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Impact of etiology, sex, diabetes mellitus and remission status on erythrocytic profile in patients with cushing's syndrome: a large population database study. 病因、性别、糖尿病和缓解状态对库欣综合征患者红细胞特征的影响:一项大型人群数据库研究。
IF 3.3 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-08-01 Epub Date: 2024-05-20 DOI: 10.1007/s11102-024-01399-8
Laura Dery, Julia Stern, Ilan Shimon, Yaron Rudman, Shiri Kushnir, Tzipora Shochat, Maria Fleseriu, Amit Akirov

Purpose: The study aimed to characterize the erythrocytic profile in patients with cushing's syndrome (CS) versus controls from the normal population according to etiology, sex, presence of diabetes mellitus (DM) and hypercortisolemia remission status.

Methods: This retrospective cohort analysis compared erythrocytic parameters between patients with CS of pituitary (CD) and adrenal (aCS) etiology and age, sex, body mass index (BMI) and socioeconomic status-matched controls in a 1:5 ratio. Laboratory values at baseline were calculated as mean values during the year preceding CS diagnosis, and over one year thereafter.

Results: The cohort included 397 CS patients (68.26% female; mean age 51.11 ± 16.85 years) and 1970 controls. Patients with CS had significantly higher baseline median levels of hemoglobin (Hgb) (13.70 g/dL vs. 13.12 g/dL [p < 0.0001]) and hematocrit (Hct) (41.64% vs. 39.80% [p < 0.0001]) compared to controls. These differences were observed for both CD and aCS and for both sexes. Patients who attained remission had Hgb and Hct levels comparable to controls (13.20 g/dL and 40.08% in patients with CD and aCS vs. 13.20 g/dL and 39.98% in controls). Meanwhile, those with persistent/recurrent disease maintained elevated levels. Patients with comorbid DM had similar Hgb but higher Hct (p = 0.0419), while patients without DM showed elevated erythrocytic values compared to controls (p < 0.0001).

Conclusion: Our data illustrates that erythrocytic parameters are directly influenced by glucocorticoid excess as Hgb and Hct are higher in patients with CS, and normalize after remission. We have identified the influence of DM on erythrocytic parameters in patients with CS for the first time.

目的:该研究旨在根据病因、性别、是否患有糖尿病(DM)和高皮质醇血症缓解状态,分析库欣综合征(CS)患者与正常人群对照组的红细胞特征:这项回顾性队列分析以 1:5 的比例比较了垂体(CD)和肾上腺(aCS)病因 CS 患者与年龄、性别、体重指数(BMI)和社会经济状况匹配的对照组之间的红细胞参数。基线实验室值按 CS 诊断前一年的平均值计算,此后一年的平均值也按此计算:组群包括 397 名 CS 患者(68.26% 为女性;平均年龄为 51.11 ± 16.85 岁)和 1970 名对照组。CS 患者的血红蛋白(Hgb)基线中位数水平明显高于对照组(13.70 g/dL vs. 13.12 g/dL [p 结论:CS 患者的血红蛋白基线中位数水平明显高于对照组(13.70 g/dL vs. 13.12 g/dL):我们的数据表明,红细胞参数直接受糖皮质激素过量的影响,因为 CS 患者的 Hgb 和 Hct 较高,并在病情缓解后趋于正常。我们首次发现了 DM 对 CS 患者红细胞参数的影响。
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引用次数: 0
Exploring potential influencing factors of inadherence to specialist aftercare and long-term medication in patients with acromegaly. 探索肢端肥大症患者不坚持专科术后护理和长期服药的潜在影响因素。
IF 3.3 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-08-01 Epub Date: 2024-05-24 DOI: 10.1007/s11102-024-01400-4
Sonja Siegel, Sabrina Giese, Jürgen Honegger, Anna Lena Friedel, Agnieszka Grzywotz, Karsten Henning Wrede, Ulrich Sure, Nicole Unger, Ilonka Kreitschmann-Andermahr

Purpose: To improve the understanding of adherence as one major factor of disease control in acromegaly patients, we systematically assessed patients' motivations to adhere to advised follow-up schedules and recommended medication for acromegaly.

Methods: Cross-sectional, postal questionnaire study on adult patients with acromegaly, operated upon a growth hormone producing pituitary adenoma more than 1 year ago in two tertiary treatment centers. We assessed demographic and clinical characteristics, disease status, adherence to acromegaly medication and/or aftercare, and the five dimensions defined by the World Health Organization influencing adherence. Wherever applicable, we included validated short scales. The answers of 63 patients (33 f, 30 m; mean age 56.1 y) were analyzed.

Results: Patients with problems in adherence to aftercare had a significantly lower subjective symptomload than those adherent to aftercare (p = 0.026) and a lower perceived need for treatment (p = 0.045). Patients with adherence problems to medication had a higher subjective symptomload than those without (p = 0.056). They also tended to have shorter consultations, were significantly more often dissatisfied with the duration of their medical consultations (42% vs 4.8%, p = 0.019) and tended to find that their physician explained potential difficulties with adherence less well than patients without adherence problems (p = 0.089).

Conclusions: To our knowledge, this is the first study which explored adherence to medication and aftercare in patients with acromegaly, taking into account potential influencing factors from all areas defined by the WHO model of adherence. Of the modifiable factors of adherence, patient-doctor relationship seemed to play a crucial role and could be one leverage point to improve adherence.

