Pituitary最新文献

{"title":"Genetic profiling of synchronous pituitary corticotroph adenomas.","authors":"Dongyun Zhang, Karen Tsai, Cristian Santana, Keanu Javaherian, Matthew Lee, Marvin Bergsneider, Won Kim, Marilene B Wang, Harry V Vinters, Weihong Yan, Anthony P Heaney","doi":"10.1007/s11102-025-01549-6","DOIUrl":"https://doi.org/10.1007/s11102-025-01549-6","url":null,"abstract":"<p><strong>Purpose: </strong>Double or multiple pituitary adenomas account for only 1.6-3.3% of all corticotroph tumors. We sought to better understand the underlying molecular pathogenesis of two distinct corticotroph adenomas in a 43-year-old female.</p><p><strong>Methods: </strong>Two histopathologically confirmed corticotroph adenomas were submitted for whole-exome sequencing along with a matched blood sample. The functional effects of identified variants of uncertain significance on corticotroph tumor pro-opiomelanocortin transcription and proliferation were characterized.</p><p><strong>Results: </strong>WES demonstrated a loss-of-function variant in the G-protein coupled receptor 162 [GPR162 (R218*)] in the right corticotroph tumor, and a novel missense variant in ubiquitin specific peptidase 8 [USP8 (P681Q)] in the left tumor. Compared to wild-type GPR162 which potently suppressed POMC transcription, the stop-gain variant (R218*) exhibited reduced inhibitory effect. The novel USP8 variant (P681Q) found in the contra-lateral tumor led to increased POMC transcription although weaker than the well characterized hotspot variant S718P, and did not affect EGFR ubiquitin. Interestingly, the patient also had a germline variant in the 21-alpha-hydroxylase gene (CYP21A2 p.A392T) though without clinical features of congenital adrenal hyperplasia.</p><p><strong>Conclusion: </strong>We report, for the first time, the genetic profiles of a patient with dual pituitary corticotroph tumors, identifying a stop-gain variant in GPR162 in one tumor and a novel USP8 variant (S718P) in the other. While both somatic variants increased POMC expression, only GPR162 R218* affected proliferation. We hypothesize that alterations in adrenal steroidogenesis due to the CYP21A1 mutation may have reduced negative feedback on corticotroph cells and acted in a permissive way to facilitate corticotroph tumorigenesis.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"77"},"PeriodicalIF":3.3,"publicationDate":"2025-06-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144369147","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"ACRODAT and SAGIT for the assessment of disease activity in acromegaly: a multicenter study of the Veneto region in Italy.","authors":"Francesca Dassie, Filippo Ceccato, Paola Sartorato, Virginia Trevisan, Maria Vittoria Davì, Silvia Camerini, Ernesto De Menis, Carla Scaroni, Pietro Maffei","doi":"10.1007/s11102-025-01543-y","DOIUrl":"https://doi.org/10.1007/s11102-025-01543-y","url":null,"abstract":"","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"76"},"PeriodicalIF":3.3,"publicationDate":"2025-06-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144369145","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Predictive factors for post-therapeutic biochemical discordance in acromegaly: a monocentric analysis of 156 cases.","authors":"Dimitrios Emmanouilidis, Witold Polanski, Tareq Juratli, Stephan B Sobottka, Elena Tsourdi, Matthias Gruber, Thomas Pinzer, Ilker Y Eyüpoglu","doi":"10.1007/s11102-025-01547-8","DOIUrl":"https://doi.org/10.1007/s11102-025-01547-8","url":null,"abstract":"<p><strong>Purpose: </strong>Biochemical remission is the primary treatment goal in acromegaly. However, some patients experience biochemical discordance between growth hormone (GH) and insulin-like growth factor-I (IGF-I) levels following multimodal therapy, complicating disease assessment and management. This study aims to identify predictive factors associated with post-therapeutic biochemical discrepancy.</p><p><strong>Methods: </strong>We conducted a retrospective monocentric analysis of 156 patients with GH-producing pituitary adenomas (PAs) who underwent transsphenoidal surgery between 1984 and 2017. Biochemical outcomes were classified into four groups: group 1 (biochemical remission), group 2 (isolated GH normalization), group 3 (isolated IGF-I normalization), and group 4 (persistent acromegaly). Predictive factors for biochemical discrepancy were assessed, including demographic data, tumor characteristics, medication, irradiation, follow up duration, and disease recurrence.</p><p><strong>Results: </strong>The median age of the cohort was 48.2 years, with a female predominance (61.5%). Most PAs were macroadenomas (79.6%) and invasive (53.9%). Biochemical remission was achieved in 69.9%, while 19.2% exhibited biochemical discrepancy. Univariate analysis identified overall medication (pre- and/or postoperative), irradiation, and invasive PAs as significant factors associated with biochemical discordance. Logistic regression confirmed medication as the most influential predictor, with irradiation as a potential contributing factor. Disease recurrence was the only distinguishing factor between persistent acromegaly and biochemical discrepancy.