case_images: PMC11828756_01
case_images: Image findings. (A,B) Abdominal plain CT finding. Arrows show right (A) and left (B) adrenal masses. 123I-metaiodobenzylguanidine scintigraphy finding from anterior side (C) and posterior side (D). Arrows show accumulation in the left adrenal gland. (E,F) Chest CT (lung window setting). CT: computed tomography
case_images: cureus-0017-00000077459-i01.jpg
case_images: PMC11828756_01_cureus-0017-00000077459-i01.jpg
case_images: FIG1
case_images: Computed tomography (CT) revealed significant stool accumulation but no ileus, and bilateral adrenal masses were detected (Figures 1A, 1B); elevated levels of urinary and plasma catecholamines, metanephrine, and normetanephrine were also detected, leading to a diagnosis of pheochromocytoma. In addition, endocrinological analysis showed elevated ACTH and cortisol levels, despite magnetic resonance imaging ruling out a pituitary tumor; this finding was initially considered to be the result of stress responses. 123I-metaiodobenzylguanidine (MIBG) scintigraphy revealed accumulation only in the left adrenal gland (Figures 1C, 1D). CT revealed ground-glass opacities and nodular infiltrates in both lungs, along with pleural effusion (Figures 1E, 1F). beta-D-glucan was elevated (208.5 pg/mL, reference range < 11.0 pg/mL), and a peripheral blood cytomegalovirus (CMV) antigen immunological assay using the horseradish peroxidase-labeled monoclonal antibody was marginally positive (eight CMV-antigen positive leukocytes per 50,000 cells).
case_images: Patient's clinical course from the time of transfer to our hospital. (A) The time courses of treatment by alpha-blockers and osilodrostat for pheochromocytoma and management of steroid hormone. Line graphs illustrating changes in potassium (B), cortisol, and ACTH (C) levels. (D) Brain CT on day 3 after CPA. ACTH: adrenocorticotropic hormone; CPA: cardiopulmonary arrest; BSC: best supportive care; DST: dexamethasone suppression test; CT: computed tomography; CIV: continuous intravenous infusion
case_images: cureus-0017-00000077459-i02.jpg
case_images: PMC11828756_01_cureus-0017-00000077459-i02.jpg
case_images: FIG2
case_images: The patient's clinical course is shown in Figure 2. Treatment included gradual escalation of doxazosin and phentolamine, tapering of methylprednisolone, and initiation of osilodrostat through a gastric tube on day 17 to control hypercortisolemia (Figure 2A). Interestingly, it lowered not only cortisol but also ACTH levels and successfully corrected potassium levels (Figures 2B, 2C), with the subsequent initiation of hydrocortisone replacement to avoid hypoadrenalism. Despite successful resuscitation after more than 10 minutes of arrest, brain CT on day 3 postarrest revealed severe cerebral edema (Figure 2D), indicating a poor prognosis for neurological recovery.
case_images: (A) Macroscopic image of the left adrenal tumor. A circumscribed mass was found in the left adrenal gland. (B-E) Microscopic images of left adrenal tumor. Atypical cells showing polymorphism grow in alveolar (B) and diffuse (C) patterns with sustentacular cells (arrows) (B), which are highlighted by immunohistochemical staining for S-100 (inset in B). Tumor cells are immunopositive for synaptophysin (D) and chromogranin A (E). Hematoxylin-eosin stain (B,C); immunostains for synaptophysin (D) and chromogranin A (E). Scale bars: 100 mum (B,C) and 40 mum (D,E)
case_images: cureus-0017-00000077459-i03.jpg
case_images: PMC11828756_01_cureus-0017-00000077459-i03.jpg
case_images: FIG3
case_images: A subsequent autopsy confirmed a 4 cm x 2.5 cm tumor in the left adrenal gland (Figure 3A). Histologically, the tumor comprised atypical cells with pleomorphic nuclei, enlarged round nuclei, and multinucleated giant cells that proliferated in alveolar (Zellballen) (Figure 3B) and diffuse (Figure 3C) patterns. At the periphery of the alveolar nests, S-100-positive sustentacular cells were observed (arrows and inset in Figure 3B). Immunohistochemically, the atypical cells were positive for synaptophysin and chromogranin A (Figures 3D, 3E).
case_images: Production of ACTH and the invasive properties of left adrenal pheochromocytoma. (A) Immunohistochemical staining showing the production of ACTH. Invasive properties of tumor cells in lymphatic vessels (arrows) (B) and veins (arrow) (C). Immunostains for ACTH (A) and D2-40 (B); Elastica van Gieson stain (C). Scale bar: 40 mum (A) and 200 mum (B,C). ACTH: adrenocorticotropic hormone
case_images: cureus-0017-00000077459-i04.jpg
case_images: PMC11828756_01_cureus-0017-00000077459-i04.jpg
case_images: FIG4
case_images: Tumor cells were immunopositive for ACTH (Figure 4A), indicating that the tumor was an ACTH-secreting pheochromocytoma. Immunostaining for D2-40 and Elastica van Gieson revealed the invasive properties of the tumor cells in the lymphatic system and veins, respectively (Figures 4B, 4C).