目的:为了更好地了解肢端肥大症患者坚持治疗是疾病控制的一个主要因素,我们系统地评估了患者坚持建议的随访时间和推荐的肢端肥大症药物治疗的动机:方法:横断面邮寄问卷调查,对象是在两家三级治疗中心接受生长激素垂体腺瘤手术超过一年的成年肢端肥大症患者。我们评估了人口统计学和临床特征、疾病状态、对肢端肥大症药物治疗和/或术后护理的依从性,以及世界卫生组织定义的影响依从性的五个方面。在适用的情况下,我们采用了经过验证的简易量表。我们对 63 名患者(33 名女性,30 名男性;平均年龄 56.1 岁)的回答进行了分析:结果:在坚持术后护理方面存在问题的患者,其主观症状负荷明显低于坚持术后护理的患者(p = 0.026),其治疗需求感知也明显低于坚持术后护理的患者(p = 0.045)。有服药问题的患者的主观症状负荷高于无服药问题的患者(p = 0.056)。他们的就诊时间往往更短,对就诊时间的不满意度明显更高(42% vs 4.8%,p = 0.019),而且他们往往认为医生对他们在坚持用药方面可能遇到的困难的解释不如没有坚持用药问题的患者(p = 0.089):据我们所知,这是第一项探讨肢端肥大症患者服药依从性和术后护理的研究,其中考虑到了世界卫生组织依从性模型所定义的所有领域的潜在影响因素。在可改变依从性的因素中,患者与医生的关系似乎起着至关重要的作用,可以成为提高依从性的一个杠杆点。
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引用次数: 0
Pituitary apoplexy: a systematic review of non-gestational risk factors. 垂体性脑瘫:非妊娠风险因素的系统回顾。
IF 3.3 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-08-01 Epub Date: 2024-06-27 DOI: 10.1007/s11102-024-01412-0
Smile Kajal, Youssef El Sayed Ahmad, Akaber Halawi, Mohammad Abraham Kazemizadeh Gol, William Ashley

Purpose: Pregnancy is a known risk factor for Pituitary Apoplexy (PA) but there is a lack of consistency in the literature regarding non-gestational risk factors responsible for PA.

Methods: We did a systematic review following PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines to identify the non-gestational risk factors associated with the development of PA in adult patients with pituitary adenoma. Also, we discuss here a case of an elderly female with pituitary macroadenoma who was initially planned for pituitary resection electively but underwent emergency surgery after she developed PA.

Results: As per screening and eligibility criteria, seven studies with 4937 study participants were included in this systematic review out of which 490 (9.92%) patients had PA, including asymptomatic subclinical PA (SPA) and symptomatic clinical PA (CPA). The macroadenomas and negative staining of the tumor were found to be a significant risk factor consistently in multivariate analysis in three and two retrospective studies, respectively. However, the results were varied for any significant difference in the risk factors for apoplexy between SPA and CPA. Similarly, there was no consistency among the studies for risk factors significantly responsible for CPA or PA compared to controls.

Conclusion: No single non-gestational risk factor is solely responsible for the development of PA in a pituitary adenoma compared to the control population. Tumor size (macroadenoma) and the non-functioning status of the adenoma are the only significant factors contributing independently toward an apoplectic event in most patients. Such patients can be prioritized for early pituitary tumor resection.

目的:众所周知,妊娠是垂体性脑瘫(PA)的一个危险因素,但有关导致垂体性脑瘫的非妊娠危险因素的文献缺乏一致性:我们按照PRISMA(系统综述和荟萃分析的首选报告项目)指南进行了系统综述,以确定与垂体腺瘤成年患者发生PA相关的非妊娠风险因素。此外,我们还讨论了一例患有垂体大腺瘤的老年女性患者,她最初计划选择性进行垂体切除术,但在出现 PA 后接受了紧急手术:根据筛选和资格标准,本系统综述纳入了7项研究,共有4937名参与者,其中490名(9.92%)患者患有PA,包括无症状亚临床PA(SPA)和有症状临床PA(CPA)。分别在三项和两项回顾性研究的多变量分析中发现,大腺瘤和肿瘤阴性染色一直是重要的风险因素。然而,SPA 和 CPA 的脑动脉骤停风险因素是否存在显著差异,结果却不尽相同。同样,与对照组相比,导致CPA或PA的风险因素也不尽相同:结论:与对照人群相比,没有任何一个非妊娠风险因素是导致垂体腺瘤发生PA的唯一原因。肿瘤大小(大腺瘤)和腺瘤的非功能状态是大多数患者发生脑溢血的唯一重要独立因素。这类患者可优先考虑早期垂体瘤切除术。
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引用次数: 0
Pituitary tumor centers of excellence (PTCOE): the next border of acromegaly treatment. 垂体瘤卓越中心(PTCOE):肢端肥大症治疗的下一个边界。
IF 3.3 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-08-01 Epub Date: 2024-06-25 DOI: 10.1007/s11102-024-01416-w
Silvia Grottoli, Ezio Ghigo
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引用次数: 0
The diverging role of O-GlcNAc transferase in corticotroph and somatotroph adenomas O-GlcNAc转移酶在皮质腺瘤和体细胞腺瘤中的不同作用
IF 3.8 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-07-27 DOI: 10.1007/s11102-024-01431-x
Roel Gonzalez, Logan Massman, Sophia Ho, Sarai Luna, Stephanie Cheok, Brandon Liang, Kelly Mrachek, Dylan Coss, Adriana G. Ioachimescu, Nathan Zwagerman, Stephanie Olivier-Van Stichelen

