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, 2019, 2530487
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A Case of Ileocecal IgG4-Related Sclerosing Mesenteritis Diagnosed by Endoscopic Ultrasound-Guided Fine Needle Aspiration Using Forward-Viewing Linear Echoendoscope

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Case Reports

A Case of Ileocecal IgG4-Related Sclerosing Mesenteritis Diagnosed by Endoscopic Ultrasound-Guided Fine Needle Aspiration Using Forward-Viewing Linear Echoendoscope

Yuichi Takano et al. Case Rep Gastrointest Med.

Abstract

A 25-year-old woman had undergone removal of a cryptogenic tumor in the left maxillary sinus 1 year prior to presentation. The patient experienced abdominal pain for 4 days with repeated vomiting episodes; therefore, she was transferred to our hospital by an ambulance. Contrast-enhanced computed tomography revealed a 3-cm tumor in the ileocecal region, which caused small bowel obstruction. Contrast imaging of the ileus tube showed extrinsic compression of the ileocecal region. Forward-viewing linear echoendoscope revealed an irregular hypoechoic tumor measuring 3 cm outside the gastrointestinal tract. Using a 25G needle, endoscopic ultrasound-guided fine needle aspiration (EUS-FNA) was performed. Pathological finding was an inflammatory fibrous tissue with diffuse lymphoplasmacytic infiltration, with more than 10 IgG4-positive cells detected in a high-power field. Re-examination of a pathology specimen of the maxillary sinus tumor provided by the previous attending physician revealed that the inflammatory tissue had diffuse lymphoplasmacytic infiltration, which were accompanied by storiform fibrosis and obliterative phlebitis. Immunostaining revealed more than 50 IgG4-positive cells in a high-power field, a finding suggestive of IgG4-related disease. The serum IgG4 level was 21 mg/dl, which was within the normal range. Treatment was initiated with prednisolone at a dose of 50 mg/day, and the dose was later tapered off. CT and MRI performed 2 months later showed complete disappearance of the ileocecal tumor. The final diagnosis was asynchronously occurring IgG4-related maxillary sinusitis and sclerosing mesenteritis.

Conflict of interest statement

The authors declare that they have no conflicts of interest.

Figures

Figure 1
Figure 1
(a), (b) CECT showing a 3-cm hypervascular tumor (arrow) in the ileocecal region accompanied by small bowel obstruction.
Figure 2
Figure 2
(a) MRI T2-weighted images showing mild high intensity lesions in the ileocecal region (arrow). (b) MRI diffusion-weighted images showing decreased diffusion in lesions (arrow).
Figure 3
Figure 3
Contrast imaging of the ileus tube showing extrinsic compression of the ileocecal region (arrow). TI: terminal ileum, C: cecum, A: Ascending colon.
Figure 4
Figure 4
(a) Endoscopy of the lower gastrointestinal tract showing no abnormalities in the mucous membrane of the cecum and ileum. (b) Extramural compression was detected in the terminal ileum (arrow).
Figure 5
Figure 5
(a) Forward-viewing linear echoendoscope, TGF-UC180J (Olympus Medical Systems Corp, Tokyo, Japan). (b) EUS revealed an irregular hypoechoic tumor measuring 3 cm outside the gastrointestinal tract.
Figure 6
Figure 6
(a) A specimen obtained by EUS-FNA. Pathologically, the lesion was found to be composed of inflammatory fibrous tissues with lymphoplasmacytic infiltration (Hematoxylin-Eosin Stain, x400). (b) IgG4 staining revealed more than 10 IgG4-positive cells in an HPF (IgG4 Stain x400).
Figure 7
Figure 7
(a) Contrast-enhanced MRI showing an irregular tumor in the left maxillary sinus (arrow). (b) Pathologically, diffuse lymphoplasmacytic infiltration and storiform fibrosis are observed (Hematoxylin-Eosin Stain, x200). (c) More than 50 IgG4-positive cells are observed in an HPF (IgG4 Stain, x400).
Figure 8
Figure 8
(a) CECT showing complete disappearance of the tumor at 2 months after steroid treatment (arrow). (b) MRI diffusion-weighted images indicate the absence of any lesion (arrow).

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