Non-Hodgkin's lymphoma with extensive emperipolesis mimicking Rosai–Dorfman disease: A rare case report

Rane, Sharada; Parkhi, Mayur; Vishwasrao, Sharvari D; Nakate, Leena

doi:10.4103/ijpm.ijpm_409_18

Cited by 4 publications

(4 citation statements)

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“…Furthermore, in late-stage RDD lesions, fibrosis becomes more prevalent, making the identification of residual RDD islands challenging [ 29 ]. While emperipolesis is a crucial indicator suggesting RDD disease, it is not entirely specific, as scattered emperipolesis can be observed in other histiocytic disorders such as Erdheim-Chester disease, yellow granuloma and malignant histiocytosis [ 30 – 32 ]. Meanwhile, immunohistochemistry plays a pivotal role in further delineating RDD disease.…”

Section: Discussion and Literature Reviewmentioning

confidence: 99%

Mediastinal Rosai-Dorfman Disease with KRAS mutation case report and literature review

Zhang,

Fang,

Wang

et al. 2024

J Cardiothorac Surg

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Background Rosai-Dorfman Disease (RDD) is a rare self-limiting histiocytosis, more prevalent in children and young adults. It typically manifests as painless bilateral massive cervical lymphadenopathy but may also extend to extra-nodal sites, with intrathoracic RDD noted in 2% of cases. Distinguishing mediastinal RDD from thymoma on imaging poses challenges, underscoring the reliance on pathological features and immunohistochemical staining for diagnosis. Case presentation Patient, male, 33 years old, underwent lung a CT revealing an enlarged round soft tissue shadow in the anterior superior mediastinum, compared to a year ago. Surgical resection removed the entire mass, thymus, and part of the pericardium, confirming RDD on pathology. Genetic testing using second-generation testing technology identified a KRAS gene point mutation. Conclusions No established treatment protocol currently exists for this disease. However, as genetic mutation research progresses, a novel therapeutic avenue is emerging: targeted therapy integrated with surgical interventions.

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Section: Discussion and Literature Reviewmentioning

confidence: 99%

Mediastinal Rosai-Dorfman Disease with KRAS mutation case report and literature review

Zhang,

Fang,

Wang

et al. 2024

J Cardiothorac Surg

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“…Their biological mechanisms and pathogenic roles are starting to emerge. Some findings suggested that lymphocytes may use the alternative cell‐in‐cell phenomenon to kill target tumour cells . Alternatively, tumour cells may destroy invading lymphocytes as a mechanism to escape immune surveillance .…”

Section: Discussionmentioning

confidence: 99%

“…Some findings suggested that lymphocytes may use the alternative cell-in-cell phenomenon to kill target tumour cells. 15,16 Alternatively, tumour cells may destroy invading lymphocytes as a mechanism to escape immune surveillance. 17 The consequences of emperipolesis were also debatable.…”

Section: Discussionmentioning

confidence: 99%

Emperipolesis mediated by CD8⁺ T cells correlates with biliary epithelia cell injury in primary biliary cholangitis

Zhao

et al. 2019

J Cellular Molecular Medi

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Primary biliary cholangitis (PBC) is an autoimmune disease characterized by chronic destruction of the bile ducts. A major unanswered question regarding the pathogenesis of PBC is the precise mechanisms of small bile duct injury. Emperipolesis is one of cell‐in‐cell structures that is a potential histological hallmark associated with chronic hepatitis B. This study aimed to clarify the pathogenesis and characteristics of emperipolesis in PBC liver injury. Sixty‐six PBC patients, diagnosed by liver biopsy combined with laboratory test, were divided into early‐stage PBC (stages I and II, n = 39) and late‐stage PBC (stages III and IV, n = 27). Emperipolesis was measured in liver sections stained with haematoxylin‐eosin. The expressions of CK19, CD3, CD4, CD8, CD20, Ki67 and apoptosis of BECs were evaluated by immunohistochemistry or immunofluorescence double labelling. Emperipolesis was observed in 62.1% of patients with PBC, and BECs were predominantly host cells. The number of infiltrating CD3+ and CD8+ T cells correlated with the advancement of emperipolesis (R2 = 0.318, P < .001; R2 = 0.060, P < .05). The cell numbers of TUNEL‐positive BECs and double staining for CK19 and Ki67 showed a significant positive correlation with emperipolesis degree (R2 = 0.236, P < .001; R2 = 0.267, P < .001). We conclude that emperipolesis mediated by CD8+ T cells appears to be relevant to apoptosis of BEC and thus may aggravate the further injury of interlobular bile ducts.

