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3.
J Cancer Res Ther ; 20(1): 496-499, 2024 Jan 01.
Article En | MEDLINE | ID: mdl-38554375

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.


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
4.
Turk Neurosurg ; 34(2): 331-342, 2024.
Article En | MEDLINE | ID: mdl-38497187

AIM: To share the surgical outcomes of 31 patients who underwent endoscopic endonasal transsphenoidal surgery (EETS) at a single center. MATERIAL AND METHODS: This retrospective analysis of 31 craniopharyngioma cases (2013-2022) with a minimum 6-month follow-up included demographic data, preoperative findings, postoperative resection volumes, recurrence rates, pathological diagnoses, and complications. RESULTS: Herein, 34 EETS surgeries were performed on 31 patients (12 males, 19 females). The presenting symptoms included visual loss (58%), hypopituitarism (54.8%), and diabetes insipidus (25.8%). Gross total resection was achieved in 87% of the patients, with 64.5% total and 22.5% near-total resection. Total resection prevented recurrences, contrasting with 75% recurrence in the subtotal resection patients (p=0.000). The primary patients showed 73.1% total resection, while only 20% of the recurrent patients achieved it (p=0.049). When comparing the first 16 cases with the last 15 cases in terms of surgical experience, the rates of resection (p=0.040) and recurrence-free survival (p=0.020) in the last 15 cases were statistically significant. Patients with preoperative visual loss demonstrated 94.4% improvement or stability postoperatively. Postoperative complications included hypopituitarism (71.4%), permanent diabetes insipidus (60.8%), worsening vision (6.5%), cerebrospinal fluid leakage (9.7%), meningitis (6.5%), and a 3.2% perioperative mortality rate. CONCLUSION: This study underscores the role of surgical resection in craniopharyngiomas, emphasizing the impact of surgical experience on recurrence-free survival. Primary surgery, with minimal complications and maximal resection, is crucial in managing recurrence challenges. Endoscopic endonasal transsphenoidal surgery, particularly in experienced centers, offers advantages such as panoramic vision and access to the third ventricle base, facilitating total and near-total resection and extending recurrence-free survival.


Craniopharyngioma , Diabetes Insipidus , Hypopituitarism , Pituitary Neoplasms , Male , Female , Humans , Craniopharyngioma/surgery , Retrospective Studies , Treatment Outcome , Pituitary Neoplasms/surgery , Pituitary Neoplasms/pathology , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Hypopituitarism/etiology , Diabetes Insipidus/etiology , Diabetes Insipidus/complications , Vision Disorders/etiology
5.
Praxis (Bern 1994) ; 113(1): 23-26, 2024 Jan.
Article De | MEDLINE | ID: mdl-38381107

INTRODUCTION: A 27-year-old man presented due to unilateral leg pain. He had a history of diabetes insipidus and panhypopituitarism. Laboratory analysis revealed hormonal undersupply. MRI showed a large contrast medium-absorbing mass in the pituitary gland extending into the hypothalamus. FDG-PET/CT examination revealed a hypermetabolic soft tissue lesion around the left femoral shaft. After biopsy of the lesion, a diagnosis of multisystemic Langerhans cell histiocytosis was made.


Diabetes Insipidus , Diabetes Mellitus , Hypopituitarism , Male , Humans , Adult , Positron Emission Tomography Computed Tomography , Hypopituitarism/diagnosis , Hypopituitarism/etiology , Diabetes Insipidus/diagnosis , Diabetes Insipidus/etiology , Pain/etiology
6.
Endocr Pract ; 30(2): 141-145, 2024 Feb.
Article En | MEDLINE | ID: mdl-38029928

OBJECTIVE: Adipsic diabetes insipidus (ADI) is a life-threatening disease. It is characterized by arginine vasopressin deficiency and thirst absence. Data about clinical characteristics of ADI were scarce. This study investigated the clinical features of hospitalized ADI patients. METHODS: A retrospective study was conducted of hospitalized ADI patients admitted to the Endocrinology Department of Huashan Hospital between January 2014 and December 2021, and compared with central diabetes insipidus (CDI) patients with normal thirst. RESULTS: During the study period, there were a total of 507 hospitalized CDI patients, among which 50 cases were ADI, accounting for 9.9%. Forty percent of ADI patients were admitted due to hypernatremia, but there were no admissions due to hypernatremia in the control group. The lesions of ADI patients were more likely to be located in the suprasellar area (100% vs 66%, P < .05). Higher prevalence of hypothalamic dysfunction (76% vs 8%, P < .001), central hypothyroidism (100% vs 90%, P = .031), hyperglycemia (66% vs 32%, P < .001), dyslipidemia (92% vs 71%, P = .006), and hyperuricemia (64% vs 37%, P = .003) was found in the ADI group than in the control group. The proportions of hypernatremia were higher in the ADI group both at admission and at discharge (90% vs 8%, 68% vs 8%, respectively, both with P < .001), contributing to higher prevalence of complications, such as renal insufficiency, venous thrombosis, and infection. CONCLUSION: ADI patients were found with higher prevalence of hypernatremia, hypopituitarism, hypothalamic dysfunction, metabolic disorders, and complications, posing a great challenge for comprehensive management.


