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1.
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
2.
Probl Endokrinol (Mosk) ; 69(6): 54-62, 2024 Jan 24.
Article in Russian | MEDLINE | ID: mdl-38311995

ABSTRACT

In recent years, there has been a significant increase in the prevalence of autoimmune endocrinopathies, which are known to affect various levels of the endocrine system, including the pituitary gland. Hypophysitis is a general term used to describe any form of sellar and suprasellar inflammation that leads to structural changes in the hypothalamic-pituitary region and manifests itself in varying degrees of hormonal deficiency of the anterior and posterior pituitary glands. To date, there is a primary form of hypophysitis, which occurs as a result of an autoimmune lesion directly to the pituitary gland, and a secondary form of hypophysitis, which occurs as a result of the presence of a systemic autoimmune disease. Regardless of the etiology, patients with hypophysitis show various signs and symptoms caused by an inflammatory process in the pituitary gland, which can lead to the development of hypopituitarism, compression of the sellar and parasellar structures. MRI is currently the best non-invasive diagnostic tool for diagnosing hypopituitarism, however, the diagnosis can be made with certainty only by histological examination of the pituitary tissue, which requires an invasive approach, which greatly reduces the feasibility of this procedure. In this article, we present a patient with MRI showing signs of hypophysitis in the absence of clear clinical symptoms.


Subject(s)
Hypophysitis , Hypopituitarism , Pituitary Diseases , Humans , Diagnosis, Differential , Hypophysitis/complications , Hypophysitis/diagnosis , Pituitary Diseases/complications , Pituitary Diseases/diagnosis , Pituitary Diseases/therapy , Pituitary Gland/diagnostic imaging , Pituitary Gland/pathology , Hypopituitarism/diagnosis
3.
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
4.
Front Endocrinol (Lausanne) ; 14: 1297146, 2023.
Article in English | MEDLINE | ID: mdl-38152125

ABSTRACT

Background: Pituitary stalk interruption syndrome (PSIS) is a congenital disease commonly found in patients with combined pituitary hormone deficiency (CPHD). Most PSIS patients manifest growth retardation and delayed puberty. We report a rare case of PSIS with tall stature, liver cirrhosis and diabetes, possibly caused by an inactivating KCNJ11 gene mutation. Case presentation: A 37-year-old female patient initially presented with liver cirrhosis and diabetes, without any secondary sexual characteristics. Endocrine investigation indicated CPHD. Small anterior pituitary, invisible pituitary stalk and no eutopic posterior lobe hypersignal in the sella turcica viewed in magnetic resonance imaging (MRI) confirmed the diagnosis of PSIS. Despite receiving no growth hormone or sex hormone therapy, she reached a final height of 186 cm. Liver histopathology revealed nonalcoholic fatty cirrhosis. Genetic testing identified a heterozygous p.Arg301Cys mutation in the KCNJ11 gene. Conclusion: This is a rare case of PSIS with liver cirrhosis and diabetes associated with an inactivating KCNJ11 gene mutation. It's supposed that early hyperinsulinism caused by the KCNJ11 gene mutation, as well as delayed epiphyseal closure due to estrogen deficiency, contributed to the patient's exceptionally tall stature. Untreated growth hormone deficiency (GHD) resulted in increased visceral fat, leading to nonalcoholic fatty liver disease (NAFLD) and cirrhosis. The decline in ß cell function with age, combined with NAFLD, may have played a role in the development of diabetes.


Subject(s)
Diabetes Mellitus , Non-alcoholic Fatty Liver Disease , Pituitary Diseases , Female , Humans , Adult , Non-alcoholic Fatty Liver Disease/pathology , Pituitary Diseases/complications , Pituitary Gland/pathology , Diabetes Mellitus/genetics , Diabetes Mellitus/pathology , Mutation , Liver Cirrhosis/complications , Liver Cirrhosis/genetics
5.
Front Endocrinol (Lausanne) ; 14: 1241145, 2023.
Article in English | MEDLINE | ID: mdl-38027203

