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1.
Eur J Endocrinol ; 183(4): 369-379, 2020 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-32621582

RESUMO

Background: The '3PAs' syndrome, associating pituitary adenoma (PA) and pheochromocytoma/paraganglioma (PPGL), is sometimes associated with mutations in PPGL-predisposing genes, such as SDHx or MAX. In '3PAs' syndrome, PAs can occur before PPGL, suggesting a new gateway into SDHx/MAX-related diseases. Objective: To determine the SDHx/MAX mutation prevalence in patients with isolated PAs and characterize PAs of patients with SDHx/MAX mutations. Design: Genes involved in PAs (AIP/MEN1/CDKN1B) or PPGLs (SDHx/MAX) were sequenced in patients with isolated PAs. We then conducted a review of cases of PA in the setting of '3PAs' syndrome. Results: A total of 263 patients were recruited. Seven (likely) pathogenic variants were found in AIP, two in MEN1, two in SDHA, and one in SDHC. The prevalence of SDHx mutations reached 1.1% (3/263). Of 31 reported patients with PAs harboring SDHx/MAX mutations (28 published cases and 3 cases reported here), 6/31 (19%) developed PA before PPGL and 8/31 (26%) had isolated PA. The age of onset was later than in patients with AIP/MEN1 mutations. PAs were mainly macroprolactinomas and showed intracytoplasmic vacuoles seen on histopathology. Conclusions: We discovered SDHx mutations in patients bearing PA who had no familial or personal history of PPGL. However, the question of incidental association remains unresolved and data to determine the benefit of SDHx/MAX screening in these patients are lacking. We recommend that patients with isolated PA should be carefully examined for a family history of PPGLs. A family history of PPGL, as well as the presence of intracytoplasmic vacuoles in PA, requires SDHx/MAX genetic testing of patients.


Assuntos
Adenoma/genética , Mutação em Linhagem Germinativa , Neoplasias Hipofisárias/genética , Succinato Desidrogenase/genética , Adenoma/epidemiologia , Adenoma/patologia , Adolescente , Neoplasias das Glândulas Suprarrenais/epidemiologia , Neoplasias das Glândulas Suprarrenais/genética , Neoplasias das Glândulas Suprarrenais/patologia , Adulto , Idade de Início , Idoso , Fatores de Transcrição de Zíper de Leucina e Hélice-Alça-Hélix Básicos/genética , Criança , Análise Mutacional de DNA/métodos , Feminino , França/epidemiologia , Predisposição Genética para Doença , Testes Genéticos , Sequenciamento de Nucleotídeos em Larga Escala , Humanos , Isoenzimas/genética , Masculino , Pessoa de Meia-Idade , Paraganglioma/epidemiologia , Paraganglioma/genética , Paraganglioma/patologia , Feocromocitoma/epidemiologia , Feocromocitoma/genética , Feocromocitoma/patologia , Neoplasias Hipofisárias/epidemiologia , Neoplasias Hipofisárias/patologia , Prolactinoma/epidemiologia , Prolactinoma/genética , Prolactinoma/patologia , Subunidades Proteicas/genética , Estudos Retrospectivos , Adulto Jovem
3.
Pituitary ; 23(1): 3-8, 2020 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-31641970

RESUMO

Lactotroph adenomas, also called prolactinomas and prolactin-secreting adenomas, constitute nearly 80% of functioning pituitary tumors and about 30-50% of all adenomas in the clinical practice. Lactotroph adenomas occur in the general population at a prevalence of 45/100,000, are more common in women, but also involve men and children of both sexes. Most lactotroph adenomas are microadenomas occurring in reproductive-age women who present with oligo/amenorrhea, galactorrhea, and infertility. In men and elderly women, lactotroph adenomas are usually macroadenomas and are most commonly associated with symptoms of a tumoral mass, including headaches, neurologic defects, and visual loss. Although clinical and laboratory features may differ depending on patient's gender and age, the histopathology of the tumors is similar. Lactotroph adenomas are histologically classified into three subtypes: the common sparsely granulated lactotroph adenoma, and the rare densely granulated lactotroph adenoma and acidophilic stem cell adenoma. We will review the main pathological features of the lactotroph adenomas and some of their characteristics that may predict biological behavior and responsiveness to treatment.


Assuntos
Prolactinoma/epidemiologia , Feminino , Humanos , Masculino , Neoplasias Hipofisárias/epidemiologia , Prevalência
4.
Sultan Qaboos Univ Med J ; 19(2): e129-e134, 2019 May.
Artigo em Inglês | MEDLINE | ID: mdl-31538011

