Risks of Ruxolitinib in STAT1 Gain-of-Function-Associated Severe Fungal Disease

被引:50
作者
Zimmerman, Ofer [1 ]
Rosler, Berenice [2 ,3 ]
Zerbe, Christa S. [1 ]
Rosen, Lindsey B. [1 ]
Hsu, Amy P. [1 ]
Uzel, Gulbu [1 ]
Freeman, Alexandra F. [1 ]
Sampaio, Elizabeth P. [1 ]
Rosenzwieg, Sergio D. [4 ]
Kuehn, Hye Sun [4 ]
Kim, Tiffany [5 ]
Brooks, Kristina M. [5 ]
Kumar, Parag [5 ]
Wang, Xiaowen [2 ,3 ,6 ]
Netea, Mihai G. [2 ,3 ]
van de Veerdonk, Frank L. [2 ,3 ]
Holland, Steven M. [1 ]
机构
[1] Natl Inst Allergy & Immunol, Immunopathogenesis Sect, Lab Clin Infect Dis, NIH, Bethesda, MD USA
[2] Radboud Univ Nijmegen, Med Ctr, Dept Internal Med, Nijmegen, Netherlands
[3] Radboud Univ Nijmegen, Med Ctr, Radboud Ctr Infect Dis RCI, Nijmegen, Netherlands
[4] NIH, Immunol Serv, Dept Lab Med, Natl Inst Clin Ctr, Bldg 10, Bethesda, MD 20892 USA
[5] NIH, Dept Pharm, Clin Ctr, Bethesda, MD 20892 USA
[6] Peking Univ, Hosp 1, Dept Dermatol, Beijing, Peoples R China
基金
美国国家卫生研究院; 欧洲研究理事会;
关键词
STAT1; gain of function; ruxolitinib; coccidiomycosis; dermatophytosis; CHRONIC MUCOCUTANEOUS CANDIDIASIS; SIGNAL TRANSDUCER; INCB018424; PHOSPHATE; HEALTHY-VOLUNTEERS; FUNCTION MUTATIONS; ACTIVATOR; PHARMACODYNAMICS; PHARMACOKINETICS; UNDERLIE;
D O I
10.1093/ofid/ofx202
中图分类号
R392 [医学免疫学]; Q939.91 [免疫学];
学科分类号
100102 ;
摘要
Heterozygous STAT1 gain-of-function (GOF) mutations are associated with chronic mucocutaneous candidiasis and a broad spectrum of infectious, inflammatory, and vascular manifestations. We describe therapeutic failures with the Janus Kinase (JAK) inhibitor ruxolitinib in 2 STAT1 GOF patients with severe invasive or cutaneous fungal infections.
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共 18 条
[1]   A study on the risk of fungal infection with tofacitinib (CP-690550), a novel oral agent for rheumatoid arthritis [J].
Chen, Yong ;
Gong, Fang-Yuan ;
Li, Zhen-Jun ;
Gong, Zheng ;
Zhou, Zhe ;
Ma, Shu-Yan ;
Gao, Xiao-Ming .
SCIENTIFIC REPORTS, 2017, 7
[2]   Ruxolitinib is a potent immunosuppressive compound: is it time for anti-infective prophylaxis? [J].
Heine, Annkristin ;
Brossart, Peter ;
Wolf, Dominik .
BLOOD, 2013, 122 (23) :3843-3844
[3]   Use of ruxolitinib to successfully treat chronic mucocutaneous candidiasis caused by gain-of-function signal transducer and activator of transcription 1 (STAT1) mutation [J].
Higgins, Eleanor ;
Al Shehri, Tariq ;
McAleer, Maeve A. ;
Conlon, Niall ;
Feighery, Conleth ;
Lilic, Desa ;
Irvine, Alan D. .
JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY, 2015, 135 (02) :551-U356
[4]   PKC Signaling Regulates Drug Resistance of the Fungal Pathogen Candida albicans via Circuitry Comprised of Mkc1, Calcineurin, and Hsp90 [J].
LaFayette, Shantelle L. ;
Collins, Cathy ;
Zaas, Aimee K. ;
Schell, Wiley A. ;
Betancourt-Quiroz, Marisol ;
Gunatilaka, A. A. Leslie ;
Perfect, John R. ;
Cowen, Leah E. .
PLOS PATHOGENS, 2010, 6 (08) :79-80
[5]   Gain-of-function human STAT1 mutations impair IL-17 immunity and underlie chronic mucocutaneous candidiasis [J].