ABSTRACT

Objective: Hypothalamic-pituitary axis dysfunction is a common complication in post-operative craniopharyngioma(CP) patients, and it greatly impacts the long-term quality of life of such patients. To better understand the effects of postoperative hypothalamic-pituitary dysfunction and long-term hormone replacement therapy in patients with childhood CP, we assessed approximately 200 patients with childhood-onset CP postoperatively. Methods: Clinical details of patients with childhood-onset CP who underwent sellar tumor resection in Beijing Children's Hospital and Beijing Tiantan Hospital from 2018 to 2019 were retrieved retrospectively. The participants were followed up to assess the effects of post-operative long-term hormone replacement therapy and assess the tumor recurrence rate. Results: The median age of admission was 8.1 (1.8, 14.3) years. Headache (45.5%), visual impairment (39.5%), and nausea (33.0%) were the most common clinical manifestations. ACP accounted for 95% of all CP cases. The incidence of central adrenal insufficiency and central hypothyroidism within the first week after surgery was 56.2% and 70.3%, respectively. At the same time 85.5% of the patients required at least one dose of desmopressin to control urine output. Total survival and tumor recurrence rates were 98.6% and 26.1%, respectively, with a median follow-up time of 29.7 (19.0, 40.3) months. During the follow-up period, 28.1% patients met the diagnostic criteria for short stature, while 54.4% fit the criteria for obesity. In addition, 94.4% of the patients were taking at least one kind of hormone substitution, and 74.7% were taking three or more. The prevalence of levothyroxine, glucocorticoid, desmopressin, and growth hormone replacement therapy was 87.3%, 77.5%, 78.9% and 31.0%, respectively. The proportion of patients treated with the substitutive combination of levothyroxine, hydrocortisone, and desmopressin was 54.9%. Conclusion: This study is a large-sample systematic postoperative endocrine function evaluation of patients with childhood-onset CP. Due to the high prevalence of post-operative hypothalamic-pituitary dysfunction, patients with CP usually require long-term multiple hormone substitution therapy. Individualized management and accurate hormone replacement dosage for postoperative childhood-onset CP patients are of great importance.


Subject(s)
Craniopharyngioma , Hypothalamic Diseases , Pituitary Diseases , Pituitary Neoplasms , Humans , Child , Craniopharyngioma/complications , Craniopharyngioma/epidemiology , Craniopharyngioma/surgery , Retrospective Studies , Thyroxine , Quality of Life , Deamino Arginine Vasopressin , Neoplasm Recurrence, Local/complications , Pituitary Neoplasms/epidemiology , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Hypothalamic Diseases/complications , Pituitary Diseases/complications , Hormone Replacement Therapy , Hydrocortisone
6.
Front Endocrinol (Lausanne) ; 14: 1199091, 2023.
Article in English | MEDLINE | ID: mdl-37409223

ABSTRACT

Introduction: Alcohol-induced hypercortisolism (AIH) is underrecognized and may masquerade as neoplastic hypercortisolism [Cushing syndrome (CS)] obscuring its diagnosis. Objective and methods: In order to characterize AIH, we performed a chart review of eight patients (4 males and 4 females; 2014-2022) referred for evaluation and treatment of neoplastic hypercortisolism - six for inferior petrosal sinus sampling, one due to persistent CS after unilateral adrenalectomy, and one for pituitary surgery for Cushing disease (CD). Five underwent dDAVP stimulation testing. Results: All eight patients had clinical features of hypercortisolism and plasma ACTH levels within or above the reference interval confirming hypothalamic-pituitary mediation. All had abnormal low-dose dexamethasone suppression test and increased late-night salivary cortisol. Only one had increased urine cortisol excretion. In contrast to CD, the 5 patients tested had blunted or absent ACTH and cortisol responses to desmopressin. Two had adrenal nodules and one had abnormal pituitary imaging. Most patients underreported their alcohol consumption and one denied alcohol use. Elevated blood phosphatidyl ethanol (PEth) was required in one patient to confirm excessive alcohol use. All patients had elevations of liver function tests (LFTs) with AST>ALT. Conclusion: AIH is an under-appreciated, reversible cause of non-neoplastic hypercortisolism that is indistinguishable from neoplastic CS. Incidental pituitary and adrenal imaging abnormalities as well as under-reporting of alcohol consumption further confound the diagnosis. Measurement of PEth helps to confirm an alcohol use disorder. Elevations of LFTs (AST>ALT) and subnormal ACTH and cortisol responses to dDAVP help to distinguish AIH from neoplastic hypercortisolism.