RESUMO

Objectives: This study aimed to evaluate the aetiologies of hyperprolactinaemia in the United Arab Emirates (UAE). Methods: This retrospective study used laboratory databases to identify all patients who underwent evaluation for prolactin at Tawam Hospital, Al Ain, UAE, between 2009 and 2015. Of those 2,280 patients, all patients with low or normal prolactin (n = 1,315) were excluded. Subsequently, charts of the remaining patients (n = 965) with hyperprolactinaemia were reviewed and those with incomplete work-ups or insufficient documentation of the hyperprolactinaemia's aetiology were excluded (n = 458). Results: A total of 507 patients were included in the study. The average age at prolactin evaluation was 36 ± 13.2 years and the majority (67.1%) of patients were female. The most common reasons for requesting prolactin were menstrual disorders (29.5%), infertility (18%), evaluation of sellar masses (14.3%), ruling out seizures (13.4 %) and monitoring while on psychiatric medications (8.7%). The most common causes of hyperprolactinaemia were prolactinoma (17%), transient hyperprolactinaemia (14.6%), drug-induced side effects (14.4%), polycystic ovarian syndrome (11.8%) and seizure disorder (7.7%). In females, common aetiologies were prolactinomas, transient and idiopathic hyperprolactinaemia, while sellar masses, seizures, chronic kidney disease and acute illnesses were common aetiologies of hyperprolactinaemia in males. The prolactin level varied between the different aetiologies and a level of >250 ng/mL was suggestive of macro-prolactinoma. Conclusion: A significant proportion of patients with hyperprolactinaemia have transient hyperprolactinaemia. Before further investigations are carried out, prolactin level assessment should be repeated, especially in patients with mild hyperprolactinaemia.


Assuntos
Hiperprolactinemia/etiologia , Adulto , Feminino , Humanos , Hiperprolactinemia/epidemiologia , Masculino , Pessoa de Meia-Idade , Prolactina/análise , Prolactina/sangue , Prolactinoma/complicações , Prolactinoma/epidemiologia , Estudos Retrospectivos , Centros de Atenção Terciária/organização & administração , Centros de Atenção Terciária/estatística & dados numéricos , Emirados Árabes Unidos/epidemiologia
5.
Endocrinol Metab Clin North Am ; 48(3): 569-581, 2019 09.
Artigo em Inglês | MEDLINE | ID: mdl-31345524

RESUMO

Pituitary adenomas are common. The impact of pituitary tumors on fertility are mainly caused by oversecretion and/or undersecretion of pituitary hormones or compression of pituitary stalk and normal pituitary tissue by the tumor. Diagnosing and managing pituitary tumors during pregnancy involve many challenges, including the effect of hormone excess or deficiency on pregnancy outcome, changes in the pituitary or pituitary-related hormones, changes in tumor size, and the impact of various treatments of pituitary tumors on maternal and fetal outcomes. This article discusses the diagnosis and treatment of patients with prolactinomas, acromegaly, Cushing disease, and other pituitary tumors during pregnancy.


Assuntos
Neoplasias Hipofisárias/diagnóstico , Neoplasias Hipofisárias/terapia , Prolactinoma/diagnóstico , Prolactinoma/terapia , Acromegalia/diagnóstico , Acromegalia/epidemiologia , Acromegalia/etiologia , Acromegalia/terapia , Feminino , Humanos , Neoplasias Hipofisárias/complicações , Neoplasias Hipofisárias/epidemiologia , Gravidez , Prolactinoma/complicações , Prolactinoma/epidemiologia
6.
Best Pract Res Clin Endocrinol Metab ; 33(2): 101290, 2019 04.
Artigo em Inglês | MEDLINE | ID: mdl-31326373

RESUMO

Prevalence and incidence of prolactinomas are approximately 50 per 100,000 and 3-5 new cases/100,000/year. The pathophysiological mechanism of hyperprolactinemia-induced gonadotropic failure involves kisspeptin neurons. Prolactinomas in males are larger, more invasive and less sensitive to dopamine agonists (DAs). Macroprolactin, responsible for pseudohyperprolactinemia is a frequent pitfall of prolactin assay. DAs still represent the primary therapy for most prolactinomas, but neurosurgery has regained interest, due to progress in surgical techniques and a high success rate in microprolactinoma, as well as to some underestimated side effects of long-term DA treatment, such as impulse control disorders or impaired quality of life. Recent data show that the suspected effects of DAs on cardiac valves in patients with prolactinomas are reassuring. Finally, temozolomide has emerged as a valuable treatment for rare cases of aggressive and malignant prolactinomas that do not respond to all other conventional treatments.


Assuntos
Neoplasias Hipofisárias/diagnóstico , Neoplasias Hipofisárias/epidemiologia , Neoplasias Hipofisárias/terapia , Prolactinoma/diagnóstico , Prolactinoma/epidemiologia , Prolactinoma/terapia , Agonistas de Dopamina/uso terapêutico , Feminino , Humanos , Hiperprolactinemia/diagnóstico , Hiperprolactinemia/epidemiologia , Hiperprolactinemia/etiologia , Hiperprolactinemia/terapia , Incidência , Kisspeptinas/metabolismo , Masculino , Neurônios/metabolismo , Neurônios/patologia , Neoplasias Hipofisárias/complicações , Prevalência , Prolactina/análise , Prolactina/sangue , Prolactinoma/complicações , Qualidade de Vida
7.
Neuroendocrinology ; 109(1): 20-27, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-30731464

RESUMO

Clinically relevant pituitary adenomas are present in about 1 per 1,000 of the general population and prolactinomas are by far the most common clinical subtype of pituitary adenomas. Usually prolactinomas affect premenopausal women and present with typical symptoms of menstrual disturbance and/or galactorrhea. They are generally managed with dopamine agonists to restore fertility and to control symptoms and tumor size. In a subset of prolactinomas, however, management remains challenging. Studies in recent years have identified the factors related to dopamine agonist resistance, such as male sex, genetic features, and aggressive tumor behavior. Certain other patient groups represent particular challenges for management, such as pediatric patients and pregnant women. Treatment with dopamine agonists is usually safe and effective, and adverse effects such as clinically relevant cardiac valvular complications and impulse control disorders may occur in isolated instances. A number of important disease characteristics of prolactinomas remain to be explained, such as the difference in sex prevalence before and after menopause, the higher prevalence of macroadenomas in older males, and the biochemical mechanisms of resistance to dopaminergic agonists.


