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  • 1
    Online Resource
    Online Resource
    Changes in the management and comorbidities of acromegaly over three decades: the French Acromegaly Registry
    Oxford University Press (OUP) ; 2017
    In:  European Journal of Endocrinology Vol. 176, No. 5 ( 2017-05), p. 645-655
    Details
    In: European Journal of Endocrinology, Oxford University Press (OUP), Vol. 176, No. 5 ( 2017-05), p. 645-655
    Abstract: Acromegaly is a rare disease associated with chronic multisystem complications. National registries have been created in several countries. Design The French Registry contains data on acromegaly epidemiology, management and comorbidities recorded over more than three decades, retrospectively until 1999 and prospectively from 1999 to 2012. Results Data could be analyzed for 999 of the 1034 patients included in the registry (46% males). Disease control, defined as IGF-I normalization (adjusted for age and sex), was achieved in 75% of patients at the last follow-up visit. Half the patients with uncontrolled disease had IGF-I levels below 1.5 times the upper limit of normal (ULN). The proportion of patients with surgically cured disease did not change markedly over time, whereas the proportion of patients with uncontrolled disease fell and the proportion of patients with medically controlled disease rose. Cardiovascular, metabolic, respiratory and rheumatologic comorbidities and their outcomes were recorded for most patients, and no noteworthy overall deterioration was noted over time. Cancer occurred in 10% of patients, for a standardized incidence ratio of 1.34 (95% CI: 0.94–1.87) in men and 1.24 (0.77–1.73) in women. Forty-one patients died during follow-up, for a standardized mortality ratio of 1.05 (0.70–1.42). Most deaths were due to cancer. Conclusions The majority of patients with acromegaly now have successful disease control thanks to the multistep management. The incidence of comorbidities following diagnosis of acromegaly is very low. Life expectancy is now close to that of the general population, probably owing to better management of the GH/IGF-I excess and comorbidities.
    Type of Medium: Online Resource
    ISSN: 0804-4643 , 1479-683X
    URL: Article
    DOI: 10.1530/EJE-16-1064
    RVK:
    WA 15000
    Language: Unknown
    Publisher: Oxford University Press (OUP)
    Publication Date: 2017
    detail.hit.zdb_id: 1485160-X
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  • 2
    Online Resource
    Online Resource
    McCune-Albright Syndrome: A Detailed Pathological and Genetic Analysis of Disease Effects in an Adult Patient
    The Endocrine Society ; 2014
    In:  The Journal of Clinical Endocrinology & Metabolism Vol. 99, No. 10 ( 2014-10), p. E2029-E2038
    Details
    In: The Journal of Clinical Endocrinology & Metabolism, The Endocrine Society, Vol. 99, No. 10 ( 2014-10), p. E2029-E2038
    Type of Medium: Online Resource
    ISSN: 0021-972X , 1945-7197
    URL: Article
    DOI: 10.1210/jc.2014-1291
    RVK:
    XA 10000
    Language: English
    Publisher: The Endocrine Society
    Publication Date: 2014
    detail.hit.zdb_id: 2026217-6
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  • 3
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    Online Resource
    Multivariable Prediction Model for Biochemical Response to First-Generation Somatostatin Receptor Ligands in Acromegaly
    The Endocrine Society ; 2020
    In:  The Journal of Clinical Endocrinology & Metabolism Vol. 105, No. 9 ( 2020-09-01), p. 2964-2974
    Details
    In: The Journal of Clinical Endocrinology & Metabolism, The Endocrine Society, Vol. 105, No. 9 ( 2020-09-01), p. 2964-2974
    Abstract: First-generation somatostatin receptor ligands (fg-SRLs) represent the mainstay of medical therapy for acromegaly, but they provide biochemical control of disease in only a subset of patients. Various pretreatment biomarkers might affect biochemical response to fg-SRLs. Objective To identify clinical predictors of the biochemical response to fg-SRLs monotherapy defined as biochemical response (insulin-like growth factor (IGF)-1 ≤ 1.3 × ULN (upper limit of normal)), partial response ( & gt;20% relative IGF-1 reduction without normalization), and nonresponse (≤20% relative IGF-1 reduction), and IGF-1 reduction. Design Retrospective multicenter study. Setting Eight participating European centers. Methods We performed a meta-analysis of participant data from 2 cohorts (Rotterdam and Liège acromegaly survey, 622 out of 3520 patients). Multivariable regression models were used to identify predictors of biochemical response to fg-SRL monotherapy. Results Lower IGF-1 concentration at baseline (odds ratio (OR) = 0.82, 95% confidence interval (CI) 0.72–0.95 IGF-1 ULN, P = .0073) and lower bodyweight (OR = 0.99, 95% CI 0.98–0.99 kg, P = .038) were associated with biochemical response. Higher IGF-1 concentration at baseline (OR = 1.40, (1.19–1.65) IGF-1 ULN, P ≤ .0001), the presence of type 2 diabetes (oral medication OR = 2.48, (1.43–4.29), P = .0013; insulin therapy OR = 2.65, (1.02–6.70), P = .045), and higher bodyweight (OR = 1.02, (1.01–1.04) kg, P = .0023) were associated with achieving partial response. Younger patients at diagnosis are more likely to achieve nonresponse (OR = 0.96, (0.94–0.99) year, P = .0070). Baseline IGF-1 and growth hormone concentration at diagnosis were associated with absolute IGF-1 reduction (β = 0.90, standard error (SE) = 0.02, P ≤ .0001 and β  = 0.002, SE = 0.001, P = .014, respectively). Conclusion Baseline IGF-1 concentration was the best predictor of biochemical response to fg-SRL, followed by bodyweight, while younger patients were more likely to achieve nonresponse.