Subject(s)
Cushing Syndrome , Pituitary ACTH Hypersecretion , Pituitary Diseases , Male , Female , Humans , Cushing Syndrome/complications , Cushing Syndrome/diagnosis , Hydrocortisone , Deamino Arginine Vasopressin , Pituitary ACTH Hypersecretion/complications , Pituitary ACTH Hypersecretion/diagnosis , Pituitary Diseases/complications , Ethanol , Adrenocorticotropic Hormone
7.
Orphanet J Rare Dis ; 18(1): 165, 2023 06 26.
Article in English | MEDLINE | ID: mdl-37365629

ABSTRACT

BACKGROUND: Pituitary abscess (PA) is a rare condition and not well understood. We aimed to describe a case and perform a comprehensive systematic review to explore presenting symptoms, radiological findings, endocrine abnormalities and mortality. AIM: To identify presenting symptoms, radiological findings, endocrinological abnormalities and predictors of mortality for PA. METHODS: We systematically reviewed the literature to identify all case reports of PA. Data regarding presentation, mortality, radiological findings, endocrinological abnormalities and treatment was extracted. RESULTS: We identified 488 patients from 218 articles meeting the inclusion criteria. Mortality was 5.1%, with days to presentation (OR 1.0005, 95% CI 1.0001-1.0008, p < 0.01) being the only identified independent predictor of mortality. Mortality rates have decreased over time, with cases published prior to 2000 having higher mortality rates (OR 6.92, 95% CI 2.80-17.90, p < 0.001). The most common symptom was headache (76.2%), followed by visual field defects (47.3%). Classical signs of infection were only present in 43%. The most common imaging feature on magnetic resonance imaging (MRI) was high T2 and low T1 signal of the pituitary gland with peripheral contrast enhancement. Over half (54.8%) were culture negative, with the most common bacterial organism being staphylococcus aureus (7.8%) and fungal organism being aspergillus (8.8%). The most common endocrine abnormality was hypopituitarism (41.1%), followed by diabetes insipidus (24.8%). Whilst symptoms resolved in most patients, persistent endocrine abnormalities were present in over half of patients (61.0%). CONCLUSION: PA is associated with significant mortality, with delayed presentation increasing risk of mortality. Ongoing endocrinological abnormalities are common. Given the non-specific clinical presentation, the appearance of high T2, low T1 and peripheral contrast enhancement of the pituitary on MRI should prompt consideration of this rare disease.


Subject(s)
Hypopituitarism , Pituitary Diseases , Pituitary Neoplasms , Humans , Abscess/complications , Pituitary Diseases/complications , Pituitary Diseases/diagnosis , Pituitary Diseases/microbiology , Hypopituitarism/complications , Pituitary Neoplasms/complications , Magnetic Resonance Imaging
8.
BMC Endocr Disord ; 23(1): 96, 2023 May 04.
Article in English | MEDLINE | ID: mdl-37143052

ABSTRACT

BACKGROUND: Some patients develop immunoglobulin G4 (IgG4)-related hypophysitis associated with systemic diseases. More than 30 cases of IgG4-related hypophysitis have been reported. However, biopsy has rarely been performed in these patients, and none have had an associated pituitary neuroendocrine tumor (PitNET). We present a case of concurrent IgG4-related hypophysitis and PitNET. CASE PRESENTATION: A 56-year-old Japanese man arrived at the hospital with visual impairment, bitemporal hemianopia, and right abducens nerve palsy. Magnetic resonance imaging revealed pituitary body and stalk swelling as well as a small poorly enhanced right anterior lobe mass. Laboratory and loading test results suggested hypopituitarism. Because IgG4 level was elevated, a systemic examination was performed; multiple nodules were found in both lung fields. The diagnosis was based on an endoscopic transnasal biopsy of the pituitary gland. A histopathological examination revealed a marked infiltration of plasma cells into the pituitary gland, which was strongly positive for IgG4. The histological features of the resected tumor were consistent with those of gonadotroph PitNET, which was immunohistochemically positive for follicle-stimulating hormone-ß and steroidogenic factor-1, and no plasma cell infiltration was observed. Based on the histopathological examination results, steroid therapy was initiated, which reduced pituitary gland size and serum IgG4 levels. DISCUSSION AND CONCLUSIONS: This is the first reported case of IgG4-related hypophysitis with PitNET. Although no pathological findings indicating a relationship between the two conditions were found, we were able to preoperatively differentiate multiple lesions via detailed diagnostic imaging.


Subject(s)
Autoimmune Hypophysitis , Gonadotrophs , Hypophysitis , Neuroendocrine Tumors , Pituitary Diseases , Pituitary Neoplasms , Male , Humans , Middle Aged , Autoimmune Hypophysitis/complications , Autoimmune Hypophysitis/diagnosis , Autoimmune Hypophysitis/pathology , Gonadotrophs/pathology , Neuroendocrine Tumors/complications , Neuroendocrine Tumors/diagnosis , Neuroendocrine Tumors/pathology , Pituitary Gland/diagnostic imaging , Pituitary Gland/pathology , Pituitary Diseases/complications , Hypophysitis/diagnosis , Hypophysitis/diagnostic imaging , Pituitary Neoplasms/diagnosis , Pituitary Neoplasms/diagnostic imaging , Immunoglobulin G
9.
Endocr Pract ; 29(6): 471-477, 2023 Jun.
Article in English | MEDLINE | ID: mdl-37004872