Assuntos
Neoplasias Hipofisárias/epidemiologia , Neoplasias Hipofisárias/terapia , Prolactinoma/epidemiologia , Prolactinoma/terapia , Feminino , Humanos , Masculino
9.
Pituitary ; 22(2): 113-123, 2019 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-30637623

RESUMO

PURPOSE: To explore the clinical characteristics of pituitary adenomas in patients with MEN1 and to summarize treatment strategies for MEN1 in a Chinese population. METHODS: We retrospectively analyzed 54 MEN1 patients with pituitary adenomas diagnosed at Peking Union Medical College Hospital from March 2003 to January 2017. Clinical data, laboratory testing results, treatments of involved glands and treatment responses were collected and analyzed. RESULTS: The mean age at pituitary adenoma diagnosis was 53.9 ± 17.8. The patients initially consulted the Endocrinology, General Surgery and Neurosurgery departments, in descending frequency. The nonfunctioning adenoma, prolactinoma, GH-secreting adenoma, cosecreting adenoma, and ACTH-secreting adenoma subtypes accounted for 48.1%, 27.8%, 9.3%, 9.3% and 5.6% of the cases, respectively. The remission rate for prolactinomas was 46.2% (6/13) treated with bromocriptine. And the remission rates were 87.5% (7/8) and 100% (3/3) for GH-secreting adenomas and ACTH-secreting adenomas respectively achieved by transsphenoidal surgery. Nineteen (35.2%) patients with asymptomatic nonfunctioning pituitary adenomas showed no progression after a 35-month follow-up with close observation. Regarding treatment priority, patients with thymic carcinoid tumors received first-line surgery, 54% of the patients with enteropancreatic tumors had these tumors treated first, and 26% of all patients had their pituitary adenomas treated first. In acromegalic patients, pituitary lesions tended to be treated first (75%, p = 0.002). PHPT and adrenocortical adenomas can be managed with elective surgery. CONCLUSIONS: The treatment of MEN1 requires cooperation between multidisciplinary teams. Individualized treatment according to the severity of glandular involvement is needed. GH-secreting and ACTH-secreting pituitary adenomas require active treatment, while nonfunctioning pituitary adenomas can be observed closely.


Assuntos
Adenoma/epidemiologia , Neoplasia Endócrina Múltipla Tipo 1/epidemiologia , Neoplasias Hipofisárias/epidemiologia , Acromegalia/epidemiologia , Acromegalia/patologia , Acromegalia/cirurgia , Adenoma/patologia , Adenoma/cirurgia , Adulto , Idoso , China , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Neoplasia Endócrina Múltipla Tipo 1/patologia , Neoplasia Endócrina Múltipla Tipo 1/cirurgia , Hipófise/patologia , Neoplasias Hipofisárias/patologia , Neoplasias Hipofisárias/cirurgia , Prolactinoma/epidemiologia , Prolactinoma/patologia , Prolactinoma/cirurgia , Estudos Retrospectivos
10.
Endocrine ; 63(2): 323-331, 2019 02.
Artigo em Inglês | MEDLINE | ID: mdl-30267354

RESUMO

PURPOSE: Recent work supports the use of T2-weighted MRI intensity as a tool for treatment stratification in acromegaly. Our study aimed to establish if the pattern of T2 intensity could be a predictor of hormonal and/or tumoral response to dopamine agonists (DAs) in prolactinomas. METHODS: This was a retrospective study performed in two academic centers. We characterized the magnetic resonance T2-weighted aspect of prolactinomas (signal intensity and homogeneity in the whole tumors) before DA therapy and correlated this pattern to the prolactin (PRL) concentration at diagnosis and to hormonal and tumoral responses after 1 year of medical treatment. We separately analyzed a subgroup of prolactinomas visually very bright in more than 50% of the surface ("cystic" tumors). RESULTS: Out of 70 prolactinomas, 80% were T2 hyperintense and 40% were heterogeneous. At diagnosis, heterogeneous prolactinomas were more frequent in men (68% vs. 28.9%, p ≤ 0.011), larger (median area 304.5 mm2 vs. 56.5 mm2, p ≤ 0.021), taller (mean height 18.6 mm vs. 9.9 mm, p < 0.001), more secreting (median PRL ULN_area 23 µg/L/cm2 vs. 12.6 µg/L/cm2, p ≤ 0.032) and had poorer hormonal response to DA as compared with homogeneous prolactinomas. "Cystic" tumors were diagnosed almost exclusively in women and secreted less prolactin, but showed similar hormonal and tumoral response as "non-cystic" tumors. In homogeneous prolactinomas, the T2-weighted intensity ratio was correlated to prolactin secretion, although not significantly, and did not predict hormonal and tumoral response to DA. CONCLUSIONS: Our study confirms that hypo/isointense prolactinoma is a rare finding and suggests for the first time that the heterogeneity of prolactinoma T2 signal at diagnosis might be correlated with a different clinical behavior and could be used as a negative predictor factor of hormonal response to DA.


