Concomitantly Diagnosed Disseminated M kansasii Infection and Hairy Cell Leukemia With Review of Pathophysiology
The association between Hairy Cell Leukemia (HCL) and non-tuberculous mycobacterial infections (NTMs) is well described, most notably Mycobacterium kansasii. The exact pathophysiology is not known. We report a case of a 31-year-old male with concomitantly diagnosed HCL and disseminated M kansasii in...
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description | The association between Hairy Cell Leukemia (HCL) and non-tuberculous mycobacterial infections (NTMs) is well described, most notably Mycobacterium kansasii. The exact pathophysiology is not known. We report a case of a 31-year-old male with concomitantly diagnosed HCL and disseminated M kansasii infection who presented with rash, pancytopenia, and bulky axillary lymphadenopathy. The M kansasii was initially diagnosed through use of cell-free DNA detection and confirmed by bone marrow and lymph node cultures. Hairy Cell Leukemia was diagnosed with peripheral flow cytometry and confirmed via the same bone marrow sample. His HCL was put into remission with a single course of cladribine and rituximab chemotherapy; however, his M kansasii infection persisted for 6 months despite aggressive antimicrobial and surgical therapy. It was finally controlled using high-dose rifampin in combination with azithromycin and ethambutol. This case highlights the known link between HCL and M kansasii. Furthermore, it hints at potential causes beyond chemotherapy-induced immunocompromise. Notable possibilities include HCL cells acting as sanctuary sites for M kansasii to evade the immune system, and subclinical M kansasii infections causing NLRP3 inflammasome overactivation to trigger the oncogenic transformation to HCL. More research into the pathophysiologic link between HCL and M kansasii infections would allow for more effective prevention, diagnosis, and treatment of these severe atypical infections which are the major cause of morbidity in the cladribine era of HCL treatment. |
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The exact pathophysiology is not known. We report a case of a 31-year-old male with concomitantly diagnosed HCL and disseminated M kansasii infection who presented with rash, pancytopenia, and bulky axillary lymphadenopathy. The M kansasii was initially diagnosed through use of cell-free DNA detection and confirmed by bone marrow and lymph node cultures. Hairy Cell Leukemia was diagnosed with peripheral flow cytometry and confirmed via the same bone marrow sample. His HCL was put into remission with a single course of cladribine and rituximab chemotherapy; however, his M kansasii infection persisted for 6 months despite aggressive antimicrobial and surgical therapy. It was finally controlled using high-dose rifampin in combination with azithromycin and ethambutol. This case highlights the known link between HCL and M kansasii. Furthermore, it hints at potential causes beyond chemotherapy-induced immunocompromise. Notable possibilities include HCL cells acting as sanctuary sites for M kansasii to evade the immune system, and subclinical M kansasii infections causing NLRP3 inflammasome overactivation to trigger the oncogenic transformation to HCL. More research into the pathophysiologic link between HCL and M kansasii infections would allow for more effective prevention, diagnosis, and treatment of these severe atypical infections which are the major cause of morbidity in the cladribine era of HCL treatment.</description><identifier>ISSN: 2324-7096</identifier><identifier>EISSN: 2324-7096</identifier><identifier>DOI: 10.1177/23247096241253343</identifier><identifier>PMID: 38767131</identifier><language>eng</language><publisher>Los Angeles, CA: SAGE Publications</publisher><subject>Adult ; Azithromycin - therapeutic use ; Bone marrow ; Case Report ; Chemotherapy ; Cladribine - therapeutic use ; Humans ; Infections ; Leukemia ; Leukemia, Hairy Cell - complications ; Leukemia, Hairy Cell - diagnosis ; Leukemia, Hairy Cell - drug therapy ; Male ; Mycobacterium Infections, Nontuberculous - complications ; Mycobacterium Infections, Nontuberculous - diagnosis ; Mycobacterium Infections, Nontuberculous - drug therapy ; Mycobacterium kansasii ; Pathophysiology ; Rifampin - therapeutic use ; Rituximab - therapeutic use</subject><ispartof>JIM - high impact case reports, 2024-01, Vol.12, p.23247096241253343-23247096241253343</ispartof><rights>2024 American Federation for Medical Research</rights><rights>2024 American Federation for Medical Research. 