ABSTRACT

OBJECTIVE: To investigate the incidence of corticotroph hyperplasia (CH) or lymphocyte infiltration in the pituitary of patients with obesity. METHODS: The pituitary and adrenal glands from 161 adult autopsies performed between 2010 and 2019 at our institution were reviewed. The clinical history, body mass index (BMI), and cause of death were recorded. Routine hematoxylin and eosin staining, reticulin staining, and immunohistochemical staining for adrenocorticotropic hormone, CD3, and CD20 were performed. The results were analyzed using the Fisher and chi-square statistics. Decedents were separated into 4 groups based on BMI (kg/m2): (1) lean (BMI, <25.0), (2) overweight (BMI, 25.0-29.9), (3) obesity class I (BMI, 30.0-34.9), and (4) obesity classes II to III (BMI, >34.9). RESULTS: CH/neoplasia was identified in 44 of 161 pituitary glands. Four (9.1%) of 53 lean patients had pituitary lesions, whereas 27.3% (12) of overweight, 22.7% (10) of obesity class I, and 40.9% (18) of obesity class II patients had hyperplasia (P < .0001). Small corticotroph tumors were identified in 15 patients; only 1 was a lean patient, and the tumor was associated with the Crooke hyaline change of nontumorous corticotrophs. The presence of CH and neoplasia was associated with adrenal cortical hyperplasia and lipid depletion. Microscopic foci of T and B lymphocytes were identified in the pituitaries of patients in each weight category; no independent association between BMI and lymphocyte inflammation was found. CONCLUSION: Our data indicate an association between CH/neoplasia and obesity. It remains unclear whether obesity is the cause or effect of adrenocorticotropic hormone and cortisol excess.


Subject(s)
Obesity, Morbid , Pituitary Diseases , Pituitary Neoplasms , Adult , Humans , Corticotrophs/metabolism , Corticotrophs/pathology , Obesity, Morbid/pathology , Hyperplasia/pathology , Overweight/complications , Overweight/epidemiology , Pituitary Gland/pathology , Adrenocorticotropic Hormone/metabolism , Pituitary Diseases/complications , Pituitary Diseases/epidemiology , Pituitary Neoplasms/complications , Pituitary Neoplasms/epidemiology , Pituitary Neoplasms/pathology , Obesity/complications , Obesity/epidemiology
10.
Rev Med Liege ; 78(4): 189-192, 2023 Apr.
Article in French | MEDLINE | ID: mdl-37067833

ABSTRACT

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.


Subject(s)
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.
Best Pract Res Clin Endocrinol Metab ; 37(4): 101752, 2023 07.
Article in English | MEDLINE | ID: mdl-36878774

ABSTRACT

There is increased interest related to the impact of coronavirus disease 19 (COVID-19) on the endocrine system and in particular on the pituitary gland. Over the course of the severe infection with acute respiratory syndrome coronavirus 2 (SARS-CoV-2), there are both acute and delayed effects on the pituitary, related to infection and/or treatment. Hypopituitarism, pituitary apoplexy and hypophysitis have been all reported, as well as arginine vasopressin deficiency (diabetes insipidus) and syndrome of inappropriate antidiuretic hormone secretion. Furthermore, patients with acromegaly, Cushing's disease and hypopituitarism are theoretically at increased risk of complications with COVID-19 and require close monitoring. Evidence regarding pituitary dysfunction in patients with COVID-19 continues to be gathered, as the breadth and depth of knowledge also continues to rapidly evolve. This review summarizes data analysis to date on the possible effects of COVID-19 and COVID-19 vaccination on patients with normal pituitary function and patients with known pituitary pathology. Though clinical systems were significantly affected, it seems there is no overall loss of biochemical control in patients with certain pituitary pathologies.


