Sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease): a rare case of subglottic narrowing

1992 ◽  
Vol 106 (1) ◽  
pp. 61-62 ◽  
Author(s):  
R. G. Courteney-Harris ◽  
M. J. Goddard

AbstractA case of subglottic narrowing caused by an extranodal deposit of sinus histiocytosis in an eight-year-old child with Rosai-Dorfman disease is presented. The airway obstruction was initially managed with a tracheostomy, and then on confirmation of the diagnosis by lymph node biopsy, by excision of the extranodal deposit with a CO2 bronchoscopic laser.

2010 ◽  
Vol 134 (2) ◽  
pp. 276-278 ◽  
Author(s):  
Mark Podberezin ◽  
Ronald Angeles ◽  
Grace Guzman ◽  
David Peace ◽  
Sujata Gaitonde

Abstract Sinus histiocytosis with massive lymphadenopathy (SHML), also called Rosai-Dorfman disease, is a rare entity. Its etiology and pathogenesis are still essentially unclear. The histologic hallmark of this disease is proliferation of distinctive histiocytes within lymph node sinuses and in extranodal sites. Approximately 23% of patients with SHML, documented in the SHML Registry, presented with disease primarily in extranodal sites, and very few cases of SHML (<1%) involving the gastrointestinal system have been described in the literature. We report an unusual case of primary pancreatic SHML with infiltration of the process into peripancreatic, perinephric, and perisplenic adipose tissue, simulating malignancy.


1970 ◽  
Vol 8 (1) ◽  
pp. 28-31
Author(s):  
Md Faizul Islam Chowdhury ◽  
MA Kashem Khandaker ◽  
Hossain M Zaid ◽  
Hazera Khatun ◽  
Md Murad Hossain ◽  
...  

Mr. Shahinoor an 18 year old male got himself admitted at DMCH in July,2007 with the complaints of bilateral neck swelling and fever for 1 month. The enlarged lymph nodes were clustered in cervical regions involving both anterior and posterior chains and largest one measuring 5 cm×2.5 cm.He was otherwise healthy on examination. Full blood count showed neutrophil leukocytosis and raised ESR.Sputum for AFB was found negative and xray chest was normal. FNA of lymph node showed non specific lymphadenitis with reactive hyperplasis. Lymph node biopsy was done and histopathology revealed Rosai Dorfman Disease, a very rare yet fascinating disorder also called Sinus Histiocytosis with Massive Lymphadenopathy.It is a benign proliferative disorder of the histiocytes. He was not given any tretament and is under follow up.   DOI = 10.3329/jom.v8i1.1376 J MEDICINE 2007; 8 : 28-31


Author(s):  
Kalpana Giri ◽  
Ashok Baral ◽  
Niva Tiwari ◽  
Krishna Sharma

Rosai-Dorfman disease (RDD) is referred to as Sinus Histiocytosis Massive Lymphadenopathy (SHML) of unknown origin. We are reporting a rare case of Rosai Dorfman disease, a case of 6 years old male with a history of multiple painless bilateral submandibular and cervical lymphadenopathy.


2011 ◽  
Vol 135 (3) ◽  
pp. 390-393
Author(s):  
Changlee S Pang ◽  
David D Grier ◽  
Michael W Beaty

Abstract Sinus histiocytosis with massive lymphadenopathy (SHML), also known as Rosai-Dorfman disease, is a rare self-limiting disorder of histiocytes with unknown etiology. Sinus histiocytosis with massive lymphadenopathy is most common in children and young adults and is characterized by painless lymphadenopathy. Histologically there is a proliferation of sinus histiocytes with lymphophagocytosis or emperipolesis. On rare occasions, SHML has been associated with lymphoma, usually involving different anatomic sites and developing at different times. We report a case of concomitant SHML and nodal marginal zone lymphoma involving the same lymph node without involvement of other nodal or extranodal sites. The presence of concomitant SHML within the lymph node involved by nodal marginal zone lymphoma may represent the responsiveness of SHML histiocytes to B-cell–derived cytokines in lymphoproliferative disorders. To our knowledge, this is the first description of concomitant occurrence of SHML and nodal marginal zone lymphoma.