Purpose

Molecular mechanisms involved in the pathogenesis and tumor progression of pituitary adenomas (PA) remain incompletely understood. Corticotroph and somatotroph PA are associated with a high clinical burden, and despite improved surgical outcomes and medical treatment options, they sometimes require multiple surgeries and radiation. Preliminary data suggested a role for O-GlcNAc Transferase (OGT), the enzyme responsible for the O-GlcNAcylation of proteins. O-GlcNAcylation and OGT have been found elevated in other types of tumors.

Methods

We evaluated 60 functioning and nonfunctioning PA (NFPA) from operated patients and postmortem normal and tumoral pituitary tissue by immunohistochemistry. We performed transcriptomic analyses to explore the relevance of the O-GlcNAc Transferase (OGT) in PAs. We detected OGT in immunobiological analysis and define its level in PA tissue in patients.

Results

OGT was strongly associated with PA hormone secretory capacity in functioning PA and with tumor growth in NFPAs. In NFPAs, OGT was positively associated with tumor size but not with cavernous sinus invasion (Knosp grading). In GH-secreting PA, OGT expression was negatively correlated with circulating Insulin-like Growth Factor 1 level. In adrenocorticotropic hormone (ACTH)-secreting PA, OGT expression was positively associated with circulating ACTH levels. OGT did not correlate with tumor size in secreting PAs. OGT levels were higher in gonadotroph PA compared to normal glands.

Conclusion

O-GlcNAcylation can be downregulated in non-cancerous tumors such as GH-secreting adenomas. Future studies are warranted to elucidate the role of OGT in the pathogenesis of PAs.

目的 垂体腺瘤(PA)的发病机制和肿瘤进展的分子机制仍不完全清楚。皮质脑垂体腺瘤和体细胞脑垂体腺瘤具有较高的临床负担,尽管手术效果和药物治疗方案有所改善,但有时仍需要多次手术和放射治疗。初步数据表明,O-GlcNAc 转化酶(OGT)是一种负责蛋白质 O-GlcNAcylation 的酶。我们用免疫组化方法评估了 60 例手术患者的功能性和非功能性 PA(NFPA),以及死后正常和肿瘤垂体组织。我们进行了转录组学分析,以探讨O-GlcNAc转移酶(OGT)在PA中的相关性。我们在免疫生物学分析中检测了OGT,并确定了其在患者PA组织中的水平。结果OGT与功能性PA的PA激素分泌能力和NFPAs的肿瘤生长密切相关。在 NFPA 中,OGT 与肿瘤大小呈正相关,但与海绵窦侵犯(Knosp 分级)无关。在分泌促肾上腺皮质激素的 PA 中,OGT 的表达与循环中胰岛素样生长因子 1 的水平呈负相关。在分泌促肾上腺皮质激素(ACTH)的 PA 中,OGT 的表达与循环 ACTH 水平呈正相关。在分泌 PA 中,OGT 与肿瘤大小无关。结论O-GlcNAcylation可在非癌症肿瘤(如分泌GH的腺瘤)中下调。未来的研究需要阐明 OGT 在 PA 发病机制中的作用。
{"title":"The diverging role of O-GlcNAc transferase in corticotroph and somatotroph adenomas","authors":"Roel Gonzalez, Logan Massman, Sophia Ho, Sarai Luna, Stephanie Cheok, Brandon Liang, Kelly Mrachek, Dylan Coss, Adriana G. Ioachimescu, Nathan Zwagerman, Stephanie Olivier-Van Stichelen","doi":"10.1007/s11102-024-01431-x","DOIUrl":"https://doi.org/10.1007/s11102-024-01431-x","url":null,"abstract":"<h3 data-test=\"abstract-sub-heading\">Purpose</h3><p>Molecular mechanisms involved in the pathogenesis and tumor progression of pituitary adenomas (PA) remain incompletely understood. Corticotroph and somatotroph PA are associated with a high clinical burden, and despite improved surgical outcomes and medical treatment options, they sometimes require multiple surgeries and radiation. Preliminary data suggested a role for <i>O</i>-GlcNAc Transferase (OGT), the enzyme responsible for the <i>O</i>-GlcNAcylation of proteins. <i>O</i>-GlcNAcylation and OGT have been found elevated in other types of tumors.</p><h3 data-test=\"abstract-sub-heading\">Methods</h3><p>We evaluated 60 functioning and nonfunctioning PA (NFPA) from operated patients and postmortem normal and tumoral pituitary tissue by immunohistochemistry. We performed transcriptomic analyses to explore the relevance of the <i>O</i>-GlcNAc Transferase (OGT) in PAs. We detected OGT in immunobiological analysis and define its level in PA tissue in patients.</p><h3 data-test=\"abstract-sub-heading\">Results</h3><p><i>OGT</i> was strongly associated with PA hormone secretory capacity in functioning PA and with tumor growth in NFPAs. In NFPAs, <i>OGT</i> was positively associated with tumor size but not with cavernous sinus invasion (Knosp grading). In GH-secreting PA, <i>OGT</i> expression was negatively correlated with circulating Insulin-like Growth Factor 1 level. In adrenocorticotropic hormone (ACTH)-secreting PA, OGT expression was positively associated with circulating ACTH levels. <i>OGT</i> did not correlate with tumor size in secreting PAs. OGT levels were higher in gonadotroph PA compared to normal glands.</p><h3 data-test=\"abstract-sub-heading\">Conclusion</h3><p><i>O</i>-GlcNAcylation can be downregulated in non-cancerous tumors such as GH-secreting adenomas. Future studies are warranted to elucidate the role of OGT in the pathogenesis of PAs.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":"18 1","pages":""},"PeriodicalIF":3.8,"publicationDate":"2024-07-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141774552","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}
引用次数: 0
The diagnosis and prevalence of hypoprolactinemia in patients with panhypopituitarism and the effects on depression and sexual functions. 泛垂体功能减退症患者低泌乳素血症的诊断和发病率,以及对抑郁和性功能的影响。
IF 3.8 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-06-01 Epub Date: 2024-05-03 DOI: 10.1007/s11102-024-01393-0
Ilknur Uzun, Zuleyha Karaca, Aysa Hacioğlu, Kursad Unluhizarci, Fahrettin Kelestimur

