与极低剂量肾上腺皮质激素治疗相关的机会性感染:两例军团菌和卡氏肺孢子菌肺炎病例

Azusa Ikeda , Megumi Tsuji , Hiroyuki Nagafuchi , Yukiko Kuroda , Kenji Kurosawa , Tomohide Goto
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引用次数: 0

摘要

背景促肾上腺皮质激素(ACTH)疗法是治疗婴儿痉挛症的一线疗法,众所周知,该疗法会按剂量增加感染性疾病的发病率。极低剂量的促肾上腺皮质激素疗法被认为是安全的,目前还没有与该疗法相关的机会性感染或其他严重感染的报道。在此,我们报告了两例与极低剂量促肾上腺皮质激素治疗相关的军团菌肺炎和卡氏肺孢子菌肺炎病例。病例 1 患有 PIGA 变异型,在接受极低剂量促肾上腺皮质激素治疗 6 个月后,于治疗开始 2 周后出现肺炎;军团菌肺炎在发病 14 天后确诊。虽然给予了米诺环素、环丙沙星、阿奇霉素和利福平治疗,但肺炎发展严重,需要长期重症监护,导致慢性呼吸衰竭。患者2患有KLHL20变异型,在3个月大时接受了为期4周的超低剂量ACTH治疗,治疗结束1周后出现卡氏肺孢子菌肺炎,经三甲双胍-磺胺甲噁唑治疗后痊愈。
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Opportunistic infections associated with extremely low-dose adrenocorticotropic hormone therapy: Two cases of Legionella and Pneumocystis carinii pneumonia

Background

Adrenocorticotropic hormone (ACTH) therapy, the first-line treatment for infantile spasms, is known to dose-dependently increase incidences of infectious diseases. Extremely low-dose ACTH therapy is considered safe, and there have been no reports of opportunistic or other serious infections associated with this therapy. Herein, we report two cases of Legionella and Pneumocystis carinii pneumonia associated with extremely low-dose ACTH therapy.

Case presentation

Patient 1 with PIGA variant was administered extremely low-dose ACTH therapy at 6 months, developing pneumonia 2 weeks after therapy initiation; Legionella pneumonia diagnosis was confirmed 14 days after onset. Although minocycline, ciprofloxacin, azithromycin, and rifampicin were administered, pneumonia progressed critically and required prolonged intensive care, resulting in chronic respiratory failure. Patient 2 with KLHL20 variant was administered extremely low-dose ACTH therapy for 4 weeks at the age of 3 months, developing Pneumocystis carinii pneumonia 1 week after completion of therapy, which recovered with trimethoprim-sulfamethoxazole therapy.

Conclusion

Extremely low-dose and short-term ACTH therapy can be associated with opportunistic infections, and early diagnosis and treatment are crucial, because delays in administering appropriate antibiotics can lead to severe complications.
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