Coinfection of Pulmonary Hydatid Cyst and Aspergilloma: Case Report and Systematic Review

Hydatidosis is one of the most common zoonotic diseases between humans and animals. The disease has a worldwide distribution, but it is endemic in the Middle East, the Mediterranean, Southern Europe, East Africa, Latin America, Australia, and New Zealand (Table 1). Aspergilloma generally can develop in the lung cavity including preexisting cavities caused by tuberculosis, sarcoidosis and only a few cases have been reported in cavities as pulmonary hydatid cyst. Aspergilloma in hydatid cyst may occur after the onset surgery or many years later, while in our case the patient had concurrent Aspergillus and Echinococcus infections. E. granulosus is the most common agent in hydatid disease, and people can be infected through contaminated food or water with feces or even by direct contact with canines [26]. Depending on the size and location of the cysts, infection may remain asymptomatic for many years. The fungus could easily grow inside the cyst, because the immune system cells are not able to penetrate into the cavities. The most common symptom of aspergilloma is hemoptysis which can be caused by vascular invasion of fungal mycelium [28]. In literature review of 22 patients including the current case, 75 % had a symptom of hemoptysis. Pulmonary infection seemed to be more prevalent in younger individuals [29]. Seventy percent of our cases were male, and the mean age of all cases was 40.8 years (Table 1). Although aspergilloma occurs in both immunocompromised and immunocompetent patients [30, 31], all patients in the literature review were immunocompetent and had no structural deformities of the lungs. Development of coinfection of hydatid cyst and aspergilloma is extremely rare, as in a retrospective study, 100 archival cases with the diagnosis of hydatid cyst were reevaluated and ultimately only two cases were found to have aspergilloma and hydatid cyst coexistence [11]. One of the most common sites of hydatid cyst occurrence is the liver (50–70 %), and lungs (10–30 %) are the second common place of involvement [32]. In this study, eighty percent of cases had lung involvement in right lobe and 54 % showed involvement in the lower lobe. Bhatia et al. reported that about 60 % of pulmonary hydatidosis could affect the right lung and also 50–60 % involved the lower lobes [33]. Radiography methods are very effective in both hydatid cyst and aspergillosis. The air crescent sign is an important specific sign in diagnosis of aspergilloma. When patient changes his position, this mass usually moves within the cavity and the sign is called a “monod sign.” Most of the patients in this review had both of these signs (Table 1), but these findings are also found in pulmonary tuberculosis, hydatid cyst, pulmonary abscess, bronchogenic carcinoma, and pneumocystis pneumonia. The specificity and sensitivity of radiographic methods to identify the Aspergillus colonies trapped in a hydatid cyst are unclear [33]. Early diagnosis and treatment are vital to prevent probable complications stemming from infection by these two pathogens, such as massive hemoptysis or even invasive disease. A definitive diagnosis is based on pathologic evaluation or a positive culture, serological tests may be useful. The treatment choice of aspergilloma is surgery and lobectomy. In our study, 76 % of all cases preferred standard lobectomy procedures followed by 24 % carried out thoracotomy, pneumonectomy, and cystectomy as the surgical methods. Al-Kattan et al. [34] choose standard thoracotomy and lobectomy procedures; however, they also declared that wedge resection could be carried out if the lung tissue surrounding the lesion was healthy. Some authors considered that efficiency of antifungal therapy for aspergilloma is still doubtful [11, 35, 36]. In our case, we did not use any antifungal therapy, whereas in the literature review, 52 % of all cases were treated with azole agents such as itraconazole and 29 % had anti-helminthic therapy like albendazole. In a 28-year experience in Taiwan, Chen et al. indicated that 14 of 19 patients who had been unsuccessfully treated with antifungal agents for at least 10 days finally had surgery because of persistent symptoms [37]. Nevertheless, some authors suggest antifungal therapy during the 3 month post-operation, especially in immunocompromised patients who might be at risk of additional Aspergillus infection [11, 35, 38]. In conclusion, to the best of our knowledge, limited data regarding the co-occurrence of hydatidosis and aspergillosis are available and only sporadic case reports described appropriate management. We conclude that in endemic areas and in individuals with underlying diseases, hydatidosis must be considered as a possibility before labeling a cavity just as aspergilloma. Surgical treatment alone or in combination with antifungal therapy is a suitable method to prevent life-threatening complications like massive haemoptysis or even invasive forms of the disease.

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