Assuntos
Agonistas de Dopamina/uso terapêutico , Imagem por Ressonância Magnética/métodos , Neoplasias Hipofisárias/diagnóstico , Neoplasias Hipofisárias/tratamento farmacológico , Prolactinoma/diagnóstico , Prolactinoma/tratamento farmacológico , Acromegalia/diagnóstico , Acromegalia/tratamento farmacológico , Acromegalia/epidemiologia , Adolescente , Adulto , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Neoplasias Hipofisárias/epidemiologia , Neoplasias Hipofisárias/patologia , Valor Preditivo dos Testes , Prognóstico , Prolactinoma/epidemiologia , Prolactinoma/patologia , Estudos Retrospectivos , Fatores de Risco , Resultado do Tratamento , Carga Tumoral , Adulto Jovem
11.
Endocrine ; 63(2): 332-340, 2019 02.
Artigo em Inglês | MEDLINE | ID: mdl-30269265

RESUMO

PURPOSE: Macroprolactinemia is characterized by predominance of macroprolactin molecules in circulation and generally has extra-pituitary origin. Macroprolactin is viewed as biologically inactive, therefore asymptomatic, and thus may not require any treatment or prolonged follow-up. In addition, data on prevalence of macroprolactinemia and its clinical manifestation are also rare. Therefore, the present study was aimed to find out prevalence of macroprolactinemia and its association, if any, with reproductive manifestations. MATERIAL AND METHODS: Macroprolactin was measured in 102 hyperprolactinemia cases (>100 ng/ml prolactin level), 135 physiological hyperprolactinemia cases (50 pregnant and 85 lactating females; >100 ng/ml prolactin level) and 24 controls. Poly ethylene glycol (PEG) precipitation method was carried out to screen macroprolactin. Prolactin recovery of <25% was considered overt macroprolactinemia. Detailed clinical data was recorded which included complete medical history, physical examination and hormone measurements besides CT/MRI for pituitary abnormalities. RESULTS: Prevalence of macroprolactinemia was 21.57% (22/102) in hyperprolactinemia (prolactin >100 ng/ml). There was no case of macroprolactinemia in physiological hyperprolactinemia, or healthy control females. Reproductive manifestations were present in 72.73% (16/22) macroprolactinemia cases, out of which macroprolactinemia was the sole cause of associated reproductive manifestations in 68.7% (11/16) cases. Reversal of reproductive dysfunction/s was observed in five cases with appropriate treatment for high macroprolactin. CONCLUSION: Macroprolactinemia prevalence was found to be 21.5%, out of which 72.73% cases had associated reproductive dysfunctions.


Assuntos
Hiperprolactinemia/epidemiologia , Hiperprolactinemia/fisiopatologia , Infertilidade/epidemiologia , Distúrbios Menstruais/epidemiologia , Reprodução/fisiologia , Disfunções Sexuais Fisiológicas/epidemiologia , Disfunções Sexuais Psicogênicas/epidemiologia , Aborto Espontâneo/sangue , Aborto Espontâneo/epidemiologia , Aborto Espontâneo/etiologia , Adulto , Estudos de Casos e Controles , Disfunção Erétil/sangue , Disfunção Erétil/epidemiologia , Disfunção Erétil/etiologia , Feminino , Humanos , Hiperprolactinemia/sangue , Hiperprolactinemia/etiologia , Infertilidade/sangue , Infertilidade/etiologia , Libido/fisiologia , Masculino , Distúrbios Menstruais/sangue , Distúrbios Menstruais/etiologia , Pessoa de Meia-Idade , Neoplasias Hipofisárias/complicações , Neoplasias Hipofisárias/epidemiologia , Gravidez , Prevalência , Prolactina/sangue , Prolactinoma/complicações , Prolactinoma/epidemiologia , Fatores de Risco , Disfunções Sexuais Fisiológicas/sangue , Disfunções Sexuais Fisiológicas/etiologia , Disfunções Sexuais Psicogênicas/sangue , Disfunções Sexuais Psicogênicas/etiologia , Adulto Jovem
12.
Genet Test Mol Biomarkers ; 22(12): 702-708, 2018 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-30461320