Diabetes Insipidus, Neurogenic , Diabetes Insipidus , Diabetes Mellitus , Hypernatremia , Humans , Hypernatremia/etiology , Hypernatremia/complications , Retrospective Studies , Diabetes Insipidus/etiology , Diabetes Insipidus/complications , Diabetes Insipidus, Neurogenic/epidemiology , Diabetes Insipidus, Neurogenic/etiology , Thirst
7.
Acta Neurochir (Wien) ; 165(9): 2435-2444, 2023 09.
Article En | MEDLINE | ID: mdl-37530890

PURPOSE: Although Rathke cleft cysts (RCC) are benign lesions of the sellar region, recurrence is frequent after surgical treatment. Nuclear translocation of ß-catenin (NTßC), a key effector of the wnt-signaling pathway that is responsible for cell renewal, has been shown to act as a proto-oncogene and is considered to be a potential risk factor for increased recurrence in RCC. In this study, we analyzed a surgically treated cohort into patients with and without NTßC expression in order to identify clinical and imaging differences and further evaluate the risk of recurrence. METHODS: Patients with resection of RCC between 04/2001 and 11/2020 were included. Histological specimens were immunohistochemically stained for ß-catenin. Study endpoints were time to cyst recurrence (TTR) and functional outcome. Functional outcome included ophthalmological and endocrinological data. Furthermore, MRI data were assessed. RESULTS: Seventy-three patients (median age 42.3 years) with RCC underwent mainly transsphenoidal cyst resection (95.9%), 4.1% via transcranial approach. Immunohistochemical staining for ß-catenin was feasible in 61/73 (83.6%) patients, with nuclear translocation detected in 13/61 cases (21.3%). Patients with and without NTßC were equally likely to present with endocrine dysfunction before surgery (p = 0.49). Postoperative new hypopituitarism occurred in 14/73 (19.2%) patients. Preoperative visual impairment was equal in both groups (p = 0.52). Vision improved in 8/21 (33.3%) patients and visual field deficits in 22/34 (64.7%) after surgery. There was no difference in visual and perimetric outcome between patients with and without NTßC (p = 0.45 and p = 0.23, respectively). On preoperative MRI, cyst volume (9.9 vs. 8.2 cm3; p = 0.4) and evidence of hemorrhage (30.8% vs. 35.4%; p = 0.99) were equal and postoperative cyst volume decreased significantly in both groups (0.7 vs. 0.5 cm3; p < 0.0001 each). Cyst progression occurred in 13/73 (17.8%) patients after 39.3 ± 60.3 months. Cyst drainage with partial removal of the cyst wall resulted in improved recurrence-free survival without increasing the risk of complications compared with cyst fenestration alone. Patients with postoperative diabetes insipidus had an increased risk for recurrence according to multivariate analysis (p = 0.005). NTßC was evident in 4/15 patients (26.7%) and was not associated with a higher risk for recurrence (p = 0.67). CONCLUSION: Transnasal transsphenoidal cyst drainage with partial removal of the cyst wall reduces the risk of recurrence without increasing the risk of complications compared with fenestration of the cyst alone. Patients with postoperative diabetes insipidus seem to have an increased risk for recurrence. In contrast, NTßC was not associated with a higher risk of recurrence and did not provide stratification for clinically distinct patients.


Carcinoma, Renal Cell , Central Nervous System Cysts , Cysts , Diabetes Insipidus , Kidney Neoplasms , Humans , Adult , beta Catenin , Central Nervous System Cysts/diagnostic imaging , Central Nervous System Cysts/surgery , Central Nervous System Cysts/complications , Diabetes Insipidus/etiology , Magnetic Resonance Imaging/adverse effects , Catenins , Retrospective Studies , Cysts/complications , Treatment Outcome
8.
J Investig Med High Impact Case Rep ; 11: 23247096231186046, 2023.
Article En | MEDLINE | ID: mdl-37431875

