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1.
Front Endocrinol (Lausanne) ; 15: 1369729, 2024.
Article in English | MEDLINE | ID: mdl-38572480

ABSTRACT

Purpose: The purpose of this study was to explore the factors influencing PRL levels in patients with prolactinoma and to investigate the correlations between anxiety, depression, sleep, self-efficacy, and PRL levels. Methods: This retrospective study included 176 patients with prolactinoma who received outpatient treatment at the Affiliated Hospital of Zunyi Medical University from May 2017 to August 2022. The general information questionnaire, Hospital Anxiety and Depression Scale (HADS), Athens Insomnia Scale (AIS), and General Self-Efficacy Scale (GSES) were used for data collection. A generalized estimating equation (GEE) model was used to analyze the factors influencing PRL levels in patients with prolactinoma. GEE single-effect analysis was used to compare PRL levels at different time points between anxiety group and nonanxiety group, between insomnia group and normal group, and between low, medium, and high self-efficacy groups. Results: The median baseline PRL level and the PRL levels at 1, 3, 6, and 12 months of follow-up were 268.50 ng/ml, 122.25 ng/ml, 21.20 ng/ml, 19.65 ng/ml, and 16.10 ng/ml, respectively. Among patients with prolactinoma, 59.10% had anxiety (HADS-A score = 7.35 ± 3.34) and 28.98% had depression (HADS-D score = 5.23 ± 3.87), 9.10% had sleep disorders (AIS score = 6.10 ± 4.31) and 54.55% had low self-efficacy (GSES score = 2.13 ± 0.83). Educational level, tumor size, number of visits, sleep quality, anxiety level, and self-efficacy level were found to be factors influencing PRL levels in patients with prolactinoma (P<0.05). Higher PRL levels were observed in the anxiety group compared to the non-anxiety group (P<0.001), in the insomnia group compared to the normal group (P<0.05), and in the low self-efficacy group compared to the medium and high self-efficacy groups (P<0.05). Conclusion: PRL levels in patients with prolactinoma are related to education level, tumor size, number of visits, anxiety, self-efficacy, and sleep but not depression. PRL levels were higher in patients with anxiety, low self-efficacy, and sleep disorders.


Subject(s)
Pituitary Neoplasms , Prolactinoma , Sleep Initiation and Maintenance Disorders , Humans , Prolactinoma/complications , Depression , Retrospective Studies , Self Efficacy , Prolactin , Sleep , Anxiety , Pituitary Neoplasms/complications
2.
Front Endocrinol (Lausanne) ; 15: 1373869, 2024.
Article in English | MEDLINE | ID: mdl-38628582

ABSTRACT

Objective: Double pituitary adenomas (DPA) are a rare clinical condition, and our knowledge of them is limited. Missing the second lesion leading to incomplete biochemical remission after surgery is an important challenge in DPA management. This study aims to analyze independent prognostic factors in DPA patients and summarize clinical experiences to prevent surgical failure. Methods: Two cases of DPA patients with Cushing's disease diagnosed and surgically treated at Peking Union Medical College Hospital are reported. A literature review was performed on the online database Pubmed, and 57 DPA patients from 22 retrieved articles were included. Demographic characteristics, endocrine manifestations, diagnostic methods, tumor size, and immunohistochemical features of 59 patients were analyzed. Binary logistic regression models were used to identify independent prognostic factors affecting postoperative biochemical remission. Results: Among 59 DPA patients, the mean ± SD age was 43.64 ± 14.42 years, with 61.02% being female (n = 36). The most common endocrine manifestations were Cushing's syndrome (23/59, 38.98%) and acromegaly (20/59, 33.90%). The most prevalent immunohistochemical types were ACTH-immunopositive (31/118, 26.27%) and GH-immunopositive (31/118, 26.27%) tumors. Microadenomas (<1cm) were the most frequent in terms of tumor size (62/92, 67.39%). The detection rate for double lesions on 3.0T MRI was 50.00% (14/28), which significantly higher than 1.5T MRI (P = 0.034). Univariate analysis revealed that female, Cushing's syndrome and only single lesion detected by surgical exploration were associated with significantly worse prognosis (P<0.05). Multivariate analysis identified double lesion detected by surgical exploration (OR = 0.08, P = 0.003) and contiguous type tumor (OR = 0.06, P = 0.017) as independent protective factors for DPA patients. Conclusions: The double lesion detected by surgical exploration is independently associated with a better prognosis for DPA patients. Comprehensive intraoperative exploration are crucial measures to avoid missing causative lesions.


