ACTH-independent Cushing's syndrome with bilateral cortisol-secreting adrenal adenomas: a case report and review of literatures.

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obesity 2 endocrinologydiseases
osteoporosis 1 endocrinologydiseases
rosuvastatin 1 endocrinologydiseasesdrugs
adrenal adenoma 8 endocrinologydiseases
cortisol 46 endocrinologydiseasesdrugs
dexamethasone 4 endocrinologydiseasesdrugs
hyperlipidemia 1 endocrinologydiseases

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cortisol 83 Title: BMC Endocrine DisordersACTH-independent Cushing’s syndrome with bilateral cortisol -secreting adrenal adenomas: a case report and review of literaturesJia WeiSheyu LiQilin LiuYuchun ZhuNianwei
cortisol 495 /2018AbstractBackgroundAdrenocorticotropic hormone (ACTH)-independent Cushing’s syndrome (CS) with bilateral cortisol -secreting adenomas has been rarely reported in the literatures. Precise recognition and management of
cortisol 932 dyspnea for over 10 years. ACTH-independent CS was diagnosed based on undetectable ACTH and unsuppressed cortisol levels by dexamethasone. Computed tomography (CT) scan indicated bilateral adrenal masses, and adrenal
cortisol 1124 masses, and adrenal venous sampling (AVS) adjusted by plasma aldosterone revealed hypersecretion of cortisol from both adrenal glands. Bilateral cortisol-secreting adrenal adenomas were suspected and confirmed
cortisol 1169 adjusted by plasma aldosterone revealed hypersecretion of cortisol from both adrenal glands. Bilateral cortisol -secreting adrenal adenomas were suspected and confirmed by the postoperative pathology in subsequent
cortisol 1764 cannulation in this situation.BackgroundCushing’s syndrome (CS), which results from prolonged excessive cortisol secretion, is a collection of complicated symptoms and associated with significant morbidity and mortality
cortisol 2120 usually induced by unilateral adrenal adenomas or adrenal carcinomas accompanied by autonomous adrenal cortisol secretion [[1]]. ACTH-independent CS is occasionally caused by bilateral adrenocortical lesions, including
cortisol 2749 a new case of a Chinese female patient with ACTH-independent Cushing’s syndrome due to bilateral cortisol -secreting adenomas, which was diagnosed through adrenal venous sampling (AVS) adjusted by plasma aldosterone
cortisol 4505 hemoglobin (HbA1c) was 6.6% (Table 1). Endocrinological examinations showed that circadian rhythm of cortisol disappeared, and the level of ACTH was less than 1.00 ng/L (Table 2). Twenty-four-hour urine free
cortisol 4614 disappeared, and the level of ACTH was less than 1.00 ng/L (Table 2). Twenty-four-hour urine free cortisol (24 h UFC) elevated to 634.8μg/24 h (reference range: 20.26-127.55μg/24 h). The next morning (8 a.m.)
cortisol 4737 elevated to 634.8μg/24 h (reference range: 20.26-127.55μg/24 h). The next morning (8 a.m.) serum cortisol level after an overnight 1 mg dexamethasone suppression test (DMST) was 787.5 nmol/L, indicated lack
cortisol 6672 ODMST–––211.6290.1ACTH adrenocorticotropic hormone, ODMST overnight dexamethasone suppression test, PTC plasma total cortisol , 24 h UFC 24 h urine free cortisolFor differential diagnosis, aldosterone-to-renin ratio (ARR) was
cortisol 6709 ODMST overnight dexamethasone suppression test, PTC plasma total cortisol, 24 h UFC 24 h urine free cortisol For differential diagnosis, aldosterone-to-renin ratio (ARR) was measured after discontinuation of irbesartan
cortisol 7911 functional lesions in this patient, AVS was performed and the concentrations of plasma aldosterone and cortisol were measured from both adrenal veins (AV) and inferior vena cava (IVC). Adrenal venous catheterization
cortisol 8126 was successful, and the hormone levels were shown in Table 3. The adrenal vein to inferior vena cava cortisol (AV: IVC) gradient was 13.57 on the right side and 13.88 on the left side. The left and right AV to
cortisol 8310 The left and right AV to IVC gradient of aldosterone were 5.58 and 6.79 respectively. Moreover, the cortisol /aldosterone ratio (CAR) in adrenal veins was 292.52 on the right and 359.29 on the left, along with
cortisol 8616 indicated non-lateralization, this patient was diagnosed with CS induced by bilateral adrenal excessive cortisol secretion.Fig. 1Adrenal computed tomography (CT) of the patient. Adrenal CT showed a right adrenal nodule
cortisol 9949 × 200)Overnight 1 mg DMST was repeated 2 weeks after surgery, which demonstrated no inhibition on the serum cortisol at 8 a.m. on the following day, despite significantly decreased cortisol level post-operation (Table 2).