Subject(s)
COVID-19 , Hypopituitarism , Pituitary Diseases , Humans , SARS-CoV-2 , COVID-19 Vaccines , COVID-19/complications , COVID-19/pathology , Pituitary Diseases/complications , Pituitary Gland/pathology , Hypopituitarism/etiology
12.
Medicina (Kaunas) ; 59(3)2023 Mar 14.
Article in English | MEDLINE | ID: mdl-36984566

ABSTRACT

Background and Objectives: Pituitary abscess (PA) is a rare occurrence, representing less than 1% of pituitary lesions, and is defined by the presence of an infected purulent collection within the sella turcica. Pas can be classified as either primary, when the underlying pituitary is normal prior to infection, or secondary, when there is associated a pre-existing sellar pathology (i.e., pituitary adenoma, Rathke's cleft cysts, or craniopharyngioma), with or without a recent history of surgery. Preoperative diagnosis, owing to both non-specific symptoms and imaging features, remains challenging. Treatment options include endonasal trans-sphenoidal pus evacuation, as well as culture and tailored antibiotic therapy. Methods: A retrospective multicenter study, conducted on a prospectively built database over a 20-year period, identified a large series of 84 patients harboring primary sellar abscess. The study aimed to identify crucial clinical and imaging features in order to accelerate appropriate management. Results: The most common clinical presentation was a symptom triad consisting of various degrees of asthenia (75%), visual impairment (71%), and headache (50%). Diagnosis was achieved in 95% of cases peri- or postoperatively. Functional recovery was good for visual disturbances and headache. Pituitary function recovery remained very poor (23%), whereas the preoperative diagnosis represented a protective factor. Conclusions: In light of the high prevalence of pituitary dysfunction following the management of PAs, early diagnosis and treatment might represent a crucial issue. Currently, there are no standard investigations to establish a conclusive preoperative diagnosis; however, new, emerging imaging methods, in particular nuclear imaging modalities, represent a very promising tool, whose potential warrants further investigations.


Subject(s)
Pituitary Diseases , Pituitary Neoplasms , Humans , Abscess , Pituitary Diseases/complications , Pituitary Diseases/diagnosis , Pituitary Diseases/surgery , Pituitary Neoplasms/complications , Pituitary Neoplasms/diagnosis , Pituitary Neoplasms/surgery , Pituitary Gland/pathology , Headache , Magnetic Resonance Imaging
13.
BMC Neurol ; 23(1): 11, 2023 Jan 12.
Article in English | MEDLINE | ID: mdl-36631799

ABSTRACT

BACKGROUND: Granulomatous hypophysitis is a rare disease that presents with chronic inflammation of the pituitary gland. In this study, we reported a case of granulomatous hypophysitis associated with a pituitary abscess. CASE PRESENTATION: A 39-year-old woman presented with a 2-year history of infertility. For the past six months, she has suffered from amenorrhea, decreased libido, headaches, and vertigo. She was referred to our hospital with a suspected diagnosis of nonfunctioning pituitary adenoma based on her presentation and brain MRI findings. She underwent trans-sphenoidal surgery (TSS). Direct observation during surgery revealed drainage of malodor pus and pituitary gland abscess. The histopathological evaluation also showed granulomatous hypophysitis and neutrophilic microabscess formation. The patient was initially treated with high doses of ceftriaxone (2 g twice daily) and metronidazole (500 mg (mg) four times per day). Also, the patient received cortisol replacement therapy after the operation. After obtaining the antibiogram and culture results, the treatment regimen was continued for 4 weeks postoperatively, followed by amoxicillin-clavulanate (500/125 mg three times daily) for a total duration of 12 weeks. CONCLUSION: The patient recovered uneventfully and the postoperative MRI was normal without any remnant lesions.


Subject(s)
Hypophysitis , Pituitary Diseases , Pituitary Neoplasms , Humans , Female , Adult , Abscess/complications , Abscess/therapy , Pituitary Diseases/complications , Pituitary Diseases/diagnosis , Pituitary Diseases/pathology , Pituitary Neoplasms/surgery , Hypophysitis/complications , Hypophysitis/diagnosis , Pituitary Gland/diagnostic imaging , Pituitary Gland/surgery , Magnetic Resonance Imaging
14.
J Clin Endocrinol Metab ; 108(2): 323-330, 2023 01 17.
Article in English | MEDLINE | ID: mdl-36201475

ABSTRACT

CONTEXT: Pituitary stalk interruption syndrome (PSIS) is rare in the pediatric population. It combines ectopic posterior pituitary stalk interruption and anterior pituitary hypoplasia with hormonal deficiencies. The phenotype is highly heterogeneous and obesity/overweight seems to be underreported in the literature. OBJECTIVE: To identify patients with PSIS and obesity or overweight, describe their phenotype, and compare them with patients with PSIS without overweight/obesity. METHODS: Sixty-nine children and young adults with PSIS in a Toulouse cohort from 1984 to 2019 were studied. We identified 25 obese or overweight patients (OB-OW group), and 44 were nonobese/overweight (NO group). Then the groups were compared. RESULTS: All cases were sporadic. The sex ratio was 1.6. The main reason for consultation in both groups was growth retardation (61% in OB-OW group, 77% in NO group). History of neonatal hypoglycemia was more common in the OB-OW than in the NO group (57% vs 14%, P = .0008), along with extrapituitary malformations (64% vs 20%, P < 0001). The incidence of caesarean section was higher in the OB-OW group (52%) than in the NO group (23%), although not significant (P = .07). CONCLUSION: Patients with PSIS who are obese/overweight display interesting phenotypic differences that suggest hypothalamic defects. Studies are needed that include additional information on hormonal levels, particularly regarding oxytocin and ghrelin.