Purpose: We aimed to investigate the prevalence and the diagnostic criteria of hypoprolactinemia in patients with panhypopituitarism and the effects of hypoprolactinemia on depression and sexual functions.

Materials and methods: Forty-eight patients with panhypopituitarism and 20 healthy volunteers were included. Basal hormone levels were measured and a TRH stimulation test was performed. For the evaluation of sexual functions, questionnaries of Female Sexual Functional Index (FSFI) for females and International Erectile Functional Index for males were performed to the subjects. Depressive symptoms were evaluated by Beck Depression Envontory score (BDI-II).

Results: The peak PRL response to TRH stimulation test at 5th percentile in the control group was 18.6 ng/ml in males and 41.6 ng/ml in females and accepted as the cut-offs for sufficient response of PRL. Prolactin was insufficient in 42(87.5%) patients. A basal PRL level of ≤ 5.7 ng/ml in males and 7.11 ng/ml in females was 100% specific in predicting an inadequate response to TRH stimulation test with 80% and 70% sensitivity respectively. A basal PRL level of ≥ 8.5 ng/dl in males was 100% specific and 76% sensitive, and in females a level of ≥ 15.2 ng/dl was 96% specific and 66% sensitive in predicting an adequate response to TRH. PRL deficient patients with panhypopituitarism had higher depression scores compared to the controls, lower sexual function scores in males.

Conclusion: PRL deficiency is prevalent among individuals with panhypopituitarism, with the potential to result in elevated depression scores in both sexes and impaired sexual functions in males. A basal PRL level seems to be sufficient for the diagnosis of hypoprolactinemia in routine clinical practice.

目的:我们旨在研究泛垂体功能减退症患者低泌乳素血症的患病率和诊断标准,以及低泌乳素血症对抑郁和性功能的影响:研究对象包括48名垂体功能减退症患者和20名健康志愿者。测量基础激素水平并进行 TRH 刺激试验。为了评估性功能,对受试者进行了女性性功能指数(FSFI)和男性国际勃起功能指数问卷调查。抑郁症状通过贝克抑郁指数(BDI-II)进行评估:结果:在对照组中,男性 PRL 对 TRH 刺激试验的峰值反应为 18.6 纳克/毫升,女性为 41.6 纳克/毫升。42名(87.5%)患者的催乳素不足。男性基础 PRL 水平≤ 5.7 纳克/毫升,女性基础 PRL 水平≤ 7.11 纳克/毫升,对预测 TRH 刺激试验反应不足的特异性为 100%,敏感性分别为 80% 和 70%。男性基础PRL水平≥8.5 ng/dl的特异性为100%,敏感性为76%;女性基础PRL水平≥15.2 ng/dl的特异性为96%,敏感性为66%,可预测对TRH的充分反应。与对照组相比,垂体前叶素缺乏症患者的抑郁评分更高,男性患者的性功能评分更低:结论:PRL 缺乏在泛垂体功能减退症患者中很普遍,可能导致男女抑郁评分升高,男性性功能受损。在常规临床实践中,基础 PRL 水平似乎足以诊断低泌乳素血症。
{"title":"The diagnosis and prevalence of hypoprolactinemia in patients with panhypopituitarism and the effects on depression and sexual functions.","authors":"Ilknur Uzun, Zuleyha Karaca, Aysa Hacioğlu, Kursad Unluhizarci, Fahrettin Kelestimur","doi":"10.1007/s11102-024-01393-0","DOIUrl":"10.1007/s11102-024-01393-0","url":null,"abstract":"<p><strong>Purpose: </strong>We aimed to investigate the prevalence and the diagnostic criteria of hypoprolactinemia in patients with panhypopituitarism and the effects of hypoprolactinemia on depression and sexual functions.</p><p><strong>Materials and methods: </strong>Forty-eight patients with panhypopituitarism and 20 healthy volunteers were included. Basal hormone levels were measured and a TRH stimulation test was performed. For the evaluation of sexual functions, questionnaries of Female Sexual Functional Index (FSFI) for females and International Erectile Functional Index for males were performed to the subjects. Depressive symptoms were evaluated by Beck Depression Envontory score (BDI-II).</p><p><strong>Results: </strong>The peak PRL response to TRH stimulation test at 5th percentile in the control group was 18.6 ng/ml in males and 41.6 ng/ml in females and accepted as the cut-offs for sufficient response of PRL. Prolactin was insufficient in 42(87.5%) patients. A basal PRL level of ≤ 5.7 ng/ml in males and 7.11 ng/ml in females was 100% specific in predicting an inadequate response to TRH stimulation test with 80% and 70% sensitivity respectively. A basal PRL level of ≥ 8.5 ng/dl in males was 100% specific and 76% sensitive, and in females a level of ≥ 15.2 ng/dl was 96% specific and 66% sensitive in predicting an adequate response to TRH. PRL deficient patients with panhypopituitarism had higher depression scores compared to the controls, lower sexual function scores in males.</p><p><strong>Conclusion: </strong>PRL deficiency is prevalent among individuals with panhypopituitarism, with the potential to result in elevated depression scores in both sexes and impaired sexual functions in males. A basal PRL level seems to be sufficient for the diagnosis of hypoprolactinemia in routine clinical practice.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":" ","pages":"277-286"},"PeriodicalIF":3.8,"publicationDate":"2024-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11150180/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140861052","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}
引用次数: 0
Repeat endoscopic endonasal transsphenoidal surgery for residual or recurrent Cushing's disease: safety, feasibility, and success. 针对残留或复发库欣病的重复内窥镜经鼻腔手术:安全性、可行性和成功率。
IF 3.3 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-06-01 Epub Date: 2024-05-15 DOI: 10.1007/s11102-024-01396-x
Sahin Hanalioglu, Muhammet Enes Gurses, Neslihan Nisa Gecici, Baylar Baylarov, Ilkay Isikay, Alper Gürlek, Mustafa Berker