Subject(s)
Acromegaly , Adenoma , Cushing Syndrome , Pituitary ACTH Hypersecretion , Pituitary Neoplasms , Humans , Female , Adult , Middle Aged , Male , Pituitary Neoplasms/diagnosis , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Cushing Syndrome/diagnosis , Adenoma/diagnosis , Pituitary ACTH Hypersecretion/complications , Acromegaly/complications
3.
Front Endocrinol (Lausanne) ; 15: 1235441, 2024.
Article in English | MEDLINE | ID: mdl-38590825

ABSTRACT

Introduction: Transsphenoidal surgery (TSS) is the preferred surgical method for most pituitary adenomas owing to high efficacy and low mortality. This study aimed to evaluate the influence of metabolic syndrome (MetS) on postoperative outcomes of TSS for pituitary adenoma. Methods: This population-based, retrospective observational study extracted data of adults 20-79 y receiving TSS for pituitary adenoma from the US Nationwide Inpatient Sample (NIS) between 2005-2018. Primary outcomes were pituitary-related complications, poor outcomes (i.e., in-hospital mortality or unfavorable discharge), prolonged length of stay (LOS), and patient safety indicators (PSIs). Univariate and multivariate regressions were performed to determine the associations between study variables and outcomes. Results: 19,076 patients (representing a 93,185 US in-patient population) were included, among which 2,109 (11.1%) patients had MetS. After adjustment, pre-existing MetS was not significantly associated with presence of pituitary-related complications and poor outcomes. In contrast, MetS was significantly associated with an increased risk for prolonged LOS (adjusted OR (aOR) = 1.19; 95% CI: 1.05-1.34), PSIs (aOR = 1.31; 95% CI: 1.07-1.59) and greater hospital costs (adjusted ß = 8.63 thousand USD; 95% CI: 4.98-12.29). Among pituitary-related complications, MetS was independently associated with increased risk of cerebrospinal fluid (CSF) rhinorrhea (aOR = 1.22, 95% CI: 1.01, 1.47) but lowered diabetes insipidus (aOR = 0.83, 95% CI: 0.71, 0.97). Discussion: MetS does not pose excessive risk of in-hospital mortality or unfavorable discharge. However, MetS independently predicted having PSIs, prolonged LOS, greater hospital costs, and CSF rhinorrhea. Study findings may help clinicians achieve better risk stratification before TSS.


Subject(s)
Adenoma , Metabolic Syndrome , Pituitary Diseases , Pituitary Neoplasms , Adult , Humans , Pituitary Neoplasms/epidemiology , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Metabolic Syndrome/epidemiology , Metabolic Syndrome/complications , Inpatients , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Pituitary Diseases/epidemiology , Pituitary Diseases/surgery , Pituitary Diseases/complications , Adenoma/surgery
4.
Front Endocrinol (Lausanne) ; 15: 1345363, 2024.
Article in English | MEDLINE | ID: mdl-38481440

ABSTRACT

X-linked acrogigantism (X-LAG) is a rare form of pituitary gigantism that is associated with growth hormone (GH) and prolactin-secreting pituitary adenomas/pituitary neuroendocrine tumors (PitNETs) that develop in infancy. It is caused by a duplication on chromosome Xq26.3 that leads to the misexpression of the gene GPR101, a constitutively active stimulator of pituitary GH and prolactin secretion. GPR101 normally exists within its own topologically associating domain (TAD) and is insulated from surrounding regulatory elements. X-LAG is a TADopathy in which the duplication disrupts a conserved TAD border, leading to a neo-TAD in which ectopic enhancers drive GPR101 over-expression, thus causing gigantism. Here we trace the full diagnostic and therapeutic pathway of a female patient with X-LAG from 4C-seq studies demonstrating the neo-TAD through medical and surgical interventions and detailed tumor histopathology. The complex nature of treating young children with X-LAG is illustrated, including the achievement of hormonal control using a combination of neurosurgery and adult doses of first-generation somatostatin analogs.


Subject(s)
Acromegaly , Genetic Diseases, X-Linked , Gigantism , Human Growth Hormone , Pituitary Neoplasms , Adult , Humans , Child , Female , Child, Preschool , Gigantism/genetics , Gigantism/therapy , Gigantism/metabolism , Acromegaly/pathology , Growth Hormone/metabolism , Genetic Diseases, X-Linked/genetics , Genetic Diseases, X-Linked/therapy , Pituitary Neoplasms/complications , Pituitary Neoplasms/genetics , Pituitary Neoplasms/pathology
5.
J Clin Densitom ; 27(2): 101479, 2024.
Article in English | MEDLINE | ID: mdl-38447349

