Dataset Information

A first case report of dapsone inducing recurrent ventricular arrhythmia.

ABSTRACT: Background:Ventricular arrhythmias (VAs) are life-threatening arrhythmias which are associated with significant morbidity and mortality. Ventricular arrhythmias are induced by a change in the myocardial environment altering cardiomyocyte electrophysiology. The substrate for VA includes myocardial scar, electrolyte disturbances, and drugs altering cellular electrophysiology. Case summary:Here, we present a case of a 52-year-old man with known ischaemic cardiomyopathy, presenting with VA storms secondary to dapsone, an anti-microbial used in this case for the prophylaxis of pneumocystis pneumonia. This is the first case linking dapsone to the development of VAs. Ventricular arrhythmias storm occurred towards the end of the course of anti-microbial therapy and the patient was referred for sympathectomy. However, following the end of treatment, no further VA occurred and sympathectomy was therefore avoided. Discussion:The underlying mechanism for the association between dapsone treatment and VA is unclear and a prolonged QTc was not observed in our case. It is important to recognize that every drug has many physiological effects and in patients with underlying diseases whereby there is already an unfavourable environment, additional drugs can lower the threshold of triggering an arrhythmia and the result can be life-threatening. In a patient with ischaemic cardiomyopathy, where underlying substrate for VA may already exist, the introduction of dapsone could lower the threshold for development of arrhythmia.

SUBMITTER: Wong B

PROVIDER: S-EPMC6939790 | biostudies-literature |

REPOSITORIES: biostudies-literature

ACCESS DATA

Similar Datasets

Project description:BackgroundAnti-mitochondrial antibody (AMA)-positive myositis is an atypical inflammatory myopathy characterized by chronic progression of muscle atrophy and cardiac involvement. Few detailed reports have shown the clinical course of the cardiac complications of AMA-positive myositis.Case summaryA 47-year-old man presented with shortness of breath on exertion. Cardiac dilatation was visible on chest X-ray, and echocardiography demonstrated diffuse hypokinesis with a reduced left ventricular (LV) ejection fraction of 30%. He had mild muscle weakness in the bilateral iliopsoas muscles, and his creatine kinase (CK) and anti-mitochondrial M2 antibody levels were elevated. A liver biopsy showed no findings of primary biliary cholangitis. Coronary angiography revealed normal coronary arteries. An endomyocardial biopsy showed interstitial fibrosis and marked degeneration of the mitochondria. Fluorodeoxyglucose (FDG)-positron emission tomography/computed tomography showed circumferential abnormal accumulation in the LV myocardium, and he was diagnosed with cardiomyopathy associated with AMA-positive myositis. Optimal drug therapy for heart failure was started, and a cardiac resynchronization therapy-defibrillator was implanted. However, his cardiac function did not improve, and he was hospitalized due to ventricular tachycardia storm 5 years after the diagnosis. Ventricular tachycardia was terminated by radiofrequency catheter ablation on the LV-anterior papillary muscle. Steroid therapy was initiated and resulted in a decreased uptake of FDG and a normalized CK level at 3 months after his second discharge; however, LV systolic dysfunction remained 1 year later.DiscussionAnti-mitochondrial antibody-positive myositis can affect the myocardium and cause severe LV dysfunction and life-threatening ventricular arrhythmia over time.KeywordsAnti-mitochondrial antibody-positive myositis • Endomyocardial biopsy • Ventricular tachycardia • Left ventricular dysfunction • Case report • Magnetic resonance imaging • Near-infrared spectroscopy-intravascular ultrasound.

Project description:INTRODUCTION: Carney complex is a multiple neoplasia syndrome involving cardiac, endocrine, neural and cutaneous tumors with a variety of pigmented skin lesions. It has an autosomal dominant mode of inheritance. Approximately 7% of cardiac myxomas are related to the Carney complex. Myxomas that occur as part of the Carney complex affect both sexes with equal frequency. Cardiac myxomas with Carney complex are reported mostly in the left side of the heart and are less common on the right side. As per our review, this is the first reported case of Carney complex with right ventricle cardiac myxoma. CASE PRESENTATION: We present a rare case of recurrent cardiac myxoma in a patient later diagnosed to have Carney complex. A 46-year-old Caucasian man with a history of thyroid hyperplasia came to out-patient cardiology department with new onset atrial fibrillation. A transthoracic echocardiogram revealed a right ventricular mass attached to his interventricular septum, which was later seen on a transesophageal echocardiogram and cardiac magnetic resonance imaging. He underwent resection of the ventricular mass which on pathology revealed myxoma. He later developed skin lesions, pituitary adenoma and Sertoli cell tumor suggesting Carney complex. Two years later he developed a new mass within his right atrium which was later resected. CONCLUSIONS: Carney complex is a rare autosomal dominant disease with variable penetrance. Since it involves multiple organs, patients diagnosed with Carney complex should undergo serial endocrine workup, neural assessments, echocardiograms and testicular ultrasounds. Of the total number of cases of Carney complex, 65% are linked to PRKAR1A gene mutation. It is important for clinicians to be cognizant of a link between cardiac myxoma and Carney complex. The use of multi-imaging modalities allows better delineation of the mass before planned resection. Carney complex-related cardiac myxoma comprises 7% of all cardiac myxomas. Right ventricular cardiac myxomas are rare. This case report is the first to describe right ventricular myxoma with Carney complex.

Dataset Information

A first case report of dapsone inducing recurrent ventricular arrhythmia.

Similar Datasets

OmicsDI is part of the ELIXIR infrastructure