Purpose: The success and outcomes of repeat endoscopic transsphenoidal surgery (ETS) for residual or recurrent Cushing's disease (CD) are underreported in the literature. This study aims to address this gap by assessing the safety, feasibility, and efficacy of repeat ETS in these patients.

Methods: A retrospective analysis was conducted on 56 patients who underwent a total of 65 repeat ETS performed by a single neurosurgeon between January 2006 and December 2020. Data including demographic, clinical, laboratory, radiological, and operative details were collected from electronic medical records. Logistic regression was utilized to identify potential predictors associated with sustained remission.

Results: Among the cases, 40 (61.5%) had previously undergone microscopic surgery, while 25 (38.5%) had prior endoscopic procedures. Remission was achieved in 47 (83.9%) patients after the first repeat ETS, with an additional 9 (16.1%) achieving remission after the second repeat procedure. During an average follow-up period of 97.25 months, the recurrence rate post repeat surgery was 6.38%. Sustained remission was achieved in 48 patients (85.7%), with 44 after the first repeat ETS and 4 following the second repeat ETS. Complications included transient diabetes insipidus (DI) in 5 (7.6%) patients, permanent (DI) in 2 (3%) patients, and one case (1.5%) of panhypopituitarism. Three patients (4.6%) experienced rhinorrhea necessitating reoperation. A serum cortisol level > 5 µg/dL on postoperative day 1 was associated with a reduced likelihood of sustained remission.

Conclusion: Repeat ETS is a safe and effective treatment option for residual or recurrent CD with satisfactory remission rates and low rates of complications.

