scholarly journals Methotrexate-associated lymphoproliferative disorder with hypopituitarism and central diabetes insipidus

2019 ◽  
Vol 2019 ◽  
Author(s):  
Misaki Aoshima ◽  
Koji Nagayama ◽  
Kei Takeshita ◽  
Hiroshi Ajima ◽  
Sakurako Orikasa ◽  
...  
Keyword(s):  
Diabetes Insipidus ◽  
Subcutaneous Tissue ◽  
Brain Magnetic Resonance Imaging ◽  
Immunosuppressive Drugs ◽  
Anterior Mediastinum ◽  
Central Diabetes Insipidus ◽  
Oculomotor Nerve Palsy ◽  
List Type ◽  
Pituitary Dysfunction ◽  
Pituitary Lesion

Summary Patients treated with immunosuppressive drugs, especially methotrexate (MTX), rarely develop lymphoproliferative disorders (LPDs), known as MTX-related LPD (MTX–LPD). The primary site of MTX–LPD is often extranodal. This is the first reported case of MTX–LPD in the pituitary. A 65-year-old woman was admitted to our hospital with symptoms of oculomotor nerve palsy and multiple subcutaneous nodules. She had been treated with MTX for 11 years for rheumatoid arthritis. Computed tomography showed multiple masses in the orbit, sinuses, lung fields, anterior mediastinum, kidney, and subcutaneous tissue. Brain magnetic resonance imaging revealed a sellar mass. She was diagnosed with hypopituitarism and central diabetes insipidus based on endocrine examination. Although pituitary biopsy could not be performed, we concluded that the pituitary lesion was from MTX–LPD, similar to the lesions in the sinuses, anterior mediastinum, and subcutaneous tissue, which showed polymorphic LPD on biopsy. MTX was discontinued, and methylprednisolone was administered to improve the neurologic symptoms. After several weeks, there was marked improvement of all lesions, including the pituitary lesion, but the pituitary function did not improve. When pituitary lesions are caused by MTX–LPD, the possibility of anterior hypopituitarism and central diabetes insipidus needs to be considered. Further studies are needed to investigate the effectiveness of early diagnosis and treatment of MTX–LPD in restoring pituitary dysfunction. Learning points Pituitary lesions from MTX–LPD may cause hypopituitarism and central diabetes insipidus. Pituitary metastasis of malignant lymphoma and primary pituitary lymphoma, which have the same tissue types with MTX–LPD, have poor prognosis, but the lesions of MTX–LPD can regress only after MTX discontinuation. In cases of pituitary lesions alone, a diagnosis of MTX–LPD may be difficult, unless pituitary biopsy is performed. This possibility should be considered in patients treated with immunosuppressive drugs. Pituitary hypofunction and diabetes insipidus may persist, even after regression of the lesions on imaging due to MTX discontinuation.

Metastatic Adenocarcinoma Presenting with Central Diabetes Insipidus

1998 ◽  
Vol 84 (1) ◽  
pp. 85-86
Author(s):  
Mark A. Marinella
Keyword(s):  
Magnetic Resonance Imaging ◽  
Magnetic Resonance ◽  
Diabetes Insipidus ◽  
Water Deprivation ◽  
Brain Magnetic Resonance Imaging ◽  
Central Diabetes Insipidus ◽  
Metastatic Adenocarcinoma ◽  
Rare Occurrence ◽  
Resonance Imaging ◽  
Recent Onset

The case of a previously healthy 63-year-old female with metastatic adenocarcinoma to the hypothalamus presenting with central diabetes insipidus is presented. The patient was found to have metastatic disease isolated to her hypothalamus on brain magnetic resonance imaging as well as a water deprivation test consistent with central diabetes insipidus. The patient had a decrease in symptoms of polyuria and polydypsia as well as a decrease in urine volumes after treatment with intranasal vasopressin. Even though a rare occurrence, physicians should consider metastatic adenocarcinoma in patients with recent-onset polyuria and polydypsia.