RESUMO

Aims: Aryl hydrocarbon receptor-interacting protein (AIP) gene mutations have long been associated with apparently sporadic pituitary adenomas (PAs) with a prevalence range of 0-12%. The aim of this study was to evaluate the frequency of germline AIP variations in a large cohort of apparently sporadic PAs diagnosed before the age of 40 years, who did not exhibit hypercalcemia and/or MEN1 syndrome components during long-term follow-up. Materials and Methods: A total of 97 patients, diagnosed with functional PAs ≤40 years old, composed of somatotropinoma (n = 55), prolactinoma (n = 25), and corticotrophinoma (n = 17), were recruited for this study. Fifty-one of these patients [somatotropinoma (n = 30), prolactinoma (n = 15), and corticotrophinoma (n = 11)] were previously reported as AIP mutation-negative by Sanger sequencing. The entire coding sequence of the AIP gene, along with exon/intron boundaries and the untranslated regions of 41 newly recruited patients, were sequenced for germline variations. In addition, all patients were subjected to multiplex ligation-dependent probe amplification to detect copy number variations in the AIP gene. Results: The AIP c.911G>A: p.Arg304Gln (rs104894190) variant was detected in only two patients with functional PA: one with somatotropinoma [in 1/55 (1.8%)] and one with prolactinoma [in 1/25 (4%)]. None of the corticotrophinomas revealed AIP gene alterations. Thus, the overall prevalence of AIP variation was 2.1% in our cohort. Conclusions: Germline AIP gene variations among Turkish patients with apparently sporadic PAs are relatively rare among patients ≤40 years old. None of the patients in our cohort revealed any obviously pathogenic AIP variants.


Assuntos
Adenoma Hipofisário Secretor de ACT/genética , Adenoma/genética , Mutação em Linhagem Germinativa , Adenoma Hipofisário Secretor de Hormônio do Crescimento/genética , Peptídeos e Proteínas de Sinalização Intracelular/genética , Polimorfismo de Nucleotídeo Único , Prolactinoma/genética , Adenoma Hipofisário Secretor de ACT/diagnóstico por imagem , Adenoma Hipofisário Secretor de ACT/epidemiologia , Acromegalia/etiologia , Acromegalia/genética , Adenoma/diagnóstico por imagem , Adenoma/epidemiologia , Adolescente , Adulto , Idade de Início , Feminino , Testes Genéticos , Adenoma Hipofisário Secretor de Hormônio do Crescimento/diagnóstico por imagem , Adenoma Hipofisário Secretor de Hormônio do Crescimento/epidemiologia , Heterozigoto , Humanos , Imagem por Ressonância Magnética , Masculino , Pessoa de Meia-Idade , Neuroimagem , Prolactinoma/diagnóstico por imagem , Prolactinoma/epidemiologia , Turquia/epidemiologia , Adulto Jovem
13.
Endocrine ; 62(2): 361-370, 2018 11.
Artigo em Inglês | MEDLINE | ID: mdl-30171518

RESUMO

PURPOSE: To review incidence, treatment and outcome of pediatric pituitary adenomas (PAs). METHODS: A follow-up study patients with the age of ≤19 years old who were treated from 1995 to 2015 in Mexico. RESULTS: Out of 1244 diagnosed PA, 43 patients were children (35 females, 8 males) with a mean age of 17.2 years. The majority were macroadenomas (70%) with prolactinomas (PRL) dominating (63%) followed by non-functioning adenomas (21%). In total, 40% were diagnosed as invasive. Growth hormone (GH) secreting adenomas, adrenocorticotropic hormone secreting and mixed GH-PRL secreting were rare. The treatment modalities were dopamine agonists and surgery. The average treatment time was 44 months with an average follow-up period of 104 months. Sixty-eight percent (27/40) of the patients had complete response after long time follow-up. Thirty-one percent did not respond to treatment whereof three patients died due to advanced disease and late intervention. The principal causes for treatment failure were treatment resistance, late intervention and poor patient compliance. CONCLUSIONS: Sixty eight percent had complete treatment response without any sign of disease, while ~31% did not respond to treatment or did not comply to follow up/treatment. Optimized early diagnose, treatment methods with early intervention, long time follow-up and with better measures for patient compliance should improve outcomes.


Assuntos
Adenoma , Neoplasias Hipofisárias , Adenoma/diagnóstico , Adenoma/epidemiologia , Adenoma/patologia , Adenoma/terapia , Adolescente , Idade de Início , Criança , Feminino , Seguimentos , Humanos , Masculino , México/epidemiologia , Neoplasias Hipofisárias/diagnóstico , Neoplasias Hipofisárias/epidemiologia , Neoplasias Hipofisárias/patologia , Neoplasias Hipofisárias/terapia , Prolactinoma/epidemiologia , Prolactinoma/patologia , Prolactinoma/terapia
14.
Hormones (Athens) ; 17(2): 261-267, 2018 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-29858852

RESUMO

PURPOSE: Pituitary tumours are slowly progressing tumours, mostly benign, with a reported global prevalence of 16.7% (22.5% in radiologic studies and 14.4% in autopsy studies). Clinical and epidemiological data on pituitary adenomas in Saudi Arabia are lacking. We aimed to utilise our database variables to determine clinical and epidemiological characteristics as well as treatment outcomes of pituitary tumours among Saudi patients. METHODS: This retrospective study was conducted in King Fahad Medical City, Riyadh, Saudi Arabia, in patients with pituitary tumours. Data were collected between 2006 and 2017. RESULTS: Overall, 284 patients (females: 38.1 ± 13.9 years; males: 44.1 ± 15.4 years) with pituitary tumours were included. Common pituitary tumours were prolactin-secreting adenomas (45%), non-functioning pituitary adenomas (NFPAs: 35.6%), growth hormone (GH)-secreting adenomas (10.6%), craniopharyngiomas (7%), and adrenocorticotropic hormone (ACTH)-secreting adenomas (1.8%). Prolactin-secreting adenomas were more frequently microadenomas and were common among females. Headaches and visual symptoms occurred commonly in NFPA patients (62.4 and 45.5%, respectively) than in those with prolactin-secreting adenomas (56.3 and 32.8%, respectively) or GH-secreting adenomas (40 and 16.6%, respectively). Medical treatment was the mainstay for prolactin-secreting adenoma patients (69%). Pituitary surgery was the primary therapy in NFPA patients (43.6%) and GH-secreting adenomas (86.7%). CONCLUSION: This study identified the pattern of pituitary tumours in Saudi patients and management strategies. Further, the study highlights the need for a nationwide registry to improve surveillance and physicians' knowledge in Saudi Arabia.