ABSTRACT

OBJECTIVE: Hyperprolactinemia has negative impacts on metabolism and musculoskeletal health. In this study, individuals with active prolactinoma were evaluated for nonalcoholic fatty liver disease (NAFLD) and musculoskeletal health, which are underemphasized in the literature. METHODS: Twelve active prolactinoma patients and twelve healthy controls matched by age, gender, and BMI were included. Magnetic resonance imaging-proton density fat fraction (MRI-PDFF) was used to evaluate hepatic steatosis and magnetic resonance elastography (MRE) to evaluate liver stiffness measurement (LSM). Abdominal muscle mass, and vertebral MRI-PDFF was also evaluated with MRI. Body compositions were evaluated by dual energy X-ray absorptiometry (DXA). The skeletal muscle quality (SMQ) was classified as normal, low and weak by using "handgrip strength/appendicular skeletal muscle mass (HGS/ASM)" ratio based on the cut-off values previously stated in the literature. RESULTS: Prolactin, HbA1c and CRP levels were higher in prolactinoma patients (p<0.001, p=0.033 and p=0.035, respectively). The median MRI-PDFF and MRE-LSM were 3.0% (2.01-15.20) and 2.22 kPa (2.0-2.5) in the prolactinoma group and 2.5% (1.65-10.00) and 2.19 kPa (1.92-2.54) in the control group, respectively and similiar between groups. In prolactinoma patients, liver MRI-PDFF showed a positive and strong correlation with the duration of disease and traditional risk factors for NAFLD. Total, vertebral and pelvic bone mineral density was similar between groups, while vertebral MRI-PDFF tended to be higher in prolactinoma patients (p=0.075). Muscle mass and strength parameters were similar between groups, but HGS/ASM tended to be higher in prolactinoma patients (p=0.057). Muscle mass was low in 33.3% of prolactinoma patients and 66.6 of controls. According to SMQ, all prolactinoma patients had normal SMQ, whereas 66.6% of the controls had normal SMQ. CONCLUSION: Prolactinoma patients demonstrated similar liver MRI-PDFF and MRE-LSM to controls despite their impaired metabolic profile and lower gonadal hormone levels. Hyperprolactinemia may improve muscle quality in prolactinoma patients despite hypogonadism.


Subject(s)
Absorptiometry, Photon , Magnetic Resonance Imaging , Muscle, Skeletal , Non-alcoholic Fatty Liver Disease , Pituitary Neoplasms , Prolactinoma , Humans , Pilot Projects , Non-alcoholic Fatty Liver Disease/diagnostic imaging , Non-alcoholic Fatty Liver Disease/physiopathology , Male , Female , Adult , Middle Aged , Muscle, Skeletal/diagnostic imaging , Muscle, Skeletal/physiopathology , Case-Control Studies , Prolactinoma/diagnostic imaging , Prolactinoma/physiopathology , Prolactinoma/complications , Pituitary Neoplasms/diagnostic imaging , Pituitary Neoplasms/complications , Pituitary Neoplasms/physiopathology , Elasticity Imaging Techniques , Hand Strength , Liver/diagnostic imaging , Liver/pathology , Glycated Hemoglobin , Bone Density , Prolactin/blood , Body Composition
7.
J Cancer Res Ther ; 20(1): 496-499, 2024 Jan 01.
Article in English | MEDLINE | ID: mdl-38554375

ABSTRACT

Metastasis to pituitary gland is a rare condition, and patients are usually asymptomatic. Diabetes insipidus (DI) is the most common presenting symptom, and breast cancer is the most common source of pituitary metastasis (PM). We report a case of PM of breast cancer presenting as DI. A 45-year-old female patient presented to our department with complaints of polyuria and polydipsia. She had a medical history of metastatic breast adenocarcinoma. Laboratory data showed normal fasting plasma glucose level and hypotonic urine. Brain magnetic resonance imaging (MRI) showed infiltration of the pituitary stalk and the absence of the posterior pituitary bright spot consistent with metastasis to the pituitary gland. The water deprivation and vasopressin challenge tests confirmed central DI. Pituitary function tests revealed disconnection hyperprolactinemia with a menopausal profile. The patient was treated with vasopressin with great clinical results. Pituitary metastases are rare but should be suspected in patients with metastatic cancer who present with DI.


Subject(s)
Breast Neoplasms , Diabetes Insipidus , Diabetes Mellitus , Pituitary Neoplasms , Female , Humans , Middle Aged , Breast Neoplasms/complications , Breast Neoplasms/diagnosis , Breast Neoplasms/pathology , Diabetes Insipidus/diagnosis , Diabetes Insipidus/etiology , Diabetes Insipidus/drug therapy , Magnetic Resonance Imaging , Pituitary Gland/pathology , Pituitary Neoplasms/complications , Pituitary Neoplasms/diagnosis , Vasopressins/therapeutic use
8.
Front Endocrinol (Lausanne) ; 15: 1338345, 2024.
Article in English | MEDLINE | ID: mdl-38370355

ABSTRACT

Prolactinomas (PRLomas) constitute approximately half of all pituitary adenomas and approximately one-fifth of them are diagnosed in males. The clinical presentation of PRLomas results from direct prolactin (PRL) action, duration and severity of hyperprolactinemia, and tumor mass effect. Male PRLomas, compared to females, tend to be larger and more invasive, are associated with higher PRL concentration at diagnosis, present higher proliferative potential, are more frequently resistant to standard pharmacotherapy, and thus may require multimodal approach, including surgical resection, radiotherapy, and alternative medical agents. Therefore, the management of PRLomas in men is challenging in many cases. Additionally, hyperprolactinemia is associated with a significant negative impact on men's health, including sexual function and fertility potential, bone health, cardiovascular and metabolic complications, leading to decreased quality of life. In this review, we highlight the differences in pathogenesis, clinical presentation and treatment of PRLomas concerning the male sex.


