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Suspected metastatic adrenocortical carcinoma revealing as pulmonary Kaposi sarcoma in adrenal Cushing's syndrome.

Bala M, Ronchi CL, Pichl J, Wild V, Kircher S, Allolio B, Hahner S - BMC Endocr Disord (2014)

Bottom Line: Adrenal CS was confirmed by standard biochemical evaluation.Post-operative follow up imaging showed complete remission of all KS-related pulmonary nodules solely after resolution of hypercortisolism.KS may occur in the setting of endogenous CS and may go into remission after cure of hypercortisolism without further specific treatment.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Medicine I, Endocrine and Diabetes Unit, University Hospital of Wuerzburg, Oberduerrbacherstrasse 6, Wuerzburg D-97080, Germany. bala_m@ukw.de.

ABSTRACT

Background: Kaposi sarcoma (KS) is a malignant disease most commonly diagnosed in the setting of a human immunodeficiency virus (HIV) infection and in patients receiving immunosuppressive treatment. Pulmonary KS has never been reported in association with endogenous Cushing's syndrome (CS).

Case presentation: A 60-year-old woman presented with symptoms and signs of CS. Adrenal CS was confirmed by standard biochemical evaluation. Imaging revealed a right adrenal lesion (diameter 3.5 cm) and multiple pulmonary nodules, suggesting a cortisol-secreting adrenal carcinoma with pulmonary metastases. The patient underwent right adrenalectomy with a pathohistological diagnosis of an adrenal adenoma. Subsequent thoracoscopic wedge resection of one lung lesion revealed pulmonary KS with positive immunostaining for human herpes virus 8 (HHV-8). HIV-serology was negative. Hydrocortisone replacement was initiated for secondary adrenal insufficiency after surgery. Post-operative follow up imaging showed complete remission of all KS-related pulmonary nodules solely after resolution of hypercortisolism.

Conclusion: KS may occur in the setting of endogenous CS and may go into remission after cure of hypercortisolism without further specific treatment.

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Related in: MedlinePlus

Imaging of the right adrenal tumor at initial evaluation. i. CT imaging revealed a 3.3 cm right adrenal tumor (white arrow) ii. High uptake of 18 F-FDG with standardized uptake value (SUV) of 5.0 for the right adrenal lesion (white arrow).
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Figure 1: Imaging of the right adrenal tumor at initial evaluation. i. CT imaging revealed a 3.3 cm right adrenal tumor (white arrow) ii. High uptake of 18 F-FDG with standardized uptake value (SUV) of 5.0 for the right adrenal lesion (white arrow).

Mentions: Laboratory tests performed at the local hospital showed failure to suppress cortisol levels after 1 mg dexamethasone and elevated 24-hour urine cortisol secretion. Plasma adrenocorticotropin (ACTH) levels were suppressed indicating an adrenal origin of the hypercortisolism (Table 1). A pulmonary nodule was suspected in the abdominal CT imaging and an additional chest CT revealed multiple bilateral pulmonary nodules which were not described in a CT imaging of the chest performed 5 years earlier. A fluorodeoxyglucose positron emission tomography (FDG-PET) was performed for further evaluation. A high uptake of 18 F-FDG for both the adrenal lesion and the pulmonary nodules was observed (Figure 1 and Figure 2).


Suspected metastatic adrenocortical carcinoma revealing as pulmonary Kaposi sarcoma in adrenal Cushing's syndrome.

Bala M, Ronchi CL, Pichl J, Wild V, Kircher S, Allolio B, Hahner S - BMC Endocr Disord (2014)

Imaging of the right adrenal tumor at initial evaluation. i. CT imaging revealed a 3.3 cm right adrenal tumor (white arrow) ii. High uptake of 18 F-FDG with standardized uptake value (SUV) of 5.0 for the right adrenal lesion (white arrow).
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4128825&req=5

Figure 1: Imaging of the right adrenal tumor at initial evaluation. i. CT imaging revealed a 3.3 cm right adrenal tumor (white arrow) ii. High uptake of 18 F-FDG with standardized uptake value (SUV) of 5.0 for the right adrenal lesion (white arrow).
Mentions: Laboratory tests performed at the local hospital showed failure to suppress cortisol levels after 1 mg dexamethasone and elevated 24-hour urine cortisol secretion. Plasma adrenocorticotropin (ACTH) levels were suppressed indicating an adrenal origin of the hypercortisolism (Table 1). A pulmonary nodule was suspected in the abdominal CT imaging and an additional chest CT revealed multiple bilateral pulmonary nodules which were not described in a CT imaging of the chest performed 5 years earlier. A fluorodeoxyglucose positron emission tomography (FDG-PET) was performed for further evaluation. A high uptake of 18 F-FDG for both the adrenal lesion and the pulmonary nodules was observed (Figure 1 and Figure 2).

Bottom Line: Adrenal CS was confirmed by standard biochemical evaluation.Post-operative follow up imaging showed complete remission of all KS-related pulmonary nodules solely after resolution of hypercortisolism.KS may occur in the setting of endogenous CS and may go into remission after cure of hypercortisolism without further specific treatment.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Medicine I, Endocrine and Diabetes Unit, University Hospital of Wuerzburg, Oberduerrbacherstrasse 6, Wuerzburg D-97080, Germany. bala_m@ukw.de.

ABSTRACT

Background: Kaposi sarcoma (KS) is a malignant disease most commonly diagnosed in the setting of a human immunodeficiency virus (HIV) infection and in patients receiving immunosuppressive treatment. Pulmonary KS has never been reported in association with endogenous Cushing's syndrome (CS).

Case presentation: A 60-year-old woman presented with symptoms and signs of CS. Adrenal CS was confirmed by standard biochemical evaluation. Imaging revealed a right adrenal lesion (diameter 3.5 cm) and multiple pulmonary nodules, suggesting a cortisol-secreting adrenal carcinoma with pulmonary metastases. The patient underwent right adrenalectomy with a pathohistological diagnosis of an adrenal adenoma. Subsequent thoracoscopic wedge resection of one lung lesion revealed pulmonary KS with positive immunostaining for human herpes virus 8 (HHV-8). HIV-serology was negative. Hydrocortisone replacement was initiated for secondary adrenal insufficiency after surgery. Post-operative follow up imaging showed complete remission of all KS-related pulmonary nodules solely after resolution of hypercortisolism.

Conclusion: KS may occur in the setting of endogenous CS and may go into remission after cure of hypercortisolism without further specific treatment.

Show MeSH
Related in: MedlinePlus