PMC8417750_01
Abdominal magnetic resonance imaging performed in March 2014 and histological analysis of the adrenal adenoma. (A) Contrast-enhanced coronal T1-weighted MRI showing a 4.5 x 4.2 cm adenoma of the left adrenal gland (white arrow). (B-D) Histological investigation of the adrenal adenoma with hematoxylin and eosin staining: (B) Solitary, well circumscribed intra-adrenal mass confined to the gland; (C) Nested/fasciculated growth pattern of uniform tumor cells; (D) Tumor cells showing lipid-rich foamy cytoplasm with small uniform nuclei. Absent mitosis or necrosis. (H.E. 6.5x, 10.0x, 40.0x).
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This revealed the presence of a left-sided adrenal lesion of 4.5 x 4.2 cm (Figure 1A). Slides from the histological investigations are exemplarily shown in Figures 1B-D. Figure 1). ACTH stimulation test revealed a persistent partial adrenal insufficiency (serum cortisol after 1 hour: 17.5 microg/dl), while the basal ACTH level was 16 ng/l. As reported in Figure 2, the biochemical workup (including 1 mg DST, UFC, and LNSC) indicated remission from CS.
Selected endocrine parameters from initial diagnosis of adrenal Cushing's syndrome until recovery after transsphenoidal surgery for Cushing's disease. Course of (A) plasma ACTH, (B) serum cortisol during the 1 mg dexamethasone suppression test, (C) late-night salivary cortisol, and (D) 24-hour urinary free cortisol. The time of adrenalectomy (in February 2015) and transsphenoidal adenomectomy (in June 2019) are illustrated with vertical broken bars. The reference range of each test is shown as a grey area. ACTH, adrenocorticotropin hormone; ADX, adrenalectomy; FU, follow up; TSS, transsphenoidal surgery.
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The 1 mg dexamethasone suppression test (DST) and the late-night salivary cortisol (LNSC) results were remarkably lower than preoperatively, but not normalized (Figure 2). The course of the four most relevant endocrine parameters (i.e., ACTH, 1 mg DST, LNSC, and 24-hour UFC) during our observation is provided in Figure 2. Biochemical workup revealed recurrent glucocorticoid excess, surprisingly without suppressed ACTH (Figure 2). At that time, the biochemical tests were once again clearly pathological (Figure 2). ACTH stimulation test revealed a persistent partial adrenal insufficiency (serum cortisol after 1 hour: 17.5 microg/dl), while the basal ACTH level was 16 ng/l. As reported in Figure 2, the biochemical workup (including 1 mg DST, UFC, and LNSC) indicated remission from CS. In the last follow up (conducted in March 2021), the patient was in complete clinical and biochemical remission (Figure 2) and reported no signs and symptoms suggestive for Cushing's syndrome.
Sellar magnetic resonance imaging (MRI) performed in April 2019 and histological analysis of the pituitary adenoma. (A) coronal and (B) sagittal contrast-enhanced T1-weighted sellar magnetic resonance imaging. The white arrows indicate a hypointense focal lesion of 0.5 x 0.4 x 0.4 cm in the dorsal part of the adenohypophysis, reaching to the right side of the gland. This lesion was radiologically regarded as compatible with a microadenoma. (C, D) hematoxylin and eosin staining of the ACTH-secreting pituitary adenoma with: fragmented parts of a good differentiated epithelial tumor; cells present with medium size and oval nuclei (with an interspersed chromatin structure); occasional detection of nucleoli; no evidence of high mitotic activity; cytoplasm predominantly chromophobe. (H.E. 10x, 20x).
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Furthermore, a pituitary MRI revealed a hypointense focal lesion of 0.5 x 0.4 x 0.4 cm in the dorsal part of the adenohypophysis compatible with a pituitary microadenoma (Figures 3A, B).