目的:文献中对残留或复发库欣病(CD)的重复内镜经蝶手术(ETS)的成功率和疗效报道不足。本研究旨在通过评估这些患者重复 ETS 的安全性、可行性和有效性来弥补这一空白:方法:我们对 2006 年 1 月至 2020 年 12 月间接受过 65 次重复 ETS 的 56 名患者进行了回顾性分析。从电子病历中收集了包括人口统计学、临床、实验室、放射学和手术细节在内的数据。利用逻辑回归确定与持续缓解相关的潜在预测因素:病例中,40 例(61.5%)曾接受过显微手术,25 例(38.5%)曾接受过内窥镜手术。47例(83.9%)患者在首次重复ETS后病情得到缓解,另有9例(16.1%)患者在第二次重复手术后病情得到缓解。在平均 97.25 个月的随访期间,重复手术后的复发率为 6.38%。48名患者(85.7%)的病情得到了持续缓解,其中44人在第一次重复ETS手术后缓解,4人在第二次重复ETS手术后缓解。并发症包括:5 例(7.6%)患者出现一过性糖尿病,2 例(3%)患者出现永久性糖尿病,1 例(1.5%)患者出现泛垂体功能减退。三名患者(4.6%)出现鼻出血,需要再次手术。术后第 1 天血清皮质醇水平大于 5 µg/dL 与持续缓解的可能性降低有关:结论:对于残留或复发的 CD,重复 ETS 是一种安全有效的治疗方法,缓解率令人满意,并发症发生率较低。
{"title":"Repeat endoscopic endonasal transsphenoidal surgery for residual or recurrent Cushing's disease: safety, feasibility, and success.","authors":"Sahin Hanalioglu, Muhammet Enes Gurses, Neslihan Nisa Gecici, Baylar Baylarov, Ilkay Isikay, Alper Gürlek, Mustafa Berker","doi":"10.1007/s11102-024-01396-x","DOIUrl":"10.1007/s11102-024-01396-x","url":null,"abstract":"<p><strong>Purpose: </strong>The success and outcomes of repeat endoscopic transsphenoidal surgery (ETS) for residual or recurrent Cushing's disease (CD) are underreported in the literature. This study aims to address this gap by assessing the safety, feasibility, and efficacy of repeat ETS in these patients.</p><p><strong>Methods: </strong>A retrospective analysis was conducted on 56 patients who underwent a total of 65 repeat ETS performed by a single neurosurgeon between January 2006 and December 2020. Data including demographic, clinical, laboratory, radiological, and operative details were collected from electronic medical records. Logistic regression was utilized to identify potential predictors associated with sustained remission.</p><p><strong>Results: </strong>Among the cases, 40 (61.5%) had previously undergone microscopic surgery, while 25 (38.5%) had prior endoscopic procedures. Remission was achieved in 47 (83.9%) patients after the first repeat ETS, with an additional 9 (16.1%) achieving remission after the second repeat procedure. During an average follow-up period of 97.25 months, the recurrence rate post repeat surgery was 6.38%. Sustained remission was achieved in 48 patients (85.7%), with 44 after the first repeat ETS and 4 following the second repeat ETS. Complications included transient diabetes insipidus (DI) in 5 (7.6%) patients, permanent (DI) in 2 (3%) patients, and one case (1.5%) of panhypopituitarism. Three patients (4.6%) experienced rhinorrhea necessitating reoperation. A serum cortisol level > 5 µg/dL on postoperative day 1 was associated with a reduced likelihood of sustained remission.</p><p><strong>Conclusion: </strong>Repeat ETS is a safe and effective treatment option for residual or recurrent CD with satisfactory remission rates and low rates of complications.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":" ","pages":"259-268"},"PeriodicalIF":3.3,"publicationDate":"2024-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11150181/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140922528","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}
引用次数: 0
Bone health and skeletal fragility in second- and third-line medical therapies for acromegaly: preliminary results from a pilot single center experience. 肢端肥大症二线和三线药物治疗中的骨骼健康和骨骼脆性:单中心试点经验的初步结果。
IF 3.8 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-06-01 Epub Date: 2024-05-07 DOI: 10.1007/s11102-024-01398-9
Sabrina Chiloiro, Antonella Giampietro, Amato Infante, Pier Paolo Mattogno, Liverana Lauretti, Alessandro Olivi, Laura De Marinis, Alfredo Pontecorvi, Francesco Doglietto, Antonio Bianchi

Introduction: Skeletal fragility is a clinically relevant and not-reversible complication of acromegaly, involving around 30-40% of patients since the disease diagnosis. Few studies have investigated the effects on skeletal health of medical therapies for acromegaly. In this retrospective longitudinal monocentre study, we investigated the outcome of skeletal fragility in patients treated with Pasireotide Lar in combination with Pegvisomant (Pasi-Lar + Peg-V), also comparing those observed in patients treated with conventional therapies.

Results: We included 6 patients treated with Pasi-Lar + Peg-V, 5 patients treated with Peg-V in monotherapy (m-Peg-V), 16 patients treated with Peg-V plus first-generation somatostatin receptor ligands (fg-SRLs + Peg-V), 9 patients treated with Pasi-Lar. None of the patients treated with Pasi-Lar + Peg-V experienced worsening of spine and femoral bone mineral density (BMD) and incident vertebral fractures (i-VFs). Eight patients experienced i-VFs. The frequency of i-VFs was significantly lower in patients treated with the Pasi-Lar + Peg-V (0/8; 0%), as compared to those observed in m-Peg-V treated patients (4/8; 50%, p = 0.02). The frequency of i-VFs was slightly but not significantly higher in Pasi-Lar treated patients (1/8; 12.5% p = 0.6) and in fg-SRLs + Peg-V treated patients (3/8; 37.5% p = 0.364), concerning those treated with Pasi-Lar + Peg-V (0/8; 0%). I-VFs occurred more frequently in patients with higher GH levels at acromegaly diagnosis (p < 0.001), and in patients who experienced a BMD worsening (p = 0.005).

Conclusion: Our preliminary data suggested that in conventional and multi-drug resistant acromegaly, the combination therapy Pasi-Lar + Peg-V may prevent the worsening of BMD and the occurrence of i-VFs. Prospective and translational studies should further validate these results and ascertain underlying physiopathology mechanisms.