</p><p><strong>Conclusion: </strong>Overall medication use is the strongest predictor of biochemical discrepancy, with irradiation potentially contributing. No clear distinguishing factors between biochemical discordance and persistent acromegaly were identified, except from disease recurrence. Managing patients with biochemical discrepancy similarly to those with persistent acromegaly may be advisable. Further research is needed to refine treatment strategies.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"74"},"PeriodicalIF":3.3,"publicationDate":"2025-06-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144369148","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Baseline testosterone levels as a predictor of hypogonadism resolution in male patients with isolated hyperprolactinemia.","authors":"Angelo Milioto, Cristian Petolicchio, Lorenzo Mattioli, Claudia Campana, Anna Arecco, Diego Ferone, Francesco Cocchiara, Federico Gatto","doi":"10.1007/s11102-025-01548-7","DOIUrl":"https://doi.org/10.1007/s11102-025-01548-7","url":null,"abstract":"<p><strong>Purpose: </strong>To evaluate the prevalence and the timing of gonadal axis restoration in men with hypogonadism secondary to hyperprolactinemia after prolactin (PRL) normalization, and to identify factors associated with testosterone (TT) recovery to normal values.</p><p><strong>Methods: </strong>We retrospectively analyzed clinical records of male patients with central hypogonadism and successfully treated isolated hyperprolactinemia. Data on PRL, TT, gonadotropins levels were retrieved for different time points: diagnosis, PRL normalization, gonadal axis restoration (if achieved) and last follow-up. Testosterone replacement therapy within 6 months of PRL normalization was an exclusion criterion.</p><p><strong>Results: </strong>Twenty-nine patients, median age 50 years (IQR 41-58), were included. The etiology of hyperprolactinemia included: prolactinoma (n = 23, 79%), non-functioning pituitary adenoma causing stalk effect (n = 5, 17%) and idiopathic cause (n = 1, 4%). After successful treatment of hyperprolactinemia, 20 patients (69%) spontaneously recovered the gonadal axis, achieving normal TT levels. Ten patients normalized PRL and TT values concurrently, while the other 10 exhibited a median delay of 6.5 months (4-9.25) after PRL normalization; the former group showed lower baseline PRL levels at diagnosis compared to the latter (p = 0.007). Patients who recovered the gonadal axis had higher baseline TT values compared to those with persistent hypogonadism (p = 0.02). At ROC curve analysis, baseline TT was a good predictor of spontaneous gonadal axis recovery after PRL normalization (AUC 0.869, p = 0.002).</p><p><strong>Conclusion: </strong>In patients with hypogonadism secondary to isolated hyperprolactinemia, gonadal axis recovery occurs frequently, particularly in those with higher baseline TT. Lower PRL levels at diagnosis are associated with a faster recovery of gonadal axis.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"75"},"PeriodicalIF":3.3,"publicationDate":"2025-06-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144369146","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Diagnosis of growth hormone deficiency: searching for Consensus along life.","authors":"Flavia Prodam, Pietro Maffei","doi":"10.1007/s11102-025-01546-9","DOIUrl":"https://doi.org/10.1007/s11102-025-01546-9","url":null,"abstract":"","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"73"},"PeriodicalIF":3.3,"publicationDate":"2025-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144294803","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Letter to the editor: Pituitary hyperplasia causing visual symptoms in a young woman planning pregnancy.","authors":"Frédérick Rault, Julia Morera, Thomas Gaberel","doi":"10.1007/s11102-025-01532-1","DOIUrl":"https://doi.org/10.1007/s11102-025-01532-1","url":null,"abstract":"","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"72"},"PeriodicalIF":3.3,"publicationDate":"2025-06-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144267133","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Effect of antineoplastic drug therapies on carcinoma and aggressive pituitary tumors: a systematic review and meta-analysis.","authors":"Ana Beatriz Ribeiro Cardoso, Amanda Cristina Zimmermann, Gerald Raverot, Vania Dos Santos Nunes-Nogueira","doi":"10.1007/s11102-025-01541-0","DOIUrl":"https://doi.org/10.1007/s11102-025-01541-0","url":null,"abstract":"<p><strong>Purpose: </strong>This systematic review aims to evaluate tumor control outcomes associated with antineoplastic drug therapies used for aggressive pituitary tumors (APTs) and pituitary carcinomas (PCs).