Either optic neuritis (neuropathy) or hypopituitarism has been known to occur separately after COVID-19 vaccination. In this report, we describe the rare combination of hypophysitis and optic neuritis which occurred after COVID-19 vaccination. A 74-year-old woman began to have thirst, polydipsia, and polyuria, and was diagnosed as central diabetes insipidus 1 month after the fourth COVID-19 mRNA vaccine. Head magnetic resonance imaging (MRI) disclosed the thickened pituitary stalk and enlarged pituitary gland with high contrast enhancement as well as the absence of high-intensity signals in the posterior pituitary lobe on the T1-weighted image, leading to the diagnosis of lymphocytic hypophysitis. She was well with desmopressin nasal spray until further 2 months later, when she developed bilateral optic neuritis, together with gait disturbance, intention tremor of the upper extremities, urinary retention, constipation, abnormal sensation in the distal part of the lower extremities, and moderate hemiplegia on the left side. Autoantibodies, including anti-aquaporin 4 (AQP4) and anti-myelin oligodendrocyte glycoprotein (MOG), were all negative. She showed multifocal spinal cord lesions on MRI and oligoclonal bands in the cerebrospinal fluid obtained by spinal tap, and so underwent steroid pulse therapy with methylprednisolone in the tentative diagnosis of multiple sclerosis, resulting in visual acuity recovery and alleviation of neurological symptoms. In the literature review, the combination of optic neuritis and hypophysitis, mostly with diabetes insipidus, was reported in 15 patients as case reports before the years of COVID-19 pandemic. The COVID-19 vaccination would trigger the onset of hypophysitis and optic neuritis in this patient.


COVID-19 , Diabetes Insipidus , Diabetes Mellitus , Hypophysitis , Optic Neuritis , Female , Humans , COVID-19 Vaccines/adverse effects , Pandemics , COVID-19/complications , Diabetes Insipidus/etiology , mRNA Vaccines
9.
Ann Endocrinol (Paris) ; 84(4): 413-423, 2023 Aug.
Article En | MEDLINE | ID: mdl-37019429

PURPOSE: Endocrine disorders are the most frequent postoperative complications in patients undergoing pituitary surgery. Given the absence of recent guidelines on the postoperative care following pituitary surgery, this article summarizes the available evidence on the topic. METHOD: We conducted a systematic search of PubMed up to 2021 and updated the search in December 2022. We retrieved 119 articles and included 53 full-text papers. RESULTS: The early postoperative care consists of the assessment for cortisol deficiency and diabetes insipidus (DI). Experts suggest that all patients should receive a glucocorticoid (GC) stress dose followed by a rapid taper. The decision for GC replacement after discharge depends on the morning plasma cortisol level on day 3 after surgery. Experts suggest that patients with a morning plasma cortisol<10 mcg/dL should receive GC replacement at discharge, and those with 10-18 mcg/dL a morning dose only, with formal assessment of the hypothalamic-pituitary-adrenal axis at week 6 postoperatively. When the cortisol level is>18 mcg/dL, the patient can be discharged safely without GC, as suggested by observational studies. Postoperative care also includes a close monitoring of water balance. If DI develops, desmopressin is used only in case of uncomfortable polyuria or hypernatremia. The assessment of other hormones is indicated at 3 months postoperatively and beyond. CONCLUSION: The evaluation and treatment of patients following pituitary surgery are based on expert opinion and a few observational studies. Further research is needed to provide additional evidence on the most appropriate approach.


Pituitary Diseases , Pituitary Neoplasms , Postoperative Complications , Humans , Diabetes Insipidus/etiology , Glucocorticoids/therapeutic use , Hydrocortisone , Hypothalamo-Hypophyseal System , Pituitary Diseases/surgery , Pituitary Neoplasms/surgery , Pituitary-Adrenal System , Clinical Protocols/standards , Postoperative Complications/diagnosis , Postoperative Complications/therapy
10.
Rev Med Liege ; 78(4): 189-192, 2023 Apr.
Article Fr | MEDLINE | ID: mdl-37067833

One of the difficult challenges in endocrinology is the etiological diagnosis of isolated thickened pituitary stalk (PS). We report the case of a woman in whom a thickened PS was diagnosed following the onset of central diabetes insipidus revealed by polyuria-polydypsia syndrome of late pregnancy and postpartum. The pituitary exploration showed panhypopituitarism with disconnecting hyperprolactinemia. An etiological investigation for an inflammatory, granulomatous or tumour cause was carried out, but was negative. Postpartum lymphocytic hypophysitis was then retained. However, the course was puzzling with a control pituitary MRI showing disappearance of the PS thickening with paradoxical appearance of a supra-pituitary tumour, the biopsy of which concluded of being a Langerhansian histiocytosis. This paradoxical sequence is unusual and has not been reported before. It called into question the autoimmune lymphocytic origin of the thickened PS, initially considered, and raised the likelihood of a causal relationship between this PS thickening and Langerhansian histiocytosis.