简介骨骼脆性是肢端肥大症的一种临床相关且不可逆转的并发症,自疾病确诊以来,约有 30%-40% 的患者会出现骨骼脆性。很少有研究调查肢端肥大症药物疗法对骨骼健康的影响。在这项回顾性纵向单中心研究中,我们调查了帕西瑞罗肽拉尔与培维索孟联合疗法(帕西拉尔+培维索孟)治疗患者的骨骼脆性结果,并对传统疗法治疗患者的骨骼脆性结果进行了比较:我们纳入了 6 名接受帕西拉尔 + Peg-V 治疗的患者、5 名接受 Peg-V 单一疗法(m-Peg-V)治疗的患者、16 名接受 Peg-V 加第一代体生长抑素受体配体(fg-SRLs + Peg-V)治疗的患者、9 名接受帕西拉尔治疗的患者。在接受 Pasi-Lar + Peg-V 治疗的患者中,没有人出现脊柱和股骨骨矿物质密度(BMD)恶化以及椎体骨折(i-VFs)事件。8名患者出现了i-VFs。与接受 m-Peg-V 治疗的患者(4/8;50%,p = 0.02)相比,接受 Pasi-Lar + Peg-V 治疗的患者发生 iVFs 的频率明显降低(0/8;0%)。接受 Pasi-Lar 治疗的患者(1/8;12.5% p = 0.6)和接受 fg-SRLs + Peg-V 治疗的患者(3/8;37.5% p = 0.364)出现 i-VFs 的频率略高,但并不明显,而接受 Pasi-Lar + Peg-V 治疗的患者(0/8;0%)出现 i-VFs 的频率更高。在确诊肢端肥大症时 GH 水平较高的患者中,I-VF 的发生率更高(P我们的初步数据表明,对于传统和耐多药的肢端肥大症患者,Pasi-Lar + Peg-V 联合疗法可预防 BMD 恶化和 i-VFs 的发生。前瞻性和转化性研究应进一步验证这些结果,并确定潜在的生理病理机制。
{"title":"Bone health and skeletal fragility in second- and third-line medical therapies for acromegaly: preliminary results from a pilot single center experience.","authors":"Sabrina Chiloiro, Antonella Giampietro, Amato Infante, Pier Paolo Mattogno, Liverana Lauretti, Alessandro Olivi, Laura De Marinis, Alfredo Pontecorvi, Francesco Doglietto, Antonio Bianchi","doi":"10.1007/s11102-024-01398-9","DOIUrl":"10.1007/s11102-024-01398-9","url":null,"abstract":"<p><strong>Introduction: </strong>Skeletal fragility is a clinically relevant and not-reversible complication of acromegaly, involving around 30-40% of patients since the disease diagnosis. Few studies have investigated the effects on skeletal health of medical therapies for acromegaly. In this retrospective longitudinal monocentre study, we investigated the outcome of skeletal fragility in patients treated with Pasireotide Lar in combination with Pegvisomant (Pasi-Lar + Peg-V), also comparing those observed in patients treated with conventional therapies.</p><p><strong>Results: </strong>We included 6 patients treated with Pasi-Lar + Peg-V, 5 patients treated with Peg-V in monotherapy (m-Peg-V), 16 patients treated with Peg-V plus first-generation somatostatin receptor ligands (fg-SRLs + Peg-V), 9 patients treated with Pasi-Lar. None of the patients treated with Pasi-Lar + Peg-V experienced worsening of spine and femoral bone mineral density (BMD) and incident vertebral fractures (i-VFs). Eight patients experienced i-VFs. The frequency of i-VFs was significantly lower in patients treated with the Pasi-Lar + Peg-V (0/8; 0%), as compared to those observed in m-Peg-V treated patients (4/8; 50%, p = 0.02). The frequency of i-VFs was slightly but not significantly higher in Pasi-Lar treated patients (1/8; 12.5% p = 0.6) and in fg-SRLs + Peg-V treated patients (3/8; 37.5% p = 0.364), concerning those treated with Pasi-Lar + Peg-V (0/8; 0%). I-VFs occurred more frequently in patients with higher GH levels at acromegaly diagnosis (p < 0.001), and in patients who experienced a BMD worsening (p = 0.005).</p><p><strong>Conclusion: </strong>Our preliminary data suggested that in conventional and multi-drug resistant acromegaly, the combination therapy Pasi-Lar + Peg-V may prevent the worsening of BMD and the occurrence of i-VFs. Prospective and translational studies should further validate these results and ascertain underlying physiopathology mechanisms.</p>","PeriodicalId":20202,"journal":{"name":"Pituitary","volume":" ","pages":"303-309"},"PeriodicalIF":3.8,"publicationDate":"2024-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140870978","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}
引用次数: 0
Appearance of fluid content in Rathke's cleft cyst is associated with clinical features and postoperative recurrence rates. 拉氏裂隙囊肿内液体含量的外观与临床特征和术后复发率有关。
IF 3.8 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-06-01 Epub Date: 2024-05-18 DOI: 10.1007/s11102-024-01395-y
Takamitsu Iwata, Satoru Oshino, Youichi Saitoh, Manabu Kinoshita, Yuji Onoda, Noriyuki Kijima, Kosuke Mukai, Michio Otsuki, Haruhiko Kishima

Purpose: The contents of Rathke's cleft cysts (RCCs) vary from clear and slightly viscous to purulent. Surgical treatment of symptomatic RCCs involves removing the cyst contents, whereas additional cyst-wall opening to prevent reaccumulation is at the surgeon's discretion. The macroscopic findings of the cyst content can reflect the nature of RCCs and would aid in surgical method selection.

Methods: We retrospectively reviewed the records of 42 patients with symptomatic RCCs who underwent transsphenoidal surgery at our institute between January 2010 and March 2022. According to the intraoperative findings, cyst contents were classified into type A (purulent), type B (turbid white with mixed semisolids), or type C (clear and slightly viscous). Clinical and imaging findings and early recurrence rate (within two years) were compared according to the cyst content type.