    Type of Medium: Online Resource
    ISSN: 0021-972X , 1945-7197
    URL: Article
    DOI: 10.1210/clinem/dgaa387
    RVK:
    XA 10000
    Language: English
    Publisher: The Endocrine Society
    Publication Date: 2020
    detail.hit.zdb_id: 2026217-6
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  • 4
    Online Resource
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    Medical Treatment in Cushing’s Syndrome: Dopamine Agonists and Cabergoline
    S. Karger AG ; 2010
    In:  Neuroendocrinology Vol. 92, No. Suppl. 1 ( 2010), p. 116-119
    Details
    In: Neuroendocrinology, S. Karger AG, Vol. 92, No. Suppl. 1 ( 2010), p. 116-119
    Abstract: Dopamine (DA) is a catecholamine with a wide range of functions and whose five subtype receptors are found in different organs where they exert a mainly inhibitory action. Since this action may also appear in a number of secretory tumors in various locations, DA agonists have elicited some interest as a medical treatment for hypercorticism. Non-iatrogenic Cushing’s syndromes are due in 70% of the cases to a pituitary adrenocorticotropic hormone (ACTH)-producing adenoma, and, less frequently, to an adrenal adenoma or an ectopic ACTH secretion by a neuroendocrine tumor. First-line treatment in Cushing’s syndrome consists of the surgical removal of the secreting tumor. However, surgery may not achieve a complete cure in a number of cases, hence emphasizing the potential benefit of a medical complementary treatment, which could also benefit patients as an alternative approach, either when waiting for, or when the patient is not eligible for surgery. Studies of corticotropic adenomas have shown that 80% of these tumors express D2 receptors. Clinical trials of DA agonists in Cushing’s disease have shown an inhibitory effect of these drugs with an inhibition of ACTH secretion and/or a decrease of tumor size. There are only a few cases of documented use of DA agonists in ectopic ACTH secretion, but when the tumor expresses DA receptors, DA agonists may represent a useful complementary treatment. DA receptors are also expressed in normal and tumoral adrenals, suggesting a potential use of DA agonists in Cushing’s syndrome secondary to adrenal tumors. However, clinical data regarding this specific situation are very scarce, maybe due to the relatively high rate of surgical cure of adrenal adenomas. In conclusion, DA agonists represent a potential preparatory or complementary treatment for endogenous Cushing’s syndrome, especially in Cushing’s disease. These compounds may be underused as suggested by the scarce number of publication and case reports in the literature. In the future, association of these drugs with somatostatin analogs may also prove beneficial.