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The exact pathophysiology is not known. We report a case of a 31-year-old male with concomitantly diagnosed HCL and disseminated M kansasii infection who presented with rash, pancytopenia, and bulky axillary lymphadenopathy. The M kansasii was initially diagnosed through use of cell-free DNA detection and confirmed by bone marrow and lymph node cultures. Hairy Cell Leukemia was diagnosed with peripheral flow cytometry and confirmed via the same bone marrow sample. His HCL was put into remission with a single course of cladribine and rituximab chemotherapy; however, his M kansasii infection persisted for 6 months despite aggressive antimicrobial and surgical therapy. It was finally controlled using high-dose rifampin in combination with azithromycin and ethambutol. This case highlights the known link between HCL and M kansasii. Furthermore, it hints at potential causes beyond chemotherapy-induced immunocompromise. Notable possibilities include HCL cells acting as sanctuary sites for M kansasii to evade the immune system, and subclinical M kansasii infections causing NLRP3 inflammasome overactivation to trigger the oncogenic transformation to HCL. More research into the pathophysiologic link between HCL and M kansasii infections would allow for more effective prevention, diagnosis, and treatment of these severe atypical infections which are the major cause of morbidity in the cladribine era of HCL treatment.</description><subject>Adult</subject><subject>Azithromycin - therapeutic use</subject><subject>Bone marrow</subject><subject>Case Report</subject><subject>Chemotherapy</subject><subject>Cladribine - therapeutic use</subject><subject>Humans</subject><subject>Infections</subject><subject>Leukemia</subject><subject>Leukemia, Hairy Cell - complications</subject><subject>Leukemia, Hairy Cell - diagnosis</subject><subject>Leukemia, Hairy Cell - drug therapy</subject><subject>Male</subject><subject>Mycobacterium Infections, Nontuberculous - complications</subject><subject>Mycobacterium Infections, Nontuberculous - diagnosis</subject><subject>Mycobacterium Infections, Nontuberculous - drug therapy</subject><subject>Mycobacterium kansasii</subject><subject>Pathophysiology</subject><subject>Rifampin - therapeutic use</subject><subject>Rituximab - therapeutic use</subject><issn>2324-7096</issn><issn>2324-7096</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2024</creationdate><recordtype>article</recordtype><sourceid>AFRWT</sourceid><sourceid>EIF</sourceid><sourceid>BENPR</sourceid><recordid>eNp1UV1vFCEUJUZjm9of4Ish8cWXrXwNLE_GrNo2WaMxGh8Jw1x2aWdgHZia-fdls7VWjbxwufecA4eD0HNKzihV6jXjTCiiJROUNZwL_ggd73uLffPxg_oIneZ8RepaNowq_RQd8aWSinJ6jHarFF0aQrGx9DN-F-wmpgxdrXKGIURb6uEjvrYx2xwCvoweXAkpYhs7fGHDOOMV9D1ew3RdCRZ_D2WLv8BNgJ84efzZlm3abeccUp828zP0xNs-w-ndfoK-fXj_dXWxWH86v1y9XS-coLosnJNKL7W2jdSeK-elFI5ZDaLxugXJQDaaAijFleq8l5xB50QrWtaSrmv4CXpz0N1N7VBHEMtoe7Mbw2DH2SQbzJ-TGLZmk24MpZQoTlVVeHWnMKYfE-RihpBdtWojpCkbThpFVMOJrNCXf0Gv0jTG6s9wKvThsyuKHlBuTDmP4O9fQ4nZZ2r-ybRyXjy0cc_4lWAFnB0A2W7g97X_V7wFLN6qwg</recordid><startdate>20240101</startdate><enddate>20240101</enddate><creator>Stanton, Daniel J.</creator><creator>Quadri, Nadia Z.</creator><creator>Tanabe, Melinda B.</creator><general>SAGE Publications</general><general>Sage Publications Ltd</general><scope>AFRWT</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>PIMPY</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0003-1042-0554</orcidid></search><sort><creationdate>20240101</creationdate><title>Concomitantly Diagnosed Disseminated M kansasii Infection and Hairy Cell Leukemia With Review of Pathophysiology</title><author>Stanton, Daniel J. ; Quadri, Nadia Z. ; Tanabe, Melinda B.