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Giant cell myocarditis with central diabetes insipidus: A case report.

ABSTRACT: A 51-year-old male, previously diagnosed with central diabetes insipidus due to lymphocytic hypophysitis, presented with fever and dyspnea for 1 week. On arrival, he exhibited hypotension (85/60 mmHg) and sinus tachycardia (110 bpm). His electrocardiogram revealed mild ST elevation on V2-V4. Echocardiography indicated a near-normal (50%) left ventricular ejection fraction (LVEF), although the inferior wall of the left ventricle exhibited severe hypokinesis. Fulminant myocarditis and circulatory insufficiency were suspected, and treatment with dobutamine, 3 ?g/kg/min, was started. His LVEF gradually decreased to 20%. On day 17, he developed cardiogenic shock due to ventricular tachycardia and underwent peripheral venous-arterial extracorporeal membrane oxygenation and intra-aortic balloon pumping. Although he did not exhibit polyuria, intravenous vasopressin infusion (0.5 U/h) was performed to maintain normonatremia. Endomyocardial biopsy results revealed the infiltration of scattered giant cells (GCs) and extensive lymphocytes. Despite immunosuppressive therapy (methylprednisolone and cyclosporine), his cardiac function did not recover. On day 36, he received a biventricular assist device; however, he died on day 47 due to the progression of sepsis and multiple organ failure. We speculate that a deficient expression of programmed cell death protein-1 was the cause of both GC myocarditis and lymphocytic hypophysitis. .

SUBMITTER: Yamaguchi S

PROVIDER: S-EPMC6951310 | biostudies-literature | 2020 Jan

REPOSITORIES: biostudies-literature

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Giant cell myocarditis with central diabetes insipidus: A case report.

Yamaguchi Shogo S Sawamura Akinori A Nakaguro Masato M Shimoyama Yoshie Y Morimoto Ryota R Kato Hiroo H Arao Yoshihito Y Oishi Hideo H Haga Tomoaki T Kuwayama Tasuku T Yokoi Tsuyoshi T Hiraiwa Hiroaki H Kondo Toru T Okumura Takahiro T Murohara Toyoaki T

Journal of cardiology cases 20190910 1

A 51-year-old male, previously diagnosed with central diabetes insipidus due to lymphocytic hypophysitis, presented with fever and dyspnea for 1 week. On arrival, he exhibited hypotension (85/60 mmHg) and sinus tachycardia (110 bpm). His electrocardiogram revealed mild ST elevation on V2-V4. Echocardiography indicated a near-normal (50%) left ventricular ejection fraction (LVEF), although the inferior wall of the left ventricle exhibited severe hypokinesis. Fulminant myocarditis and circulatory ...[more]

PMID: 31933698

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Project description:The treatment of asymptomatic hydronephrosis due to ureteropelvic junction obstruction (UPJO), as well as the timing and indications for surgical intervention, remain controversial. Diabetes insipidus (DI) is a rare disease in infants that is known to cause non-obstructive hydronephrosis, while its association with obstructive hydronephrosis has not been reported. Some studies have found that increased water intake is a predisposing risk factor of developing hydronephrosis. However, there are no studies or guidelines that indicate the specific follow-up intervals and surgical indications for mild hydronephrosis if predisposing risk factors persist. A 46-month-old boy was admitted to our Urology Department with a history of Society of Fetal Urology (SFU) Grade 2 prenatal left hydronephrosis, which was stable at SFU Grade 1-2 at regular postnatal follow-ups. The patient developed polydipsia and polyuria three months prior to admission, then he was considered as primary polydipsia by endocrinology and was treated with fluid restriction while the examination was negative. Renal ultrasound at follow-up demonstrated severe left hydronephrosis with an anterior-posterior diameter (APD) of 6.25 cm three months after symptom onset. Diuretic renography (DR) revealed a renal function of 13.7% with a glomerular filtration rate (GFR) of 11.25 mL/min. The patient was otherwise asymptomatic without any abdominal pain or vomiting. He underwent left pyelostomy immediately as well as laparoscopic left dismembered ureteropelvioplasty after three months, and a diagnosis of UPJO was confirmed. The patient had an uncomplicated postoperative recovery and the result of follow-up renal ultrasound was stable. However, the symptoms of polydipsia and polyuria did not improve significantly. He underwent pituitary magnetic resonance imaging (MRI) and pathological examination, the results of which were consistent with central DI caused by Langerhans cell hyperplasia four months postoperatively. This case indicates the need to pay attention to children with mild hydronephrosis undergoing regular observation and conservative treatment, as a sudden aggravation of the hydronephrosis and a rapid decline of renal function may occur if DI persists.

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Giant cell myocarditis with central diabetes insipidus: A case report.

Publications

Giant cell myocarditis with central diabetes insipidus: A case report.

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