Subject(s)
Adenoma , Hyperprolactinemia , Pituitary Neoplasms , Prolactinoma , Female , Male , Humans , Prolactinoma/therapy , Prolactinoma/drug therapy , Hyperprolactinemia/diagnosis , Hyperprolactinemia/etiology , Hyperprolactinemia/therapy , Quality of Life , Pituitary Neoplasms/diagnosis , Pituitary Neoplasms/therapy , Pituitary Neoplasms/complications , Adenoma/diagnosis , Adenoma/etiology , Adenoma/therapy
9.
Front Endocrinol (Lausanne) ; 15: 1337741, 2024.
Article in English | MEDLINE | ID: mdl-38390203

ABSTRACT

This report describes a rare case of a 20-year-old man with an ACTH- and prolactin-secreting invasive pituitary macroadenoma causing hyperprolactinemia and Cushing's disease. He was later found to have an AIP mutation. Treatment with cabergoline (1.5 mg weekly) normalized prolactin concentrations and induced a major shrinkage of the adenoma. Not only was urinary free cortisol normalized for more than 14 years, but also the treatment induced normal hypothalamo-pituitary-adrenal (HPA) axis function as illustrated by the reappearance of a normal cortisol/ACTH circadian rhythm, cortisol suppression to dexamethasone, and disappearance of the excessive and aberrant responses to CRH and desmopressin, respectively. This case is the first description of complete restoration of the physiological characteristics of the HPA axis by a medication during the treatment of Cushing's disease. Although exceptional, it illustrates that drugs targeting the pituitary adenoma can bring true complete remission of Cushing's disease.


Subject(s)
Pituitary ACTH Hypersecretion , Pituitary Neoplasms , Male , Humans , Young Adult , Adult , Hypothalamo-Hypophyseal System , Pituitary ACTH Hypersecretion/complications , Pituitary ACTH Hypersecretion/drug therapy , Hydrocortisone , Prolactin , Pituitary-Adrenal System , Pituitary Neoplasms/complications , Pituitary Neoplasms/drug therapy , Adrenocorticotropic Hormone
10.
World Neurosurg ; 184: e659-e673, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38342172

ABSTRACT

BACKGROUND: Giant pituitary adenomas (GPAs) are defined as tumors with ≥40 mm in any maximum diameter, and these tend to invade multiple intracranial compartments. Hence, treatment remains a surgical challenge. OBJECTIVE: To describe the clinical and surgical outcomes of the endoscopic endonasal approach (EEA) for GPA in a pituitary referral center in Latin America and to analyze associated predictive factors. METHODS: 37 patients with histologically-confirmed GPA treated solely through the EEA between a 2-year period were included. Preoperative and postoperative clinical and neuroimaging findings; surgical morbidity and mortality; and binary logistic regression analysis to assess predictive factors were analyzed. RESULTS: Preoperative visual impairment prevalence was 97.3%. Mean tumor volume was 32 cc and gross total resection rate was 40.5%. Favorable visual acuity and visual fields outcome rate was 75% and 82.9%, respectively. In the multivariate analysis, bilateral cavernous sinus invasion (P = 0.018) and postoperative cerebrospinal fluid (CSF) leak (P = 0.036) were associated with an unfavorable visual acuity outcome, while radiation therapy (P = 0.035) was for visual fields. Similarly, intraoperative CSF leak was a predictive factor for postoperative CSF leak (10.8%) (P = 0.042) and vascular injury (13.5%) (P = 0.048). CONCLUSIONS: In this first Mexican clinical series, we demonstrated that the EEA is a safe and effective technique for GPA, although early diagnosis and prompt intervention may promote further visual function preservation without significant endocrine morbidity.


Subject(s)
Adenoma , Pituitary Diseases , Pituitary Neoplasms , Humans , Pituitary Neoplasms/diagnostic imaging , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Adenoma/diagnostic imaging , Adenoma/surgery , Adenoma/complications , Nose/surgery , Postoperative Complications/epidemiology , Treatment Outcome , Retrospective Studies
11.
BMC Endocr Disord ; 24(1): 20, 2024 Feb 08.
Article in English | MEDLINE | ID: mdl-38326790

ABSTRACT

BACKGROUND: Lymphocytic hypophysitis is a rare autoimmune condition that usually presents during pregnancy and causes inflammation of the pituitary gland. Although the pathophysiology is not well understood, it often presents with headaches, visual disturbances, and symptoms of hypopituitarism. However, not all cases may present with hypopituitarism which can make this rare disease with an incidence of ~ 1 in 9 million much more difficult to diagnose. CASE PRESENTATION: We present a 35-year-old G4P4 woman with progressive vision loss and intermittent frontal headaches during her first trimester through 2 months postpartum. She presented with no symptoms of hypopituitarism and her hormone panel only showed elevated prolactin, possibly due to her breastfeeding. She was treated with a right pterional craniotomy with decompression of both optic nerves, partial resection of the suprasellar mass, and glucocorticoid therapy for headaches and visual disturbances. CONCLUSION: This case is notable for a presentation of lymphocytic hypophysitis without symptoms of hypopituitarism. This is important for outpatient providers to be aware of, especially those that care for pregnant patients so that unfavorable outcomes can be avoided.


