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1.
Artigo em Inglês | MEDLINE | ID: mdl-39077973

RESUMO

Nerve sheath myxomas are extremely rare myxoid peripheral nerve sheath tumors with a predilection for the distal extremities, particularly common in the fingers and knees. Here, we report a 60-year-old male patient with a subconjunctival epibulbar nerve sheath myxoma, which was clinically diagnosed as an orbital fat prolapse. The lesion was successfully debulked without clinical recurrence over more than 3 years. To our knowledge, this is the first case with subconjunctival presentation and fourth orbital reported case.

2.
J Clin Endocrinol Metab ; 106(6): 1783-1792, 2021 05 13.
Artigo em Inglês | MEDLINE | ID: mdl-33544833

RESUMO

CONTEXT: Transsphenoidal surgery (TSS) is the primary treatment of choice in acromegaly. It is important to identify patients in whom surgical cure is not attainable at an early stage, both to inform patients on expected treatment outcome and to select those who are more likely to need additional therapy. OBJECTIVE: To identify predictors for remission after TSS in acromegaly. METHODS: Large multicenter study with retrospective data collection from 3 tertiary neurosurgical referral centers in The Netherlands. We analyzed clinical data since 2000 from 3 cohorts (Groningen, Nijmegen, and Rotterdam, total n = 282). Multivariate regression models were used to identify predictors of early biochemical remission (12 weeks to 1 year postoperatively) according to the 2010 consensus criteria, long-term remission (age- and sex-normalized insulin-like growth factor 1 [IGF-1] and the absence of postoperative treatment until last follow-up), and relative IGF-1 and growth hormone [GH] reduction. RESULTS: A larger maximum tumor diameter (odds ratio [OR] 0.91, 95% CI 0.87-0.96, P ≤ .0001) was associated with a lower chance of early biochemical remission. A larger maximum tumor diameter (OR 0.93, 95% CI 0.89-0.97, P = .0022) and a higher random GH concentration at diagnosis (OR 0.98, 95% CI 0.96-0.99, P = .0053) were associated with a lower chance of long-term remission. CONCLUSION: Maximum tumor diameter and random GH concentration at diagnosis are the best predictors for remission after TSS in acromegaly.


Assuntos
Acromegalia/diagnóstico , Acromegalia/cirurgia , Procedimentos Neurocirúrgicos , Acromegalia/epidemiologia , Acromegalia/metabolismo , Adenoma/diagnóstico , Adenoma/epidemiologia , Adenoma/metabolismo , Adenoma/cirurgia , Adulto , Estudos de Coortes , Feminino , Adenoma Hipofisário Secretor de Hormônio do Crescimento/diagnóstico , Adenoma Hipofisário Secretor de Hormônio do Crescimento/epidemiologia , Adenoma Hipofisário Secretor de Hormônio do Crescimento/metabolismo , Adenoma Hipofisário Secretor de Hormônio do Crescimento/cirurgia , Humanos , Masculino , Pessoa de Meia-Idade , Países Baixos/epidemiologia , Procedimentos Neurocirúrgicos/métodos , Procedimentos Neurocirúrgicos/estatística & dados numéricos , Período Pós-Operatório , Prognóstico , Indução de Remissão/métodos , Estudos Retrospectivos , Osso Esfenoide/cirurgia , Resultado do Tratamento
3.
J Clin Endocrinol Metab ; 105(9)2020 09 01.
Artigo em Inglês | MEDLINE | ID: mdl-32589751

RESUMO

CONTEXT: 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 (>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 (ß = 0.90, standard error (SE) = 0.02, P ≤ .0001 and ß â€…= 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.


Assuntos
Acromegalia/tratamento farmacológico , Biomarcadores Farmacológicos , Modelos Teóricos , Receptores de Somatostatina/agonistas , Somatostatina/análogos & derivados , Acromegalia/sangue , Acromegalia/diagnóstico , Adulto , Biomarcadores/análise , Biomarcadores/metabolismo , Biomarcadores Farmacológicos/análise , Biomarcadores Farmacológicos/metabolismo , Estudos de Coortes , Europa (Continente) , Feminino , Hormônio do Crescimento Humano/sangue , Humanos , Fator de Crescimento Insulin-Like I/metabolismo , Ligantes , Masculino , Pessoa de Meia-Idade , Análise Multivariada , Octreotida/uso terapêutico , Peptídeos Cíclicos/uso terapêutico , Prognóstico , Estudos Retrospectivos , Somatostatina/uso terapêutico , Resultado do Tratamento
4.
Arch Endocrinol Metab ; 63(6): 646-652, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-31939490