Author(s):  
Uppin Narayan Reddy ◽  
Swathi Chacham ◽  
Janampally Ravikiran ◽  
Jillalla Narsing Rao ◽  
Jakkampudi Nagasravani ◽  
...  

1970 ◽  
Vol 52 (195) ◽  
pp. 955-957
Author(s):  
Rakesh Digambar Waghmare

Sinus histiocytosis with massive lymphadenopathy (SHML), Rosai-Dorfman Disease, is a rare histiocytic syndrome first described by Rosai and Dorfman, most frequently seen in children and young adults.The disease is more common in males and in individuals of African descent but rare in Asians. It is mainly characterized by painless bilateral cervical lymph node enlargement and is often associated with fever and leucocytosis. This case is being reported for its rarity in presentation in an elderly female with both generalized nodal as well as extranodal manifestations. Without the awareness about RDD, the diagnosis of RDD is unexpected especially in South East Asian Countries where certain lymphadenopathies such as tuberculosis, metastatic malignancies and lymphomas are common. Keywords: emperipolesis; rosai-dorfman disease; sinus histiocytosis with massive lymphadenopathy.


2000 ◽  
Vol 114 (7) ◽  
pp. 565-567 ◽  
Author(s):  
P. Aluffi ◽  
A. Prestinari ◽  
A. Ramponi ◽  
M. Castri ◽  
F. Pia

We present a case of an 81-year-old man with a history of worsening dysphonia of six months duration. A year before a sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease) was diagnosed on a submandibular lymph node biopsy. On presentation the patient showed a mass in the left subglottic area with a modest reduction in the airway space. The patient underwent an endoscopic CO2 laser excision of the mass, without post-operative complications. Histopathological examination and electron microscope images confirmed the previous diagnosis of Rosai-Dorfman disease. The clinical and pathological features of this entity are discussed.


1986 ◽  
Vol 95 (4) ◽  
pp. 331-335 ◽  
Author(s):  
Vanessa G. Schweitzer ◽  
Gregg D. Bobier

Sinus histiocytosis with massive cervical lymphadenopathy (SHML) was originally described in 1969 as a benign clinicopathologic entity characterized by massive bilateral cervical lymphadenopathy, fever, leukocytosis, elevated ESR, and hypergammaglobulinemia, usually occurring within the first two decades of life. We present an illustrated case of an elderly patient with polyclonal hypergammaglobulinemia and a 2-year history of multilobulated cervical and submandibular lymphadenopathy. The etiology and pathogenesis of SHML are not known. Diagnosis requires lymph node biopsy to exclude other causes of cervical lymphadenopathy such as malignant lymphoma, malignant histiocytosis, metastatic carcinoma, and tuberculous lymphadenitis. Histologic examination shows marked dilatation of subcapsular and medullary lymph node sinuses containing large, foamy or vacuolated histiocytes. Although no curative treatment is known, corticosteroids, radiation therapy, vinblastine and oral cyclophosphamide, and surgery have been used to palliate constitutional symptoms and mechanical obstruction from massive lymphadenopathy. Since one third of SHML patients have evidence of disease for 5 years, and a mortality rate of 7% exists with benign histologic disease, all patients with SHML should be carefully screened for evidence of immunodeficiencies that may precipitate a fatal outcome.


Author(s):  
Aya Al-Rashdi ◽  
Badryia Al Qassabi ◽  
Radiya Al Ajmi ◽  
Suad Al Aghbari

Breast cancer (BC) and malignant lymphoma (ML) each of them are considered as a common primary malignant disease worldwide. Synchronous occurrences of BC and ML are rare. We are presenting a case of a 34 year old patient with breast cancer and Hodgkin lymphoma detected in non-sentinel lymph node biopsy. This represents a rare case of coexistence of dual malignancies. We review the literature and discuss possible etiologies for these synchronous tumors.


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