Subject(s)
Pituitary Diseases , Pituitary Gland , Child , Female , Humans , Pregnancy , Cesarean Section , Obesity/complications , Obesity/epidemiology , Overweight/complications , Overweight/epidemiology , Pituitary Diseases/complications , Pituitary Diseases/epidemiology , Pituitary Diseases/genetics , Pituitary Gland/abnormalities , Young Adult
15.
Neurol India ; 70(5): 2093-2099, 2022.
Article in English | MEDLINE | ID: mdl-36352614

ABSTRACT

Background: Snakebite envenomation (SE) is an important tropical disease in India, causing significant morbidity and mortality among patients. The hormonal deficiencies due to the involvement of the pituitary in case of SE can present in either acute or delayed setting. Hypopituitarism (HP) is often an underrecognized and relatively rarely reported complication of this neglected disease. Methods: We present here the data of 15 patients diagnosed to have HP following systemic SE and are being currently followed-up in the Endocrinology outpatient department of a tertiary care hospital of South India. The study was approved by the Institute ethics committee, and informed onsent was taken from all the study patients. The study was a record-based retrospective analysis of the patients with HP following SE. Clinical data including lag time in diagnosis and type of snake were determined. Further, hormonal data including all the anterior pituitary functions (thyroid stimulating hormone, free T4, cortisol, insulin-like growth factor (IGF-1) luteinizing hormone, follicular-stimulating hormone, testosterone; prolactin) and water deprivation test to determine diabetes insipidus (DI) in patients with polyuria on follow-up were extracted from the records and the hospital information system. An experienced neuroradiologist examined the magnetic resonance imaging (MRI) findings of the pituitary. Results: The mean age of the patients was 43 ± 9 years and 80% were male. Around 90% of patients belonged to upper-lower socioeconomic status according to the modified Kuppuswamy scale. The commonest snake species reported was Russell's viper. Thirteen patients had delayed HP. The median duration from snakebite to onset of HP symptoms was 1 year (range 0.33-10 years). However, the median time from snakebite to the diagnosis of HP was 7 years (range 1-13 years). Central hypothyroidism and hypogonadism were present in all subjects. However, central hypocortisolism was noted in 93% of patients. Low IGF-1 was noted in all the six patients where data were available. One patient had partial central DI. Thirteen out of 15 patients had reduction of pituitary volume in MRI. Conclusion: HP in patients with SE can appear slowly and the diagnosis is frequently delayed for years. Following snakebite, multiple pituitary hormone deficiencies associated with radiological abnormalities like a significant reduction in the pituitary volume are common.


Subject(s)
Daboia , Diabetes Insipidus , Hypopituitarism , Hypothalamic Diseases , Pituitary Diseases , Snake Bites , Animals , Humans , Male , Adult , Middle Aged , Female , Snake Bites/complications , Retrospective Studies , Hypopituitarism/diagnostic imaging , Hypopituitarism/etiology , Hypothalamic Diseases/complications , Diabetes Insipidus/etiology , Pituitary Diseases/complications
16.
Pituitary ; 25(6): 1004-1014, 2022 Dec.
Article in English | MEDLINE | ID: mdl-36322283