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c419t-cc679899a569f37cf664c2a9e45f9be62e6591ee77377dff632edc4b4b2b0dd53</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2024</creationdate><topic>Adult</topic><topic>Azithromycin - therapeutic use</topic><topic>Bone marrow</topic><topic>Case Report</topic><topic>Chemotherapy</topic><topic>Cladribine - therapeutic use</topic><topic>Humans</topic><topic>Infections</topic><topic>Leukemia</topic><topic>Leukemia, Hairy Cell - complications</topic><topic>Leukemia, Hairy Cell - diagnosis</topic><topic>Leukemia, Hairy Cell - drug therapy</topic><topic>Male</topic><topic>Mycobacterium Infections, Nontuberculous - complications</topic><topic>Mycobacterium Infections, Nontuberculous - diagnosis</topic><topic>Mycobacterium Infections, Nontuberculous - drug therapy</topic><topic>Mycobacterium kansasii</topic><topic>Pathophysiology</topic><topic>Rifampin - therapeutic use</topic><topic>Rituximab - therapeutic use</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Stanton, Daniel J.</creatorcontrib><creatorcontrib>Quadri, Nadia Z.</creatorcontrib><creatorcontrib>Tanabe, Melinda B.</creatorcontrib><collection>Sage Journals GOLD Open Access 2024</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Publicly Available Content Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>JIM - high impact case reports</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Stanton, Daniel J.</au><au>Quadri, Nadia Z.</au><au>Tanabe, Melinda B.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Concomitantly Diagnosed Disseminated M kansasii Infection and Hairy Cell Leukemia With Review of Pathophysiology</atitle><jtitle>JIM - high impact case reports</jtitle><addtitle>J Investig Med High Impact Case Rep</addtitle><date>2024-01-01</date><risdate>2024</risdate><volume>12</volume><spage>23247096241253343</spage><epage>23247096241253343</epage><pages>23247096241253343-23247096241253343</pages><issn>2324-7096</issn><eissn>2324-7096</eissn><abstract>The association between Hairy Cell Leukemia (HCL) and non-tuberculous mycobacterial infections (NTMs) is well described, most notably Mycobacterium kansasii. The exact pathophysiology is not known. We report a case of a 31-year-old male with concomitantly diagnosed HCL and disseminated M kansasii infection who presented with rash, pancytopenia, and bulky axillary lymphadenopathy. The M kansasii was initially diagnosed through use of cell-free DNA detection and confirmed by bone marrow and lymph node cultures. Hairy Cell Leukemia was diagnosed with peripheral flow cytometry and confirmed via the same bone marrow sample. His HCL was put into remission with a single course of cladribine and rituximab chemotherapy; however, his M kansasii infection persisted for 6 months despite aggressive antimicrobial and surgical therapy. It was finally controlled using high-dose rifampin in combination with azithromycin and ethambutol. This case highlights the known link between HCL and M kansasii. Furthermore, it hints at potential causes beyond chemotherapy-induced immunocompromise. Notable possibilities include HCL cells acting as sanctuary sites for M kansasii to evade the immune system, and subclinical M kansasii infections causing NLRP3 inflammasome overactivation to trigger the oncogenic transformation to HCL. More research into the pathophysiologic link between HCL and M kansasii infections would allow for more effective prevention, diagnosis, and treatment of these severe atypical infections which are the major cause of morbidity in the cladribine era of HCL treatment.</abstract><cop>Los Angeles, CA</cop><pub>SAGE Publications</pub><pmid>38767131</pmid><doi>10.1177/23247096241253343</doi><orcidid>https://orcid.org/0000-0003-1042-0554</orcidid><oa>free_for_read</oa></addata></record> |
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subjects | Adult Azithromycin - therapeutic use Bone marrow Case Report Chemotherapy Cladribine - therapeutic use Humans Infections Leukemia Leukemia, Hairy Cell - complications Leukemia, Hairy Cell - diagnosis Leukemia, Hairy Cell - drug therapy Male Mycobacterium Infections, Nontuberculous - complications Mycobacterium Infections, Nontuberculous - diagnosis Mycobacterium Infections, Nontuberculous - drug therapy Mycobacterium kansasii Pathophysiology Rifampin - therapeutic use Rituximab - therapeutic use |
title | Concomitantly Diagnosed Disseminated M kansasii Infection and Hairy Cell Leukemia With Review of Pathophysiology |
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