Subject(s)
Autoimmune Hypophysitis , Hypopituitarism , Pituitary Diseases , Pituitary Neoplasms , Humans , Pregnancy , Female , Adult , Pituitary Neoplasms/complications , Pituitary Neoplasms/diagnosis , Pituitary Neoplasms/surgery , Autoimmune Hypophysitis/diagnosis , Autoimmune Hypophysitis/complications , Pituitary Diseases/diagnosis , Pituitary Diseases/complications , Hypopituitarism/diagnosis , Hypopituitarism/etiology , Pituitary Hormones , Headache/etiology , Headache/complications , Magnetic Resonance Imaging
12.
Neurol Med Chir (Tokyo) ; 64(4): 160-167, 2024 Apr 15.
Article in English | MEDLINE | ID: mdl-38355129

ABSTRACT

Prolactin-producing pituitary tumor (PRLoma) is the most prevalent functional pituitary tumor. If the tumor becomes large, vision can be impaired. In contrast to other pituitary tumors, cabergoline (CAB) is extremely effective for PRLoma and has become the first-line treatment. In this study, we examined our experience with the pharmacological and surgical management of PRLomas with visual impairment (VI) to determine whether VI could be a surgical indication. Further, we discussed the function of surgery in situations where the gold standard of PRLoma treatment was CAB administration. Of the 159 patients with PRLomas (age, 13-77 [mean = 36.3] years; men, 29; women, 130) at Tokyo Women's Medical University Hospital from 2009 to 2021, 18 (age, 15-67 [mean = 35.8] years; men, 12; woman, 6) had VI (subjectively, 12; objectively, 6). They started CAB treatment immediately (maximum dose: 0.5 to 6 mg/week; average: 2.17 mg/week). VI improved in 16 patients (88.9%) but did not improve in 2 (11.1%) requiring surgeries. One of the two patients had a parenchymal tumor resistant to CAB, and the other had a cystic tumor due to intratumoral bleeding. Consequently, CAB is the first-line treatment for PRLomas with VI because of its significantly high rate of improvement. However, close and rigorous surveillance is necessary for cases resistant to CAB, and the correct decision is required regarding surgical interventions at proper timing and appropriate surgical approaches considering the purpose of surgery.


Subject(s)
Antineoplastic Agents , Pituitary Neoplasms , Prolactinoma , Male , Humans , Female , Adolescent , Young Adult , Adult , Middle Aged , Aged , Pituitary Neoplasms/complications , Pituitary Neoplasms/surgery , Prolactinoma/complications , Prolactinoma/drug therapy , Prolactinoma/surgery , Prolactin/therapeutic use , Ergolines/adverse effects , Antineoplastic Agents/therapeutic use , Cabergoline/therapeutic use , Vision Disorders/chemically induced , Vision Disorders/drug therapy , Dopamine Agonists/therapeutic use
13.
Ann Intern Med ; 177(3): 315-323, 2024 Mar.
Article in English | MEDLINE | ID: mdl-38373302

ABSTRACT

BACKGROUND: Glucocorticoids suppress inflammation. Autoimmune disease may occur after remission of Cushing's disease (CD). However, the development of autoimmune disease in this context is not well described. OBJECTIVE: To determine 1) the incidence of autoimmune disease in patients with CD after surgical remission compared with patients with nonfunctioning pituitary adenomas (NFPAs) and 2) the clinical presentation of and risk factors for development of autoimmune disease in CD after remission. DESIGN: Retrospective matched cohort analysis. SETTING: Academic medical center/pituitary center. PATIENTS: Patients with CD with surgical remission and surgically treated NFPA. MEASUREMENTS: Cumulative incidence of new-onset autoimmune disease at 3 years after surgery. Assessment for hypercortisolemia included late-night salivary cortisol levels, 24-hour urine free cortisol (UFC) ratio (UFC value divided by the upper limit of the normal range for the assay), and dexamethasone suppression tests. RESULTS: Cumulative incidence of new-onset autoimmune disease at 3 years after surgery was higher in patients with CD (10.4% [95% CI, 5.7% to 15.1%]) than in those with NFPAs (1.6% [CI, 0% to 4.6%]) (hazard ratio, 7.80 [CI, 2.88 to 21.10]). Patients with CD showed higher prevalence of postoperative adrenal insufficiency (93.8% vs. 16.5%) and lower postoperative nadir serum cortisol levels (63.8 vs. 282.3 nmol/L) than patients with NFPAs. Compared with patients with CD without autoimmune disease, those who developed autoimmune disease had a lower preoperative 24-hour UFC ratio (2.7 vs. 6.3) and a higher prevalence of family history of autoimmune disease (41.2% vs. 20.9%). LIMITATION: The small sample of patients with autoimmune disease limited identification of independent risk factors. CONCLUSION: Patients achieving surgical remission of CD have higher incidence of autoimmune disease than age- and sex-matched patients with NFPAs. Family history of autoimmune disease is a potential risk factor. Adrenal insufficiency may be a trigger. PRIMARY FUNDING SOURCE: Recordati Rare Diseases Inc.


