Case reportSuccessful control of exacerbation of Allergic Bronchopulmonary Aspergillosis due to Aspergillus terreus in a cystic fibrosis patient with short-term adjunctive therapy with voriconazole: A case report
Introduction
Allergic bronchopulmonary aspergillosis (ABPA) is a hypersensitivity lung disorder characterized by airway inflammation, elevated levels of Aspergillus specific IgE and IgG, elevated total IgE and positive skin sensitivity to Aspergillusallergens/proteins [1], [2]. Aspergillus fumigatus was reported as the most common etiologic agent of ABPA, however other species of Aspergillusare rarely implicated [3]. ABPA typically occurs in patients with asthma or cystic fibrosis (CF) with a 2–15% rate in CF patients [4], [5]. The global burden of ABPA in CF patients is estimated at about 7000 cases annually [6]. Glucocorticoids are the mainstay of management of ABPA; however, the use of antifungals (usually itraconazole) in patients with slow or poor response to steroids, frequent relapses, steroid-dependent ABPA, or steroid toxicity has been recommended by a consensus expert panel [5].
Here, we report an unusual case of ABPA exacerbation due to two different non-fumigatus Aspergillus species that was successfully controlled by short-term course of voriconazole therapy after a failed itraconazole treatment.
Section snippets
Case report
In this report, a 12-year-old boy with ABPA was presented. The CF was diagnosed at age 1.5 years old by positive sweet chloride concentration, homozygous genotype for CFTR mutation (p.F508del/p.F508del) and pancreatic insufficiency. Since childhood, he showed symptoms of moderate lung problems, severe cough, chest tightness and antibiotic therapy (3–4 times per year). At the age of 6 years, he showed a respiratory colonization with Pseudomonas aeruginosa and Aspergillus flavus. ABPA was
Discussion
This report showed a successful control of ABPA exacerbation with short-term VOR plus corticosteroids therapy in a CF patient with previously failed ITC therapy. A. fumigatus, A. niger, and A. terreus are commonly implicated species in ABPA. However, A. ochraceus is a rare cause of ABPA. The first case report of ABPA due to A. ochraceus was presented by Novey and Wells in 1978 [9]. Nakahara et al. [10] reported an ABPA cases without CF caused by A. terreus with extensive collapse of the left
Conclusion
This case presentation showed that other species of Aspergillusthan A. fumigatus can be involved in ABPA. It could be recommended VOR as an alternative adjunctive therapy along with corticosteroid for ABPA patients who failed or may not tolerate ITC. The antifungal susceptibility test is helpful in choosing an alternative medicine, in patients who failed to respond to initial therapy.
Ethical statement
Informed consent was obtained from all individual participants included in the study.
Authors’ contribution
Conceptualisation and design of the study: Dr. Vida Mortezaee, Dr. Maryam Hassanzad.
Sample collection: Dr. Vida Mortezaee, Maedeh Maleki.
Acquisition of data: Dr. Vida Mortezaee, Dr. Somayeh Sharifynia, Dr. Mihan Poorabdollah.
Analysis and interpretation of data: Dr. Felix Bongomin, Prof. Mohammad Taghi Hedayati, Dr. Vida Mortezaee.
Drafting of manuscript: Dr. Vida Mortezaee, Maryam Hassanzad.
Revising it critically for important intellectual content: Prof Ali Akbar Velayati, Prof. Mohammad Taghi
Patient consent
Obtained.
Ethics committee code
IR.MAZUMS.REC.95.235.
Disclosure of interest
The authors declare that they have no competing interest.
Acknowledgements
This research was supported by Invasive Fungi Research Center (IFRC) (fund number: 2060), Mazandaran University of Medical Sciences, Sari, Iran. We thank of Milad Medical Laboratory, Isfahan, Iran, and our colleagues from Paediatric Respiratory Diseases Research Center, National Research Institute of Tuberculosis and Lung Diseases (NRITLD), Shahid Beheshti University of Medical Sciences, Tehran, Iran, who provided insight and expertise that greatly assisted the research.
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