PMC7864275_01
ultrasound of the lacrimal glands before treatment: the left (A) and right (B) lacrimal glands were enlarged (left: 30 x 14 mm and right: 26 x 14 mm) before treatment, with multiple hypoechoic areas; C, D) both glands displayed hypervascularity
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F1
ultrasound of the lacrimal glands after treatment: left (A) and right (B) lacrimal glands decreased in size (left: 9 x 4 mm and right: 8 x 5 mm), and the vascularity and hypoechoic areas disappeared in response to corticosteroid therapy
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F2
Figure 2, Figure 3). Facial magnetic resonance imaging (MRI) demonstrated the diffuse enlargement of the lacrimal and submandibular glands, without evidence of focal lesion or nodularity.
ultrasound of the submandibular glands: A, B) the submandibular glands before treatment showed bilateral enlargement with multiple hypoechoic, hypervascular areas; C) the submandibular glands after treatment showed a decrease in the size and vascularity of the glands, in addition to the disappearance of hypoechoic areas
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F3
Figure 2, Figure 3). Facial magnetic resonance imaging (MRI) demonstrated the diffuse enlargement of the lacrimal and submandibular glands, without evidence of focal lesion or nodularity.
MRI showed enlarged lacrimal glands before treatment: A, B) fat-suppressed T2-weighted images demonstrated the hypointense bilateral swelling of the lacrimal glands; C) the lacrimal glands were isointense to skeletal muscle on T1-weighted images; D) T1-weighted images post-contrast showed that the lacrimal glands are remarkably and homogeneously enhanced
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F4
No evidence of sialolithiasis or dilatation of the parotid duct was detected (Figure 4, Figure 5).
facial MRI before treatment: A) fat-suppressed T2-weighted images showed that the submandibular glands were diffusely enlarged and hypointense; B) T1-weighted images demonstrated the low signal intensity of submandibular glands; C) T1-weighted images with contrast showed the remarkably homogeneous enhancement of these glands
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F5
No evidence of sialolithiasis or dilatation of the parotid duct was detected (Figure 4, Figure 5).
microscopic images of the lacrimal gland visualized with hematoxylin and eosin stain (A-C) and CD138 antibody stain (D); A) lymphoplasmacytic infiltration and fibrosis; B) fibrosclerosis was found in the gland interstitium; C) lacrimal ducts were observed with collagenous sheaths; D) CD38 immunoreactivity was observed, confirming plasma cell infiltration
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F6
The immunohistochemical findings revealed CD138-positive cells (Figure 6) and the absence of malignant cells.
microscopic images of lacrimal gland with anti-IgG antibody stain x100 (A); anti-IgG4 antibody stain x100 (B) and anti-IgG4 antibody stain x400 (C); A, B) representative section of the lacrimal gland showed infiltration of IgG and IgG4 cells; C) markedly increased IgG4-positive plasma cells (the number of IgG4+ cells >50/ high-power field)
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F7
Immunostaining results demonstrated that there were remarkably increases of IgG and IgG4 positive plasma cells with the IgG4 positive plasma cells > 50/ high-power field (Figure 7).