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JAMA Netw Open 2021 May 3;4(5):e219878. Epub 2021 May 3.

Department of Pediatrics, Ribeirao Preto Medical School, University of Sao Paulo, Sao Paulo, Brazil.

Importance: The Zika virus infects progenitor neuron cells, disrupts cerebral development, and, in mice, drives hypothalamic defects. Patients with microcephaly caused by congenital Zika infection present with midline cerebral defects, which may result in hypopituitarism.

Objective: To analyze postnatal growth and the presence of clinical and biochemical features associated with hypopituitarism in children with congenital Zika infections. Read More

Endocrinol Diabetes Metab Case Rep 2021 May 1;2021. Epub 2021 May 1.

Department of Endocrinology and Diabetes, Royal Brisbane and Women's Hospital, Brisbane, Australia.

Summary: A 34-year-old woman presented 18 months post-partum with blurred vision, polyuria, amenorrhoea, headache and general malaise. Comprehensive clinical examination showed left superior temporal visual loss only. Initial investigations revealed panhypopituitarism and MRI demonstrated a sellar mass involving the infundibulum and hypothalamus. Read More

Endocrinol Diabetes Metab Case Rep 2021 May 1;2021. Epub 2021 May 1.

Department of Endocrinology and Metabolism, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito-shi, Ibaraki, Japan.

Summary: A 61-year-old man developed central diabetes insipidus caused by mixed histiocytosis (MH) representing Langerhans cell histiocytosis overlapping with Erdheim-Chester disease. Bone, skin, vascular, and retroperitoneal involvements were also observed. Dynamic hormonal testing showed normal responses for anterior pituitary hormones, except for impaired secretion of growth hormone (GH). Read More

Radiol Case Rep 2021 Jun 17;16(6):1514-1517. Epub 2021 Apr 17.

Department of Radiology, University of Missouri, Columbia, MO.

We presented the late magnetic resonance imaging characteristics in a 47-year-old male who diagnosed with a permanent trauma-induced diabetes insipidus. The patient developed polyuria following a deceleration injury which has been diagnosed as central diabetes insipidus based on the water deprivation test. Computed tomography or magnetic resonance evaluation of the pituitary gland is usually normal in such cases. Read More

Int J Environ Res Public Health 2021 Apr 30;18(9). Epub 2021 Apr 30.

Department of Biomedical Sciences and Human Oncology (DIMO), Division of Medical Genetics, University of Bari "Aldo Moro", 70124 Bari, Italy.

Wolfram syndrome is a rare neurodegenerative disorder that is typically characterized by diabetes mellitus and optic atrophy. Other common features are diabetes insipidus and hearing loss, but additional less-frequent findings may also be present. The phenotype spectrum is quite wide, and penetrance may be incomplete. Read More

J Endocrinol Invest 2021 May 3. Epub 2021 May 3.

Neuromed Institute, IRCCS, Pozzilli, IS, Italy.

Purpose: Salivary gland (SG) tissue and derived neoplasms may occur in the sellar region. As the current literature is mostly limited to case reports, the puzzling case of an inflammatory SG removed by transsphenoidal surgery (TS) and mimicking a prolactinoma prompted us to perform the first systematic review of these unusual conditions.

Methods: A systematic literature search was conducted according to the PRISMA guidelines. Read More

Case Rep Neurol Med 2021 13;2021:6690372. Epub 2021 Apr 13.

Department of Pathology, Uonuma Kikan Hospital, Uonuma Institute of Community Medicine, Niigata University Medical and Dental Hospital, Niigata 949-7302, Japan.

Prolonged postoperative pyrexia (PPP) due to Mollaret's meningitis following endoscopic transsphenoidal surgery (eTSS) for an intracranial epidermoid cyst can be confused with postoperative meningeal infection after transsphenoidal resection, especially in the middle of the COVID-19 pandemic. Anosmia, as well as dysgeusia, cannot be evaluated in patients of eTSS for a while after surgery. We report a case of an infundibular epidermoid cyst with post-eTSS Mollaret's meningitis (MM). Read More

Clin Case Rep 2021 Apr 27;9(4):1937-1942. Epub 2021 Feb 27.

Division of Pediatric Endocrinology KidZ Health Castle UZ Brussel Vrije Universiteit Brussel Brussels Belgium.