cortisol 10023 no inhibition on the serum cortisol at 8 a.m. on the following day, despite significantly decreased cortisol level post-operation (Table 2). Therefore, it can be inferred that the autonomous cortisol secretion
cortisol 10115 decreased cortisol level post-operation (Table 2). Therefore, it can be inferred that the autonomous cortisol secretion from left adrenal masses was persistent. The left adrenal gland was then removed and two adenomas
cortisol 10305 then removed and two adenomas were confirmed by pathological examination (Fig. 2). The 8 a.m. plasma cortisol after 3 days of bilateral adrenalectomy was 37.30 nmol/L. Hydrocortisone replacement therapy (from
cortisol 10884 performed to screen the case reports relating to ACTH-independent Cushing’s syndrome caused by bilateral cortisol -secreting adenomas. Searching words included “Cushing’s syndrome” and “bilateral adrenocortical
cortisol 15601 revealed bilateral adrenal uptake. AVS was performed in eight cases to evaluate the hypersecretion of cortisol , and only two of them applied cortisol gradient adjusted by plasma aldosterone [[9], [10]]. All patients
cortisol 15640 was performed in eight cases to evaluate the hypersecretion of cortisol, and only two of them applied cortisol gradient adjusted by plasma aldosterone [[9], [10]]. All patients underwent surgical resection of adenomas,
cortisol 16351 adrenalectomy [[21]–[23]].Discussion and conclusionACTH-independent Cushing’s syndrome with bilateral cortisol -secreting adenomas has been rarely reported in the literature [[6], [8]–[24]]. This disorder should
cortisol 18140 by international guidelines to differentiate unilateral from bilateral primary aldosteronism through cortisol -corrected aldosterone ratio [[30], [32], [33]]. During AVS, blood was collected from bilateral AV and
cortisol 18306 AVS, blood was collected from bilateral AV and IVC or peripheral vein (PV) to measure aldosterone and cortisol levels, and the comparison of AV and IVC cortisol concentration was further used to assess whether successful
cortisol 18356 or peripheral vein (PV) to measure aldosterone and cortisol levels, and the comparison of AV and IVC cortisol concentration was further used to assess whether successful cannulation was achieved in PA. In this
cortisol 19151 robust assay for this purpose, after the aldosterone overproduction being excluded.We used AV: IVC cortisol ratio in both side and left-to-right CAR gradient to differentiate unilateral from bilateral cortisol
cortisol 19253 cortisol ratio in both side and left-to-right CAR gradient to differentiate unilateral from bilateral cortisol overproduction. Young et al. [[8]] suggested that if the cortisol gradient of AV to PV or IVC was greater
cortisol 19319 differentiate unilateral from bilateral cortisol overproduction. Young et al. [[8]] suggested that if the cortisol gradient of AV to PV or IVC was greater than 6.5, cortisol-secreting adenoma should be considered. Although
cortisol 19378 Young et al. [[8]] suggested that if the cortisol gradient of AV to PV or IVC was greater than 6.5, cortisol -secreting adenoma should be considered. Although the accuracy and applicability remain to be proved
cortisol 19619 this area, we can at least assume that a larger ratio represents a greater likelihood of spontaneous cortisol secretion. The authors also proposed cutoff values of high- to low-side AV cortisol gradient ratio to
cortisol 19703 of spontaneous cortisol secretion. The authors also proposed cutoff values of high- to low-side AV cortisol gradient ratio to determine the lateralization of cortisol hypersecretion, and suggested that predominant
cortisol 19762 proposed cutoff values of high- to low-side AV cortisol gradient ratio to determine the lateralization of cortisol hypersecretion, and suggested that predominant cortisol secretion was considered if the cortisol lateralization
cortisol 19818 gradient ratio to determine the lateralization of cortisol hypersecretion, and suggested that predominant cortisol secretion was considered if the cortisol lateralization ratio was ≥2.3 [[8]]. However, unadjusted
cortisol 19859 of cortisol hypersecretion, and suggested that predominant cortisol secretion was considered if the cortisol lateralization ratio was ≥2.3 [[8]]. However, unadjusted cortisol lateralization ratio might confound
cortisol 19927 secretion was considered if the cortisol lateralization ratio was ≥2.3 [[8]]. However, unadjusted cortisol lateralization ratio might confound the interpretation of AVS results because of the cortisol concentration
cortisol 20021 unadjusted cortisol lateralization ratio might confound the interpretation of AVS results because of the cortisol concentration was 1.8 times higher in the right AV than in the left side, which was the result of dilution
cortisol 21036 throughout the duration of treatment and follow-up.The optimal treatment for patients with bilateral cortisol -secreting adenomas remains uncertain [[27], [38]]. Two-step bilateral adrenalectomy was performed on
cortisol 21203 Two-step bilateral adrenalectomy was performed on our patient and resulted in remarkable remission of hyper cortisol ism, and further confirmed the cortisol-secreting feature of each adrenal mass. Even though lifelong