Le diagnostic étiologique d'un épaississement isolé de la tige pituitaire (TP) constitue l'un des grands défis en endocrinologie. Nous rapportons le cas d'une patiente chez qui un épaississement de la TP a été diagnostiqué suite à la survenue d'un diabète insipide central révélé par un syndrome polyuro-polydypsique de fin de grossesse et du post-partum. Le bilan hypophysaire a montré un panhypopituitarisme avec une hyperprolactinémie de déconnexion. Une enquête étiologique à la recherche d'une cause inflammatoire, granulomateuse ou tumorale a été menée et s'est avérée négative. Une hypophysite lymphocytaire du post-partum a alors été retenue. Cependant, l'évolution a été déroutante avec, à l'IRM hypophysaire de contrôle, la disparition de l'épaississement de la TP et l'apparition paradoxale d'une tumeur suprahypophysaire dont la biopsie a conclu à une histiocytose langerhansienne. Cette évolution paradoxale est inhabituelle et n'a pas été rapportée auparavant. Elle a remis en question l'origine lymphocytaire auto-immune de l'épaississement de la TP, retenue initialement, et a soulevé la possibilité d'une relation de cause à effet entre cet épaississement de la TP et l'histiocytose langerhansienne.


Diabetes Insipidus , Histiocytosis , Pituitary Diseases , Female , Humans , Pregnancy , Diabetes Insipidus/diagnosis , Diabetes Insipidus/etiology , Pituitary Diseases/complications , Pituitary Diseases/diagnosis , Pituitary Diseases/pathology , Pituitary Gland/diagnostic imaging , Pituitary Gland/pathology , Histiocytosis/complications , Histiocytosis/pathology , Magnetic Resonance Imaging/adverse effects
11.
World Neurosurg ; 175: e636-e643, 2023 Jul.
Article En | MEDLINE | ID: mdl-37030477

OBJECTIVE: To analyze and find risk factors associated with developing transient diabetes insipidus (DI) using a multicenter case series after trans-sphenoidal surgery. METHODS: Medical records of patients who underwent trans-sphenoidal surgery for pituitary adenoma resection between 2010 and 2021 at 3 different neurosurgical centers by 4 experienced neurosurgeons were retrospectively analyzed. The patients were divided into 2 groups (DI group or control group). Logistic regression analysis was conducted to identify risk factors associated with postoperative DI. Univariate logistic regression was performed to identify variables of interest. Covariates with a P value <0.05 were incorporated into multivariate logistic regression models to identify independently associated risk factors for DI. All statistical tests were conducted using RStudio. RESULTS: A total of 344 patients were included; 68% were women, the mean age was 46.5 years, and nonfunctioning adenomas were the most frequent (171, 49.7%). The mean tumor size was 20.3 mm. Covariates associated with postoperative DI were age, female gender, and gross total resection. The multivariable model showed that age (odds ratio [OR] 0.97, CI 0.95-0.99, P = 0.017) and female gender (OR 2.92, CI 1.50-6.03, P = 0.002) remained significant predictors of DI development. Gross total resection was no longer a significant predictor of DI in the multivariable model (OR 1.86, CI 0.99-3.71, P = 0.063), suggesting that this variable may be confounded by other factors. CONCLUSIONS: The independent risk factors for the development of transient DI were female and young patients.


Adenoma , Diabetes Insipidus , Diabetes Mellitus , Pituitary Neoplasms , Humans , Female , Middle Aged , Male , Pituitary Neoplasms/pathology , Retrospective Studies , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Adenoma/pathology , Diabetes Insipidus/epidemiology , Diabetes Insipidus/etiology , Risk Factors , Treatment Outcome
12.
World Neurosurg ; 175: e55-e63, 2023 Jul.
Article En | MEDLINE | ID: mdl-36907270

BACKGROUND: Diabetes insipidus (DI) is a common complication after endoscopic transsphenoidal surgery (TSS) for pituitary adenoma (PA), which affects the quality of life in patients. Therefore, there is a need to develop prediction models of postoperative DI specifically for patients who undergo endoscopic TSS. This study establishes and validates prediction models of DI after endoscopic TSS for patients with PA using machine learning algorithms. METHODS: We retrospectively collected information about patients with PA who underwent endoscopic TSS in otorhinolaryngology and neurosurgery departments between January 2018 and December 2020. The patients were randomly split into a training set (70%) and a test set (30%). The 4 machine learning algorithms (logistic regression, random forest, support vector machine, and decision tree) were used to establish the prediction models. Area under the receiver operating characteristic curves were calculated to compare the performance of the models. RESULTS: A total of 232 patients were included, and 78 patients (33.6%) developed transient DI after surgery. Data were randomly divided into a training set (n = 162) and a test set (n = 70) for development and validation of the model, respectively. The area under the receiver operating characteristic curve was highest in the random forest model (0.815) and lowest in the logistic regression model (0.601). Invasion of pituitary stalk was the most important feature for model performance, closely followed by macroadenomas, size classification of PA, tumor texture, and Hardy-Wilson suprasellar grade. CONCLUSIONS: Machine learning algorithms identify preoperative features of importance and reliably predict DI after endoscopic TSS for patients with PA. Such a prediction model may enable clinicians to develop individualized treatment strategy and follow-up management.