RESUMO

Advances in combination medical treatment have offer new perspectives for acromegaly patients with persistent disease activity despite receiving the available medical monotherapies. The outcomes of combination medical treatment may reflect both additive and synergistic effects. This review focuses on combination medical treatment and its current position in acromegaly, based on clinical studies evaluating the efficacy and safety of combined medical treatment(s) and our own experiences with combination therapy. Arch Endocrinol Metab. 2019;63(6):646-52.


Assuntos
Agonistas de Dopamina/administração & dosagem , Hormônio do Crescimento Humano/análogos & derivados , Receptores de Somatostatina/administração & dosagem , Receptores da Somatotropina/antagonistas & inibidores , Somatostatina/análogos & derivados , Acromegalia/tratamento farmacológico , Quimioterapia Combinada , Hormônio do Crescimento Humano/administração & dosagem , Humanos , Qualidade de Vida , Somatostatina/administração & dosagem
5.
Eur J Endocrinol ; 181(2): K21-K27, 2019 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-31167168

RESUMO

Prolactinomas are the most commonly encountered pituitary adenomas in the clinical setting. While most can be controlled by dopamine agonists, a subset of prolactinomas are dopamine-resistant and very aggressive. In such tumors, the treatment of choice is neurosurgery and radiotherapy, with or without temozolomide. Here, we report a patient with an highly aggressive, dopamine-resistant prolactinoma, who only achieved biochemical and tumor control during pasireotide long-acting release (PAS-LAR) therapy, a second-generation somatostatin receptor ligand (SRL). Interestingly, cystic degeneration, tumor cell necrosis or both was observed after PAS-LAR administration suggesting an antitumor effect. This case shows that PAS-LAR therapy holds clinical potential in selective aggressive, dopamine-resistant prolactinomas that express somatostatin (SST) receptor subtype 5 and appears to be a potential new treatment option before starting temozolomide. In addition, PAS-LAR therapy may induce cystic degeneration, tumor cell necrosis or both in prolactinomas.


Assuntos
Adenoma/tratamento farmacológico , Agonistas de Dopamina/administração & dosagem , Resistencia a Medicamentos Antineoplásicos/efeitos dos fármacos , Neoplasias Hipofisárias/tratamento farmacológico , Prolactinoma/tratamento farmacológico , Somatostatina/análogos & derivados , Adenoma/diagnóstico por imagem , Resistencia a Medicamentos Antineoplásicos/fisiologia , Feminino , Hormônios/administração & dosagem , Humanos , Pessoa de Meia-Idade , Invasividade Neoplásica/diagnóstico por imagem , Neoplasias Hipofisárias/diagnóstico por imagem , Prolactinoma/diagnóstico por imagem , Somatostatina/administração & dosagem , Resultado do Tratamento , Carga Tumoral/efeitos dos fármacos , Carga Tumoral/fisiologia
6.
Arch. endocrinol. metab. (Online) ; 63(6): 646-652, Nov.-Dec. 2019.
Artigo em Inglês | LILACS | ID: biblio-1055016

RESUMO

ABSTRACT Advances in combination medical treatment have offer new perspectives for acromegaly patients with persistent disease activity despite receiving the available medical monotherapies. The outcomes of combination medical treatment may reflect both additive and synergistic effects. This review focuses on combination medical treatment and its current position in acromegaly, based on clinical studies evaluating the efficacy and safety of combined medical treatment(s) and our own experiences with combination therapy. Arch Endocrinol Metab. 2019;63(6):646-52


Assuntos
Humanos , Somatostatina/análogos & derivados , Receptores de Somatostatina/administração & dosagem , Receptores de Somatostatina/antagonistas & inibidores , Agonistas de Dopamina/administração & dosagem , Hormônio do Crescimento Humano/análogos & derivados , Qualidade de Vida , Acromegalia/tratamento farmacológico , Somatostatina/administração & dosagem , Hormônio do Crescimento Humano/administração & dosagem , Quimioterapia Combinada
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