ABSTRACT

PURPOSE: Central diabetes insipidus is a complication that may occur after pituitary surgery and has been difficult to predict. This study aimed to identify the cutoff levels of serum copeptin and its optimal timing for predicting the occurrence of central diabetes insipidus in patients who underwent transsphenoidal surgery. METHODS: This was a prospective observational study of patients who underwent transsphenoidal surgery for pituitary gland or stalk lesions. Copeptin levels were measured before surgery, 1 h after extubation, and on postoperative days 1, 2, 7, and 90. RESULTS: Among 73 patients, 14 (19.2%) and 13 (17.8%) patients developed transient and permanent central diabetes insipidus, respectively. There was no significant difference in copeptin levels before surgery and 1 h after extubation; copeptin levels on postoperative days 1, 2, 7, and 90 were significantly lower in patients with permanent central diabetes insipidus than in those without central diabetes insipidus. Copeptin measurement on postoperative day 2 exhibited the highest performance for predicting permanent central diabetes insipidus among postoperative days 1, 2, and 7 (area under the curve [95% confidence interval] = 0.754 [0.632-0.876]). Serum copeptin level at postoperative day 2(< 3.1 pmol/L) showed a sensitivity of 92.3% and a negative predictive value of 97.1%. The ratio of copeptin at postoperative day 2 to baseline (< 0.94) presented a sensitivity of 84.6% and a negative predictive value of 94.9%. The copeptin levels > 3.4 and 7.5 pmol/L at postoperative day 2 and 7 may have ruled out the occurrence of CDI with a negative predictive value of 100%. CONCLUSION: The copeptin level at postoperative day 2 and its ratio to baseline can predict the occurrence of permanent central diabetes insipidus after pituitary surgery.


Subject(s)
Diabetes Insipidus, Neurogenic , Diabetes Mellitus , Pituitary Diseases , Humans , Diabetes Insipidus, Neurogenic/etiology , Pituitary Diseases/complications , Pituitary Gland/surgery , Glycopeptides , Postoperative Complications/epidemiology
17.
J Clin Neurosci ; 106: 180-184, 2022 Dec.
Article in English | MEDLINE | ID: mdl-36369079

ABSTRACT

BACKGROUND: Hyponatremia is a common and potentially dangerous complication of transsphenoidal surgery. Prophylactic postoperative fluid restriction has been trialled as a method to reduce the incidence of postoperative hyponatremia. METHODS: A systematic review of the literature was performed in accordance with the PRISMA statement. Risk of bias was assessed using the MINORS criteria. Meta-analysis was performed using the random-effects model. RESULTS: A total of 6 retrospective cohort studies were available for analysis. Fluid restriction was commonly between 1000 and 1500 ml/day and limited to the first postoperative week. Overall, the rate postoperative hyponatremia was fourfold less in the fluid restricted cohorts (3.4 % vs 11.2 %, OR 0.24 (95 %CI 0.15-0.38), p < 0.01). There was no difference in readmission rates (1.4 % vs 3.9 %, OR 0.32 (95 %CI 0.09-1.13), p = 0.08) or postoperative diabetes insipidus (14.5 % vs 18.6 %, OR 0.82 (95 %CI 0.50-1.36), p = 0.45) between fluid restricted and control cohorts. CONCLUSION: Prophylactic postoperative fluid restriction is a cheap, easily implemented intervention that appears to reduce the rate of postoperative hyponatremia, but not necessarily re-admission rates. Whether these prevented cases of hyponatremia are clinically significant remains to be demonstrated.


Subject(s)
Hyponatremia , Pituitary Diseases , Pituitary Neoplasms , Humans , Hyponatremia/etiology , Hyponatremia/prevention & control , Hyponatremia/epidemiology , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Retrospective Studies , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Postoperative Complications/prevention & control , Pituitary Gland/surgery , Pituitary Diseases/complications
18.
Medicine (Baltimore) ; 101(43): e31338, 2022 Oct 28.
Article in English | MEDLINE | ID: mdl-36316923

ABSTRACT

Rheumatic diseases have been reported to sometimes involve the pituitary gland. This study aims to characterize the clinical features and outcomes of patients with rheumatic disease-associated hypophysitis. We used the electronic medical record system in our hospital to identify nine patients with pituitary involvement in rheumatoid disease. We summarized the clinical characteristics, radiographic findings, treatments, and clinical outcomes of the 9 patients. We also performed a systematic literature review of systemic lupus erythematosus (SLE) cases with pituitary involvement published in PubMed and Wanfang databases from 1995 to 2021, and eight patients with complete information were selected. In the nine-patient cohort, the median age was 54 years, and the spectrum of rheumatic diseases included immunoglobulin G4-related disease (IgG4RD) (4/9), SLE (2/9), vasculitis (2/9), and Sjögren syndrome (SS) (1/9). All patients had pituitary abnormalities on radiological assessment, 6 developed diabetes insipidus (DI), and 8 presented with anterior pituitary hormone deficiencies in the disease duration. All the patients had multisystem involvement. As compared to hypophysitis with IgG4RD (IgG4-H), the age at onset of hypophysitis with SLE (SLE-H) patients was younger [(30.4 ± 16.4) years vs. (56.0 ± 0.8) years] and the disease duration was shorter [(14.0 ± 17.5) months vs. (71.0 ± 60.9) months] (P < .05). All patients were managed with glucocorticoids (GC) in combination with another immunosuppressant, and the majority of patients improved within 4 months. Six patients achieved disease remission while four required at least one hormone replacement therapy. Hypophysitis is a rare complication secondary to a variety of various rheumatic diseases that can occur at any stage. GC combined with additional immunosuppressants could improve patients' symptoms; however some patients also required long-term hormone replacement therapy in pituitary disorders.