    Type of Medium: Online Resource
    ISSN: 0028-3835 , 1423-0194
    URL: Article
    DOI: 10.1159/000317716
    Language: English
    Publisher: S. Karger AG
    Publication Date: 2010
    detail.hit.zdb_id: 1483028-0
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  • 5
    Online Resource
    Online Resource
    Genetic susceptibility in pituitary adenomas: from pathogenesis to clinical implications
    Informa UK Limited ; 2011
    In:  Expert Review of Endocrinology & Metabolism Vol. 6, No. 2 ( 2011-03), p. 195-214
    Details
    In: Expert Review of Endocrinology & Metabolism, Informa UK Limited, Vol. 6, No. 2 ( 2011-03), p. 195-214
    Type of Medium: Online Resource
    ISSN: 1744-6651 , 1744-8417
    URL: Article
    DOI: 10.1586/eem.10.87
    Language: English
    Publisher: Informa UK Limited
    Publication Date: 2011
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  • 6
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    Dutch founder SDHB exon 3 deletion in patients with pheochromocytoma-paraganglioma in South Africa
    Bioscientifica ; 2022
    In:  Endocrine Connections Vol. 11, No. 1 ( 2022-01-01)
    Details
    In: Endocrine Connections, Bioscientifica, Vol. 11, No. 1 ( 2022-01-01)
    Abstract: Screening studies have established genetic risk profiles for diseases such as multiple endocrine neoplasia type 1 (MEN1) and pheochromocytoma–paraganglioma (PPGL). Founder effects play an important role in the regional/national epidemiology of endocrine cancers, particularly PPGL. Founder effects in the Netherlands have been described for various diseases, some of which established themselves in South Africa due to Dutch emigration. The role of Dutch founder effects in South Africa has not been explored in PPGL. Design We performed a single-center study in South Africa of the germline genetic causes of isolated/syndromic neuroendocrine tumors. Methods Next-generation panel, Sanger sequencing and multiplex ligand-dependent probe amplification for endocrine neoplasia risk genes. Results From a group of 13 patients, we identified 6 with PPGL, 4 with sporadic or familial isolated pituitary adenomas, and 3 with clinical MEN1; genetic variants were identified in 9/13 cases. We identified the Dutch founder exon 3 deletion in SDHB in two apparently unrelated individuals with distinct ethnic backgrounds that had metastatic PPGL. Asymptomatic carriers with this Dutch founder SDHB exon 3 deletion were also identified. Other PPGL patients had variants in SDHB , and SDHD and three MEN1 variants were identified among MEN1 and young-onset pituitary adenoma patients. Conclusions This is the first identification of a Dutch founder effect for PPGL in South Africa. Awareness of the presence of this exon 3 SDHB deletion could promote targeted screening at a local level. Insights into PPGL genetics in South Africa could be achieved by studying existing patient databases for Dutch founder mutations in SDHx genes.
    Type of Medium: Online Resource
    ISSN: 2049-3614
    URL: Article
    DOI: 10.1530/EC-21-0560
    Language: Unknown
    Publisher: Bioscientifica
    Publication Date: 2022
    detail.hit.zdb_id: 2668428-7
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  • 7
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    Online Resource
    Pituitary MRI in Cushing's disease ‐ an update
    Wiley ; 2022
    In:  Journal of Neuroendocrinology Vol. 34, No. 8 ( 2022-08)
    Details
    In: Journal of Neuroendocrinology, Wiley, Vol. 34, No. 8 ( 2022-08)
    Abstract: Pituitary MRI is essential in the diagnosis of ACTH‐dependent Cushing's syndrome, but its results are inconsistent. The demonstration of a sellar image compatible with the diagnosis of corticotropinoma varies from 40% to 90%, depending on the centre where the imaging is performed. In fact, the expertise of the neuroradiologist, use of a Tesla 3.0 MRI and choice of sequences are fundamental. The T2 and 3D gradient echo sequences after gadolinium injection are the most informative and today allow the detection of macro‐ and microadenomas in almost all cases. The diagnosis of numerous picoadenomas ( 〈 3–4 mm) is more challenging. The 2D and 3D spin echo or delayed T1 SE or FLAIR sequences after gadolinium can be used as a complement or to confirm a suspicious image. Characterization of corticotropinomas remains problematic. However, the correct assessment of so‐called incidentalomas by recognizing artifacts, anatomical variants and frequent Rathke's cleft cysts eliminates around 90% of the incidentalomas that mimic pituitary adenomas, as repetitively reported in the literature. For the time being, there is reason to believe that hybrid imaging combining PET and MRI such as 11C‐methionine PET coregistered with volumetric MRI will solve the diagnosis of corticotropinomas in the near future.
    Type of Medium: Online Resource
    ISSN: 0953-8194 , 1365-2826
    URL: Issue
    URL: Article
    DOI: 10.1111/jne.v34.8
    DOI: 10.1111/jne.13123
    Language: English
    Publisher: Wiley
    Publication Date: 2022
    detail.hit.zdb_id: 2007386-0
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  • 8
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    Online Resource
    T2-signal intensity, SSTR expression, and somatostatin analogs efficacy predict response to pasireotide in acromegaly
    Oxford University Press (OUP) ; 2020
    In:  European Journal of Endocrinology Vol. 182, No. 6 ( 2020-06), p. 595-605
    Details
    In: European Journal of Endocrinology, Oxford University Press (OUP), Vol. 182, No. 6 ( 2020-06), p. 595-605
    Abstract: T2-signal intensity and somatostatin (SST) receptor expression are recognized predictors of therapy response in acromegaly. We investigated the relationship between these predictors and the hormonal and tumoral responses to long-acting pasireotide (PAS-LAR) therapy, which were also compared with responsiveness to first-generation somatostatin receptor ligands (SRLs). Design The PAPE study is a cohort study. Methods We included 45 acromegaly patients initially receiving SRLs, followed by combination therapy with pegvisomant, and finally PAS-LAR. We assessed tumor volume reduction (≥25% from baseline), IGF-1 levels (expressed as the upper limit of normal), and T2-weighted MRI signal and SST receptor expression of the adenoma. Results Patients with significant tumor shrinkage during PAS-LAR showed higher IGF-1 levels during PAS-LAR (mean (S.D.): 1.36 (0.53) vs 0.93 (0.43), P  = 0.020), less IGF-1 reduction after first-generation SRLs (mean (S.D.): 0.55 (0.71) vs 1.25 (1.07), P  = 0.028), and lower SST 2 receptor expression (median (IQR): 2.0 (1.0–6.0) vs 12.0 (7.5–12.0), P  = 0.040). Overall, T2-signal intensity ratio was increased compared with baseline (mean (S.D.): 1.39 (0.56) vs 1.25 (0.52), P  = 0.017) and a higher T2-signal was associated with lower IGF-1 levels during PAS-LAR (β: −0.29, 95% CI: −0.56 to −0.01, P  = 0.045). A subset of PAS-LAR treated patients with increased T2-signal intensity achieved greater reduction of IGF-1 (mean (S.D.): 0.80 (0.60) vs 0.45 (0.39), P  = 0.016). Conclusions Patients unresponsive to SRLs with a lower SST 2 receptor expression are more prone to achieve tumor shrinkage during PAS-LAR. Surprisingly, tumor shrinkage is not accompanied by a biochemical response, which is accompanied with a higher T2-signal intensity.