Early Copeptin Determination Allows Prompt Diagnosis of Post-Neurosurgical Central Diabetes Insipidus

2019 ◽  
Vol 110 (6) ◽  
pp. 525-534 ◽  
Author(s):  
Alessandro Maria Berton ◽  
Filippo Gatti ◽  
Federica Penner ◽  
Emanuele Varaldo ◽  
Nunzia Prencipe ◽  
...  
Keyword(s):  
Diabetes Insipidus ◽  
Surrogate Marker ◽  
Pituitary Surgery ◽  
Central Diabetes Insipidus ◽  
Optimal Timing ◽  
Vasopressin Release ◽  
Biochemical Alterations ◽  
Post Surgery ◽  
Patients At Risk ◽  
Pituitary Lesion

Introduction: Central diabetes insipidus (CDI) is a frequent complication of pituitary surgery, but its diagnosis lacks standardized criteria. Copeptin, a surrogate marker of arginine vasopressin release, is triggered by psycho-physical stresses such as pituitary surgery. Low postoperative copeptin could predict CDI onset. The aims of this study were the validation of copeptin as a predictor of post-neurosurgical CDI and the identification of the optimal timing for its determination. Methods: Sixty-six consecutive patients operated for a hypothalamic-pituitary lesion were evaluated. Copeptin was determined preoperatively and at 1, 6, 12, 24 and 48 h post-extubation. Fifty-eight patients were reassessed after 3–6 months post-surgery to confirm transient (3 cases) or permanent CDI (5 cases) diagnosis. Results: A marked copeptin peak was identified at 1 h after extubation, when a value below or equal to 12.8 pmol/L had a good accuracy in identifying CDI cases (AUC 0.866, 95% CI 0.751–0.941). Moreover, a copeptin peak above 4.2 pmol/L excluded permanent forms (AUC 1, 95% CI 0.629–1). Regression analysis identified copeptin as the only significant predictor of CDI (OR 0.86, 95% CI 0.75–0.98, p = 0.02). A copeptin T1/T0 ratio below or equal to 1.47 identified patients at risk of isolated biochemical alterations even in the absence of an overt CDI. Conclusions: A prompt increase of copeptin is expected at 1 h after extubation. The absence of this peak is a reliable predictor of post-neurosurgical CDI.

scholarly journals Five cases with central diabetes insipidus and hypogonadism as first presentation of neurosarcoidosis

2000 ◽  
Vol 142 (4) ◽  
pp. 365-372 ◽  
Author(s):  
C Bullmann ◽  
M Faust ◽  
A Hoffmann ◽  
C Heppner ◽  
F Jockenhovel ◽  
...  
Keyword(s):  
Diabetes Insipidus ◽  
Case Reports ◽  
Lymphocytic Hypophysitis ◽  
Central Diabetes Insipidus ◽  
Presenting Symptoms ◽  
Pituitary Dysfunction ◽  
Mri Scans ◽  
Primary Evaluation ◽  
Therapy Result

OBJECTIVES: We retrospectively reviewed 5 patients with neurosarcoidosis, who all presented with central diabetes insipidus and hypogonadism. DESIGN: This was a single-centre, retrospective analysis of 5 cases with a minimum follow-up of 2 years. METHODS: Case analysis included clinical, biochemical, and endocrinological evaluation and frequent CT/MRI scans of involved organs as primary evaluation and in response to immunosuppressive therapy. RESULT: Neurosarcoidosis was diagnosed in all patients. Two patients had no proven extracerebral manifestation and had a stable disease over 3 and 5 years. One patient showed deterioration with corticosteroids alone but partial remission after additional cyclophosphamide. Pituitary dysfunction remained unchanged in all patients, despite total clinical and radiological remission in two patients. However, one of these patients died of acute granulomatous meningoencephalitis after two years of follow-up. CONCLUSION: Although the presenting symptoms of neurosarcoidosis may vary, the occurrence of central diabetes insipidus associated with typical radiological features is suggestive of neurosarcoidosis. However, there is an increasing number of case reports on lymphocytic hypophysitis. Without the bioptic diagnosis, the differentiation between potentially lethal isolated neurosarcoidosis and lymphocytic hypophysitis is difficult. These cases demonstrate the difficulties in diagnosing neurosarcoidosis and reflect experiences with follow-up parameters.

scholarly journals Anterior Pituitary Dysfunction in Children with Idiopathic Central Diabetes Insipidus

1995 ◽  
Vol 4 (Supple6) ◽  
pp. 109-114
Author(s):  
Yoko Nomura ◽  
Kazumichi Onigata ◽  
Ryo Ogawa ◽  
Tomohisa Nagashima ◽  
Hiroshi Miyake ◽  
...  
Keyword(s):  
Diabetes Insipidus ◽  
Anterior Pituitary ◽  
Central Diabetes Insipidus ◽  
Pituitary Dysfunction

scholarly journals One too many diabetes: the combination of hyperglycaemic hyperosmolar state and central diabetes insipidus

