TY - JOUR
AU - Safavi,, Moeinadin
AB - Abstract Co-infection of cystic echinococcosis and aspergillosis is a rare finding. The patient was a 10-year-old boy complaining of chest pain and malaise with a cyst in the right lung who underwent surgical resection of the cyst. Medical therapy with albendazole was started for the patient with the initial clinical impression of cystic echinococcosis. Later, the microscopic evaluation revealed a laminated layer of cystic echinococcosis involved by acute angle branching hyaline hyphae with septate. Thus, the diagnosis of a cystic echinococcosis and aspergillosis co-infection was established. Surgery is the mainstay of treatment along with anthelminthic and anti-fungal therapy, especially in immunocompromised patients. cystic echinococcosis, aspergillosis, co-infection INTRODUCTION Aspergillus infection happens in immunocompromised patients while the cystic echinococcosis or hydatid cyst caused by the larval stage of Echinococcus is highly endemic in western China, Central Asia, Mediterranean countries, eastern Africa and South America [1–4]. The infestation of cystic echinococcosis (hydatid cyst) with Aspergillus is extremely rare. There are only a few case reports in the literature mostly occurring in adults with ages ranging from 20 to 60 years [5–10]. To the best of the authors’ knowledge, this is the first case of pulmonary cystic echinococcosis and aspergillosis co-infection in a child. A written consent was also obtained from the patient’s guardian before preparing the article. CASE PRESENTATION The patient was a 10-year-old boy from Alborz province in Central Iran. He presented with chest pain, low grade fever, non-productive cough and malaise for three months, not responding to antibiotics. He had no history of immunodeficiency like recurrent infections. Chest X-ray (CXR) showed a complicated cyst in the lower lobe of right lung containing air. The liver had no cystic lesion (Fig. 1). Chest computed tomography (CT) scan also revealed a complicated, ruptured cyst containing air (Fig. 2). Laboratory tests revealed mild leukocytosis (11 750/ μl) with neutrophil dominance in differential count (neutrophils = 68%, monocyte = 5%, eosinophil = 2% and lymphocyte = 25%). The patient underwent surgery with a clinical impression of cystic echinococcosis. The cyst had adhesion to inner thorax wall and diaphragm. The adhesions were dissected, total cystectomy was done and the bronchi which had communication with pericyst space were sutured. Finally, capitonnage was performed (for air leak prevention from residual bronchial openings). Albendazole was started for him with 15 mg/kg/day in two divided doses for 4 weeks. The liver function tests were monitored twice every 2 weeks which were in normal limits. Fig. 1. Open in new tabDownload slide CXR revealed a complicated cyst containing air in the lower lobe of right lung. Fig. 1. Open in new tabDownload slide CXR revealed a complicated cyst containing air in the lower lobe of right lung. Fig. 2. Open in new tabDownload slide CT scan without contrast exhibited a complicated and ruptured cyst with an irregular wall containing air. Fig. 2. Open in new tabDownload slide CT scan without contrast exhibited a complicated and ruptured cyst with an irregular wall containing air. Subsequently, a previously opened cyst with a shiny milky white color was received to the pathology laboratory measuring 15 cm × 10 cm in area and 0.2 cm in thickness. A part of fibrotic tissue measuring 5 cm × 5 cm × 1 cm was also received separately in another container designated as peripheral tissue of cyst. The microscopic examination of the cyst wall showed an acellular laminated eosinophilic membrane and an outer fibrous pericyst. No protoscolices were seen (inactive lesion). Surprisingly, the pericyst also showed the presence of numerous septate hyphae with acute angle branching, conforming to the morphology of Aspergillus surrounded by mixed inflammatory cells (Fig. 3). Cut section of the separate tissue also exhibited a fibrous wall containing necrotic material and dichotomous branching septate fungal hyphae admixed with neutrophilic infiltration. No evidence of invasion was identified and findings were more in favor of aspergilloma. Thus, a diagnosis of dual infection by Echinococcus and Aspergillus was established. The patient’s symptoms were resolved after surgery and he was well during a 6-month follow-up. Fig. 3. Open in new tabDownload slide Laminated layers of cystic echinococcosis (white arrow) containing no protoscolices infiltrated by Aspergillus hyphae in the outer aspect toward the pericyst layer (black arrow) (hematoxylin and eosin stain, magnification ×200). Fig. 3. Open in new tabDownload slide Laminated layers of cystic echinococcosis (white arrow) containing no protoscolices infiltrated by Aspergillus hyphae in the outer aspect toward the pericyst layer (black arrow) (hematoxylin and eosin stain, magnification ×200). DISCUSSION Echinococcosis is one of the most prevalent zoonotic diseases. Its distribution is worldwide and is especially endemic in western China, Central Asia, Mediterranean countries, the Middle East, southern Europe, East Africa and South America. According to a systematic review, the overall prevalence of human echinococcosis was 4.2% in Iran (5.8% in southern Iran, 4.4% in northern Iran and 2.2% in central Iran). The prevalence was twice higher in rural areas than urban regions. Aspergillosis can occur in the background of pre-existing lung cavities due to underlying tuberculosis, sarcoidosis and cystic echinococcosis. Cystic echinococcosis is commonly caused by Echinococcus granulosus which might be asymptomatic for many years depending on its size and location. Hemoptysis is the most common symptom of aspergillosis due to angioinvasion or fungal mycelium. Aspergillosis superinfection on cystic echinococcosis may rarely happen and its incidence is higher in pulmonary cystic echinococcosis as a saprophytic fungal infection [2, 11–14]. Although radiologic modalities are highly diagnostic in cystic echinococcosis and aspergillosis, their findings are not accurate for the detection of their co-infection. Most of these cases have been reported as infected or complicated cystic echinococcosis in CT scans and MRI [15, 16]. Serologic tests like hydatid cyst IgM/IgG and Aspergillus galactomannan antigen have a diagnostic utility in echinococcosis and invasive aspergillosis, respectively (not performed for this patient). Cytological examination of cyst contents might help the diagnosis of cystic echinococcosis co-infection with Aspergillus species if there is no contraindication for the procedure. Although light microscopic examination of the cystic echinococcosis has an expectable result, it may show unexpected additional findings like coexistence of Aspergillus. Therefore proper sampling and meticulous evaluation are necessary [10]. Surgical treatment is the main treatment for cystic echinococcosis and aspergillosis co-infection followed by anthelminthic and anti-fungal drugs especially in immunocompromised patients [4]. REFERENCES 1 Soubani AO , Chandrasekar PH. The clinical spectrum of pulmonary aspergillosis . Chest 2002 ; 121 : 1988 – 99 . 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Google Scholar Crossref Search ADS PubMed WorldCat © The Author(s) [2020]. Published by Oxford University Press. All rights reserved. For permissions, please email: journals.permissions@oup.com This article is published and distributed under the terms of the Oxford University Press, Standard Journals Publication Model (https://academic.oup.com/journals/pages/open_access/funder_policies/chorus/standard_publication_model)
TI - Concomitant Pulmonary Cystic Echinococcosis and Aspergillosis in a Male Child
JF - Journal of Tropical Pediatrics
DO - 10.1093/tropej/fmaa020
DA - 2020-12-01
UR - https://www.deepdyve.com/lp/oxford-university-press/concomitant-pulmonary-cystic-echinococcosis-and-aspergillosis-in-a-ef3NX0TH1X
SP - 645
EP - 647
VL - 66
IS - 6
DP - DeepDyve
ER -