Pituitary最新文献

{"title":"A core outcome set for pituitary surgery research: an international delphi consensus study.","authors":"Alexandra Valetopoulou, Nicola Newall, Danyal Z Khan, Anouk Borg, Pierre M G Bouloux, Fion Bremner, Michael Buchfelder, Simon Cudlip, Neil Dorward, William M Drake, Juan C Fernandez-Miranda, Maria Fleseriu, Mathew Geltzeiler, Joy Ginn, Mark Gurnell, Steve Harris, Zane Jaunmuktane, Márta Korbonits, Michael Kosmin, Olympia Koulouri, Hugo Layard Horsfall, Adam N Mamelak, Richard Mannion, Pat McBride, Ann I McCormack, Shlomo Melmed, Katherine A Miszkiel, Gerald Raverot, Thomas Santarius, Theodore H Schwartz, Inma Serrano, Gabriel Zada, Stephanie E Baldeweg, Hani J Marcus, Angelos G Kolias","doi":"10.1007/s11102-025-01553-w","DOIUrl":"10.1007/s11102-025-01553-w","url":null,"abstract":"<p><strong>Purpose: </strong>This study aimed to develop a core outcome set (COS) for pituitary surgery to enhance the quality, efficiency and effectiveness of future pituitary adenoma surgery research.</p><p><strong>Methods: </strong>Thirty-three outcomes were identified through a systematic review of pituitary adenoma surgery outcomes and a study on patient-reported measures. These were presented in an online survey to healthcare professionals (HCPs), patients and caregivers. In the first round, participants scored each outcome's importance on a 5-point scale (1-strongly disagree; 5-strongly agree) and could also suggest additional outcomes, which were reviewed and, if appropriate, added to existing domains. In the second round, participants re-scored the updated the list, considering group median and interquartile range scores from the previous round. Outcomes with a median score of 5 were included in the COS. A final live online consensus meeting discussed and voted on borderline outcomes (median scores 3-4).</p><p><strong>Results: </strong>The first round received 95 responses (52% HCPs, 48% patients/caregivers). Of the 33 outcomes, 16 received a median score of 5 (strongly agree), three received 4.5 and 14 received 4 (agree). Round two received 87 responses (52% HCPs, 48% patients and caregivers). Of the 33 outcomes, 14 received a median ranking of 5, two received 4.5, 15 received 4 and two received 3 (neutral). The live meeting (attended by 12 participants: 5 HCPs, 6 patients, 1 caregiver), reached consensus on the final COS, which includes 7 domains: short-term surgical outcomes; nasal outcomes; ophthalmic outcomes; endocrine outcomes; quality of life and psychological outcomes; other short-term outcomes; and disease control outcomes.</p><p><strong>Conclusion: </strong>We advocate for use of the COS in future pituitary surgery research.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"88"},"PeriodicalIF":3.4,"publicationDate":"2025-07-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12287136/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144699338","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":"Response to letter to the editor \"proposed consideration for targeted immunotherapy in pituitary carcinomas and aggressive pituitary tumors\".","authors":"Ana Beatriz Ribeiro Cardoso, Amanda Cristina Zimmermann, Gerald Raverot, Vania Dos Santos Nunes-Nogueira","doi":"10.1007/s11102-025-01558-5","DOIUrl":"10.1007/s11102-025-01558-5","url":null,"abstract":"","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"86"},"PeriodicalIF":3.4,"publicationDate":"2025-07-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144699340","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":"Non-penetrant Xq26.3 duplication involving the invariant TAD border: clinical evidence for the VGLL1 region as the GPR101 pituitary enhancer of X-linked acrogigantism.","authors":"Cathie Hilditch, Samuel Curtis, Samuel Cotton, Shannon LeBlanc, Sunita De Sousa","doi":"10.1007/s11102-025-01559-4","DOIUrl":"10.1007/s11102-025-01559-4","url":null,"abstract":"<p><strong>Introduction: </strong>X-linked acrogigantism (X-LAG; OMIM: 300942) is a rare X-linked dominant, fully penetrant form of infancy-onset pituitary gigantism caused by Xq26.3 tandem duplications involving the GPR101 gene. All previously reported X-LAG-associated duplications disrupt the integrity of the resident topologically associating domain (TAD). This creates a neo-TAD, permitting ectopic chromatin interactions between GPR101 and centromeric pituitary enhancers postulated to lie between RBMX and VGLL1, and culminating in pituitary GPR101 misexpression and growth hormone excess. Conversely, none of the few previously reported cases of Xq26.3 duplications in unaffected individuals include the tissue-invariant TAD border that shields GPR101 from its centromeric enhancers. Preservation of this boundary has thus been considered synonymous with non-penetrance of X-LAG.