2018 ◽  
Vol 2018 ◽  
Author(s):  
Snezana Burmazovic ◽  
Christoph Henzen ◽  
Lukas Brander ◽  
Luca Cioccari
Keyword(s):  
Diabetes Mellitus ◽  
Glycaemic Control ◽  
Specific Gravity ◽  
Diabetes Insipidus ◽  
Urine Osmolality ◽  
Central Diabetes Insipidus ◽  
Urine Specific Gravity ◽  
List Type ◽  
Poor Glycaemic Control ◽  
History Of

Summary The combination of hyperosmolar hyperglycaemic state and central diabetes insipidus is unusual and poses unique diagnostic and therapeutic challenges for clinicians. In a patient with diabetes mellitus presenting with polyuria and polydipsia, poor glycaemic control is usually the first aetiology that is considered, and achieving glycaemic control remains the first course of action. However, severe hypernatraemia, hyperglycaemia and discordance between urine-specific gravity and urine osmolality suggest concurrent symptomatic diabetes insipidus. We report a rare case of concurrent manifestation of hyperosmolar hyperglycaemic state and central diabetes insipidus in a patient with a history of craniopharyngioma. Learning points: In patients with diabetes mellitus presenting with polyuria and polydipsia, poor glycaemic control is usually the first aetiology to be considered. However, a history of craniopharyngioma, severe hypernatraemia, hyperglycaemia and discordance between urine-specific gravity and osmolality provide evidence of concurrent diabetes insipidus. Therefore, if a patient with diabetes mellitus presents with severe hypernatraemia, hyperglycaemia, a low or low normal urinary-specific gravity and worsening polyuria despite correction of hyperglycaemia, concurrent diabetes insipidus should be sought.

scholarly journals Rare occurrence of central diabetes insipidus with dermatomyositis in a young male

2020 ◽  
Vol 2020 ◽  
Author(s):  
Aishah Ekhzaimy ◽  
Afshan Masood ◽  
Seham Alzahrani ◽  
Waleed Al-Ghamdi ◽  
Daad Alotaibi ◽  
...  
Keyword(s):  
Diabetes Insipidus ◽  
Inflammatory Myopathy ◽  
Young Male ◽  
Central Diabetes Insipidus ◽  
Endocrine Disorders ◽  
Extensive Literature ◽  
List Type ◽  
Autoimmune Conditions ◽  
History Of ◽  
Autoimmune Condition

Summary Central diabetes insipidus (CDI) and several endocrine disorders previously classified as idiopathic are now considered to be of an autoimmune etiology. Dermatomyositis (DM), a rare autoimmune condition characterized by inflammatory myopathy and skin rashes, is also known to affect the gastrointestinal, pulmonary, and rarely the cardiac systems and the joints. The association of CDI and DM is extremely rare. After an extensive literature search and to the best of our knowledge this is the first reported case in literature, we report the case of a 36-year-old male with a history of CDI, who presented to the hospital’s endocrine outpatient clinic for evaluation of a 3-week history of progressive facial rash accompanied by weakness and aching of the muscles. Learning points: Accurate biochemical diagnosis should always be followed by etiological investigation. This clinical entity usually constitutes a therapeutic challenge, often requiring a multidisciplinary approach for optimal outcome. Dermatomyositis is an important differential diagnosis in patients presenting with proximal muscle weakness. Associated autoimmune conditions should be considered while evaluating patients with dermatomyositis. Dermatomyositis can relapse at any stage, even following a very long period of remission. Maintenance immunosuppressive therapy should be carefully considered in these patients.

Central diabetes insipidus caused by a pituitary stalk germinoma resembling infundibuloneurohypophysitis

2020 ◽  
Vol 13 (9) ◽  
pp. e234724
Author(s):  
Daniela Dias ◽  
Helena Vilar ◽  
João Passos ◽  
Valeriano Leite
Keyword(s):  
Diabetes Insipidus ◽  
Brain Mri ◽  
Correct Diagnosis ◽  
Central Diabetes Insipidus ◽  
Pituitary Stalk ◽  
Initial Assessment ◽  
Complete Disappearance ◽  
Close Monitoring ◽  
Initial Symptoms ◽  
Pituitary Lesion

We report the case of a pituitary stalk germinoma initially misdiagnosed and treated as infundibuloneurohypophysitis (INH). A 27-year-old man presented with a 1-year history of polydipsia, polyuria, nycturia consistent with central diabetes insipidus and a hyperintense pituitary stalk lesion on MRI. A possible INH diagnosis was considered, after excluding other pathologies. Lesion biopsy was discarded at that time on the ground of a small target and the high risk of added morbidity. Oral desmopressin led to initial symptoms resolution but, in the following months, an anterior panhypopituitarism developed, in spite of appropriate treatment and, by that time, the brain MRI also revealed lesion growth, which prompted a biopsy recommendation. The pathology analysis revealed a germinoma. After chemotherapy and radiotherapy, there was complete disappearance of the pituitary lesion, but the panhypopituitarism persisted. In conclusion, this case highlights the importance and difficulty of precise diagnosis in the initial assessment of pituitary stalk lesions and the need for close monitoring of treatment response. Diagnostic reassessment and biopsy in atypical cases is the only path to achieve the correct diagnosis and treatment.

