Epidemiology and blood parameter changes in Cushing’s syndrome — a population-based study

Mangion, Jessica; Imbroll, Miriam Giordano; Craus, Sarah; Vassallo, Josanne; Gruppetta, Mark

doi:10.1007/s42000-022-00384-9

Cited by 5 publications

(2 citation statements)

References 38 publications

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“…To improve the prognostic assessment of PitNETs, a clinicopathological classification was proposed [37], and validated in further studies [37,[40][41][42][43][44]. Other markers of aggressive PitNET disease have been investigated, including in the following domains: (i) genetics, with germline mutations in AIP and MEN1 genes, or duplications in GPR101, as well as somatic mutations in genes ATRX or TP53, leading to more invasive and resistant PitNETs [45][46][47][48][49][50]; (ii) transcriptional and post-transcriptional regulation [51][52][53][54]; (iii) serum inflammation-based biomarkers [55][56][57]; and (iv) tumour microenvironment [58][59][60]. Nevertheless, no single marker is able to predict aggressive or recurrent PitNETs, and most of the above-mentioned markers still need validation in predicting aggressive or malignant PitNET disease.…”

Section: Aggressive and Metastatic Pitnetsmentioning

confidence: 99%

The Effects of Peptide Receptor Radionuclide Therapy on the Neoplastic and Normal Pituitary

Marques

2023

Cancers

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Pituitary neuroendocrine tumours (PitNETs) are usually benign and slow-growing; however, in some cases, they may behave aggressively and become resistant to conventional treatments. Therapeutic options for aggressive or metastatic PitNETs are limited, and currently mainly consist of temozolomide, with little experience of other emerging approaches, including peptide receptor radionuclide therapy (PRRT). Somatostatin receptor expression in PitNETs explains the effectiveness of somatostatin analogues for treating PitNETs, particularly those hypersecreting pituitary hormones, such as growth hormone or adrenocorticotropic hormone. The expression of such receptors in pituitary tumour cells has provided the rationale for using PRRT to treat patients with aggressive or metastatic PitNETs. However, the PRRT efficacy in this setting remains unestablished, as knowledge on this today is based only on few case reports and small series of cases, which are reviewed here. A total of 30 PRRT-treated patients have been thus far reported: 23 aggressive PitNETs, 5 carcinomas, and 2 of malignancy status unspecified. Of the 27 published cases with information regarding the response to PRRT, 5 (18%) showed a partial response, 8 (30%) had stable disease, and 14 (52%) had progressive disease. No major adverse effects have been reported, and there is also no increased risk of clinically relevant hypopituitarism in patients with pituitary or non-pituitary neuroendocrine tumours following PRRT. PRRT may be regarded as a safe option for patients with aggressive or metastatic PitNETs if other treatment approaches are not feasible or have failed in controlling the disease progression, with tumour shrinkage occurring in up to a fifth of cases, while about a third of aggressive pituitary tumours may achieve stable disease. Here, the data on PRRT in the management of patients with aggressive pituitary tumours are reviewed, as well as the effects of PRRT on the pituitary function in other PRRT-treated cancer patients.

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Section: Aggressive and Metastatic Pitnetsmentioning

confidence: 99%

The Effects of Peptide Receptor Radionuclide Therapy on the Neoplastic and Normal Pituitary

Marques

2023

Cancers

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“…In addition to the classic constellation of signs and symptoms, CS has been shown to in uence patients' hematological pro les: anemia [9,10], polycythemia [11][12][13], leukocytosis [9,14,15], lymphopenia [16], and proliferation of in ammatory markers [17] have all been reported. Most trends were identi ed in particular demographic groups of a patient population.…”

Section: Introductionmentioning

confidence: 99%

Impact of etiology, sex, diabetes mellitus and remission status on erythrocytic profile in patients with cushing’s syndrome: a large population database study

