scholarly journals Pulmonary infiltration as the initial manifestation of chronic lymphoproliferative disorder of natural killer cells: a case report and literature review

2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Jinjing Zhang ◽  
Pingping Wang ◽  
Xiaojing Yan

Abstract Background Chronic lymphoproliferative disorder of natural killer cells (CLPD-NK) is an extremely rare haematological disease. To the best of our knowledge, pulmonary infiltration in CLPD-NK has not been reported before. Our case study aimed to present the clinical characteristics, chest computed tomography (CT) findings, and flow cytometry immunophenotyping (FCI) results of an unusual case of migratory pulmonary infiltration in a patient with CLPD-NK. Case presentation A 51-year-old female patient was admitted to our hospital on October 8, 2019. Eight months before this visit, she had been diagnosed with pneumonia in a community hospital with 1 month of low-grade fever and had recovered after oral antibiotic administration. During follow-up, the patient presented with persistent peripheral blood (PB) lymphocytosis and ground-glass opacities on lung CT scans without any symptoms and signs or any evidence of infectious, allergic or autoimmunity pulmonary diseases. Abnormal NK cells were identified in the PB, bone marrow and bronchoalveolar lavage fluid (BALF) using FCI in our hospital. Eventually, the patient was diagnosed with pulmonary infiltration of CLPD-NK. The patient had an indolent clinical course without symptoms, hepatosplenomegaly or palpable lymphadenopathy and did not receive any therapy. The patient has remained in a good performance status 13 months after the diagnosis. Conclusions Our study described a unique case of pulmonary infiltration in a patient with CLPD-NK. The present case highlights the importance of FCI of the BALF in patients with lymphocytosis and pulmonary shadows to avoid misdiagnosis.

2020 ◽  
Vol 10 (4) ◽  
Author(s):  
Vanessa Rebecca Gasparini ◽  
Andrea Binatti ◽  
Alessandro Coppe ◽  
Antonella Teramo ◽  
Cristina Vicenzetto ◽  
...  

Cureus ◽  
2020 ◽  
Author(s):  
Shafain Sheikh ◽  
Sehreen Jahangir ◽  
Samreen Khan ◽  
Imran Ahmed

2019 ◽  
Vol 109 (5) ◽  
pp. 563-571 ◽  
Author(s):  
Toru Kawakami ◽  
Nodoka Sekiguchi ◽  
Jun Kobayashi ◽  
Taku Yamane ◽  
Sayaka Nishina ◽  
...  

2021 ◽  
Vol 10 ◽  
Author(s):  
Edoardo Giussani ◽  
Andrea Binatti ◽  
Giulia Calabretto ◽  
Vanessa Rebecca Gasparini ◽  
Antonella Teramo ◽  
...  

Large granular lymphocyte leukemias (LGLL) are sustained by proliferating cytotoxic T cells or NK cells, as happens in Chronic Lymphoproliferative Disorder of Natural Killer cells (CLPD-NK), whose etiology is only partly understood. Different hypotheses have been proposed on the original events triggering NK cell hyperactivation and transformation, including a role of viral agents. In this perspective, we revise the lines of evidence that suggested a pathogenetic role in LGLL of the exposure to retroviruses and that identified Epstein Barr Virus (EBV) in other NK cell leukemias and lymphomas and focus on the contrasting data about the importance of viral agents in CLPD-NK. EBV was detected in aggressive NK leukemias but not in the indolent CLPD-NK, where seroreactivity against HTLV-1 retrovirus envelope BA21 protein antigens has been reported in patients, although lacking clear evidence of HTLV infection. We next present original results of whole exome sequencing data analysis that failed to identify viral sequences in CLPD-NK. We recently demonstrated that proliferating NK cells of patients harbor several somatic lesions likely contributing to sustain NK cell proliferation. Thus, we explore whether “neoantigens” similar to the BA21 antigen could be generated by aberrancies present in the leukemic clone. In light of the literature and new data, we evaluated the intriguing hypothesis that NK cell activation can be caused by retroviral agents located outside the hematopoietic compartment and on the possible mechanisms involved with the prospects of immunotherapy-based approaches to limit the growth of NK cells in CLPD-NK disease.


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