Adenoma , Diabetes Insipidus , Diabetes Mellitus , Pituitary Neoplasms , Humans , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Retrospective Studies , Quality of Life , Adenoma/surgery , Adenoma/complications , Diabetes Insipidus/diagnosis , Diabetes Insipidus/etiology , Machine Learning , Postoperative Complications/etiology
13.
J Neurosurg ; 139(3): 790-797, 2023 09 01.
Article En | MEDLINE | ID: mdl-36738460

OBJECTIVE: Surgery is the primary treatment for craniopharyngioma with the preservation of hypothalamic function of paramount importance. Infundibular preservation is debated, as maximal resection decreases recurrence rates but causes hypopituitarism. A triphasic response of diabetes insipidus (DI), syndrome of inappropriate antidiuretic hormone secretion (SIADH), and recurrent DI has been described after pituitary surgery, but the impact of infundibular preservation on the triphasic response following craniopharyngioma resection has not been well established. The authors' objective was to assess postoperative fluid and sodium balance and differences in ADH imbalance management following endonasal craniopharyngioma resection based on infundibular transection status. METHODS: This is a retrospective cohort study of 19 patients with craniopharyngioma treated with endoscopic endonasal resection between 2014 and 2021. Resection was dichotomized into infundibular transection or preservation. Postoperative triphasic response, time to DI, and time to ADH replacement were compared using Fisher's exact test and Kaplan-Meier analysis. RESULTS: Based on surgeon impression, 10 patients had infundibular transection and 9 had infundibular preservation. Overall, 16 patients experienced DI, 12 experienced persistent DI, and 6 experienced SIADH. A postoperative triphasic response occurred in 40% (n = 4) of transection patients without preoperative DI and 11% (n = 1) of preservation patients without preoperative DI. The median time to postoperative DI (0.5 vs 18.0 hours, p = 0.022) and median time to ADH replacement therapy (4.5 vs 24 hours, p = 0.0004) were significantly shorter in the transection group than in the preservation group. CONCLUSIONS: Following endonasal craniopharyngioma resection, the triphasic response occurs in nearly half of infundibular transection cases. DI begins earlier with infundibular transection. On the basis of the study findings in which no patients met the criteria for SIADH or were endocrinologically unstable after postoperative day 6, it is reasonable to suggest that otherwise stable patients can be discharged at or before postoperative day 6 when ADH fluctuations have normalized and endocrinopathy is appropriately managed with oral desmopressin. Infundibular transection status may impact postoperative hormonal replacement strategies, but additional studies should evaluate their efficacies.


Craniopharyngioma , Diabetes Insipidus , Inappropriate ADH Syndrome , Pituitary Neoplasms , Humans , Craniopharyngioma/surgery , Craniopharyngioma/complications , Inappropriate ADH Syndrome/complications , Retrospective Studies , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Pituitary Gland/surgery , Diabetes Insipidus/etiology , Postoperative Complications/etiology
14.
World Neurosurg ; 172: e667-e678, 2023 Apr.
Article En | MEDLINE | ID: mdl-36739895

OBJECTIVE: Central diabetes insipidus (CDI) is the most common complication of endoscopic endonasal surgery (EES) for craniopharyngioma. However, some cases of CDI could spontaneously resolve during the follow-up period. Hence, this study aimed to determine the predictive factors for the spontaneous resolution of CDI. METHODS: Data of patients with CDI who underwent EES for craniopharyngioma between February 2009 and June 2021 were retrospectively reviewed. All patients were divided into 2 groups based on the resolution of CDI during follow-up: the recovery and no recovery groups. The baseline characteristic, surgical, and follow-up results of patients were compared. RESULTS: We identified 84 patients with CDI (35 in the recovery group and 49 in the no recovery group). A direct comparison showed that retaining the pituitary stalk (57.1% vs. 14.3%, P = 0.000) and no-hypothalamic injury (HI) (68.6% vs. 20.4%, P = 0.000) were more common in the recovery group, whereas hydrocephalus at diagnosis (8.6% vs. 46.9%, P = 0.000) was significantly more common in the no recovery group. Subsequently, we found through univariate and multivariate analysis that the spontaneous resolution of CDI was associated with hydrocephalus at diagnosis (yes vs. no: odds ratio [OR], 0.198; P = 0.045), pituitary stalk injury (retaining vs. sectioning: OR, 7.055; P = 0.004), and the Hong et al HI pattern (mild-HI vs. no-HI: OR, 0.183; P = 0.038; unilateral-HI vs. no-HI: OR, 0.147; P = 0.017; bilateral-HI vs. no-HI: OR, 0.154; P = 0.044). CONCLUSIONS: Hydrocephalus at diagnosis, pituitary stalk injury, and the Hong et al HI pattern might be predictors of the spontaneous resolution of CDI following EES for craniopharyngioma.


