-limiting disease of unknown etiology. The salivary gland involvement, indicating the extranodal character of the disease, often presents a diagnostic dilemma requiring immunohistochemical staining of surgically removed tumor to confirm the diagnosis. Case report. We report a 43-year-old man presented with an asymptomatic mass in the left mandibular angle. On physical examination, the lesion was described as a painless, mobile, firm-elastic consistency nodule, which measured 4 ? 3 cm in diameter, with normal overlying skin. A mass with the same characteristics, dimensions 2 ? 2 cm, was also noted in the right parotid region. No other changes in regional lymph nodes were detected. On macroscopic examination the lesion was firm, multilobulated, yellowish and rounded, while on microscopic examination the lesion was composed almost entirely of polygonal histiocytes with abundant cytoplasm, emperipolesis, plasma cells arranged in sheets, and lymphocytes scattered or within clusters. The observed histiocytes were found to be CD68 and S100 protein positive. Conclusion. Rosai-Dorfman disease is a beningn and frequently overlooked clinical and pathological entity that may be misinterpreted as a neoplastic disease.

scholarly journals Cutaneous Rosai-Dorfman disease: A case report

2021 ◽  
Vol 12 (e) ◽  
pp. e84-e84
Author(s):  
Soukaina Maghfour ◽  
Sana Mokni ◽  
Marouane Ben Kahla ◽  
Rima Gammoudi ◽  
Amina Aounallah ◽  
...  
Keyword(s):  
Differential Diagnosis ◽  
Case Report ◽  
Plasma Cells ◽  
Histopathological Examination ◽  
Inflammatory Cells ◽  
Left Cheek ◽  
Skin Involvement ◽  
Rosai Dorfman Disease ◽  
History Of ◽  
Systemic Symptoms

Pure cutaneous Rosai-Dorfman is distinguished from classic Rosai-Dorfman disease by isolated skin involvement without lymphadenopathy or systemic symptoms. Herein, we report a case of a 30 year-old-man with 16 months history of a slowly enlarging, asymptomatic, purple plaque on her left cheek following cutaneous leishmaniasis successfully treated. The histopathological examination showed dense infiltration of inflammatory cells involving the entire dermis, consisting of large macrophages with emperipolesis, S100 and CD68 positive, lymphocytes and plasma cells. The patient was treated with intralesional triamcinolone with a significant improvement. The present article aimed to emphasise the clinical, histological differential diagnosis and to share the tumoral presentation of cutaneous Rosai-Dorfman disease.

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.

Skin limited Rosai-Dorfman disease with elevated IgG4 positive plasma cells

2018 ◽  
Author(s):  
Iñigo Gorostiaga ◽  
ÁLVARO PÉREZ RODRÍGUEZ ◽  
ÁLVARO ['Noelia'] ◽  
jose javier aguirre
Keyword(s):  
Plasma Cells ◽  
Rosai Dorfman Disease

A Serological Storm

1970 ◽  
Vol 9 (2) ◽  
Author(s):  
Bertha Wong MD ◽  
Maria Bagovich MD ◽  
Ivan Blasutig PhD ◽  
Simon Carette MD MPhil
Keyword(s):  
Differential Diagnosis ◽  
Hepatitis C Virus ◽  
Immune System ◽  
Hepatitis C ◽  
Hcv Infection ◽  
Clinical Context ◽  
Autoantibody Profile ◽  
Life Threatening ◽  
Clinically Significant ◽  
Sensory Polyneuropathy

This article describes a patient presenting with a sensory polyneuropathy and multiple autoantibodies, leading to the diagnosis of hepatitis C virus (HCV) infection. His widely positive autoantibody profile in the absence of clinically significant rheumatic disease illustrates the importance of interpreting autoimmune serology in the appropriate clinical context and the concept of HCV being a non-specific activator of the immune system. In addition, it highlights the importance of considering untreated HCV infection in the differential diagnosis of rheumatic complaints, particularly if the workup reveals multiple autoantibodies, as HCV is a potentially severe and life-threatening disease, which can be appropriately managed with effective antiviral therapy.

scholarly journals Saprochaete clavata Infection in Immunosuppressed Patients: Systematic Review of Cases and Report of the First Oral Manifestation, Focusing on Differential Diagnosis

2021 ◽  
Vol 18 (5) ◽  
pp. 2385
Author(s):  
Carlo Lajolo ◽  
Rupe Cosimo ◽  
Schiavelli Anna ◽  
Gioco Gioele ◽  
Metafuni Elisabetta ◽  
...  
Keyword(s):  
Risk Factors ◽  
Systematic Review ◽  
Acute Myeloid Leukemia ◽  
Differential Diagnosis ◽  
Myeloid Leukemia ◽  
Oral Manifestation ◽  
First Case ◽  
Immunosuppressed Patients ◽  
Meta Analyses ◽  
Acute Myeloid