Assuntos
Adenoma/epidemiologia , Neoplasias Hipofisárias/epidemiologia , Prolactinoma/epidemiologia , Adenoma/tratamento farmacológico , Adenoma/fisiopatologia , Adenoma/cirurgia , Adulto , Idoso , Feminino , Hospitais de Ensino/estatística & dados numéricos , Humanos , Masculino , Pessoa de Meia-Idade , Neoplasias Hipofisárias/tratamento farmacológico , Neoplasias Hipofisárias/fisiopatologia , Neoplasias Hipofisárias/cirurgia , Prolactinoma/tratamento farmacológico , Prolactinoma/fisiopatologia , Prolactinoma/cirurgia , Estudos Retrospectivos , Arábia Saudita/epidemiologia , Centros de Atenção Terciária/estatística & dados numéricos
15.
Horm Res Paediatr ; 89(3): 157-165, 2018.
Artigo em Inglês | MEDLINE | ID: mdl-29455199

RESUMO

BACKGROUND: Paediatric prolactinomas are rare. The aim of this study was to investigate the clinical features and outcome of paediatric patients with prolactinomas. METHODS: In this single-centre retrospective analysis, clinical, biochemical, and radiological features of all paediatric patients with pituitary adenomas diagnosed between 2000 and 2016 were evaluated. RESULTS: Among 21 patients with pituitary adenomas, 12 patients with prolactinomas (median age 14.2 years, range 11-16.6 years, 8 females, 4 males) were identified (7 macro- and 5 microprolactinomas). The most common clinical symptoms were headaches (67%) and pubertal delay (67%). All patients with macroprolactinomas with prolactin concentrations >10,000 mU/L had at least 1 pituitary hormone deficiency. Cabergoline as first-line treatment (n = 11, median follow-up of 37 months, range 12-89 months) induced normoprolactinemia (n = 8), reduced the mean tumour volume by 80%, and ameliorated headaches (p = 0.016) and pubertal delay (p = 0.031), whereas intermittent moderate side effects occurred in 55%. CONCLUSION: Adolescents with headaches and pubertal delay should be investigated for prolactinomas. Treatment with cabergoline is well tolerated and effective in reducing clinical symptoms and prolactin concentrations was well as inducing tumour shrinkage. Further clinical prospective studies are needed to standardize paediatric treatment modalities.


Assuntos
Neoplasias Hipofisárias/epidemiologia , Prolactinoma/epidemiologia , Adolescente , Antineoplásicos/uso terapêutico , Cabergolina , Criança , Ergolinas/uso terapêutico , Feminino , Alemanha/epidemiologia , Humanos , Masculino , Neoplasias Hipofisárias/tratamento farmacológico , Prolactinoma/tratamento farmacológico , Estudos Retrospectivos
16.
Endocrine ; 59(1): 50-61, 2018 01.
Artigo em Inglês | MEDLINE | ID: mdl-29043560

RESUMO

PURPOSE: Dopamine agonists (DAs) are recommended as first-line treatment for patients with hyperprolactinemia. Generally, it is accepted that patients with hyperprolactinemia do not need lifelong medication, but the optimal timing for DA withdrawal has not been determined. The aim of this systematic review and meta-analysis is to assess the impact of DA withdrawal on the clinical outcomes of patients with hyperprolactinemia, and to explore possible factors affecting successful DA withdrawal. METHODS: The databases of PubMed, Cochrane and EMBASE were searched up to May 2016. RESULTS: The proportion of patients with persisting normoprolactinemia after DA withdrawal reached 36.6% in a random effects model (95% CI, 29.4-44.2%; I-squared: 82.5%). Data of stratified analysis showed that the success rate of drug withdrawal was high in patients using cabergoline (CAB) as the only treatment (41.2%; 95% CI 32.3-50.4%) and those using CAB over 24 months (48.7%; 95% CI 38.9-58.5%), especially in patients with idiopathic hyperprolactinemia (73.2%; 95% CI 55.6-87.7%). In addition, patients who received a low maintenance dose of CAB, and had a significant reduction in tumor size (over 50%) before withdrawal, were more likely to achieve success (51.5 and 49.4%, respectively). CONCLUSION: The success rate of DA withdrawal has increased in recent years. Further, the success rate of CAB withdrawal was higher than that of bromocriptine, especially in patients with a duration of treatment longer than 24 months. Conclusively, the probability of success was higher in patients who received low-dose CAB maintenance treatment and those who achieved a significant reduction in tumor size before withdrawal.