Endocrine Outcomes In Central Diabetes Insipidus: the Predictive Value of Neuroimaging “Mismatch Pattern”

2020 ◽  
Vol 105 (11) ◽  
Author(s):  
Deborah Bianco ◽  
Flavia Napoli ◽  
Giovanni Morana ◽  
Angela Pistorio ◽  
Anna Elsa Maria Allegri ◽  
...  
Keyword(s):  
Pituitary Gland ◽  
Diabetes Insipidus ◽  
Contrast Enhancement ◽  
Anterior Pituitary ◽  
Magnetic Resonance Images ◽  
Central Diabetes Insipidus ◽  
Pituitary Hormone ◽  
Gland Size ◽  
Pituitary Dysfunction

Abstract Context The etiology of central diabetes insipidus (CDI) in children is often unknown. Clinical and radiological features at disease onset do not allow discrimination between idiopathic forms and other conditions or to predict anterior pituitary dysfunction. Objective To evaluate the evolution of pituitary stalk (PS) thickening and the pattern of contrast-enhancement in relation with etiological diagnosis and pituitary function. Methods We enrolled 39 children with CDI, 29 idiopathic and 10 with Langerhans cell histiocytosis (LCH). Brain magnetic resonance images taken at admission and during follow-up (332 studies) were examined, focusing on PS thickness, contrast-enhancement pattern, and pituitary gland size; T2-DRIVE and postcontrast T1-weighted images were analyzed. Results Seventeen of 29 patients (58.6%) with idiopathic CDI displayed “mismatch pattern,” consisting in a discrepancy between PS thickness in T2-DRIVE and postcontrast T1-weighted images; neuroimaging findings became stable after its appearance, while “mismatch” appeared in LCH patients after chemotherapy. Patients with larger PS displayed mismatch more frequently (P = 0.003); in these patients, reduction of proximal and middle PS size was documented over time (P = 0.045 and P = 0.006). The pituitary gland was smaller in patients with mismatch (P < 0.0001). Patients with mismatch presented more frequently with at least one pituitary hormone defect, more often growth hormone deficiency (P = 0.033). Conclusions The PS mismatch pattern characterizes patients with CDI, reduced pituitary gland size, and anterior pituitary dysfunction. The association of mismatch pattern with specific underlying conditions needs further investigation. As patients with mismatch show stabilization of PS size, we assume a prognostic role of this peculiar pattern, which could be used to lead follow-up.

scholarly journals Syndrome of Inappropriate Antidiuretic Hormone Secretion (SIADH) and Subsequent Central Diabetes Insipidus: A Rare Presentation of Pituitary Apoplexy

2019 ◽  
Vol 2019 ◽  
pp. 1-4
Author(s):  
S. B. Smedegaard ◽  
J. O. Jørgensen ◽  
N. Rittig
Keyword(s):  
Diabetes Insipidus ◽  
Antidiuretic Hormone ◽  
Pituitary Apoplexy ◽  
Hormone Secretion ◽  
Central Diabetes Insipidus ◽  
Plasma Sodium ◽  
Fluid Restriction ◽  
Inappropriate Antidiuretic Hormone Secretion ◽  
Antidiuretic Hormone Secretion ◽  
Pituitary Dysfunction

Pituitary apoplexy (PA) is a rare endocrine emergency that occasionally presents with sodium disturbances. Here we present a rare case with a previously healthy 41-year-old female who presented with acute onset headache and nausea without visual impairment or overt pituitary dysfunction. Plasma sodium concentrations declined abruptly during the first two days of admission to a nadir of 111 mmol/l. Urine and blood chemistry were consistent with syndrome of inappropriate antidiuretic hormone secretion (SIADH). Magnetic resonance imaging revealed recent bleeding into a pituitary cystic process. Hyponatremia was successfully corrected with fluid restriction and both visual function and anterior pituitary function remained intact. Subsequently, the patient developed central diabetes insipidus (CDI), which responded well to desmopressin substitution. To our knowledge, this is the first case of PA presenting predominantly with posterior pituitary dysfunction that transitioned from SIADH to permanent CDI.

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