</p><p><strong>Methods: </strong>We examined a series of four family members from the same kindred with an incidentally detected GPR101-containing Xq26.3 duplication involving the invariant TAD border.</p><p><strong>Results: </strong>Chromosome microarray demonstrated an interstitial chromosome Xq26.3 duplication: arr[GRCh37] Xq26.3(135,954,223 - 136,224,319)x2, including GPR101, the TAD invariant border and RBMX, but not VGLL1. None of the relatives with the Xq26.3 duplication exhibited evidence of growth hormone excess, making this the first unaffected family with a GPR101-containing Xq26.3 duplication involving the invariant TAD border. The predicted neo-TAD in this kindred excludes the VGLL1 region, which is present in all previously described X-LAG patients and absent in all previously described unaffected individuals with Xq26.3 duplications.</p><p><strong>Conclusion: </strong>Our clinical findings suggest that TAD border involvement is not sufficient for X-LAG to develop, and implicates the VGLL1 region as likely the sole pituitary enhancer responsible for GPR101 misexpression and the X-LAG phenotype. Pending corroborative studies, this new insight into X-LAG pathogenesis may guide interpretation of future Xq26.3 duplications and counselling of families in whom such duplications are found.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"85"},"PeriodicalIF":3.4,"publicationDate":"2025-07-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12277222/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144668131","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":"Commentary: The updated European society of endocrinology clinical practice guideline for the management of aggressive pituitary tumors and pituitary carcinomas.","authors":"Roberto Salvatori, Shlomo Melmed, Andrea Giustina","doi":"10.1007/s11102-025-01555-8","DOIUrl":"10.1007/s11102-025-01555-8","url":null,"abstract":"","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"84"},"PeriodicalIF":3.4,"publicationDate":"2025-07-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144560927","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":"Microbiome and metabolic disorder in prolactinoma: intrinsic gender differences and extrinsic therapy effects.","authors":"Zhen Ye, Yifei Yu, Zhan Cao, Zhao Ye, Xufang Gu, Xiaoxu Shen, Boyu Cai, Ben Lin, Chenxing Ji, Nidan Qiao, Zerui Wu, Zhengyuan Chen, Zengyi Ma, Long Chen, Buqing Liang, Yankai Liao, Wenqiang He, Qing Shen, Jiaxin Han, Xiaoyun Cao, Xiang Zhou, Xuefei Shou, Ming Shen, Yongfei Wang, Zhaoyun Zhang, Hongying Ye, Qilin Zhang, Renyuan Gao, Yichao Zhang","doi":"10.1007/s11102-025-01544-x","DOIUrl":"10.1007/s11102-025-01544-x","url":null,"abstract":"","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"83"},"PeriodicalIF":3.4,"publicationDate":"2025-07-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144554249","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":"Proposed consideration for targeted immunotherapy in pituitary carcinomas and aggressive pituitary tumors.","authors":"Aaron Kakazu, Jeff F Zhang, Jonathan A Forbes","doi":"10.1007/s11102-025-01551-y","DOIUrl":"10.1007/s11102-025-01551-y","url":null,"abstract":"","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"79"},"PeriodicalIF":3.4,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144542001","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":"Endoscopic endonasal transcavernous surgery for a contemporary series of 59 prolactinomas.","authors":"Taishi Nakase, Vladimir A Ljubimov, Julia J Chang, Hannes Vogel, Vera Vigo, Laurence Katznelson, Juan Carlos Fernandez-Miranda","doi":"10.1007/s11102-025-01545-w","DOIUrl":"10.1007/s11102-025-01545-w","url":null,"abstract":"<p><strong>Purpose: </strong>To assess surgical outcomes in patients with prolactinomas treated surgically with contemporary endoscopic endonasal techniques within the context of recent advances in transcavernous approaches and shifts towards surgery as a primary treatment option alongside dopamine agonists.</p><p><strong>Methods: </strong>Surgical outcomes were retrospectively analyzed for 59 consecutive patients with prolactinomas who underwent endoscopic endonasal surgery between October 2018 and December 2024.