Assuntos
Agonistas de Dopamina/administração & dosagem , Hiperprolactinemia/tratamento farmacológico , Suspensão de Tratamento , Agonistas de Dopamina/efeitos adversos , Esquema de Medicação , Humanos , Hiperprolactinemia/epidemiologia , Neoplasias Hipofisárias/complicações , Neoplasias Hipofisárias/tratamento farmacológico , Neoplasias Hipofisárias/epidemiologia , Prolactinoma/complicações , Prolactinoma/tratamento farmacológico , Prolactinoma/epidemiologia , Fatores de Tempo
17.
Clin Endocrinol (Oxf) ; 88(1): 71-76, 2018 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-29044586

RESUMO

OBJECTIVE: To investigate whether the risk of incident cardiovascular disease (CVD) is increased in patients with prolactinoma. DESIGN: Population-based, retrospective, open-cohort study using The Health Improvement Network (THIN) database. PATIENTS: A total of 2233 patients with prolactinoma and 10 355 matched controls (1:5 ratio) from UK General Practices contributing to THIN were included. Sex, age, body mass index and smoking status were used as matching parameters. The primary outcome was any incident CVD, defined by Read codes suggesting myocardial infarction, angina pectoris, stroke, transient ischaemic attack or heart failure. Sex-specific-adjusted incidence rate ratios (aIRRs) were calculated with Poisson regression, using clinically relevant parameters as model covariates. Sensitivity analyses were performed to check whether a change in the initial assumptions could have an impact on the findings. RESULTS: During the 6-year observation period, the composite CVD outcome was recorded in 54 patients with prolactinoma and 180 "nonexposed" individuals. The incidence rate was 1.8 and 14.8 per 1000 person-years for the females and males with prolactinoma, respectively. The aIRRs for CVD were estimated at 0.99 [95% confidence interval (CI): 0.61-1.61, P = .968)] in female patients and 1.94 (95% CI: 1.29-2.91, P = .001) in male patients. These findings remained robust in sensitivity analyses restricting to patients with documented record of dopamine agonist treatment and those with newly diagnosed prolactinoma. CONCLUSIONS: In contrast to females, men with prolactinoma have increased risk for incident CVD; the aetiology of this gender-specific finding remains to be elucidated.


Assuntos
Doenças Cardiovasculares/etiologia , Prolactinoma/complicações , Estudos de Casos e Controles , Feminino , Humanos , Masculino , Prolactinoma/epidemiologia , Estudos Retrospectivos , Fatores de Risco , Sensibilidade e Especificidade , Fatores Sexuais
18.
Endocrine ; 59(1): 72-81, 2018 01.
Artigo em Inglês | MEDLINE | ID: mdl-29067608

RESUMO

PURPOSE: We report the efficacy and safety of transsphenoidal microsurgery in a large and homogeneous cohort of patients with pituitary adenomas (PAs) treated at a single Institute by a single neurosurgeon. METHODS: A total of 2145 consecutive patients undergoing first surgery for a PA were included: 795 (37.1%) had a nonfunctioning pituitary adenoma (NFPA), 595 (27.7%) acromegaly, 496 (23.1%) Cushing's disease, 208 (9.7%) a PRL-secreting adenoma, and 51 patients (2.4%) a TSH-secreting adenoma. Remission was achieved when strict hormonal and radiological criteria were met. RESULTS: Early surgical remission was achieved in 66% of acromegalic patients, 79.6% of patients with Cushing's disease, 64.4% of prolactinomas, 74.5% of patients with a TSH-secreting adenoma, and 66.9% of NFPAs. The mean (±SE) follow-up was 60.1 ± 1.3 months. The recurrence-free survival at 10 years was 78.2% in acromegalic patients, 68.1% in prolactinomas, 74.3% in Cushing's disease, 70.3% in TSH-secreting adenomas, and 75.3% in NFPAs. Preoperative hypoadrenalism recovered in 35.3%, hypogonadism in 43.3% and hypothyroidism in 37.4% of patients with impaired function before surgery. The mortality rate was 0.2% and major morbidity 2.1%. New onset hypoadrenalism occurred after surgery in 2.5% of patients at risk, hypogonadism in 4.1%, and hypothyroidism in 1.8%. Permanent diabetes insipidus (DI) occurred in 0.9% of patients. CONCLUSIONS: In experienced hands, transsphenoidal microsurgery for PAs achieves remission in most patients with a low complication rate. Pituitary function is preserved in most cases and can recover in more than one-third of patients with preoperative hypopituitarism.