Psychological stress is a risk factor for primary polydipsia in adolescents without psychiatric comorbidity. Taking a detailed family and social history can help to distinguish primary polydipsia from diabetes insipidus in adolescents with challenging presentations of polydipsia and polyuria. Read More

J Investig Med High Impact Case Rep 2021 Jan-Dec;9:23247096211012191

Children's Healthcare of Atlanta, Emory University, Atlanta, GA, USA.

Neurosarcoidosis is a rare phenomenon in the pediatric population, with only a few cases reported in the literature worldwide. While hypothalamo-pituitary involvement is known to occur, direct infiltration of the pituitary gland and isolated anterior pituitary dysfunction without diabetes insipidus is seldom observed. A high index of suspicion is required for diagnosis of neurosarcoidosis, and treatment can be challenging due to lack of standardized guidelines. Read More

J Neurosci Rural Pract 2021 Apr 1;12(2):343-349. Epub 2021 Apr 1.

Department of Neuroimaging and Interventional Radiology, National Institute of Mental Health and Neurosciences, Bengaluru, Karnataka, India.

 The treatment of cystic craniopharyngioma in children is varied. The treatment ranges from radical excision to direct radiotherapy. As the morbidity of excision is high, more conservative approaches are used. Read More

J Clin Med 2021 Apr 14;10(8). Epub 2021 Apr 14.

Department of Endocrinology, Faculty of Medicine, Medical College, Jagiellonian University, 31-008 Krakow, Poland.

Pituitary stalk lesions (PSL) are a very rare pathology. The majority of conditions affecting the infundibulum do not present with clinically apparent symptoms, what makes the diagnosis difficult. The recognition might be also complicated by the non-specific and transient characteristics of hormonal insufficiencies. Read More

Healthcare (Basel) 2021 Apr 1;9(4). Epub 2021 Apr 1.

Erode College of Pharmacy, Tamil Nadu Dr. M.G.R. Medical University, Tamilnadu 638112, India.

Dipsogenic diabetes insipidus (DDI) is a subtype of primary polydipsia (PP), which occurs mostly in healthy people without psychiatric disease. In contrast, PP is characterized by a polyuria polydipsia syndrome (PPS) associated with psychiatric illness. However, the pathogenesis of DDI is not well established and remains unexplored. Read More

Endocrinol Diabetes Metab Case Rep 2021 Apr 1;2021. Epub 2021 Apr 1.

Department of Pediatrics, Keio University School of Medicine, shinjyuku, Tokyo, Japan.

Summary: We report a male infant with congenital nephrogenic diabetes insipidus (NDI) who presented with hypercalcemia and hyperphosphatemia since birth. Serum sodium started to increase at 39 days. Although there was no polyuria, urine osmolality was 71 mOsm/kg, when serum osmolality was 296 mOsm/kg with plasma arginine vasopressin 22. Read More

Pan Afr Med J 2021 26;38:86. Epub 2021 Jan 26.

Department of Pediatric Endocrinology, Faculty of Medicine, University of Khartoum, khartoum, Sudan.

Adipsia is a rare condition characterized by a lack of thirst due to a defect in specific osmoreceptors located in the hypothalamus. The disorder is characterized by failure to maintain the body's normal plasma osmolality (POSM), resulting in chronic or recurrent severe hypernatremia and dehydration. Adipsia is usually accompanied by central diabetes insipidus (DI). Read More

BMC Endocr Disord 2021 Apr 21;21(1):78. Epub 2021 Apr 21.

Department of Endocrinology, Zhejiang Provincial People's Hospital, Affiliated People's Hospital, Hangzhou Medical College, Hangzhou, 310003, Zhejiang province, China.

Background: Diabetes insipidus (DI) can be a common cause of polydipsia and polyuria. Here, we present a case of congenital nephrogenic diabetes insipidus (CNDI) accompanied with central diabetes insipidus (CDI) secondary to pituitary surgery.

Case Presentation: A 24-year-old Chinese woman came to our hospital with the complaints of polydipsia and polyuria for 6 months. Read More

J Pediatr Endocrinol Metab 2021 Apr 21. Epub 2021 Apr 21.

Department of Pediatric Endocrinology, Istanbul University-Cerrahpasa Cerrahpasa Faculty of Medicine, Istanbul, Turkey.

Objectives: Wolfram syndrome (WS) is a rarely seen autosomal recessive multisystem neurodegenerative disorder caused by mutations in the gene.

Case Presentation: Three sisters with WS had diabetes mellitus (DM) at 4 years of age and optic atrophy. In addition, the first case had hearing impairment, and the second case had diabetes insipidus and urinary incontinence. Read More

J Hum Genet 2021 Apr 21. Epub 2021 Apr 21.

