{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2023,11,21]],"date-time":"2023-11-21T00:19:54Z","timestamp":1700525994646},"reference-count":24,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2023,9,1]],"date-time":"2023-09-01T00:00:00Z","timestamp":1693526400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2023,9,1]],"date-time":"2023-09-01T00:00:00Z","timestamp":1693526400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Neurol"],"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Chronic lymphoproliferative disorders of natural killer cells (CLPD-NK) is a rare lymphoproliferative disease. Peripheral neuropathy is an unusual symptom of CLPD-NK. We report a case of peripheral neuropathy associated with CLPD-NK and perform a review of literatures.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>a 62-year\u2010old woman presented with progressive numbness and weakness in both extremities. Electrophysiological examinations indicated a sensorimotor polyneuropathy. Peripheral blood examination revealed that the number of white blood cells (WBC) and lymphocytes were significantly increased. Flow cytometry analysis identified that 84% of the lymphocytes are NK cells that mainly expressed CD56, combined with variable expression of CD16, CD2, CD7, CD94, granzyme B, perforin, and CD158 but negative for CD3. Sural nerve biopsy revealed that a plethora of NK cells infiltrated into nerve fascicles. On treatment with combined cyclophosphamide and corticosteroids, her symptoms rapidly improved. Moreover, the absolute lymphocyte count and its proportion recovered to normal range after 3 months\u2019 treatment.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusion<\/jats:title>\n                <jats:p>To the best of our knowledge, this is the first case report of peripheral neuropathy associated with CLPD-NK from Chinese. This rare lymphoproliferative disease should be considered if peripheral neuropathy combines with increased WBC or lymphocytes. Immunosuppressive drugs are the major treatment and most patients can achieve a good prognosis.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s12883-023-03310-7","type":"journal-article","created":{"date-parts":[[2023,9,1]],"date-time":"2023-09-01T07:02:50Z","timestamp":1693551770000},"update-policy":"http:\/\/dx.doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Peripheral neuropathy associated with chronic lymphoproliferative disorders of natural killer cells (CLPD-NK): a case report and literature review"],"prefix":"10.1186","volume":"23","author":[{"given":"Hong","family":"An","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jiaxiang","family":"Guo","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hongliang","family":"Guo","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wenli","family":"Hu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ming","family":"Lu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2023,9,1]]},"reference":[{"key":"3310_CR1","doi-asserted-by":"publisher","first-page":"960","DOI":"10.1046\/j.1365-2141.1999.01624.x","volume":"106","author":"GR Rabbani","year":"1999","unstructured":"Rabbani GR, Phyliky RL, Tefferi A. A long-term study of patients with chronic natural killer cell lymphocytosis. Br J Haematol. 1999;106:960\u20136.","journal-title":"Br J Haematol"},{"key":"3310_CR2","doi-asserted-by":"publisher","first-page":"65","DOI":"10.1007\/s12308-009-0034-z","volume":"2","author":"MS Lim","year":"2009","unstructured":"Lim MS, de Leval L, Quintanilla-Martinez L. Commentary on the 2008 who classification of mature t- and nk-cell neoplasms. J Hematop. 2009;2:65\u201373.","journal-title":"J Hematop"},{"key":"3310_CR3","doi-asserted-by":"publisher","first-page":"e455","DOI":"10.1038\/bcj.2016.59","volume":"6","author":"MV Shah","year":"2016","unstructured":"Shah MV, Hook CC, Call TG, Go RS. A population-based study of large granular lymphocyte leukemia. Blood Cancer J. 2016;6:e455.","journal-title":"Blood Cancer J"},{"key":"3310_CR4","doi-asserted-by":"publisher","first-page":"1449","DOI":"10.1038\/leu.2016.68","volume":"30","author":"AG Dinmohamed","year":"2016","unstructured":"Dinmohamed AG, Brink M, Visser O, Jongen-Lavrencic M. Population-based analyses among 184 patients diagnosed with large granular lymphocyte leukemia in the netherlands between 2001 and 2013. Leukemia. 