PMC10185866_01
(a) Ultrasonographic illustrations of the focal thickening of the proximal duodenum
(sagittal plane view) and (b) ultrasonographic picture of the lesion (transversal plane
view). White arrow indicates the lumen of duodenum, and the crosses indicate focal
thickening of the duodenal wall with loss of layering
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ALT (207 IU/l; RI 15-123) and slight hypercreatininaemia (creatinine 159 micromol/l; RI 46-157). Abdominal ultrasound revealed severe focal thickening (up to 5.8 mm) of the proximal duodenum, displaying a loss of intes-tinal layering (Figure 1), in association with discrete pancreaticoduodenal lymphadenopathy (thickness 3.4 mm).
(a,b) Gastroduodenoscopy showed an abnormal and proliferative aspect of the proximal
duodenal mucosa associated with ulcerated areas exhibiting a yellow-green discoloration.
(c) Colonoscopy revealed a diffuse oedematous mucosa and many circumscribed
white-to-grey areas embedded in the mucosa
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The proximal duodenal mucosa showed a proliferative and marked lesion associated with multiple ulcerative areas, exhibiting yellow-green discoloration (Figure 2a,b). Colonoscopy revealed diffuse oedematous mucosa in the distal colon and numerous white-to-grey circumscribed regions embedded in the mucosa (Figure 2c).
(a) Histopathology showed the infiltration of eosinophils (arrowheads) into duodenal
tissue with intralesional fungi (arrows) and branching and anastomosing trabecular
pattern of dense sclerotic collagen (asterisk; haematoxylin-eosin-saffron). (b)
Confirmation of the presence of fungi in duodenal lesions (arrows; periodic
acid-Schiff, x 400)
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Histological examination of the duodenal tissue revealed branching and anastomosing trabeculae of dense sclerotic collagen fibres, interspersed with moderately pleomorphic fibroblasts and a mixed population of inflammatory cells predominantly composed of eosinophils (Figure 3a). Periodic acid-Schiff (PAS) and Gomori-Grocott stains confirmed the presence of fungal filaments in the FGESF lesions (Figure 3b). Some lesions were ulcerative and spilled over the mucosal surface (Figure 3a,b).
(a) Gastroduodenoscopy before immunomodulatory treatments (T0) and (b)
gastroduodenoscopy after 71 days of immunomodulatory treatments (T1: T0 + 71 days). A
marked improvement of the lesions is identified, but yellow-green discoloration areas
are persistent
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Gastroduodenoscopy was performed 71 days after the FGESF diagnosis, showing marked improvement in the lesions (Figure 4).
Rhizopus microsporus identified in duodenal endoscopic biopsies: (a)
macroscopic aspect of the colonies; (b) binocular image of sporangiophores, rhizoids and
pigmented filaments (x 10 magnification); (c) germinating sporangium and columella (x
100 magnification); and (d) growing stolons (x 100 magnification)
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Microflex LT/HS (MBT SMART; Bruker Daltonics) and matched against a mass spectrophotometry identification online database application.   Fungal culture yielded a fast-growing mould with woolly colonies that were initially white and became grey-to-brown later. Microscopically, broad aseptate hyphae produced rhizoids and sporangiophores, with dark sporangia and globose columellae (Figure 5).
(a) Gastroduodenoscopy before immunomodulatory treatment (T0), (b) gastroduodenoscopy
after immunomodulatory treatment and before antifungal treatment (T1: T0 + 71 days) and
(c) gastroduodenoscopy after antifungal treatment and ciclosporin withdrawal (T2:
T0 + 159 days)
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Treatment compliance was only possible for 18 days; nevertheless, 2 months after ciclosporin withdrawal, endoscopic examination showed a relapse of the lesions (Figure 6).