Waki Imoto1,2,3,4, Koichi Yamada1,2,4, Yuriko Hajika5, Kousuke Okamoto6, Yuka Myodo4, Makoto Niki4, Gaku Kuwabara1,2,3,4, Kazushi Yamairi1,2,4, Wataru Shibata1,2,4, Naoko Yoshii1,2,3,4, Kiyotaka Nakaie1,4, Kazutaka Yoshizawa7, Hiroki Namikawa1,8, Tetsuya Watanabe3, Kazuhisa Asai3, Hiroshi Moro7, Yukihiro Kaneko9, Tomoya Kawaguchi3, Yoshiaki Itoh6, Hiroshi Kakeya10,11,12. 1. Department of Infection Control Science, Osaka City University Graduate School of Medicine, 1-4-3, Asahi-machi, Abeno-ku, Osaka, 545-8585, Japan. 2. Department of Infectious Disease Medicine, Osaka City University Hospital, 1-5-7 Asahi-machi, Abeno-ku, Osaka, 545-8586, Japan. 3. Department of Respiratory Medicine, Osaka City University Graduate School of Medicine, 1-4-3 Asahi-machi, Abeno-ku, Osaka, 545-8585, Japan. 4. Department of Infection Control and Prevention, Osaka City University Hospital, 1-5-7 Asahi-machi, Abeno-ku, Osaka, 545-8586, Japan. 5. Department of Metabolism, Endocrinology, Osaka City University Graduate School of Medicine, 1-4-3 Asahi-machi, Abeno-ku, Osaka, 545-8585, Japan. 6. Department of Neurology, Osaka City University Graduate School of Medicine, 1-4-3 Asahi-machi, Abeno-ku, Osaka, 545-8585, Japan. 7. Department of Respiratory Medicine and Infectious Diseases, Niigata University Graduate School of Medical and Dental Sciences, 1-757 Asahimachi-dori, Chuo-ku, Niigata, 951-8510, Japan. 8. Department of Medical Education and General Practice, Osaka City University Graduate School of Medicine, 1-4-3 Asahi-machi, Abeno-ku, Osaka, 545-8585, Japan. 9. Department of Bacteriology, Osaka City University Graduate School of Medicine, 1-4-3 Asahi-machi, Abeno-ku, Osaka, 545-8585, Japan. 10. Department of Infection Control Science, Osaka City University Graduate School of Medicine, 1-4-3, Asahi-machi, Abeno-ku, Osaka, 545-8585, Japan. kakeya@med.osaka-cu.ac.jp. 11. Department of Infectious Disease Medicine, Osaka City University Hospital, 1-5-7 Asahi-machi, Abeno-ku, Osaka, 545-8586, Japan. kakeya@med.osaka-cu.ac.jp. 12. Department of Infection Control and Prevention, Osaka City University Hospital, 1-5-7 Asahi-machi, Abeno-ku, Osaka, 545-8586, Japan. kakeya@med.osaka-cu.ac.jp.
Abstract
BACKGROUND: Good's syndrome (GS) is characterized by immunodeficiency, and can lead to severe infection, which is the most significant complication. Although Mycobacterium rarely causes infection in patients with GS, disseminated nontuberculous mycobacterial (NTM) infection frequently occurs in GS patients that are also positive for the human immunodeficiency virus (HIV) or anti-interferon (IFN)-γ autoantibodies. Here, we report a rare case of GS with NTM without HIV or IFN-γ autoantibodies. CASE PRESENTATION: A 57-year-old Japanese male with GS and myasthenia gravis (treated with prednisolone and tacrolimus) was diagnosed with disseminated NTM infection caused by Mycobacterium abscessus subsp. massiliense. He presented with fever and back pain. Blood, lumbar tissue, urine, stool, and sputum cultures tested positive for M. abscessus. Bacteremia, spondylitis, intestinal lumber abscess, and lung infection were confirmed by bacteriological examination and diagnostic imaging; urinary and intestinal tract infections were suspected by bacteriological examination but not confirmed by imaging. Despite multidrug combination therapy, including azithromycin, imipenem/cilastatin, levofloxacin, minocycline, linezolid, and sitafloxacin, the patient ultimately died of the infection. The patient tested negative for HIV and anti-IFN-γ autoantibodies. CONCLUSIONS: Since myasthenia gravis symptoms interfere with therapy, patients with GS and their physicians should carefully consider the antibacterial treatment options against disseminated NTM.
BACKGROUND:Good's syndrome (GS) is characterized by immunodeficiency, and can lead to severe infection, which is the most significant complication. Although Mycobacterium rarely causes infection in patients with GS, disseminated nontuberculous mycobacterial (NTM) infection frequently occurs in GSpatients that are also positive for the human immunodeficiency virus (HIV) or anti-interferon (IFN)-γ autoantibodies. Here, we report a rare case of GS with NTM without HIV or IFN-γ autoantibodies. CASE PRESENTATION: A 57-year-old Japanese male with GS and myasthenia gravis (treated with prednisolone and tacrolimus) was diagnosed with disseminated NTM infection caused by Mycobacterium abscessus subsp. massiliense. He presented with fever and back pain. Blood, lumbar tissue, urine, stool, and sputum cultures tested positive for M. abscessus. Bacteremia, spondylitis, intestinal lumber abscess, and lung infection were confirmed by bacteriological examination and diagnostic imaging; urinary and intestinal tract infections were suspected by bacteriological examination but not confirmed by imaging. Despite multidrug combination therapy, including azithromycin, imipenem/cilastatin, levofloxacin, minocycline, linezolid, and sitafloxacin, the patient ultimately died of the infection. The patient tested negative for HIV and anti-IFN-γ autoantibodies. CONCLUSIONS: Since myasthenia gravis symptoms interfere with therapy, patients with GS and their physicians should carefully consider the antibacterial treatment options against disseminated NTM.
Authors: Sophie Steiner; Tatjana Schwarz; Victor M Corman; Laura Gebert; Malte C Kleinschmidt; Alexandra Wald; Sven Gläser; Jan M Kruse; Daniel Zickler; Alexander Peric; Christian Meisel; Tim Meyer; Olga L Staudacher; Kirsten Wittke; Claudia Kedor; Sandra Bauer; Nabeel Al Besher; Ulrich Kalus; Axel Pruß; Christian Drosten; Hans-Dieter Volk; Carmen Scheibenbogen; Leif G Hanitsch Journal: Front Immunol Date: 2022-03-10 Impact factor: 7.561