Chronic pulmonary aspergillosis complicating pulmonary cystic echinococcosis: a global systematic review.

Background: Human cystic echinococcosis frequently involves the lungs, and colonization of a residual cavity with Aspergillus spores can lead to chronic pulmonary aspergillosis (CPA). Objectives: We pooled and characterized cases of CPA co-existing with pulmonary cystic echinococcosis (PCE) through a systematic review of globally published data. Design: Systematic review. Data sources and methods: Primary literature was identified through searches of various electronic databases, including CINAHL, Ovid MEDLINE, MEDLINE (PubMed), EMBASE, Google Scholar, the Cochrane Database of Systematic Reviews, and African Journal Online. The search encompassed articles from inception to February 2024, using medical subject heading search terms "chronic pulmonary aspergillosis" OR "Aspergillus" OR "aspergilloma" OR "aspergillosis" AND "hydatid disease" OR "hydatidosis" OR "echinococcosis" OR "Echinococcus." Two reviewers independently assessed titles, abstracts, and full texts for eligibility using the Rayyan web-based software. The eligible studies comprised original observational research, single case reports, and case series that reported on CPA in patients with PCE based on established criteria (microbiological and/or radiological diagnosis of PCE) without language or geographic restrictions. Cochrane Risk of Bias Tools (ROB2 and ROBINS-I) were used to assess study quality and risk of bias, and the quality of the evidence was rated using the Grading of Recommendations, Assessment, Development, and Evaluations tool. The extracted data were summarized using descriptive statistics. Results: A total of 148 studies were initially identified from a literature search, and after the screening, 52 articles were selected for full-text review. We included 41 studies published from 1996 to 2023 that met the eligibility criteria and involved 3035 participants. The studies comprised case reports (75.6%, n = 31), case series (12.2%, n = 5), retrospective cohort studies (7.3%, n = 3), and one (2.4%) each of prospective cohort and cross-sectional study, respectively. More than two-thirds were conducted in India (36.6%, n = 15), followed by Turkey (14.6%, n = 6) and Iran (12.2%, n = 5). Among the 72 CPA cases, 50% were male, and 38.9% were female, with a median age of 38 years. Common comorbidities included pulmonary tuberculosis (n = 5) and diabetes mellitus (n = 3). The most frequent symptoms were cough (n = 32), hemoptysis (n = 21), and fever (n = 16). Radiological examinations were conducted in 74.4% of hydatid disease cases, while histological examinations were performed in 82.1% of aspergillosis cases. Cavitary lesions were noted in 21.4% of radiological findings. Treatment involved both surgical and medical management for 43.1% of patients, with antifungals (50%) and anthelminthic drugs (50%) commonly used. Surgical treatments included cystectomy (57.9%) and lobectomy (55.3%). All patients with reported outcomes were cured. Conclusion: CPA complicates PCE and has both similar clinical and radiological features. Diagnostic approaches primarily include radiology and histology, while treatment often involves a combination of surgical and medical management, with antifungals and anthelminthic drugs commonly used. Despite the complexity of CPA, patients generally achieve favorable outcomes with appropriate treatment. Further research is needed to optimize diagnostic and therapeutic strategies for this condition. Trial registration: PROSPERO (CRD42024510441).