PMC7272571_01
T1 pre-contrast sagittal images (left column), T1 post-contrast sagittal images (center column) and T1 post-contrast coronal images (right column). (A) Pre-operative; (B) 2 weeks post-operative; (C) 15 weeks (3.5 months) post-operative, 3 weeks post-corticosteroid taper; (D) 26 weeks (6 months) post-operative, 12 weeks post-TNF-alpha inhibitor initiation (infliximab) immediately prior to azathioprine addition; (E) 17 months post-TNF-alpha inhibitor (infliximab combined with azathioprine for 15 months, followed by adalimumab combined with azathioprine).
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F1
It was reported as a pituitary adenoma, but infundibular thickening was visible upon closer examination (Figure 1A). The presumed posterior pituitary "bright spot" was visible on the MRI T1 pre-contrast sagittal view (Figure 1A, left panel). She underwent endoscopic transsphenoidal biopsy and partial resection of the pituitary lesion, with the surgical cavity visible on the post-operative MRI (Figure 1B). MRI revealed relapse of her mass lesion on MRI (Figure 1C). Within 3 months of therapy, she experienced a marked improvement in symptoms and resolution of the mass on MRI (Figure 1D). Subsequent MRI at nearly one and a half years after initiation of immunosuppressive therapy, including 2 months on adalimumab, revealed sustained resolution of the pituitary lesion (Figure 1E).
Hematoxylin and eosin staining (Left): Upper, non-caseating granulomatous inflammation interrupting adenohypophyseal glandular cytoarchitecture; Middle, higher magnification showing multinucleate giant cells; Lower, lymphohistiocytic inflammation and fibrosis. Immunoperoxidase staining (Right): Upper and Middle, anti-CD68 macrophage marker highlighting macrophage and multinucleated giant cells; Lower, anti-CD45 lymphoid marker for lymphocytes.
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F2
The first five surgical specimens contained adenohypophysis with lymphocytic infiltrates (anti-CD45 positive) and multiple foci of epithelioid histiocytes with focal multinucleation (anti-CD68 positive) as shown in Figure 2. Pathology was consistent with a diagnosis of GrHy (Figure 2).
The timeline of the patient's clinical course, treatment and MRI monitoring are shown from the time of surgery (0 weeks) through to the most recent imaging at 20 months from surgery.
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F3
A timeline of therapeutic interventions after surgery is depicted in Figure 3. She was switched to a fully humanized anti-TNF-alpha inhibitor adalimumab at 18 months post-operatively, and azathioprine was continued (Figure 3).