Background: Serum periostin levels have been reported to be an indicator of Th2 inflammation in asthmatic patients. Objective: This study aimed to investigate serum levels of periostin in patients with allergic bronchopulmonary aspergillosis (ABPA) and to evaluate its diagnostic and monitoring value in the disease. Methods: Patients with ABPA (n = 19) and asthma (n = 24), including severe asthma with fungal sensitisation (SAFS, n = 11) and severe asthma without fungal sensitization (SAwFS, n = 13), were enrolled. Serum levels of periostin were analysed by enzyme-linked immunosorbent assay. Serum total IgE and Aspergillus fumigatus specific IgE, IgG were measured by ImmunoCAP. Levels of cytokines (IFN-γ, IL-4, IL-5, IL-8, IL-10, IL-13 and IL-17A) were measured by Meso Scale Discovery (MSD). Results: Serum levels of periostin in ABPA patients (85.55 ng/mL, [68.28-166] ng/mL) were higher than those in SAFS (50.99 ng/mL, [32.02-71.80] ng/mL; P < 0.01). Among the analysed cytokines, IL-5 levels in ABPA (1.55 pg/mL, [0.96-3.33] pg/mL) were higher than those in SAFS (0.31 pg/mL, [0.26-0.56] pg/mL; P < 0.05) or SAwFS (0.34 pg/mL, [0.21-0.56] pg/mL; P < 0.01). Serum periostin levels was positively associated with total IgE levels (r = 0.319, P < 0.05), serum IL-5 levels (r = 0.484, P < 0.01) and blood eosinophil counts (r = 0.428, P < 0.05). In ROC analysis, the clinical reference value of periostin was 68.8 ng/mL for differential diagnosis of ABPA and SAFS, with the area under the curve (AUC) of 0.81. Longitudinally, serum periostin levels did not change significantly after treatment in ABPA. Conclusions: These findings suggested that serum levels of periostin were up-regulated in ABPA patients, which may be associated with eosinophilic inflammation.