Background: Saprochaete clavata infection is an emerging issue in immunosuppressed patients, causing fulminant fungaemia. The purpose of this systematic review of cases is to retrieve all cases of S. clavata infection and describe oral lesions as the first manifestation of S. clavata infection. Methods: We report the first case of intraoral S. clavata infection in Acute Myeloid Leukemia (AML) affected subject, presenting as multiple grayish rapidly growing ulcerated swellings, and provide a review of all published cases of infection caused by S. clavata, according to PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines, conducted by searching SCOPUS, Medline, and CENTRAL databases. Only articles in English were considered. Individual patient data were analyzed to identify risk factors for S. clavata infection. Results: Seventeen of 68 retrieved articles were included in the review reporting data on 96 patients (mean age 51.8 years, 57 males and 38 females). Most cases were disseminated (86) with a 60.2% mortality rate. Ninety-five were hematological patients, with AML being the most common (57 cases). Conclusions:S. clavata infection in immunosuppressed patients has a poor prognosis: middle-age patients, male gender and Acute Myeloid Leukemia should be considered risk factors. In immunosuppressed patients, the clinical presentation can be particularly unusual, imposing difficult differential diagnosis, as in the reported case.

Extra-nodal Rosai-Dorfman Disease Confined to Breast

2020 ◽  
Vol 154 (Supplement_1) ◽  
pp. S41-S42
Author(s):  
E Ozluk ◽  
R Shackelford
Keyword(s):  
Plasma Cells ◽  
African American Woman ◽  
American Woman ◽  
Lymphocytic Infiltration ◽  
Needle Aspiration ◽  
Left Breast ◽  
Unknown Etiology ◽  
Diagnostic Challenge ◽  
Ductal Cells ◽  
Rosai Dorfman Disease

Abstract Introduction/Objective First described by Rosai and Dorfman in 1969, Rosai-Dorfman Disease (RDD) is an uncommon, idiopathic, reactive lymph node process with an unknown etiology. It may involve extra-nodal organs including the skin, bone, soft tissue, and eyes. However, breast involvement is rare and RDD confined to breast without nodal involvement is extremely uncommon. Methods Here we present a case of RDD confined to breast. The patient was a 51-year-old African American woman who was found to have an irregular, solid left breast mass on routine mammogram, which had speculated irregular margins and measured of 37.0 x 32.0 x 32.0 mm. She did not have any symptoms, nor any palpable lymph nodes. The clinician stated that the lesion was highly suspicious for a breast malignancy and requested a fine needle aspiration (FNA) of the mass, followed by total excision. Results An FNA was interpreted as atypical histiocytic cells in a mixed lymphoid background. Histopathologic examination revealed an ill-defined mass with sheets of histiocytes, plasma cells, and a mixed lymphocytic population, with occasional germinal centers. Some of the histiocytes were spindle shaped and associated with storiform collagen deposition. The histiocytes had single and multiple nuclei and exhibited occasional emperipolesis. Immunohistochemical staining with S100 diffusely highlighted the histiocytes, whereas CD1a was negative. CD3 and CD20 immunostains were positive for mixed-type lymphocytic infiltration. Cytokeratin staining was performed and reassuringly stained only the benign ductal cells. A diagnosis of RDD of the breast was made, based on these histopathologic findings. Conclusion We report an example of an extra-nodal RDD involving the mammary gland that was initially suspected to be breast carcinoma. RDD may still be a diagnostic challenge, especially in a patient with suspected carcinoma. It is the pathologist’s role to lead the clinician to the proper diagnosis and render a correct histopathologic diagnosis.

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.

BRAIN TUMORS IN INFANCY

PEDIATRICS ◽  
1948 ◽  
Vol 1 (3) ◽  
pp. 346-363
Author(s):  
I. WALLACE LEIBNER
Keyword(s):  
Nervous System ◽  
Differential Diagnosis ◽  
Brain Tumors ◽  
Correct Diagnosis ◽  
Full Recovery ◽  
The Other ◽  
Congenital Hydrocephalus ◽  
First Case ◽  
Clinical Patterns

Seven cases of brain tumors occurring in infants are presented. Six occurred in males and one was in a female. Three of them arose in the cerebellum while the other four were supratentorial. The tumors included two medulloblastomas, one [See Table II in Source Pdf] spongioblastoma polare, one ependymoma, one astrocytoma and one hemangioblastoma. The seventh case probably was also one of medulloblastoma. One of the proven medulloblastomas was supratentorial, which is unusual so faras location is concerned. The hemangioblastoma arose in the cerebrum. The location of this tumor is also a rare one and to my knowledge, it is the first case of its type reported in an infant in whom full recovery occurred following removal of the tumor. The diagnosis was unsuspected in four cases. Three of these infants were believed to be suffering from congenital hydrocephalus while the fourth was thought to be afflicted with encephalitis. The ways in which the correct diagnosis might have been made are discussed. Since bizarre clinical patterns are sometimes produced by brain tumors in infants and the diagnosis frequently overlooked, the possibility should be kept in mind by the pediatrician in considering the differential diagnosis of conditions producing signs referable to the nervous system.

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