</p><p><strong>Results: </strong>The cohort included 42 (71%) patients with macroprolactinomas and 32 (54%) patients with cavernous sinus (CS) invasion, including 14 (24%) with isolated medial wall invasion and 18 (31%) with CS compartment invasion. Median follow-up was 19 months (interquartile range = 10-38). Overall, 82% of patients demonstrated normoprolactinemia within three days of surgery and 80% (74% macroprolactinoma, 94% microprolactinoma) achieved biochemical remission at last follow-up. Adjuvant dopamine agonist treatment and/or radiation increased the long-term remission rate to 86% overall and to 83% for macroprolactinomas. Among patients for whom total resection (vs. debulking) was the primary surgical goal, long-term biochemical remission was achieved in 84% of patients (88% with adjuvant therapy). One operative complication with no neurological sequelae occurred in a patient with a giant invasive adenoma. Permanent arginine vasopressin deficiency was observed in three patients and transient diplopia was observed in four patients.</p><p><strong>Conclusion: </strong>The addition of endoscopic transcavernous approaches for prolactinoma resection can be safe and effective in selected patients after multidisciplinary evaluation when performed by an experienced neurosurgical team, providing further support for the wider adoption of surgery in the management of prolactinomas.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"81"},"PeriodicalIF":3.4,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144541998","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":"FAP as a prognostic biomarker and radiomics-based predictor of angiogenesis-associated recurrence in Adamantinomatous craniopharyngioma.","authors":"Qin Huang, Xiaorong Yan, Bo Zhang, Zelin Liu, Yongjian Chen, Xiaohai Liu, Mingchu Li, Xin Su, Xianlong Wang, Bowen Wu, Ge Chen, Jun Pan, Zhixiong Lin, Yiguang Chen","doi":"10.1007/s11102-025-01552-x","DOIUrl":"10.1007/s11102-025-01552-x","url":null,"abstract":"<p><strong>Background: </strong>Adamantinomatous craniopharyngioma (ACP) is a histologically benign yet clinically aggressive intracranial tumor characterized by high recurrence rates. Fibroblast activation protein (FAP), a marker of cancer-associated fibroblasts (CAFs), is implicated in tumor progression and microenvironment remodeling. This study evaluates the prognostic significance of FAP and develops a radiomics-based model for non-invasive preoperative risk stratification.</p><p><strong>Methods: </strong>Immunohistochemical analysis was performed on 54 ACP cases to assess FAP expression levels. Transcriptomic data from 110 ACP cases across two external databases were analyzed for differential gene expression and pathway enrichment. Immunofluorescence was conducted to determine the spatial correlation of FAP with angiogenic markers (VEGF, CD31, CD34). A radiomics model was developed using preoperative MRI data to predict FAP expression and validated for predictive performance.</p><p><strong>Results: </strong>High FAP expression was associated with extracellular matrix remodeling, angiogenesis, and inflammatory pathway activation. Clinically, high-FAP tumors exhibited larger volumes (P = 0.044), more severe hypothalamic dysfunction (P = 0.001), and shorter progression-free survival (P = 0.03). Multivariate analysis identified high FAP expression (HR = 9.890, P = 0.0340) as an independent predictor of recurrence. Immunofluorescence confirmed the co-localization of FAP⁺ CAFs with angiogenic markers, suggesting a role in tumor recurrence. The radiomics model integrating T1/T2-weighted MRI features demonstrated robust performance in predicting FAP expression, with AUCs of 0.742 (training set),0.822 (internal validation set) and 0.686(external validation set).</p><p><strong>Conclusions: </strong>FAP is a prognostic biomarker in ACP, with high expression indicative of increased recurrence risk. The radiomics model offers a non-invasive approach for preoperative risk stratification, potentially guiding surgical decision-making and anti-angiogenic therapeutic strategies.