Results: There were 42 patients classified into three types. Patients with type C were the oldest (65.4 ± 10.4 years), and type A included more females (92.9%). For magnetic resonance imaging, type-A patients showed contrast-enhanced cyst wall (92.9%), type-B patients had intracystic nodules (57.1%), and all type-C patients showed low T1 and high T2 intensities with larger cyst volumes. Fewer asymptomatic patients had type C. Preoperative pituitary dysfunction was most common in type A (71.4%). Early recurrence was observed in types A and C, which were considered candidates for cyst-wall opening.

Conclusion: The clinical characteristics and surgical prognosis of RCCs depend on the nature of their contents.

目的:拉氏裂隙囊肿(RCC)的内容物从透明、略带粘性到脓性不等。有症状的 RCC 的手术治疗包括清除囊内容物,而额外的囊壁开窗以防止再次积聚则由外科医生决定。囊肿内容物的宏观检查结果可以反映 RCC 的性质,有助于手术方法的选择:我们回顾性地查看了 2010 年 1 月至 2022 年 3 月期间在我院接受经蝶手术的 42 例无症状 RCC 患者的病历。根据术中发现,囊肿内容物被分为A型(化脓性)、B型(混有半溶液的混浊白色)和C型(清澈略带粘性)。根据囊肿内容物类型比较临床和影像学结果以及早期复发率(两年内):结果:42 名患者被分为三种类型。C型患者年龄最大(65.4 ± 10.4 岁),A型患者中女性较多(92.9%)。在磁共振成像中,A 型患者的囊壁呈对比增强(92.9%),B 型患者有囊内结节(57.1%),而所有 C 型患者均呈低 T1 和高 T2 强化,囊肿体积较大。术前垂体功能障碍最常见于A型患者(71.4%)。A型和C型患者出现早期复发,被认为是囊壁开裂的候选者:结论:RCC 的临床特征和手术预后取决于其内容物的性质。
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引用次数: 0
Changes in multi-modality management of acromegaly in a tertiary centre over 2 decades. 一家三级医疗中心二十年来对肢端肥大症多模式治疗的变化。
IF 3.8 2区 医学 Q2 ENDOCRINOLOGY & METABOLISM Pub Date : 2024-06-01 Epub Date: 2024-03-23 DOI: 10.1007/s11102-024-01387-y
V Amodru, N Sahakian, C Piazzola, R Appay, T Graillon, T Cuny, I Morange, F Albarel, M Vermalle, J Regis, H Dufour, T Brue, F Castinetti

Purpose: Acromegaly is a rare disease associated with chronic multisystem complications. New therapeutic strategies have emerged in the last decades, combining pituitary transsphenoidal surgery (TSS), radiotherapy or radiosurgery (RXT) and medical treatments.

Methods: This retrospective monocentric study focused on presentation, management and outcome of acromegaly patients diagnosed between 2000 and 2020, still followed up in 2020, with a minimum follow-up of 1 year, and comparison of the first vs. second decade of the study.

Results: 275 patients were included, 50 diagnosed before 2010 and 225 after 2010. 95% of them had normal IGF-1 levels (with or without treatment) at the last follow-up. Transsphenoidal surgery was more successful after 2010 (75% vs. 54%; p < 0.01), while tumor characteristics remained the same over time. The time from first treatment to biochemical control was shorter after 2010 than before (8 vs. 16 months; p = 0.03). Since 2010, RT was used less frequently (10% vs. 32%; p < 0.01) but more rapidly after surgery (26 vs. 53 months; p = 0.03). In patients requiring anti-secretory drugs after TSS, the time from first therapy to biochemical control was shorter after 2010 (16 vs. 29 months; p < 0.01). Tumor size, tumor invasiveness, baseline IGF-1 levels and Trouillas classification were identified as predictors of remission.

Conclusion: The vast majority of patients with acromegaly now have successful disease control with a multimodal approach. They reached biochemical control sooner in the most recent half of the study period. Future work should focus on those patients who are still uncontrolled and on the sequelae of the disease.

目的:肢端肥大症是一种与慢性多系统并发症相关的罕见疾病。过去几十年中,出现了新的治疗策略,将垂体经蝶窦手术(TSS)、放射治疗或放射外科手术(RXT)与药物治疗相结合:这项回顾性单中心研究主要关注2000年至2020年期间确诊的肢端肥大症患者的表现、管理和预后,2020年仍对其进行随访,随访时间至少1年,并对研究的第一个十年与第二个十年进行比较。结果:共纳入275例患者,其中50例在2010年之前确诊,225例在2010年之后确诊。其中95%的患者在最后一次随访时IGF-1水平正常(无论是否接受治疗)。2010年后,经蝶窦手术的成功率更高(75%对54%;P 结论:绝大多数肢端肥大症患者的IGF-1水平都在正常范围内:目前,绝大多数肢端肥大症患者都能通过多模式治疗成功控制病情。在最近的一半研究期间,他们更快地达到了生化控制。今后的工作应重点关注那些仍未得到控制的患者以及疾病的后遗症。
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引用次数: 0
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Pituitary
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