PMC9420510_01
Clinical course of the patient.. With prednisolone (PSL) treatment, peripheral blood eosinophil count decreased. The clinical course remained stable as PSL was gradually tapered and discontinued after 1 year. After discontinuance on September 2021, recurrence of ABPA was observed and mepolizumab was started. After 3 months of mepolizumab treatment, ABPA recurred a second time, so mepolizumab was replaced with dupilumab. Subsequently, dramatic improvement in ABPA was observed. Bronchoscopy was performed 2 months after the original referral (May 2019: Bronchoscopy #1), 4 months after treatment with prednisolone (September 2019: Bronchoscopy #2), and at the recurrence of ABPA (September 2021: Bronchoscopy #3). Chest computed tomography (CT) was performed on referral (March 2019: CT #1), 2 months after referral (May 2019: CT #2), 2 months after treatment with prednisolone (July 2019: CT #3), at recurrence of ABPA before treatment with mepolozumab (September 2021: #4), 3 months after treatment with mepolizumab (November 2021: CT #5), and 3 months after treatment with dupilumab (February 2022: CT #6).
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Fig. 1 shows the patient's clinical course.
Chest CT imaging during the clinical course.. Non-enhanced chest CT revealed high attenuation of the mucoid impaction in the left-lingular lobe on referral (March 2019: CT #1) (a, b). Mucoid impaction in the left-lingular lobe has spread 2 months after referral (May 2019: CT #2) (c). Mucoid impaction in the left-lingular lobe has disappeared 2 months after treatment predonisolone (July 2019: CT #3) (d). Mucoid impaction newly occluded in the left-upper lobe on recurrence of ABPA (September 2021: CT #4) (e). The improvement of mucoid impaction in the left-upper lobe is poor and newly mucoid impactions can be seen in the right-upper lobe, right-lower lobe, and left-upper lobe 3 months after treatment with mepolizumab (November 2021: CT #5) (f, g,and h). All mucoid impactions have disappeared 3 months after treatment with dupilumab (Feburuary 2022: CT #6) (i, j, and k).
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Chest computed tomography (CT) demonstrated pulmonary infiltrates and left-lingular lung lobe bronchiectasis with high-attenuation mucoid impaction (Fig. 2a and b). Two months after the referral, pulmonary infiltrates had increased (Fig. 2c). Two months later, her respiratory symptoms and chest radiography findings showed improvement (Fig. 2d). Chest CT revealed new bronchial mucus impaction in the left-upper-lung lobe without recurrence in the left-lingular lobe (Fig. 2e). However, after 3 months on mepolizumab, a second recurrence of ABPA occurred with a remarkable increase in IgE (Fig. 2f, g, h) (Table 1). ABPA with low serum total IgE levels (1660 U/L) (Table 1). After 3 months of dupilumab treatment, her peripheral eosinophil count had decreased to 96/muL, and mucus impaction was no longer visible on chest CT (Fig. 2i, j, k).
Bronchoscopic and histologic imaging.. Bronchoscopy showing that the orifice of the left-lingular lobe bronchus is occluded by mucoid impaction accompanied by mucosal edema 2 months after referral (May 2019: Bronchoscopy #1) (a). The mucoid impaction has disappeared 4 months after treatment with predonisolone. (September 2019: Bronchoscopy #2) (b). Bronchoscopic findings showing mucoid impaction in the orifice of the left B3b accompanied by mucosal edema on recurrence (September 2021: Bronchoscopy #3) (c). Hematoxylin and eosin staining showing that the biopsies from the mucoid impaction contain abundant eosinophils and Charcot-Leyden crystals (arrowhead) (May 2019) (d). Filamentous fungal hyphae are noted on the Grocott-stained specimen (e).. Scale bar = 20 mum.
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Bronchoscopy revealed that the mucosa of the orifice of the left-lingular lobe bronchus was reddened and edematous, with a mucous plug (Fig. 3a). A biopsy specimen from the lesion was pathologically proven to be a mucoid impaction containing abundant eosinophils and Charcot-Leyden crystals (Fig. 3d). Filamentous fungi suggesting Aspergillus spp. were also observed (Fig. 3e). Four months later, bronchoscopy showed that the mucoid impaction had disappeared (Fig. 3b). Bronchoscopy revealed bronchial mucus plugs in B3b of the left-upper lobe (Fig. 3c).