Assuntos
Adenoma/cirurgia , Microcirurgia/métodos , Procedimentos Neurocirúrgicos/métodos , Neoplasias Hipofisárias/cirurgia , Acromegalia/epidemiologia , Acromegalia/cirurgia , Adenoma/epidemiologia , Adolescente , Adulto , Idoso , Criança , Feminino , Humanos , Masculino , Microcirurgia/estatística & dados numéricos , Pessoa de Meia-Idade , Neurocirurgiões , Procedimentos Neurocirúrgicos/estatística & dados numéricos , Neoplasias Hipofisárias/epidemiologia , Prolactinoma/epidemiologia , Prolactinoma/cirurgia , Estudos Retrospectivos , Resultado do Tratamento , Adulto Jovem
19.
Endocrine ; 59(1): 16-29, 2018 01.
Artigo em Inglês | MEDLINE | ID: mdl-29177641

RESUMO

Prolactinomas are the most common secretory pituitary adenoma. They typically occur in women in the 3rd-6th decade of life and rarely in the pediatric population or after menopause. Most women present with irregular menses and/or infertility. Dopamine (DA) agonists, used in their treatment, are safe during pregnancy, but in most cases are discontinued at conception with close monitoring for signs or symptoms of tumor growth. Breastfeeding is safe postpartum, provided there was no significant growth during pregnancy. Some women will experience normalization of prolactin levels postpartum. Menopause may also decrease prolactin levels and even those with macroprolactinomas may consider discontinuing their DA agonist with close follow-up. Prolactinomas may be associated with decreased quality of life scores in women, and play a role in bone health and cardiovascular risk factors. This review discusses the current literature and clinical understanding of prolactinomas throughout the entirety of the female life cycle.


Assuntos
Envelhecimento/fisiologia , Neoplasias Hipofisárias , Prolactinoma , Adulto , Criança , Feminino , Humanos , Menopausa/sangue , Menopausa/fisiologia , Hipófise/embriologia , Neoplasias Hipofisárias/diagnóstico , Neoplasias Hipofisárias/epidemiologia , Gravidez , Complicações Neoplásicas na Gravidez/epidemiologia , Complicações Neoplásicas na Gravidez/etiologia , Prolactina/sangue , Prolactinoma/diagnóstico , Prolactinoma/epidemiologia , Qualidade de Vida , Fatores de Risco
20.
Obstet Gynecol ; 129(1): 185-194, 2017 01.
Artigo em Inglês | MEDLINE | ID: mdl-27926659

RESUMO

OBJECTIVE: To examine the monitoring, management, and outcomes of pituitary tumors in pregnancy. METHODS: A national, prospective, observational, population-based case series study was conducted in all U.K. consultant-led obstetric units over 3 years using the U.K. Obstetric Surveillance System. To evaluate rates of adverse pregnancy outcomes, women with a macroprolactinoma (10 mm or greater) or nonfunctioning pituitary adenoma, diagnosed before or during pregnancy, were compared with two comparison groups: 1) a U.K. Obstetric Surveillance System cohort with singleton (n=2,205) or twin (n=27) pregnancy; and 2) data from the Office of National Statistics (n=2,703,102). Main outcome measures were the incidence, management, and frequency of adverse maternal and offspring outcomes of pituitary tumors in pregnancy. RESULTS: There were 71 confirmed cases of pituitary tumors in pregnancy (49 macrolactinoma, 16 nonfunctioning adenomas, three acromegaly, three Cushing's disease). The women with pituitary tumors were 4 years older than comparison women (P<.001). None of the nine women treated with surgery or radiotherapy before pregnancy had symptomatic tumor expansion. This occurred in 6 of 40 women with macroprolactinomas and one of seven nonfunctioning adenomas diagnosed before conception and in three of five women with nonfunctioning adenomas diagnosed in pregnancy. Two women had pituitary apoplexy, both of whom also had symptoms of expansion of tumor or surrounding pituitary tissue. To within the level of accuracy possible, there was no evidence that pituitary tumors were associated with adverse pregnancy outcomes (pregnancy-induced hypertension, preeclampsia, preterm labor, stillbirth). Women with nonfunctioning adenomas were more likely to have cesarean delivery compared with women in a control group (relative risk 2.06, confidence interval 1.26-3.36, P=.035). CONCLUSION: The majority of women with macroprolactinomas and nonfunctioning adenomas have good pregnancy outcomes. Nonfunctioning pituitary adenomas occur more commonly in pregnancy than previously thought and can present de novo with symptoms of pituitary expansion in pregnancy.


Assuntos
Adenoma/epidemiologia , Neoplasias Hipofisárias/epidemiologia , Complicações Neoplásicas na Gravidez/epidemiologia , Prolactinoma/epidemiologia , Adenoma/complicações , Adenoma/patologia , Adenoma/terapia , Adulto , Amenorreia/etiologia , Antineoplásicos/uso terapêutico , Bromocriptina/uso terapêutico , Cabergolina , Estudos de Casos e Controles , Cesárea/estatística & dados numéricos , Agonistas de Dopamina/uso terapêutico , Ergolinas/uso terapêutico , Feminino , Galactorreia/etiologia , Humanos , Incidência , Neoplasias Hipofisárias/complicações , Neoplasias Hipofisárias/patologia , Neoplasias Hipofisárias/terapia , Pré-Eclâmpsia/epidemiologia , Cuidado Pré-Concepcional , Gravidez , Complicações Neoplásicas na Gravidez/patologia , Complicações Neoplásicas na Gravidez/terapia , Nascimento Prematuro/epidemiologia , Prolactinoma/complicações , Prolactinoma/patologia , Prolactinoma/terapia , Estudos Prospectivos , Natimorto/epidemiologia , Reino Unido/epidemiologia , Transtornos da Visão/etiologia , Adulto Jovem
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