Center for Statistical Genetics, Gertrude H. Sergievsky Center, and the Department of Neurology, Columbia University Medical Center, New York, NY, USA.

Background: Wolfram syndrome (WFS) is characterized by deafness, diabetes mellitus, and diabetes insipidus along with optic atrophy. WFS has an autosomal recessive mode of inheritance and is due to variants in WFS1 and CISD2.

Methods: We evaluated the underlying molecular etiology of three affected members of a consanguineous family with hearing impairment, bicuspid aortic valve, diabetes mellitus and insipidus, clinodactyly, and gastrointestinal tract abnormalities via exome sequencing approach. Read More

BMC Endocr Disord 2021 Apr 21;21(1):76. Epub 2021 Apr 21.

Department of Endocrinology, The First Affiliated Hospital of Sun Yat-sen University, 58 Zhongshan Road 2, Guangzhou, 510080, P. R. China.

Background: Wolfram syndrome (WS) is a rare autosomal recessive disorder characterized by diabetes insipidus, diabetes mellitus, optic atrophy and deafness. Mutations in Wolfram syndrome 1 (WFS1) gene may cause dysregulated endoplasmic reticulum (ER)-stress and cell apoptosis, contributing to WS symptoms. The aim of this study was to identify the molecular etiology of a case of WS and to explore the functional consequence of the mutant WFS1 gene in vitro. Read More

Clin Pediatr Endocrinol 2021 3;30(2):115-118. Epub 2021 Apr 3.

Department of Gastroenterology, Nutrition and Endocrinology, Osaka Women's and Children's Hospital, Osaka, Japan.

Endocrinol Diabetes Metab Case Rep 2021 Feb 26;2021. Epub 2021 Feb 26.

Endocrinology Department, Centro Hospitalar e Universitário do Porto, EPE, Porto, Portugal.

Summary: Pituitary metastasis (PM) can be the initial presentation of an otherwise unknown malignancy. As PM has no clinical or radiological pathognomonic features, diagnosis is challenging. The authors describe the case of a symptomatic PM that revealed a primary lung adenocarcinoma. Read More

World Neurosurg 2021 Apr 12. Epub 2021 Apr 12.

Affiliations: Department of Neurosurgery, Nanfang Hospital of Southern Medical University, Guangzhou, China.

Objective: Serum sodium abnormalities are one of the most common manifestations following radical CP excision. The aim of this study was to report the incidence and possible predictors of serum sodium disturbance and explore features of sodium destabilization manifestation among QST classification results following craniopharyngioma resection.

Methods: A retrospective analysis of clinical, biochemical, radiological and operative data for 134 successive patients who underwent primary craniopharyngioma removal between September 2016 and March 2018 was performed. Read More

J Korean Neurosurg Soc 2021 Apr 16. Epub 2021 Apr 16.

Department of Neurosurgery, Bakirkoy Research & Training Hospital for Psychiatry, Neurology, and Neurosurgery, Health Sciences University, Istanbul, Turkey.

Objective: The endoscopic endonasal transsphenoidal approach is a widely-used method for the surgical treatment of pituitary adenomas. We aimed to evaluate the results of endoscopic surgery by comparing preoperative classification methods and investigating their relationship with postoperative resection and remission rates and complications.

Methods: We retrospectively reviewed the medical records of 236 patients (118 males) who underwent surgery for pituitary adenomas. Read More

Endocrine 2021 Apr 14. Epub 2021 Apr 14.

Department of Endocrinology and Nutrition, Cliniques Universitaires Saint-Luc, Université Catholique de Louvain, Brussels, Belgium.

Objectives: Nonfunctionning pituitary macroadenomas (NFPMA) are benign tumors that cause symptoms of mass effects including hypopituitarism. Their primary treatment is transsphenoidal surgery. We aimed to determine the outcome of pituitary hormone deficits after surgical treatment of NFPMA and to identify factors predicting hormonal recovery. Read More

AACE Clin Case Rep 2021 Jan-Feb;7(1):47-50. Epub 2020 Dec 28.

Department of Endocrinology, Townsville University Hospital, Douglas, Queensland, Australia.

Objective: To evaluate the case of a woman who presented with central hypogonadism and diabetes insipidus and further developed a persistent cough leading to an unexpected diagnosis of burnt-out Langerhans cell histiocytosis (LCH).