Craniopharyngioma , Diabetes Insipidus, Neurogenic , Diabetes Insipidus , Diabetes Mellitus , Hydrocephalus , Pituitary Neoplasms , Humans , Craniopharyngioma/surgery , Craniopharyngioma/complications , Retrospective Studies , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Hydrocephalus/complications , Diabetes Insipidus/epidemiology , Diabetes Insipidus/etiology
15.
Neurosurgery ; 92(6): 1112-1129, 2023 06 01.
Article En | MEDLINE | ID: mdl-36639856

BACKGROUND: Craniopharyngiomas account for 1.2% to 4.6% of all intracranial tumors. Although age at presentation is distributed bimodally, with a pediatric peak occurring between 5 and 15 years and an adult peak between 50 and 70 years, presentation, treatment, and outcome differences between these two craniopharyngioma populations have not been thoroughly characterized. OBJECTIVE: To compare treatments and outcomes between adult and pediatric craniopharyngiomas. METHODS: This is a systematic review and meta-analysis. Web of Science, MEDLINE, and Scopus databases were searched for primary studies reporting postoperative complications, functional outcomes, recurrence, and overall survival in patients with craniopharyngioma undergoing surgery. RESULTS: The search yielded 1,202 unique articles, of which 106 (n=4,202 patients) met criteria for qualitative synthesis and 23 (n=735 patients) met criteria for meta-analysis. Compared with adult, pediatric craniopharyngiomas were less likely to present with visual defects (odds ratio [OR] 0.54, 95% CI 0.36-0.80) or cognitive impairment (OR 0.29, 95% CI 0.12-0.71) and more likely with headaches (OR 2.08, 95% CI 1.16-3.73). Children presented with significantly larger tumors compared with adults (standardized mean difference 0.68, 95% CI 0.38-0.97). Comparing functional outcomes, pediatric patients sustained higher rates of permanent diabetes insipidus (OR 1.70, 95% CI 1.13-2.56), obesity (OR 3.15, 95% CI 1.19-8.31), and cranial nerve and/or neurological defects (OR 4.87, 95% CI 1.78-13.31) than adults. No significant differences were found in rates of postoperative cerebrospinal fluid leak, overall or progression-free survival, or recurrence. CONCLUSION: Adult and pediatric craniopharyngiomas seem to have fundamental differences in clinical presentation and functional outcomes. These patients frequently require multimodality treatment and are best managed with a multidisciplinary team and an individualized approach.


Craniopharyngioma , Pituitary Neoplasms , Adolescent , Adult , Child , Child, Preschool , Humans , Combined Modality Therapy , Craniopharyngioma/surgery , Diabetes Insipidus/etiology , Neurosurgical Procedures/adverse effects , Pituitary Neoplasms/surgery , Pituitary Neoplasms/drug therapy , Postoperative Complications/etiology , Treatment Outcome
16.
J Immunother Cancer ; 11(1)2023 01.
Article En | MEDLINE | ID: mdl-36690387

BACKGROUND: Immune effector cell-associated neurotoxicity syndrome (ICANS) is a common adverse event of CD19-directed chimeric antigen receptor (CAR) T cell therapy. Other neurological adverse events, however, have not methodically been described and studied. Furthermore, safety data on CAR-T cell therapy in patients with central nervous system (CNS) lymphoma remain limited. MAIN BODY: We here report occurrence of a Guillain-Barré-like syndrome (GBS) and central diabetes insipidus (cDI) following tisagenlecleucel therapy for relapsed high-grade lymphoma with CNS involvement. Both complications were refractory to standard treatment of ICANS. Weakness of respiratory muscles required mechanical ventilation and tracheostomy while cDI was treated with desmopressin substitution for several weeks. Muscle-nerve biopsy and nerve conduction studies confirmed an axonal pattern of nerve damage. T cell-rich infiltrates and detection of the CAR transgene in muscle-nerve sections imply a direct or indirect role of CAR-T cell-mediated inflammation. In line with current treatment guidelines for GBS, intravenous immunoglobulin was administered and gradual but incomplete recovery was observed over the course of several months. CONCLUSIONS: This case report highlights the risk of rare but severe neurological adverse events, such as acute GBS or cDI, in patients treated with CAR-T cells. It further underlines the importance of appropriate patient surveillance and systematic reporting of rare complications to eventually improve treatment.