    Type of Medium: Online Resource
    ISSN: 0804-4643 , 1479-683X
    URL: Article
    DOI: 10.1530/EJE-19-0840
    RVK:
    WA 15000
    Language: Unknown
    Publisher: Oxford University Press (OUP)
    Publication Date: 2020
    detail.hit.zdb_id: 1485160-X
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  • 9
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    Online Resource
    Prolactinomas resistant to standard doses of cabergoline: a multicenter study of 92 patients
    Oxford University Press (OUP) ; 2012
    In:  European Journal of Endocrinology Vol. 167, No. 5 ( 2012-11), p. 651-662
    Details
    In: European Journal of Endocrinology, Oxford University Press (OUP), Vol. 167, No. 5 ( 2012-11), p. 651-662
    Abstract: Dopamine agonist resistance in prolactinoma is an infrequent phenomenon. Doses of cabergoline (CAB) of up to 2.0 mg/week are usually effective in controlling prolactin (PRL) secretion and reducing tumor size in prolactinomas. The clinical presentation, management, and outcome of patients that are not well controlled by such commonly used doses of CAB-resistant patients are poorly understood. Design and methods A multicenter retrospective study was designed to collect a large series of resistant prolactinoma patients, defined by uncontrolled hyperprolactinemia on CAB ≥2.0 mg weekly. Results Ninety-two patients (50 F, 42 M) were analyzed. At diagnosis, most had macroprolactinomas (82.6%); males were significantly older than females ( P =0.0003) and presented with a more aggressive disease. A genetic basis was identified in 12 patients. Thirty-six patients (39.1%) received only medical therapy, most underwent surgery (60.9%, including multiple interventions in 10.9%), and 14.1% received postoperative radiotherapy. Eight patients developed late CAB resistance (8.7%). The median maximal weekly dose of CAB (CAB max/w ) was 3.5 mg (2.0–10.5). Despite a higher CAB max/w in patients treated with multimodal therapy ( P =0.003 vs exclusive pharmacological treatment), a debulking effect of surgery was shown in 14 patients, with a higher rate of PRL control ( P =0.006) and a significant reduction in CAB max/w ( P =0.001) postoperatively. At last follow-up (median 88 months), PRL normalization and tumor disappearance were achieved in 28 and 19.9% of the patients respectively, with no significant sex-related difference observed in CAB max/w or disease control. Mortality was 4.8%, with four patients developing aggressive tumors (4.3%) and three a pituitary carcinoma (3.3%). Conclusion CAB-resistant prolactinomas remain a serious concern. Surgical debulking, newer therapeutic strategies, and early diagnosis of genetic forms could help to improve their outcome.
    Type of Medium: Online Resource
    ISSN: 0804-4643 , 1479-683X
    URL: Article
    DOI: 10.1530/EJE-12-0236
    RVK:
    WA 15000
    Language: Unknown
    Publisher: Oxford University Press (OUP)
    Publication Date: 2012
    detail.hit.zdb_id: 1485160-X
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  • 10
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    Cardiovascular complications in pituitary gigantism (results of an international study)
    Bioscientifica ; 2018
    In:  Endocrine Abstracts ( 2018-10-12)
    Details
    In: Endocrine Abstracts, Bioscientifica, ( 2018-10-12)
    Type of Medium: Online Resource
    ISSN: 1479-6848
    URL: Article
    DOI: 10.1530/endoabs.57.021
    Language: Unknown
    Publisher: Bioscientifica
    Publication Date: 2018
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