cortisol 21242 on our patient and resulted in remarkable remission of hypercortisolism, and further confirmed the cortisol -secreting feature of each adrenal mass. Even though lifelong steroid supplementation was required, the
cortisol 21927 [23]].In summary, we reported a Chinese female patient with ACTH-independent CS caused by bilateral cortisol -secreting adenomas. She was diagnosed through aldosterone-adjusted AVS and successfully treated with
cortisol 22121 successfully treated with bilateral adrenalectomy. It confirmed that selective AVS with aldosterone-corrected cortisol ratio could be a useful technique to evaluate the cortisol-secreting function of each adrenal mass and
cortisol 22180 selective AVS with aldosterone-corrected cortisol ratio could be a useful technique to evaluate the cortisol -secreting function of each adrenal mass and further guide therapeutic decision-making. Owing to the
cortisol 22400 dispute over the interpretation of the AVS results, the definite cut-off values for lateralization of cortisol hypersecretion requires further confirmation
dexamethasone 951 years. ACTH-independent CS was diagnosed based on undetectable ACTH and unsuppressed cortisol levels by dexamethasone . Computed tomography (CT) scan indicated bilateral adrenal masses, and adrenal venous sampling (AVS)
dexamethasone 4777 range: 20.26-127.55μg/24 h). The next morning (8 a.m.) serum cortisol level after an overnight 1 mg dexamethasone suppression test (DMST) was 787.5 nmol/L, indicated lack of normal suppression (Table 2). The diagnosis
dexamethasone 6270 plasma renin activity, TG triglyceride, WBC white blood cell countTable 2Results of hormone levels and dexamethasone suppression tests08:00 ACTH (ng/L)24 h UFC (ug/24 h)24:00 PTC (nmol/L)08:00 PTC (nmol/L)08:00 PTC
dexamethasone 6623 1.040.8202.1223.9–1 mg ODMST–––211.6290.1ACTH adrenocorticotropic hormone, ODMST overnight dexamethasone suppression test, PTC plasma total cortisol, 24 h UFC 24 h urine free cortisolFor differential diagnosis,
rosuvastatin 9234 vein, IVC inferior vena cavaTreatment and follow-upThe patient was treated with metoprolol succinate, rosuvastatin , insulin, calcium and vitamin D supplements during the investigation. Considering her poor cardiac function,
Select Disease Character Offset Disease Term Instance
adrenal adenoma 102 Title: BMC Endocrine DisordersACTH-independent Cushing’s syndrome with bilateral cortisol-secreting adrenal adenoma s: a case report and review of literaturesJia WeiSheyu LiQilin LiuYuchun ZhuNianwei WuYing TangQianrui
adrenal adenoma 1188 aldosterone revealed hypersecretion of cortisol from both adrenal glands. Bilateral cortisol-secreting adrenal adenoma s were suspected and confirmed by the postoperative pathology in subsequent two-step bilateral laparoscopic
adrenal adenoma 1544 by plasma aldosterone could be a useful technique in diagnosing ACTH-independent CS with bilateral adrenal adenoma s prior to surgery. And the aldosterone ratio could be used to confirm the success of adrenal vein cannulation
adrenal adenoma 2047 ACTH-independent etiologies, the latter accounts for 15~ 20% of the cases and is usually induced by unilateral adrenal adenoma s or adrenal carcinomas accompanied by autonomous adrenal cortisol secretion [[1]]. ACTH-independent
adrenal adenoma 9809 glands. Pathological examination of the right (a) and left adrenal gland (b and c) indicated bilateral adrenal adenoma s (HE stain, × 200)Overnight 1 mg DMST was repeated 2 weeks after surgery, which demonstrated no
adrenal adenoma 11803 onset (the mean age was 39.6 ± 8.6 years; ranged from 24 to 53 years); 2) the size of bilateral adrenal adenoma s ranged from 1.0 to 5.0 cm in diameter, the majority of which were solitary in both sides (12 out of
adrenal adenoma 12428 of study selection for the literature reviewTable 4The available case reports of CS with bilateral adrenal adenoma sStudy IDCountryAge (years)GenderLesions size (cm)Preoperative diagnostic techniqueOnsetDiag.rightleft1963
adrenal adenoma 17284 several previous studies, precise diagnosis and treatment of patients with bilateral ACTH-independent adrenal adenoma s remain challenging [[8], [23], [24]].The diagnostic value of AVS and 131I-6β-iodomethyl-19-norcholesterol
hyperlipidemia 3500 she had been using irbesartan, metoprolol and nifedipine XR since then. She was also diagnosed with hyperlipidemia and prescribed with statins for 5 years. The patient reported no history of alcohol or drug abuse, in
obesity 3172 hospital complaining of exertional dyspnea for more than 10 years. She had been developing truncal obesity and facial rounding over the past 2 years, without evidence of acne, hirsutism or wide purple striae.
obesity 3984 respectively, with a body mass index (BMI) of 20.96 kg/m2. She had a plethoric moon-shaped face, centripetal obesity , buffalo hump, accompanied by ecchymosis and slight edema at both lower limbs. Neurological examination
osteoporosis 7771 femoral neck and the total hip was − 3.0, − 3.2 and − 3.3, respectively, which indicated osteoporosis . In order to locate the functional lesions in this patient, AVS was performed and the concentrations

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