Liu, Luyan ;
Okada, Satoshi ;
Kong, Xiao-Fei ;
Kreins, Alexandra Y. ;
Cypowyj, Sophie ;
Abhyankar, Avinash ;
Toubiana, Julie ;
Itan, Yuval ;
Audry, Magali ;
Nitschke, Patrick ;
Masson, Cecile ;
Toth, Beata ;
Flatot, Jerome ;
Migaud, Melanie ;
Chrabieh, Maya ;
Kochetkov, Tatiana ;
Bolze, Alexandre ;
Borghesi, Alessandro ;
Toulon, Antoine ;
Hiller, Julia ;
Eyerich, Stefanie ;
Eyerich, Kilian ;
Gulacsy, Vera ;
Chernyshova, Ludmyla ;
Chernyshov, Viktor ;
Bondarenko, Anastasia ;
Cortes Grimaldo, Rosa Maria ;
Blancas-Galicia, Lizbeth ;
Madrigal Beas, Ileana Maria ;
Roesler, Joachim ;
Magdorf, Klaus ;
Engelhard, Dan ;
Thumerelle, Caroline ;
Burgel, Pierre-Regis ;
Hoernes, Miriam ;
Drexel, Barbara ;
Seger, Reinhard ;
Kusuma, Theresia ;
Jansson, Annette F. ;
Sawalle-Belohradsky, Julie ;
Belohradsky, Bernd ;
Jouanguy, Emmanuelle ;
Bustamante, Jacinta ;
Bue, Melanie ;
Karin, Nathan ;
Wildbaum, Gizi ;
Bodemer, Christine ;
Lortholary, Olivier ;
Fischer, Alain ;
Blanche, Stephane .
JOURNAL OF EXPERIMENTAL MEDICINE, 2011, 208 (08) :1635-1648
[6]   Ruxolitinib Induces Interleukin 17 and Ameliorates Chronic Mucocutaneous Candidiasis Caused by STAT1 Gain-of-Function Mutation [J].
Moessner, Rotraut ;
Diering, Nina ;
Bader, Oliver ;
Forkel, Susann ;
Overbeck, Tobias ;
Gross, Uwe ;
Grimbacher, Bodo ;
Schoen, Michael P. ;
Buhl, Timo .
CLINICAL INFECTIOUS DISEASES, 2016, 62 (07)
[7]   Risk Factors for Disseminated Coccidioidomycosis, United States [J].
Odio, Camila D. ;
Marciano, Beatriz E. ;
Galgiani, John N. ;
Holland, Steven M. .
EMERGING INFECTIOUS DISEASES, 2017, 23 (02) :308-311
[8]   NEW ALKALOID AM-2282 OF STREPTOMYCES ORIGIN TAXONOMY, FERMENTATION, ISOLATION AND PRELIMINARY CHARACTERIZATION [J].
OMURA, S ;
IWAI, Y ;
HIRANO, A ;
NAKAGAWA, A ;
AWAYA, J ;
TSUCHIYA, H ;
TAKAHASHI, Y ;
MASUMA, R .
JOURNAL OF ANTIBIOTICS, 1977, 30 (04) :275-282
[9]   Signal transducer and activator of transcription 1 (STAT1) gain-of-function mutations and disseminated coccidioidomycosis and histoplasmosis [J].
Sampaio, Elizabeth P. ;
Hsu, Amy P. ;
Pechacek, Joseph ;
Bax, Hannelore I. ;
Dias, Dalton L. ;
Paulson, Michelle L. ;
Chandrasekaran, Prabha ;
Rosen, Lindsey B. ;
Carvalho, Daniel S. ;
Ding, Li ;
Vinh, Donald C. ;
Browne, Sarah K. ;
Datta, Shrimati ;
Milner, Joshua D. ;
Kuhns, Douglas B. ;
Priel, Debra A. Long ;
Sadat, Mohammed A. ;
Shiloh, Michael ;
De Marco, Brendan ;
Alvares, Michael ;
Gillman, Jason W. ;
Ramarathnam, Vivek ;
de la Morena, Maite ;
Bezrodnik, Liliana ;
Moreira, Ileana ;
Uzel, Gulbu ;
Johnson, Daniel ;
Spalding, Christine ;
Zerbe, Christa S. ;
Wiley, Henry ;
Greenberg, David E. ;
Hoover, Susan E. ;
Rosenzweig, Sergio D. ;
Galgiani, John N. ;
Holland, Steven M. .
JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY, 2013, 131 (06) :1624-+
[10]   The Effect of CYP3A4 Inhibition or Induction on the Pharmacokinetics and Pharmacodynamics of Orally Administered Ruxolitinib (INCB018424 Phosphate) in Healthy Volunteers [J].
Shi, Jack G. ;
Chen, Xuejun ;
Emm, Thomas ;
Scherle, Peggy A. ;
McGee, Ryan F. ;
Lo, Yvonne ;
Landman, Robert R. ;
McKeever, Edward G., Jr. ;
Punwani, Naresh G. ;
Williams, William V. ;
Yeleswaram, Swamy .
JOURNAL OF CLINICAL PHARMACOLOGY, 2012, 52 (06) :809-818