2016;30:1449\u201351.","journal-title":"Leukemia"},{"key":"3310_CR5","doi-asserted-by":"publisher","first-page":"2030","DOI":"10.1093\/annonc\/mdu369","volume":"25","author":"E Poullot","year":"2014","unstructured":"Poullot E, Zambello R, Leblanc F, Bareau B, De March E, Roussel M, et al. Chronic natural killer lymphoproliferative disorders: characteristics of an international cohort of 70 patients. Ann Oncol. 2014;25:2030\u20135.","journal-title":"Ann Oncol"},{"key":"3310_CR6","doi-asserted-by":"publisher","first-page":"616","DOI":"10.1200\/EDBK_200689","volume":"38","author":"A Moignet","year":"2018","unstructured":"Moignet A, Lamy T. Latest advances in the diagnosis and treatment of large granular lymphocytic leukemia. Am Soc Clin Oncol Educ Book. 2018;38:616\u201325.","journal-title":"Am Soc Clin Oncol Educ Book"},{"key":"3310_CR7","doi-asserted-by":"publisher","first-page":"1534","DOI":"10.3324\/haematol.2009.018481","volume":"95","author":"B Bareau","year":"2010","unstructured":"Bareau B, Rey J, Hamidou M, Donadieu J, Morcet J, Reman O, et al. Analysis of a french cohort of patients with large granular lymphocyte leukemia: a report on 229 cases. Haematologica. 2010;95:1534\u201341.","journal-title":"Haematologica"},{"key":"3310_CR8","doi-asserted-by":"publisher","first-page":"2764","DOI":"10.1182\/blood-2010-07-296962","volume":"117","author":"T Lamy","year":"2011","unstructured":"Lamy T, Loughran TP. Jr. How i treat lgl leukemia. Blood. 2011;117:2764\u201374.","journal-title":"Blood"},{"key":"3310_CR9","doi-asserted-by":"publisher","first-page":"207","DOI":"10.1016\/j.beha.2019.06.006","volume":"32","author":"G Barila","year":"2019","unstructured":"Barila G, Calabretto G, Teramo A, Vicenzetto C, Gasparini VR, Semenzato G, et al. T cell large granular lymphocyte leukemia and chronic nk lymphocytosis. Best Pract Res Clin Haematol. 2019;32:207\u201316.","journal-title":"Best Pract Res Clin Haematol"},{"key":"3310_CR10","doi-asserted-by":"publisher","first-page":"911","DOI":"10.1111\/bjh.14487","volume":"180","author":"A Dutta","year":"2018","unstructured":"Dutta A, Yan D, Hutchison RE, Mohi G. Stat3 mutations are not sufficient to induce large granular lymphocytic leukaemia in mice. Br J Haematol. 2018;180:911\u20135.","journal-title":"Br J Haematol"},{"key":"3310_CR11","doi-asserted-by":"publisher","first-page":"119","DOI":"10.1016\/j.jns.2005.01.013","volume":"232","author":"M Noguchi","year":"2005","unstructured":"Noguchi M, Yoshita M, Sakai K, Matsumoto Y, Arahata M, Ontachi Y, et al. Peripheral neuropathy associated with chronic natural killer cell lymphocytosis. J Neurol Sci. 2005;232:119\u201322.","journal-title":"J Neurol Sci"},{"key":"3310_CR12","doi-asserted-by":"publisher","first-page":"85","DOI":"10.1097\/01.mph.0000152571.06437.3f","volume":"27","author":"H Wex","year":"2005","unstructured":"Wex H, Aumann V, Hausler M, Vorwerk P, Mittler U. Chronic natural killer cell lymphocytosis is associated with elevated cytotoxic activity of natural killer cells. J Pediatr Hematol Oncol. 2005;27:85\u20139.","journal-title":"J Pediatr Hematol Oncol"},{"key":"3310_CR13","doi-asserted-by":"publisher","first-page":"1501","DOI":"10.1007\/s00277-018-3310-0","volume":"97","author":"NY Saini","year":"2018","unstructured":"Saini NY, Bathini V. Large granular lymphocytic leukemia-associated peripheral neuropathy. Ann Hematol. 2018;97:1501\u20134.","journal-title":"Ann Hematol"},{"key":"3310_CR14","doi-asserted-by":"publisher","first-page":"588","DOI":"10.1212\/01.WNL.0000133410.41914.AD","volume":"63","author":"WY Au","year":"2004","unstructured":"Au WY, Mak W, Ho SL, Leung SY, Kwong YL. Reversible paraneoplastic neuropathy associated with t-cell large granular lymphocyte leukemia. Neurology. 2004;63:588\u20139.","journal-title":"Neurology"},{"key":"3310_CR15","doi-asserted-by":"publisher","first-page":"1759","DOI":"10.2169\/internalmedicine.56.8881","volume":"56","author":"K Oshimi","year":"2017","unstructured":"Oshimi K. Clinical features, pathogenesis, and treatment of large granular lymphocyte leukemias. Intern Med. 2017;56:1759\u201369.","journal-title":"Intern Med"},{"key":"3310_CR16","doi-asserted-by":"publisher","first-page":"416","DOI":"10.1080\/10428194.2017.1339880","volume":"59","author":"SR Sanikommu","year":"2018","unstructured":"Sanikommu SR, Clemente MJ, Chomczynski P, Afable MG 2nd, Jerez A, Thota S, et al. Clinical features and treatment outcomes in large granular lymphocytic leukemia (lgll). Leuk Lymphoma. 