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"80"},"PeriodicalIF":3.4,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144541999","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":"High recurrence of rathke's cleft cysts with anterior-Inferior pituitary displacement despite standard surgical approaches.","authors":"Kenta Nakase, Fumihiko Nishimura, Yudai Morisaki, Shohei Yokoyama, Masashi Kotsugi, Yasuhiro Takeshima, Ryosuke Matsuda, Shuichi Yamada, Young-Soo Park, Ichiro Nakagawa","doi":"10.1007/s11102-025-01554-9","DOIUrl":"10.1007/s11102-025-01554-9","url":null,"abstract":"<p><strong>Purpose: </strong>Although endonasal endoscopic surgery (EES) is widely used to treat symptomatic Rathke's cleft cysts (RCCs), the optimal surgical strategy remains unclear. We previously proposed that the anatomical relationship between RCCs and the anterior pituitary lobe may predict recurrence. This study aimed to evaluate clinical characteristics and long-term outcomes based on anatomical classification and to assess the impact of surgical method (marsupialization vs. reconstruction) within each subtype.</p><p><strong>Methods: </strong>We retrospectively analyzed 40 patients who underwent EES for symptomatic RCCs between 2008 and 2024. RCCs were classified into four types based on displacement: type 1 (anterior-superior), type 2 (anterior-inferior), type 3 (posterior-superior), and type 4 (posterior-inferior). Clinical, imaging, and surgical outcomes were compared across subtypes.</p><p><strong>Results: </strong>he mean follow-up duration was 112 ± 53.2 months. Recurrence occurred in 16 patients (40.0%), and 6 (15.0%) patients required reoperation. Type 2 was independently associated with a higher recurrence rate (p = 0.019), more frequent preoperative visual disturbances (p = 0.0059), and lower T1-weighted signal intensity (p = 0.027). There was no significant difference in the recurrence rate between the surgical methods within each subtype.</p><p><strong>Conclusion: </strong>The anterior-inferior subtype is more likely to recur regardless of the surgical method. The identification of high-risk subtypes may support the use of tailored strategies, including drainage-preserving techniques, to improve long-term outcomes.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"82"},"PeriodicalIF":3.4,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144542000","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":"Weight gain reversibility and BMI following treatment for Cushing's syndrome: long-term outcomes and potential predictors.","authors":"Liat Sasson, Laura Dery, Julia Stern, Ilan Shimon, Yaron Rudman, Idit Dotan, Shiri Kushnir, Tzipora Shochat, Maria Fleseriu, Amit Akirov","doi":"10.1007/s11102-025-01550-z","DOIUrl":"10.1007/s11102-025-01550-z","url":null,"abstract":"<p><strong>Background: </strong>Patients with Cushing's syndrome (CS) have higher weight and body mass index (BMI) compared to matched controls. We aimed to assess reversibility of weight gain (absolute weight and BMI decrease) following treatment for CS and identify predictors for weight loss.</p><p><strong>Methods: </strong>A retrospective study using the Clalit Health Services database analyzed a cohort of patients with CS and age-, sex- and BMI-matched controls (up to five controls per case). Weight and BMI were assessed at baseline, one-year post-diagnosis and at the end of follow-up. Patients taking GLP-1 have been excluded from the analysis.</p><p><strong>Results: </strong>The cohort included 308 CS patients (67.2% female, mean age 51.6 ± 17.4 years) and 1,020 controls (69.7% female, mean age 54.8 ± 16.9 years). After one year, CS patients who achieved remission experienced significant weight reduction from 85.1 ± 21.0 to 80.8 ± 22.7 kg and BMI decrease from 31.6 ± 7.4 to 29.9 ± 7.4 kg/m² (p < 0.01). By the end of 8.6 years of follow-up, CS patients continued to show significant reductions in both weight (85.0 ± 22.3 to 81.3 ± 22.8 kg, p < 0.01) and BMI (31.2 ± 7.6 to 29.9 ± 7.6 kg/m², p < 0.01), while controls showed slight increases in both. Among patients in remission by the end-of-follow-up, 44.0% (77/175) achieved ≥ 5% and 30.3% (53/175) achieved ≥ 10% weight loss. Patients with persistent CS exhibited no significant weight changes. Multivariate analysis identified female gender, baseline BMI ≥ 30 and a BMI decrease ≥ 1 kg/m² at one year, as predictors for ≥ 5% weight loss.</p><p><strong>Conclusion: </strong>Approximately half of CS patients in remission achieved at least 5% decrease in BMI with long-term follow-up, and a third achieved more than 10% decrease. Disease remission, female gender, baseline BMI ≥ 30, and early BMI reduction, predicted a clinically significant weight loss.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"28 4","pages":"78"},"PeriodicalIF":3.4,"publicationDate":"2025-06-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144497878","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}