Subject(s)
Adrenal Insufficiency , Autoimmune Diseases , Pituitary ACTH Hypersecretion , Pituitary Neoplasms , Humans , Cohort Studies , Hydrocortisone , Retrospective Studies , Pituitary ACTH Hypersecretion/complications , Pituitary ACTH Hypersecretion/surgery , Pituitary Neoplasms/complications , Pituitary Neoplasms/surgery , Adrenal Insufficiency/complications , Autoimmune Diseases/complications
15.
Pituitary ; 27(2): 91-128, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38183582

ABSTRACT

PURPOSE: Pituitary adenoma surgery is a complex procedure due to critical adjacent neurovascular structures, variations in size and extensions of the lesions, and potential hormonal imbalances. The integration of artificial intelligence (AI) and machine learning (ML) has demonstrated considerable potential in assisting neurosurgeons in decision-making, optimizing surgical outcomes, and providing real-time feedback. This scoping review comprehensively summarizes the current status of AI/ML technologies in pituitary adenoma surgery, highlighting their strengths and limitations. METHODS: PubMed, Embase, Web of Science, and Scopus were searched following the PRISMA-ScR guidelines. Studies discussing the use of AI/ML in pituitary adenoma surgery were included. Eligible studies were grouped to analyze the different outcomes of interest of current AI/ML technologies. RESULTS: Among the 2438 identified articles, 44 studies met the inclusion criteria, with a total of seventeen different algorithms utilized across all studies. Studies were divided into two groups based on their input type: clinicopathological and imaging input. The four main outcome variables evaluated in the studies included: outcome (remission, recurrence or progression, gross-total resection, vision improvement, and hormonal recovery), complications (CSF leak, readmission, hyponatremia, and hypopituitarism), cost, and adenoma-related factors (aggressiveness, consistency, and Ki-67 labeling) prediction. Three studies focusing on workflow analysis and real-time navigation were discussed separately. CONCLUSION: AI/ML modeling holds promise for improving pituitary adenoma surgery by enhancing preoperative planning and optimizing surgical strategies. However, addressing challenges such as algorithm selection, performance evaluation, data heterogeneity, and ethics is essential to establish robust and reliable ML models that can revolutionize neurosurgical practice and benefit patients.


Subject(s)
Adenoma , Hypopituitarism , Pituitary Neoplasms , Humans , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Artificial Intelligence , Adenoma/surgery , Adenoma/complications , Hypopituitarism/etiology , Algorithms
16.
Endocr J ; 71(3): 285-293, 2024 Mar 28.
Article in English | MEDLINE | ID: mdl-38281757

ABSTRACT

Pituitary xanthogranulomatomas (XG) are a rare pathological entity caused by accumulation of lipid laden macrophages and reactive granuloma formation usually triggered by cystic fluid leakage or hemorrhage. Our aim was to compare clinical characteristics and presenting features of patients with secondary etiology of XG and those with no identifiable founding lesion (primary -"pure" XG) in order to gain new insights into this rare pituitary pathology. In a retrospective review of 714 patients operated for sellar masses, at tertiary center, we identified 16 (2.24%) with histologically confirmed diagnosis of pituitary XG over the period of 7 years (2015-2021). Patients were further analyzed according to XG etiology: "pure"- XG (n = 8) with no identifiable founding lesion were compared to those with histological elements of pituitary tumor or cyst - secondary XG (n = 8). We identified 16 patients (11 male), mean age 44.8 ± 22.3 years, diagnosed with pituitary XG. Secondary forms were associated with Ratke's cleft cyst (RCC, n = 2) and pituitary adenoma (PA, n = 6). The most common presenting features in both groups were hypopituitarism (75%), headache (68.5%) and visual disturbances (37.5%). Predominance of male sex was noted (males 68.75%, females 31.25%), especially in patients with primary forms. Patients with primary pituitary XG were all males (p = 0.0256) and more frequently affected by panhypopituitarism (87.5% vs. 25%, p = 0.0406) compared to patients with secondary causes. Hyperprolactinemia was noted in pituitary tumor group with secondary etiology only (p = 0.0769). Majority of lesions were solid on magnetic resonance imaging - MRI (81.25%). Distinct clinical phenotype was observed dependent on the etiology of XG.


