Primary cerebral cystic echinococcosis in a child from Roman countryside: Source attribution and scoping review of cases from the literature.

IF 3.8 2区医学 Q1 Medicine

PLoS Neglected Tropical Diseases Pub Date : 2023-09-05 eCollection Date: 2023-09-01 DOI:10.1371/journal.pntd.0011612

Adriano Casulli, Stefania Pane, Franco Randi, Paola Scaramozzino, Andrea Carvelli, Carlo Efisio Marras, Andrea Carai, Azzurra Santoro, Federica Santolamazza, Francesca Tamarozzi, Lorenza Putignani

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Abstract

Background: Human cystic echinococcosis (CE) is a zoonotic parasitic infection caused by the larval stage of the species belonging to the Echinococcus granulosus sensu lato (s.l.) complex. Parasitic cysts causing human CE are mainly localized in the liver and in the lungs. In a smaller number of cases, larvae may establish in any organ or tissue, including the central nervous system (CNS). Cerebral CE (CCE) is rare but poses serious clinical challenges.

Methods: This study presents a case of CCE in a child living in the countryside near Rome (Italy), along with a comparative molecular analysis of the isolated cyst specimens from the patient and sheep of local farms. We also systematically searched the literature to summarize the most relevant epidemiological and clinical aspects of this uncommon localization.

Findings: The comparative molecular analysis confirmed that the infection was caused by E. granulosus sensu stricto (s.s.) (G3 genotype), and most likely acquired in the family farm. The literature search identified 2,238 cases of CCE. In 80.51% of cases, brain was the only localization and single CCE cysts were present in 84.07% of cases. Mean patients' age was 20 years and 70.46% were children. Cyst rupture was reported in 12.96% and recurrence of CCE after treatment in 9.61% of cases. Permanent disability was reported in 7.86% of cases, while death occurred in 6.21%. In case series reporting all CE localization, CCE represented 1.5% of all CE cases. In the few reports that identified at molecular level the CCE cyst, E. granulosus s.s. was found in 40% and E. canadensis in 60% of cases.

Conclusions: We report a rare case of CCE and evidenced the probable local origin of infection. The proportions of CE cases with uncommon localizations and with high impact on patients' lives have been globally neglected and should be included in the computation of the global burden of CE.

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罗马农村一名儿童的原发性脑囊性棘球蚴病：文献中病例的来源、归因和范围回顾。

背景：人类囊性棘球蚴病（CE）是一种人畜共患寄生虫感染，由细粒棘球蚴（s.l.）复合体的幼虫期引起。引起人类CE的寄生虫囊肿主要局限于肝脏和肺部。在少数情况下，幼虫可能在任何器官或组织中建立，包括中枢神经系统（CNS）。脑CE（CCE）是罕见的，但带来了严重的临床挑战。方法：本研究报告了一例生活在罗马（意大利）附近农村的儿童CCE病例，并对患者和当地农场绵羊的分离囊肿标本进行了比较分子分析。我们还系统地检索了文献，总结了这种罕见定位最相关的流行病学和临床方面。研究结果：比较分子分析证实，感染是由细颗粒E.granularus senso stricto（s.s.）（G3基因型）引起的，很可能是在家庭农场获得的。文献检索发现2238例CCE。在80.51%的病例中，大脑是唯一的定位，84.07%的病例中存在单个CCE囊肿。平均年龄为20岁，70.46%为儿童。12.96%的病例报告囊肿破裂，9.61%的病例报告CCE治疗后复发。7.86%的病例报告永久性残疾，6.21%的病例报告死亡。在报告所有CE定位的病例系列中，CCE占所有CE病例的1.5%。在为数不多的在分子水平上鉴定CCE囊肿的报告中，40%的病例中发现颗粒E.granularus s.s.，60%的病例中出现加拿大E.canadensis。结论：我们报告了一例罕见的CCE病例，并证明了感染的可能局部起源。具有罕见定位和对患者生活有重大影响的CE病例的比例在全球范围内被忽视，应纳入CE全球负担的计算中。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

PLoS Neglected Tropical Diseases Medicine-Infectious Diseases

CiteScore

7.40

自引率

10.50%

发文量

723

审稿时长

2-3 weeks

期刊介绍： PLOS Neglected Tropical Diseases publishes research devoted to the pathology, epidemiology, prevention, treatment and control of the neglected tropical diseases (NTDs), as well as relevant public policy. The NTDs are defined as a group of poverty-promoting chronic infectious diseases, which primarily occur in rural areas and poor urban areas of low-income and middle-income countries. Their impact on child health and development, pregnancy, and worker productivity, as well as their stigmatizing features limit economic stability. All aspects of these diseases are considered, including: Pathogenesis Clinical features Pharmacology and treatment Diagnosis Epidemiology Vector biology Vaccinology and prevention Demographic, ecological and social determinants Public health and policy aspects (including cost-effectiveness analyses).