2018;59:416\u201322.","journal-title":"Leuk Lymphoma"},{"key":"3310_CR17","doi-asserted-by":"publisher","first-page":"1082","DOI":"10.1182\/blood-2016-08-692590","volume":"129","author":"T Lamy","year":"2017","unstructured":"Lamy T, Moignet A, Loughran TP. Jr. Lgl leukemia: from pathogenesis to treatment. Blood. 2017;129:1082\u201394.","journal-title":"Blood"},{"key":"3310_CR18","doi-asserted-by":"publisher","first-page":"772","DOI":"10.1002\/ajh.26183","volume":"96","author":"N Dong","year":"2021","unstructured":"Dong N, Castillo Tokumori F, Isenalumhe L, Zhang Y, Tandon A, Knepper TC, et al. Large granular lymphocytic leukemia - a retrospective study of 319 cases. Am J Hematol. 2021;96:772\u201380.","journal-title":"Am J Hematol"},{"key":"3310_CR19","doi-asserted-by":"publisher","first-page":"3500","DOI":"10.1182\/blood-2009-07-233403","volume":"114","author":"CE Chee","year":"2009","unstructured":"Chee CE, Warrington KJ, Tefferi A. Chronic natural killer-cell lymphocytosis successfully treated with alemtuzumab. Blood. 2009;114:3500\u20132.","journal-title":"Blood"},{"key":"3310_CR20","doi-asserted-by":"publisher","first-page":"e22","DOI":"10.1016\/S2352-3026(15)00227-6","volume":"3","author":"B Dumitriu","year":"2016","unstructured":"Dumitriu B, Ito S, Feng X, Stephens N, Yunce M, Kajigaya S, et al. Alemtuzumab in t-cell large granular lymphocytic leukaemia: interim results from a single-arm, open-label, phase 2 study. Lancet Haematol. 2016;3:e22\u201329.","journal-title":"Lancet Haematol"},{"key":"3310_CR21","doi-asserted-by":"publisher","first-page":"2427","DOI":"10.1038\/leu.2015.280","volume":"29","author":"B Bilori","year":"2015","unstructured":"Bilori B, Thota S, Clemente MJ, Patel B, Jerez A, Afable Ii M, et al. Tofacitinib as a novel salvage therapy for refractory t-cell large granular lymphocytic leukemia. Leukemia. 2015;29:2427\u20139.","journal-title":"Leukemia"},{"key":"3310_CR22","doi-asserted-by":"crossref","unstructured":"Rivero A, Mozas P, Jimenez L, Lopez-Guerra M, Colomer D, Bataller A et al. Clinicobiological characteristics and outcomes of patients with t-cell large granular lymphocytic leukemia and chronic lymphoproliferative disorder of natural killer cells from a single institution. Cancers (Basel). 2021;13.","DOI":"10.3390\/cancers13153900"},{"key":"3310_CR23","doi-asserted-by":"publisher","first-page":"51","DOI":"10.1172\/JCI114168","volume":"84","author":"K Kawa-Ha","year":"1989","unstructured":"Kawa-Ha K, Ishihara S, Ninomiya T, Yumura-Yagi K, Hara J, Murayama F, et al. Cd3-negative lymphoproliferative disease of granular lymphocytes containing epstein-barr viral DNA. J Clin Invest. 1989;84:51\u20135.","journal-title":"J Clin Invest"},{"key":"3310_CR24","doi-asserted-by":"publisher","first-page":"673","DOI":"10.1182\/blood-2011-10-381921","volume":"119","author":"H Kimura","year":"2012","unstructured":"Kimura H, Ito Y, Kawabe S, Gotoh K, Takahashi Y, Kojima S, et al. Ebv-associated t\/nk-cell lymphoproliferative diseases in nonimmunocompromised hosts: prospective analysis of 108 cases. Blood. 2012;119:673\u201386.","journal-title":"Blood"}],"container-title":["BMC Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12883-023-03310-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12883-023-03310-7\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12883-023-03310-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,11,20]],"date-time":"2023-11-20T11:19:56Z","timestamp":1700479196000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcneurol.biomedcentral.com\/articles\/10.1186\/s12883-023-03310-7"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,9,1]]},"references-count":24,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2023,12]]}},"alternative-id":["3310"],"URL":"https:\/\/doi.org\/10.1186\/s12883-023-03310-7","relation":{},"ISSN":["1471-2377"],"issn-type":[{"value":"1471-2377","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,9,1]]},"assertion":[{"value":"3 April 2023","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"27 June 2023","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"1 September 2023","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"This study has been approved by the Medical Ethical Research Committee of Beijing Chaoyang Hospital Affiliated to Capital Medical University. The Ethical Approval Code is 2022\u2009\u2212\u2009205.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient for publication of the details of their medical case and any accompanying images.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"314"}}