Subject(s)
Central Nervous System Cysts , Cysts , Pituitary Diseases , Pituitary Neoplasms , Xanthomatosis , Female , Humans , Male , Young Adult , Adult , Middle Aged , Aged , Pituitary Neoplasms/complications , Pituitary Neoplasms/diagnostic imaging , Pituitary Neoplasms/epidemiology , Pituitary Diseases/epidemiology , Pituitary Gland/diagnostic imaging , Pituitary Gland/pathology , Magnetic Resonance Imaging , Central Nervous System Cysts/complications , Cysts/pathology , Granuloma/complications , Granuloma/pathology , Xanthomatosis/epidemiology , Xanthomatosis/pathology
17.
Neuro Endocrinol Lett ; 45(1): 76-80, 2024 Jan 31.
Article in English | MEDLINE | ID: mdl-38295430

ABSTRACT

INTRODUCTION: Recurrent Cushing's disease (recurrent CD) is an uncommon and intricate clinical form of Cushing's syndrome. However, the connection between the pathological types of ACTH-secreting PitNETs and the clinical signs of recurrent CD remains uncertain. CASE DESCRIPTION: A 64-year-old woman, previously diagnosed with renal carcinoma, was admitted to our hospital due to recent weight gain. Previous endocrine tests indicated fluctuating hypercortisolemia and a recurrent pituitary tumor over the past six years. She underwent two transsphenoidal hypophysectomies, and histopathological analysis of the tumor revealed it as a densely granulated corticotroph tumor (DGCT), a subtype of TPIT-lineage PitNET, accompanied by tumor apoplexy. CONCLUSION: This case highlights the connection between recurrent CD and the pathological subtypes of TPIT-lineage DGCT-PitNETs.


Subject(s)
Kidney Neoplasms , Neuroendocrine Tumors , Pituitary ACTH Hypersecretion , Pituitary Neoplasms , Female , Humans , Middle Aged , Pituitary ACTH Hypersecretion/complications , Pituitary ACTH Hypersecretion/surgery , Neuroendocrine Tumors/complications , Neuroendocrine Tumors/surgery , Corticotrophs/pathology , Pituitary Neoplasms/complications , Pituitary Neoplasms/diagnostic imaging , Pituitary Neoplasms/surgery , Kidney Neoplasms/complications
18.
PLoS One ; 19(1): e0296387, 2024.
Article in English | MEDLINE | ID: mdl-38236816

ABSTRACT

Strong lines of evidence in the neuroscience literature indicate that (a) healthy sleep facilitates cognitive processing, and (b) sleep disruption is associated with cognitive dysfunction. Despite the fact that patients with pituitary disease often display both disrupted sleep and cognitive dysfunction, few previous studies investigate whether these clinical characteristics in these patients might be related. Hence, we explored whether sleep disruption in patients with pituitary disease mediates their cognitive dysfunction. We recruited 18 patients with non-functioning pituitary adenomas (NFPA) and 19 sociodemographically matched healthy controls. They completed the Global Sleep Assessment Questionnaire (thus providing self-report data regarding sleep disruption) and were administered the Brief Test of Adult Cognition by Telephone, which assesses cognitive functioning in the domains of processing speed, working memory, episodic memory, inhibition, and reasoning. We found no significant differences in cognition between patients and controls. Furthermore, spectra of sleep disturbance did not differ significantly between patients and controls. Our data suggest that NFPA patients' cognition and sleep quality is relatively intact, and that sleep disruption does not mediate cognitive dysfunction. Larger studies should characterize sleep and cognition in patients with NFPA (and other pituitary diseases) to confirm whether disruption of the former mediates impairment in the latter.


Subject(s)
Cognition Disorders , Pituitary Neoplasms , Adult , Humans , Pituitary Neoplasms/complications , South Africa/epidemiology , Cognition Disorders/psychology , Cognition , Sleep , Neuropsychological Tests
19.
J Neurooncol ; 166(1): 51-57, 2024 Jan.
Article in English | MEDLINE | ID: mdl-38224403

ABSTRACT

PURPOSE: Craniopharyngiomas can be aggressive leading to significant complications and morbidity. It is not clear whether there are any predictive factors for incidence or outcomes. Our aim was therefore to record the incidence, presentation, characteristics and progression of paediatric craniopharyngiomas in the West of Scotland. METHOD: Retrospective case note review for children diagnosed with paediatric craniopharyngiomas at the Royal Hospital for Children Glasgow, from 1995 to 2021 was conducted. All analyses were conducted using GraphPad Prism 9.4.0. RESULTS: Of 21 patients diagnosed with craniopharyngiomas, the most common presenting symptoms were headaches (17/21, 81%); visual impairment (13/21, 62%); vomiting (9/21, 43%) and growth failure (7/21, 33%). Seventeen (81%) patients underwent hydrocephalus and/or resection surgery within 3 months of diagnosis, usually within the first 2 weeks (13/21, 62%). Subtotal resection surgeries were performed in 71% of patients, and median time between subsequent resection surgeries for tumour recurrence was 4 years (0,11). BMI SDS increased at 5 year follow-up (p = 0.021) with 43% being obese (BMI > + 2SD). More patients acquired hypopituitarism post-operatively (14/16, 88%) compared to pre-operatively (4/15, 27%). A greater incidence of craniopharyngiomas were reported in more affluent areas (10/21, 48%) (SIMD score 8-10) compared to more deprived areas (6/10, 29%) (SIMD score 1-3). Five patients (24%) died with a median time between diagnosis and death of 9 years (6,13). CONCLUSION: Over 25 years the management of craniopharyngioma has changed substantially. Co-morbidities such as obesity are difficult to manage post-operatively and mortality risk can be up to 25% according to our cohort.