Central Nervous System Neoplasms , Diabetes Insipidus , Diabetes Mellitus , Lymphoma, Non-Hodgkin , Receptors, Chimeric Antigen , Humans , Immunotherapy, Adoptive/adverse effects , Diabetes Insipidus/etiology , Cell- and Tissue-Based Therapy
17.
World Neurosurg ; 172: e476-e482, 2023 Apr.
Article En | MEDLINE | ID: mdl-36681322

BACKGROUND: Langerhans cell histiocytosis (LCH) is a rare idiopathic disease characterized by the clonal proliferation of Langerhans histiocytes in various parts of the body and capable of leading to organ damage and tumor formation. Reports of cranial LCH in the adult population are extremely rare. Although surgery remains the preferred option for localized LCH lesions, the role of stereotactic radiosurgery (SRS) is emerging. OBJECTIVE: To retrospectively review a rare case series to determine the safety and effectiveness of SRS for patients with localized cranial LCH. METHODS: We retrospectively reviewed histopathologically confirmed cases of localized cranial LCH treated with SRS at our institute in the adult population between January 2005 and September 2022. Five patients were identified with a median age of 34 years (19-54 years). The tumor location was in the pituitary stalk in 3 patients, the orbit in one patient, and the parietal skull in one patient. The median target volume was 2.8 cc (range: 0.37-6.11). Treatment was delivered in a single fraction in 4 patients (median margin dose of 8 Gy, range: 7-10 Gy) and in 3 fractions (22.5 Gy) in 1 patient. The median follow-up was 12 years (range: 4-17). None of the patients required craniotomy for tumor debulking before or after SRS. RESULTS: The local tumor control rate for the lesions was 100%. All 3 patients with LCH in the pituitary stalk had diabetes insipidus at the initial presentation and developed panhypopituitarism after SRS. Diabetes insipidus was not improved after SRS. The other 2 patients presented no adverse radiation effects. Based on the literature review, our case series was the largest retrospective series on SRS for localized cranial LCH, with the longest median follow-up. CONCLUSIONS: SRS for patients with localized cranial LCH was a safe and effective treatment modality in this case series. Larger studies are encouraged to validate the role of SRS in the treatment of localized cranial LCH.


Diabetes Insipidus , Diabetes Mellitus , Histiocytosis, Langerhans-Cell , Radiosurgery , Adult , Humans , Retrospective Studies , Radiosurgery/adverse effects , Treatment Outcome , Diabetes Insipidus/epidemiology , Diabetes Insipidus/etiology , Skull/pathology , Histiocytosis, Langerhans-Cell/radiotherapy , Histiocytosis, Langerhans-Cell/surgery , Histiocytosis, Langerhans-Cell/drug therapy , Follow-Up Studies
18.
World Neurosurg ; 172: e357-e363, 2023 Apr.
Article En | MEDLINE | ID: mdl-36640831

BACKGROUND: We implemented a streamlined care pathway for patients undergoing endoscopic transsphenoidal (TSA) pituitary surgery. Select patients are recovered in the postanesthesia care unit and transferred to a step-down unit for intermediate neurologic care (INCU), with clinicians trained to manage cerebrospinal fluid leak, diabetes insipidus (DI), and other complications. METHODS: We evaluated all TSA surgeries performed at 1 academic medical center from 7th January, 2017 to 30th March, 2020, collecting patient factors, tumor characteristics, cost variables, and outcomes. The INCU pathway was implemented on 7th January 2018. Pathway patients were compared with nonpathway patients across the study period. Outcomes were assessed using multivariate regression, adjusting for patient and surgical characteristics, including intraoperative cerebrospinal fluid leak, postoperative DI, and tumor dimensions. RESULTS: One hundred eighty-seven patients were identified. Seventy-nine were on the INCU pathway. Mean age was 53.5 years. Most patients were male (66%), privately insured (62%), and white (66%). Mean total cost of admission was $27,276. Mean length of stay (LOS) was 3.97 days. Use of the INCU pathway was associated with total cost reduction of $6376.33 (P < 0.001, 95% confidence interval [CI]: $3698.21-$9054.45) and LOS reduction by 1.27 days (P = 0.008, 95% CI: 0.33-2.20). In-hospital costs were reduced across all domains, including $1964.87 in variable direct labor costs (P < 0.001, 95% CI: $1142.08-$2787.64) and $1206.52 in variable direct supply costs (P < 0.001, 95% CI: $762.54-$1650.51). Pathway patients were discharged earlier despite a higher rate of postoperative DI (25% vs. 11%, P = 0.011), with fewer readmissions (0% vs. 6%, P = 0.021). CONCLUSIONS: A streamlined care pathway following TSA surgery can reduce in-hospital costs and LOS without compromising patient outcomes.