Dery,

Stern,

Shimon

et al. 2024

Pituitary

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The study aimed to characterize the erythrocytic pro le in patients with Cushing's syndrome (CS) versus controls from the normal population according to etiology, sex, presence of diabetes mellitus (DM) and hypercortisolemia remission status. MethodsThis retrospective cohort analysis compared erythrocytic parameters between patients with CS of pituitary (CD) and adrenal (aCS) etiology and age, sex, body mass index (BMI) and socioeconomic status-matched controls in a 1:5 ratio. Laboratory values at baseline were calculated as mean values during the year preceding CS diagnosis, and over one year thereafter. ResultsThe cohort included 397 CS patients (68.26% female; mean age 51.11 ± 16.85 years) and 1970 controls. Patients with CS had signi cantly higher baseline median levels of hemoglobin (Hgb) (13.70 g/dL vs. 13.12 g/dL [p < 0.0001]) and hematocrit (Hct) (41.64% vs. 39.80% [p < 0.0001]) compared to controls. These differences were observed for both CD and aCS and for both sexes. Patients who attained remission had Hgb and Hct levels comparable to controls (13.20 g/dL and 40.08% in patients with CD and aCS vs. 13.20 g/dL and 39.98% in controls). Meanwhile, those with persistent/recurrent disease maintained elevated levels. Patients with comorbid DM had similar Hgb but higher Hct (p = 0.0419), while patients without DM showed elevated erythrocytic values compared to controls (p < 0.0001). ConclusionOur data illustrates that erythrocytic parameters are directly in uenced by glucocorticoid excess as Hgb and Hct are higher in patients with CS, and normalize after remission. We have identi ed the in uence of DM on erythrocytic parameters in patients with CS for the rst time. ParticipantsAfter the initial screening of patients with an ICD-10 diagnosis of CS, cases had to meet one of the following criteria: (a) any recorded 24-hour urinary free cortisol (UFC) > 4 × upper limit of normal (ULN), (b) 24-hour UFC > 3 × ULN and a surgical intervention to remove the pituitary or adrenal adenoma, (c) a 24hour UFC > 2 × ULN and treatment with metyrapone, ketoconazole, osilodrostat, cabergoline, or pasireotide.Each diagnosis was retrospectively con rmed by the authors AA and YR during data collection, by a thorough review of documented biochemical and imaging test results, as well as information about management and follow-up. For each patient, the time of diagnosis was de ned as the earliest occurrence of either an elevated UFC result, the rst pituitary or adrenal gland resection, or the initial diagnosis of CS. Each patient with CS was paired with ve, age, sex, BMI and socioeconomic status-matched controls.All patients with adrenocorticotropic hormone (ACTH)-dependent CS were screened to con rm exclusion of ectopic CS. Patients with adrenocortical carcinoma that were identi ed within 5 years of CS detection or patients with ectopic CS were excluded from the study. De nitionsThe etiology of CS was established based on the available biochemical and radiological data, pharmacotherapy, radiotherapy, and surgical interventions for C...

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Inflammation-based scores in a large cohort of adrenocortical carcinoma and adrenocortical adenoma: role of the hormonal secretion pattern

Mangone,

Favero,

Prete

et al. 2024

J Endocrinol Invest

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Background Serum inflammation-based scores can predict clinical outcome in several cancer types, including adrenocortical carcinoma (ACC). It is unclear whether the extent of inflammation-based scores alterations in ACC reflects malignancy, steroid excess, or both. Methods We investigated a large retrospective cohort of adrenocortical adenomas (ACA, n = 429) and ACC (n = 61) with available baseline full blood count and hormonal evaluation. We examined the relationship between different inflammation-based scores [neutrophil-to-lymphocyte ratio (NLR), platelet-to-lymphocyte ratio (PLR), lymphocyte-to-monocyte ratio (LMR), systemic immune-inflammation index (SII), and prognostic nutrition index (PNI)] and both malignancy and steroid secretion patterns. Results All inflammation-based scores differed between ACC and ACA: patients with ACC had higher NLR, PLR, SII and lower LMR and PNI levels compared to ACA (all p values < 0.001). NLR showed a positive correlation with cortisol levels after overnight 1 mg-dexamethasone suppression test (1 mg-DST), both in ACC and ACA (p < 0.01). The ROC curve analysis determined NLR > 2.6 as the best cut-off to discriminate ACC from ACA [AUC = 0.846, p < 0.01]. At multivariable analysis, NLR > 2.6 was independently associated with ACC, 1 mg-DST cortisol levels and age, but not with tumour size. Considering the ACC, NLR and SII were higher and PNI was lower in patients with cortisol excess compared to those without cortisol excess (p = 0.002, p = 0.007, and p = 0.044 respectively). Finally, LMR and NLR differed between inactive-ACC (n = 10) and inactive-ACA (n = 215) (p = 0.040 and p = 0.031, respectively). Conclusion Inflammation-based scores are related to steroid secretion both in ACC and ACA. ACCs present a higher grade of inflammation regardless of their hormonal secretion, likely as a feature of malignancy itself.

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Epidemiology and blood parameter changes in Cushing’s syndrome — a population-based study

Cited by 5 publications

References 38 publications

The Effects of Peptide Receptor Radionuclide Therapy on the Neoplastic and Normal Pituitary

The Effects of Peptide Receptor Radionuclide Therapy on the Neoplastic and Normal Pituitary

Impact of etiology, sex, diabetes mellitus and remission status on erythrocytic profile in patients with cushing’s syndrome: a large population database study

Inflammation-based scores in a large cohort of adrenocortical carcinoma and adrenocortical adenoma: role of the hormonal secretion pattern

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