Methods: Clinical and laboratory endocrine evaluation, magnetic resonance imaging, high-resolution computed tomography, and open-lung biopsy results are discussed.

Results: A 28-year-old woman presented at 10 months postpartum with polydipsia, polyuria, and amenorrhea for 3 months. Read More

Front Vet Sci 2021 24;8:603262. Epub 2021 Mar 24.

Pharmaceutical Sciences Research Center, Shiraz University of Medical Sciences, Shiraz, Iran.

Lithium (Li) is prescribed against a wide range of neurological disorders. Besides its excellent therapeutic properties, there are several adverse effects associated with Li. The impact of Li on renal function and diabetes insipidus is the most common adverse effect of this drug. Read More

Endocrine 2021 Apr 10. Epub 2021 Apr 10.

Department of Neurological Surgery, Imam Khomeini Hospital Complex, Tehran University of Medical Sciences, Tehran, Iran.

Hypophysitis is a rare pituitary inflammatory disorder classified in different ways. Immunoglobulin G4-related disease (IgG4-RD), also a rare disease is a systemic fibro-inflammatory condition characterized by infiltration of tissue with IgG4-positive plasma cells; however prevalence of both of them probably is underestimated. In this paper, we present an Iranian patient with biopsy-proven IgG4-related hypophysitis and then review the clinical characteristics, laboratory, imaging, pathologic findings and therapeutic management as well as prognosis of 115 published cases of hypophysitis secondary to IgG4-related disease. Read More

BMJ Case Rep 2021 Apr 9;14(4). Epub 2021 Apr 9.

Department of Otolaryngology and Head and Neck Surgery, Guy's and Saint Thomas' NHS Foundation Trust, London, UK.

Cranial diabetes insipidus (DI), which can cause life-threatening dehydration, is treated with desmopressin, often intranasally. This is challenging in patients whose nasal airflow is altered, such as those requiring tracheostomy. We report the case of a patient, taking intranasal desmopressin for cranial DI, who underwent partial glossectomy, free-flap reconstruction and tracheostomy. Read More

Aging (Albany NY) 2021 Mar 26;13(7):10087-10098. Epub 2021 Mar 26.

Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.

Hypothalamic nuclei in the preoptic and anterior hypothalamic region (POAH) are critically involved in thermoregulation and neuroendocrine regulation and can be displaced by craniopharyngiomas (CPs). We aimed to locate the POAH by visualizing hypothalamic thermoregulation through task-related functional magnetic resonance imaging (fMRI) to guide hypothalamus protection intraoperatively. Nine adult healthy volunteers (HVs) and thirty-two adult primary CP patients underwent task-related fMRI for POAH localization by warm (60° C) and cold (0° C) cutaneous thermoreceptor stimulation. Read More

Best Pract Res Clin Endocrinol Metab 2021 Feb 6:101489. Epub 2021 Feb 6.

USC Pituitary Center, Department of Neurosurgery, Keck School of Medicine of University of Southern California, 1300 N. State Street, Suite 3300, Los Angeles, CA, 90033, USA; Department of Medicine, Division of Endocrinology and Diabetes, Keck School of Medicine of University of Southern California, 1333 San Pablo Street, BMT-B11, Los Angeles, CA, 90033, USA. Electronic address:

Surgical failure or recurrence of Cushing's disease can be treated with medical therapy, radiotherapy, adrenalectomy, and/or repeat transsphenoidal surgery, all of which have their respective benefits and drawbacks. Redo transsphenoidal surgery has been shown to achieve at least short-term remission in about 40-80% of patients and is associated with low rates of morbidity and near-zero mortality, albeit higher rates of postoperative hypopituitarism, diabetes insipidus, and cerebrospinal fluid leak than initial resection. Despite this, recurrence may ensue in 50% of patients. Read More

Biomedicines 2021 Mar 15;9(3). Epub 2021 Mar 15.

Department of Biochemistry, School of Medicine, Tzu Chi University, Hualien 97004, Taiwan.

Congenital nephrogenic diabetes insipidus (CNDI) is a genetic disorder caused by mutations in arginine vasopressin receptor 2 () or aquaporin 2 genes, rendering collecting duct cells insensitive to the peptide hormone arginine vasopressin stimulation for water reabsorption. This study reports a first identified mutation in Taiwan and demonstrates our effort to understand the pathogenesis caused by applying computational structural analysis tools. The CNDI condition of an 8-month-old male patient was confirmed according to symptoms, family history, and DNA sequence analysis. Read More