WHO Collaborating Centre for Prevention and Treatment of Human Echinococcosis, University Hospital, University of Franche-Comté, Besançon 25030, France.
Ann Clin Microbiol Antimicrob. 2013 Jan 2;12:1. doi: 10.1186/1476-0711-12-1.
We report the 30-yr history of a well-documented human case of alveolar echinococcosis, with a lung lesion at presentation followed by the discovery of a liver lesion, both removed by surgery. Subsequently, within the 13 years following diagnosis, metastases were disclosed in eye, brain and skull, as well as additional lung lesions. This patient had no immune suppression, and did not have the genetic background known to predispose to severe alveolar echinococcosis; it may thus be hypothesized that iterative multi-organ involvement was mostly due to the poor adherence to benzimidazole treatment for the first decade after diagnosis. Conversely, after a new alveolar echinococcosis recurrence was found in the right lung in 1994, the patient accepted to take albendazole continuously at the right dosage. After serology became negative and a fluoro-deoxy-glucose-Positron Emission Tomography performed in 2005 showed a total regression of the lesions in all organs, albendazole treatment could be definitively withdrawn. In 2011, the fluoro-deoxy-glucose-Positron Emission Tomography showed a total absence of parasitic metabolic activity and the patient had no clinical symptoms related to alveolar echinococcosis.The history of this patient suggests that multi-organ involvement and alveolar echinococcosis recurrence over time may occur in non-immune suppressed patients despite an apparently "radical" surgery. Metastatic dissemination might be favored by a poor adherence to chemotherapy. Combined surgery and continuous administration of albendazole at high dosage may allow alveolar echinococcosis patients to survive more than 30 years after diagnosis despite multi-organ involvement.
我们报告了一例经过充分记录的人类细粒棘球蚴病肺泡病例的 30 年历史,该病例最初表现为肺部病变,随后发现肝脏病变,均通过手术切除。随后,在诊断后的 13 年内,眼部、脑部和颅骨以及其他肺部病变均出现了转移。该患者没有免疫抑制,也没有已知的易患严重细粒棘球蚴病的遗传背景;因此,可以假设反复发生多器官受累主要是由于在诊断后的第一个十年内未能坚持使用苯并咪唑治疗。相反,1994 年在右肺发现新的细粒棘球蚴病复发后,患者接受了连续服用阿苯达唑的治疗,剂量正确。在血清学转为阴性后,2005 年进行的氟脱氧葡萄糖正电子发射断层扫描显示所有器官的病变完全消退,阿苯达唑治疗可以被明确停用。2011 年,氟脱氧葡萄糖正电子发射断层扫描显示寄生虫代谢活性完全消失,患者没有与细粒棘球蚴病相关的临床症状。该患者的病史表明,尽管进行了明显的“根治性”手术,非免疫抑制患者仍可能出现多器官受累和细粒棘球蚴病复发。转移性播散可能是由于对化疗的依从性差所致。联合手术和持续高剂量服用阿苯达唑可能使细粒棘球蚴病患者在多器官受累后能够存活 30 年以上。