PDF(Pubmed)
Abstract：
BACKGROUND: Lagenidium deciduum is an oomycete that can cause infections in mammals that present similarly to pythiosis and mucormycosis. Most of the existing case reports have occurred in canines and have been fatal. In animals, medical therapy has not been successful, so surgical excision is the mainstay of treatment. Lagenidium sp. infections in humans are rare. There is only one case of a human Lagenidium sp. infection in the literature, and it presented as an ocular infection. The human ocular infection was resistant to medical therapy and required a penetrating keratoplasty for cure. Additional reports of effective therapy are needed to guide management of this emerging pathogen. We present the first case of a cutaneous Lagenidium deciduum infection in a human patient, which is also the first documented case of a Lagenidium deciduum infection in an immunocompromised host of any species.
METHODS: An 18-year-old female with relapsed acute myeloid leukemia, awaiting a haploidentical stem cell transplant, presented with erythematous cutaneous lesions on her left hip and bilateral buttocks that enlarged and blackened over several days. About 1 week later, boil-like lesions appeared on her bilateral buttocks. The skin lesions were initially presumed to be bacterial in origin, so the patient was treated with clindamycin and cefepime with little improvement. Upon further investigation, fungal cultures and skin biopsies revealed aseptate hyphae, so the patient was switched to isavuconazole and amphotericin B due to concern for mucormycosis. Phenotypic characterization and DNA sequencing were performed by the Fungus Testing Laboratory, University of Texas Health Science Center at San Antonio, which identified the causal fungal organism as Lagenidium deciduum. All of her cutaneous lesions were surgically excised, and the patient was treated with micafungin, terbinafine, doxycycline, and azithromycin. Micafungin and terbinafine were continued until she achieved engraftment post-transplant.
CONCLUSIONS: We report the first successful treatment of a human Lagenidium infection in an immunocompromised host through a combination of aggressive surgical excision and prolonged antifungal therapy during the prolonged neutropenia associated with allogeneic stem cell transplant. Prompt diagnosis and management may prevent disseminated oomycosis.
METHODS: An 18-year-old female with relapsed acute myeloid leukemia, awaiting a haploidentical stem cell transplant, presented with erythematous cutaneous lesions on her left hip and bilateral buttocks that enlarged and blackened over several days. About 1 week later, boil-like lesions appeared on her bilateral buttocks. The skin lesions were initially presumed to be bacterial in origin, so the patient was treated with clindamycin and cefepime with little improvement. Upon further investigation, fungal cultures and skin biopsies revealed aseptate hyphae, so the patient was switched to isavuconazole and amphotericin B due to concern for mucormycosis. Phenotypic characterization and DNA sequencing were performed by the Fungus Testing Laboratory, University of Texas Health Science Center at San Antonio, which identified the causal fungal organism as Lagenidium deciduum. All of her cutaneous lesions were surgically excised, and the patient was treated with micafungin, terbinafine, doxycycline, and azithromycin. Micafungin and terbinafine were continued until she achieved engraftment post-transplant.
CONCLUSIONS: We report the first successful treatment of a human Lagenidium infection in an immunocompromised host through a combination of aggressive surgical excision and prolonged antifungal therapy during the prolonged neutropenia associated with allogeneic stem cell transplant. Prompt diagnosis and management may prevent disseminated oomycosis.
摘要：
背景：Lagenidiumdeduum是一种卵菌，可在哺乳动物中引起类似于化脓症和毛霉菌病的感染。现有的大多数病例报告都发生在犬科动物中，并且是致命的。在动物中,药物治疗没有成功，所以手术切除是治疗的主要手段。Lagenidiumsp.人类感染很少见。只有一例人类Lagenidiumsp。文献中的感染，它表现为眼部感染。人类眼部感染对药物治疗有抵抗力，需要进行穿透性角膜移植术才能治愈。需要其他有效治疗的报告来指导这种新兴病原体的管理。我们介绍了人类患者的第一例皮肤Lagenium蜕膜感染，这也是在任何物种的免疫受损宿主中记录的首例Lagenidium蜕膜感染病例。
方法：一名18岁女性复发性急性髓系白血病，等待单倍体干细胞移植，她的左臀部和双侧臀部出现红斑皮肤病变，几天后增大并变黑。大约一周后，她的双侧臀部出现沸腾样病变。皮肤损伤最初被认为是细菌起源，因此，患者接受克林霉素和头孢吡肟治疗，改善不大。经进一步调查,真菌培养和皮肤活组织检查显示出无菌菌丝，因此，由于担心毛霉菌病，患者改用伊沙康康唑和两性霉素B。表型表征和DNA测序由真菌测试实验室进行，德克萨斯大学圣安东尼奥分校健康科学中心，将致病真菌生物鉴定为Lageniumdeduum。她所有的皮肤损伤都是手术切除的,病人接受了米卡芬净治疗,特比萘芬,多西环素,和阿奇霉素.继续使用米卡芬净和特比萘芬，直到她在移植后实现植入。
结论：我们报告了在与异基因干细胞移植相关的长期中性粒细胞减少期间，通过积极的手术切除和长期的抗真菌治疗相结合，首次成功治疗了免疫功能低下的宿主中的人Lagenium感染。及时的诊断和治疗可以预防播散性真菌病。
方法：一名18岁女性复发性急性髓系白血病，等待单倍体干细胞移植，她的左臀部和双侧臀部出现红斑皮肤病变，几天后增大并变黑。大约一周后，她的双侧臀部出现沸腾样病变。皮肤损伤最初被认为是细菌起源，因此，患者接受克林霉素和头孢吡肟治疗，改善不大。经进一步调查,真菌培养和皮肤活组织检查显示出无菌菌丝，因此，由于担心毛霉菌病，患者改用伊沙康康唑和两性霉素B。表型表征和DNA测序由真菌测试实验室进行，德克萨斯大学圣安东尼奥分校健康科学中心，将致病真菌生物鉴定为Lageniumdeduum。她所有的皮肤损伤都是手术切除的,病人接受了米卡芬净治疗,特比萘芬,多西环素,和阿奇霉素.继续使用米卡芬净和特比萘芬，直到她在移植后实现植入。
结论：我们报告了在与异基因干细胞移植相关的长期中性粒细胞减少期间，通过积极的手术切除和长期的抗真菌治疗相结合，首次成功治疗了免疫功能低下的宿主中的人Lagenium感染。及时的诊断和治疗可以预防播散性真菌病。