Subject(s)
Autoimmune Hypophysitis , Collagen Diseases , Hypophysitis , Hypopituitarism , Lupus Erythematosus, Systemic , Pituitary Diseases , Rheumatic Diseases , Humans , Middle Aged , Adolescent , Young Adult , Adult , Hypophysitis/complications , Pituitary Diseases/complications , Pituitary Diseases/diagnosis , Hypopituitarism/etiology , Pituitary Gland/diagnostic imaging , Glucocorticoids/therapeutic use , Rheumatic Diseases/complications , Rheumatic Diseases/drug therapy , Immunosuppressive Agents/therapeutic use , Collagen Diseases/drug therapy , Lupus Erythematosus, Systemic/complications , Lupus Erythematosus, Systemic/drug therapy , Autoimmune Hypophysitis/complications , Autoimmune Hypophysitis/diagnosis , Autoimmune Hypophysitis/drug therapy
19.
Pituitary ; 25(6): 982-987, 2022 Dec.
Article in English | MEDLINE | ID: mdl-36260240

ABSTRACT

Central Diabetes Insipidus (CDI) is mainly associated with structural pathologies of the hypothalamic-pituitary area. Etiologies underlying CDI are identified in most patients, however idiopathic CDI is reported in 13-17% of cases after excluding other etiologies. The Hypopituitarism ENEA Rare Observational Study (HEROS study) retrospectively collected data of patients with idiopathic CDI from 14 pituitary centers in 9 countries. The cohort included 92 patients (59 females 64%), mean age at diagnosis was 35.4 ± 20.7 years, and a mean follow up of 19.1 ± 13.5 years following CDI diagnosis. In 6 women, diagnosis was related to pregnancy. Of 83 patients with available data on pituitary imaging, 40(48%) had normal sellar imaging, and 43(52%) had pathology of the posterior pituitary or the stalk, including loss of the bright spot, posterior pituitary atrophy or stalk enlargement. Anterior pituitary hormone deficiencies at presentation included hypogonadism in 6 (6.5%) patients (5 females), and hypocortisolism in one; during follow-up new anterior pituitary deficiencies developed in 6 patients. Replacement treatment with desmopressin was given to all patients except one, usually with an oral preparation. During follow up, no underlying disease causing CDI was identified in any patient. Patients with idiopathic CDI following investigation at baseline are stable with no specific etiology depicted during long-term follow-up.


Subject(s)
Diabetes Insipidus, Neurogenic , Diabetes Insipidus , Diabetes Mellitus , Hypopituitarism , Pituitary Diseases , Humans , Female , Diabetes Insipidus, Neurogenic/drug therapy , Diabetes Insipidus, Neurogenic/diagnosis , Retrospective Studies , Magnetic Resonance Imaging , Diabetes Insipidus/etiology , Hypopituitarism/complications , Pituitary Diseases/complications , Pituitary Gland/pathology
20.
J Clin Endocrinol Metab ; 108(1): 198-208, 2022 12 17.
Article in English | MEDLINE | ID: mdl-36300330

ABSTRACT

Transsphenoidal surgery is the first-line treatment for many clinically significant pituitary tumors and sellar lesions. Although complication rates are low when performed at high-volume centers, disorders of salt and water balance are relatively common postoperatively. Both, or either, central diabetes insipidus (recently renamed arginine vasopressin deficiency - AVP-D), caused by a deficiency in production and/or secretion of arginine vasopressin, and hyponatremia, most commonly secondary to the syndrome of inappropriate antidiuresis, may occur. These conditions can extend hospital stay and increase the risk of readmission. This article discusses common presentations of salt and water balance disorders following pituitary surgery, the pathophysiology of these conditions, and their diagnosis and management.


Subject(s)
Hyponatremia , Inappropriate ADH Syndrome , Pituitary Diseases , Pituitary Neoplasms , Water-Electrolyte Balance , Humans , Arginine Vasopressin/metabolism , Hyponatremia/etiology , Hyponatremia/therapy , Inappropriate ADH Syndrome/therapy , Inappropriate ADH Syndrome/complications , Pituitary Diseases/complications , Pituitary Neoplasms/surgery , Pituitary Neoplasms/complications , Sodium Chloride , Water
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