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“…For cytological specimen evaluation, the focus is on detection of atypical cells, and background cell characteristics, such as components of inflammatory cells, are rarely considered (14), for example in special tumor cell types with lymphocyte infiltration such as Warthin tumor of the salivary glands (15) and seminoma of the testis/dysgerminoma of the ovary (16). Numerous studies describing emperipolesis and cell cannibalism are either about experiments that utilized cytological specimens or case reports (5,8,(17)(18)(19)(20). However, in light of recent advancements in other techniques, including immunocytochemistry, in-situ hybridization and reverse transcription-quantitative PCR (21), cytological evaluations should consider other characteristics, focusing on less frequently observed findings such as CiCP, in addition to routinely observed characteristics, including cellular atypia (22).…”

Section: Introductionmentioning

confidence: 99%

High neutrophil incorporation rate of ascitic fluid cytology as an indicator of cancerous ascites

et al. 2020

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The cell-in-cell phenomenon (CiCP) involves the incorporation of a viable cell by other cells (host cells) and includes two concepts: Emperipolesis and cell cannibalism. The former involves the incorporation of hematopoietic cells as the incorporated cells, while the latter involves cell incorporation by tumor cells as host cells. A total of 239 peritoneal cavity fluid cytology specimens were evaluated for CiCP and the number of singly detectable nuclei (SDN) were measured by examining virtual slide image files. The rates of CiCP-positive cases (RCPCs) and CiCP emergence rate (CER)/SDN were significantly higher in ascites samples than in peritoneal washing samples (P<0.0001 and P=0.0026, respectively), although the numbers of SDN were not significantly different between the groups (P=0.8063). Both the RCPCs and CER/SDN were significantly higher in tumor-positive specimens than in tumor-negative specimens (P=0.0220 and P=0.0312, respectively), although the numbers of SDN were not significantly different between the samples (P=0.2471). Most of the incorporated cells were lymphocytes and the host cells were macrophages; however, the rate of neutrophil incorporation (NI) by host cells in the total CiCP cells in a sample was significantly higher in tumor-positive specimens than in tumor-negative specimens (P=0.0288). NI was mainly performed via emperipolesis by macrophages, with only six examples not by macrophages observed among all CiCP samples. The threshold NI rate/total CiCP (NI/CiCP) between tumor-positive and tumor-negative groups was 11.1% (P=0.0115). Using this threshold, the peripheral blood leukocyte count was significantly higher in the high-NI/CiCP group than in the low-NI/CiCP group (P=0.0022). The present findings revealed novel aspects of less frequently observed CiCP in ascitic fluid cytology by utilizing combined manual and computer assisted image analysis evaluation of samples. Notably, the present study indicated the importance of increased NI as an indicator of cancerous ascites.

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Non-Hodgkin's lymphoma with extensive emperipolesis mimicking Rosai–Dorfman disease: A rare case report

Cited by 4 publications

References 0 publications

Mediastinal Rosai-Dorfman Disease with KRAS mutation case report and literature review

Mediastinal Rosai-Dorfman Disease with KRAS mutation case report and literature review

Emperipolesis mediated by CD8⁺ T cells correlates with biliary epithelia cell injury in primary biliary cholangitis

High neutrophil incorporation rate of ascitic fluid cytology as an indicator of cancerous ascites

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Non-Hodgkin's lymphoma with extensive emperipolesis mimicking Rosai–Dorfman disease: A rare case report

Cited by 4 publications

References 0 publications

Mediastinal Rosai-Dorfman Disease with KRAS mutation case report and literature review

Mediastinal Rosai-Dorfman Disease with KRAS mutation case report and literature review

Emperipolesis mediated by CD8+ T cells correlates with biliary epithelia cell injury in primary biliary cholangitis

High neutrophil incorporation rate of ascitic fluid cytology as an indicator of cancerous ascites

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Emperipolesis mediated by CD8⁺ T cells correlates with biliary epithelia cell injury in primary biliary cholangitis