</p><p><strong>Methods: </strong>We included studies on patients with PC or APT who received one of the following therapies: temozolomide (TMZ), peptide receptor radionuclide therapy (PRRT), everolimus, immune checkpoint inhibitors (ICIs), lapatinib, bevacizumab, capecitabine plus temozolomide (CAPTEM). Search strategies were applied to MEDLINE, EMBASE, LILACS and CENTRAL. Two independent reviewers selected studies, assessed the risk of bias, and extracted data. Proportional meta-analyses were used to calculate overall frequencies of complete response (CR), partial response (PR), stable disease (SD), and progressive disease (PD).</p><p><strong>Results: </strong>Seventy eight studies were included. TMZ was the most commonly used therapy, followed by ICIs, bevacizumab, PRRT, CAPTEM, lapatinib, and everolimus. Among 434 patients treated with TMZ in studies involving three or more participants, CR occurred in 4% (95% confidence interval [95% CI], 1-13), PR in 33% (95% CI, 28-37), SD in 32% (95% CI, 28-36), and PD in 29% (95% CI, 25-34). For ICIs, PR occurred in 24% (95% CI, 11-44), SD in 12% (95% CI, 4-31), and PD in 67% (95% CI, 24-93).</p><p><strong>Conclusion: </strong>TMZ was the most frequently reported therapy, with PR as the predominant outcome. However, the limited data on ICIs, PRRT, bevacizumab, lapatinib, and everolimus yielded imprecise results, highlighting the need for further research with the aim of gaining more insights into treatment effects of antineoplastic drug therapies for APTs and PCs.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 3","pages":"70"},"PeriodicalIF":3.3,"publicationDate":"2025-06-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144209228","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Impulse control disorders in pituitary adenoma: What do we know and what we still don't know in almost two decades?","authors":"P Kadioglu, A Glezer","doi":"10.1007/s11102-025-01533-0","DOIUrl":"https://doi.org/10.1007/s11102-025-01533-0","url":null,"abstract":"","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 3","pages":"71"},"PeriodicalIF":3.3,"publicationDate":"2025-06-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144209229","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Enhancing diagnostic tools for vasopressin deficiency: insights from a single-center cohort study.","authors":"Alessandro Mondin, Giulia Bovo, Giorgia Antonelli, Diego Faggian, Pierluigi Mazzeo, Alessandro Bavaresco, Filippo Ceccato, Mattia Barbot","doi":"10.1007/s11102-025-01538-9","DOIUrl":"10.1007/s11102-025-01538-9","url":null,"abstract":"<p><strong>Context: </strong>A recent multicenter trial confirmed that hypertonic saline-stimulated copeptin is superior to the arginine stimulation test (AST) for diagnosing vasopressin deficiency (AVP-D). The latter, though less accurate, is cheaper, better tolerated, and easier to perform. We aimed to improve AST diagnostic accuracy by incorporating additional parameters alongside copeptin.</p><p><strong>Methods: </strong>We retrospectively analysed ASTs from patients evaluated for suspected AVP-D. Final diagnosis was defined based on clinical, biochemical, radiological and follow-up data. We evaluated the test diagnostic accuracy based on either literature reported or ROC-based thresholds of several variables even in combination.</p><p><strong>Results: </strong>Nineteen patients were included and 8 were diagnosed with AVP-D. Copeptin response to AST was flat in AVP-D compared to primary polydipsia (PP) but showed limited discriminatory power with the maximal accuracy for copeptin-based parameters reaching 73.7%. AVP-D patients had lower urinary osmolarity (UOsm) and higher plasma osmolarity and serum sodium (Na) at AST end. Na at AST end was the best predictor of AVP-D (≥ 141 mmol/L: sensitivity 87.5%, specificity 100%, accuracy 94.7%, AUC 0.989). A multistep approach initially assessing Na at AST end and, in dubious cases (140-142 mmol/l), also either copeptin peak (≤ 4.1 pmol/L), UOsm (≤ 428 mOsm/kg), or absent posterior pituitary hyperintense signal achieved 100% diagnostic accuracy. Logistic regression using Na at AST end values combined with any of these aforementioned additional variables also reached complete discrimination between AVP-D and PP.</p><p><strong>Discussion: </strong>Combining multiple parameters after AST improved diagnostic accuracy, even without measuring copeptin. Despite the study's retrospective design, small sample, and absence of hypertonic saline testing, findings support the potential utility of a multivariable approach to AST interpretation.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 3","pages":"65"},"PeriodicalIF":3.3,"publicationDate":"2025-05-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12122565/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144182059","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Artificial intelligence in acromegaly: Why, when and how.","authors":"Alessandro Giustina, Andrea Giustina","doi":"10.1007/s11102-025-01534-z","DOIUrl":"https://doi.org/10.1007/s11102-025-01534-z","url":null,"abstract":"","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 3","pages":"66"},"PeriodicalIF":3.3,"publicationDate":"2025-05-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144181858","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}