Diabetes Insipidus , Pituitary Diseases , Pituitary Neoplasms , Humans , Male , Middle Aged , Female , Length of Stay , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Critical Pathways , Postoperative Complications/etiology , Pituitary Diseases/surgery , Diabetes Insipidus/etiology , Cerebrospinal Fluid Leak/complications , Retrospective Studies
19.
Neurosurgery ; 92(6): 1269-1275, 2023 06 01.
Article En | MEDLINE | ID: mdl-36700759

BACKGROUND: Endoscopic transsphenoidal surgery remains the technique of choice for resection of pituitary adenoma. Postoperative diabetes insipidus (DI) is most often transient and observed in 1.6% to 34% of patients, whereas permanent DI has been reported in 0% to 2.7% of patients. The proposed mechanism was the transduction of traction forces exerted by the surgeon on the descended diaphragma sellae and through the pituitary stalk. OBJECTIVE: To quantify and correlate the degree of pituitary gland descent with postoperative DI. METHODS: Of 374 patients who underwent transsphenoidal resection of a pituitary adenoma between 2010 and 2020 at our institution, we report a cohort of 30 patients (Group A) DI. We also report a matched cohort by tumor volume of 30 patients who did not develop DI (Group B). We quantified the tension on the pituitary stalk by calculating pituitary descent interval (PDI) by comparing preoperative and postoperative position of the pituitary gland and using Pythagoras' formula where , with craniocaudal (CC) and anterior-posterior (AP) representing measurements of pituitary translation in respective directions after resection. RESULTS: Patients who developed DI had significantly greater pituitary gland translations in the craniocaudal (23.0 vs 16.3 mm, P = .0015) and anteroposterior (2.4 vs 1.5 mm, P = .0168) directions. Furthermore, Group A had a statistically greater PDI, which was associated with development of DI (23.2 vs 16.6 mm, P = .0017). CONCLUSION: We were able to quantify pituitary descent and subsequent tension on the pituitary stalk, while also associating it with development of postoperative DI after pituitary adenoma resection.


Adenoma , Diabetes Insipidus , Diabetes Mellitus , Pituitary Diseases , Pituitary Neoplasms , Humans , Pituitary Neoplasms/surgery , Pituitary Neoplasms/pathology , Postoperative Complications , Pituitary Gland/surgery , Pituitary Gland/pathology , Diabetes Insipidus/etiology , Adenoma/surgery , Adenoma/pathology , Retrospective Studies
20.
Oper Neurosurg (Hagerstown) ; 24(3): 248-255, 2023 03 01.
Article En | MEDLINE | ID: mdl-36701694

BACKGROUND: Manipulation of the pituitary stalk, posterior pituitary gland, and hypothalamus during transsphenoidal pituitary adenoma resection can cause disruption of water electrolyte regulation leading to diabetes insipidus (DI). OBJECTIVE: To determine whether pituitary stalk stretch is an independent risk factor for postoperative DI after pituitary adenoma resection. METHODS: A retrospective review was performed of patients undergoing endoscopic endonasal resection of pituitary macroadenoma between July 2010 and December 2016 by a single neurosurgeon. We analyzed preoperative and postoperative imaging metrics to assess predictors for postoperative DI. RESULTS: Of the 234 patients undergoing resection, 41 (17.5%) developed postoperative DI. DI was permanent in 10 (4.3%) and transient in 31 (13.2%). The pituitary stalk stretch, measured as the change in stalk length from preoperative to postoperative imaging, was greater in the DI compared with the non-DI group (10.1 mm vs 5.9 mm, P < .0001). The pituitary stalk stretch was associated with DI with significant difference in mean pituitary stalk stretch between non-DI group vs DI group (5.9 mm vs 10.1 mm, P < .0001). Multivariate analysis revealed that pituitary stalk stretch >10 mm was a significant independent predictor of postoperative DI [odds ratios = 2.56 (1.10-5.96), P = .029]. When stratified into transient and permanent DI, multivariable analysis showed that pituitary stalk stretch >10 mm was a significant independent predictor of transient DI [odds ratios = 2.71 (1.0-7.1), P = .046] but not permanent DI. CONCLUSION: Postoperative pituitary stalk stretch after transsphenoidal pituitary adenoma surgery is an important factor for postoperative DI. We propose a reconstruction strategy to mitigate stalk stretch.


Adenoma , Diabetes Insipidus , Diabetes Mellitus , Pituitary Neoplasms , Humans , Pituitary Neoplasms/diagnostic imaging , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Postoperative Complications/diagnostic imaging , Postoperative Complications/etiology , Pituitary Gland/diagnostic imaging , Pituitary Gland/surgery , Diabetes Insipidus/etiology , Hypothalamus , Adenoma/complications , Adenoma/diagnostic imaging , Adenoma/surgery
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