Subject(s)
Craniopharyngioma , Pituitary Neoplasms , Child , Humans , Craniopharyngioma/complications , Craniopharyngioma/epidemiology , Craniopharyngioma/surgery , Treatment Outcome , Retrospective Studies , Pituitary Neoplasms/complications , Pituitary Neoplasms/epidemiology , Pituitary Neoplasms/surgery , Neoplasm Recurrence, Local/surgery , Postoperative Complications/etiology
20.
J Neurosurg Pediatr ; 33(1): 73-84, 2024 Jan 01.
Article in English | MEDLINE | ID: mdl-37948683

ABSTRACT

OBJECTIVE: The optimal surgical approach for pediatric craniopharyngiomas (CPs) remains a matter of debate, with selection bias classically precluding a fair comparison of outcomes between the transcranial approach (TCA) and endoscopic endonasal approach (EEA). The purpose of this systematic review was to analyze the current role of EEA in the treatment of pediatric CPs and to determine whether, upon expansion of its indications, a comparison with TCA is valid. METHODS: A systematic review of English-language articles published between February 2010 and June 2022 was performed to identify studies in the MEDLINE (PubMed) and Embase databases reporting on the resection of pediatric CPs. Included were articles reporting on pediatric CPs removed through TCA or EEA. Case reports, review articles, and earlier or less comprehensive series by the same center were excluded. Baseline characteristics and outcomes were analyzed. Prediction intervals (PIs), heterogeneity (Q, I2, and τ2 statistics), and publication bias (funnel plot analysis) were assessed. RESULTS: A total of 835 patients underwent TCA (18 articles) and 403 patients underwent EEA (19 articles). Preoperatively, the mean patient age (p = 0.055, PI = 5.05-15.11), visual impairment (p = 0.08, PI = 19.1-90.5, I2 = 80%), and hypothalamic syndrome (p = 0.17, PI = 6.5-52.2, I2 = 62%) did not significantly differ between the EEA and TCA groups. Endocrine deficit (anterior pituitary deficit [p < 0.001, PI = 16.5-92.9, I2 = 81%] and diabetes insipidus [p < 0.001, PI = 6.3-60.6, I2 = 43%]) was more frequent in the EEA group. Hydrocephalus and signs/symptoms of raised intracranial pressure were significantly higher (p < 0.001, PI = 5.2-73.3, I2 = 70% vs p < 0.001, PI = 4.6-73, I2 = 62%, respectively) in the TCA group. Recurrent lesions (p = 0.52, PI = 2.7-87.3, I2 = 13%), tumor size (p = 0.25, PI = 22.1-56.8), third ventricle involvement (p = 0.053, PI = 10.9-81.3, I2 = 69%), and hypothalamic involvement (p = 0.06, PI = 8.5-83.6, I2 = 79%) did not differ significantly between the approaches. EEA was preferred (p = 0.006, PI = 26.8-70.8, I2 = 40%) for sellar-suprasellar CPs, whereas TCA was preferred for purely suprasellar CPs (p = 0.007, PI = 13.5-81.1, I2 = 61%). There was no difference between the approaches for purely intrasellar lesions (p = 0.94, PI = 0-62.7, I2 = 26%). The breadth of PIs, I2 values, and analysis of publication bias showed substantial variability among the pooled data, hindering the possibility of outcome meta-analyses. CONCLUSIONS: With the adoption of extended approaches, the use of EEA became appropriate for a wider spectrum of pediatric CPs, with associated excellent outcomes. Although a fair comparison between outcomes in the EEA and TCA groups was hindered because of the differences in patient populations and tumor subtypes, given the increased versatility of EEA and improved expertise in its use, surgeons can now select the optimal surgical approach based on the unique benefits and drawbacks of each pediatric CP.


Subject(s)
Craniopharyngioma , Hydrocephalus , Neuroendoscopy , Pituitary Neoplasms , Humans , Child , Craniopharyngioma/diagnostic imaging , Craniopharyngioma/surgery , Craniopharyngioma/complications , Neuroendoscopy/adverse effects , Pituitary Neoplasms/diagnostic imaging , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Neurosurgical Procedures/adverse effects , Hydrocephalus/surgery , Postoperative Complications/etiology , Postoperative Complications/surgery , Treatment Outcome , Retrospective Studies
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