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- Danielsson, Christian, et al.
(författare)
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Haemophagocytic lymphohistiocytosis after heart transplantation: a case report.
- 2020
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Ingår i: European heart journal. Case reports. - : Oxford University Press (OUP). - 2514-2119. ; 4:3, s. 1-4
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Tidskriftsartikel (refereegranskat)abstract
- Haemophagocytic lymphohistiocytosis (HLH) is an uncommon but serious systemic inflammatory response with high mortality rates. It can be triggered by malignancy or infectious agents, often in the context of immunosuppression. Literature covering HLH in heart transplantation (HTx) is scarce.A 25-year-old male with a history of celiac disease underwent HTx at Sahlgrenska Hospital in 2011 due to giant cell myocarditis and was treated with tacrolimus, mycophenolate mofetil (MMF), and prednisolone. He developed several episodes of acute cellular rejections (ACR) during the first 3 post-HTx years, which subsided after addition of everolimus. In May 2017, the patient was admitted to the hospital due to fever without focal symptoms. He had an extensive inflammatory reaction, but screening for infectious agents was negative. Haemophagocytic lymphohistiocytosis was discussed early, but first dismissed since two bone marrow biopsies revealed no signs of haemophagocytosis. Increasing levels of soluble IL-2 were considered confirmative of the diagnosis. Even with intense immunosuppressant treatment, the patient deteriorated and died in progressive multiorgan failure within 2weeks of the symptom onset.A 25-year-old HTx recipient with an extensive inflammatory response, fulfilled criteria for HLH, but the diagnosis was delayed due to normal bone marrow biopsies. A background with autoimmune reactivity and immunosuppressive therapy may have contributed to HLH, but the actual trigger was not identified. Haemophagocytic lymphohistiocytosis can occur in HTx recipients in the absence of malignancy, identifiable infectious triggers and signs of haemophagocytosis. Early diagnosis and intervention are likely to be of importance for a favourable outcome.
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- Galanti, Kristian, et al.
(författare)
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Burnt-out Cori–Forbes cardiomyopathy
- 2023
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Ingår i: European Heart Journal - Case Reports. - 2514-2119. ; 7:8
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Tidskriftsartikel (refereegranskat)
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- Karlsson, Lars O, et al.
(författare)
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Catheter ablation of ventricular tachycardia in a patient with a left endoventricular patch : a case report
- 2017
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Ingår i: European Heart Journal - Case Reports. - : Oxford Academic. - 2514-2119. ; 1:2, s. 1-4
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Tidskriftsartikel (refereegranskat)abstract
- Surgical resection of a left ventricular aneurysm in the setting of ventricular tachycardia (VT) was first described by Couch in 1959. The technique was further developed by Dor et al. with performance of endocardiectomy and complete myocardial revascularization. Despite an attempt to remove the arrhythmogenic substrate, however, recurrences of VT remain an issue. Furthermore, the surgical technique used entails limited access to the potential area of interest with regard to a percutaneous catheter ablation procedure. We present a case report of a 65-year-old man who was referred for catheter ablation due to recurrent episodes of VT. He had undergone a coronary artery bypass surgery 8 years previously. During surgery, resection of an apical thrombus and reconstruction of an apical aneurysm with a Fontan stitch and an endoventricular patch were performed. The mapping and ablation procedure was aided by intracardiac echocardiography. During mapping, the ablation catheter was noticed to enter the apical pouch from the inferoseptal border of the endoventricular patch. During the ablation procedure, one of the VTs was successfully ablated in the inferior aspect of the apical pouch. This report confirms that the arrhythmogenic substrate underneath an endoventricular patch may be accessed in some instances and that these complex catheter ablation procedures may benefit from the use of intracardiac echocardiography.
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- Littmann, K, et al.
(författare)
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Lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report
- 2020
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Ingår i: European heart journal. Case reports. - : Oxford University Press (OUP). - 2514-2119. ; 4:1, s. 1-6
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Tidskriftsartikel (refereegranskat)abstract
- BackgroundHomozygous familial hypercholesterolaemia (FH) is an autosomal-dominant inherited disease presenting with highly elevated low-density lipoprotein cholesterol (LDL-C) levels. Untreated, the patient can develop atherosclerosis and cardiovascular disease already in adolescence. Treatment with statins and ezetimibe is usually not sufficient and LDL apheresis is often required. Lomitapide, an inhibitor of the microsomal triglyceride transfer protein, reduces LDL-C and triglyceride levels and can be used alone or in combination with other therapies in homozygous FH. However, experience with this agent is still limited.Case summaryWe present a young female who was diagnosed with homozygous FH at 6 years of age. She shows a complete lack of normal LDL receptor activity and no cholesterol-lowering effect from statins. The patient was treated with LDL apheresis from 7 years of age. When LDL apheresis treatment extended to twice a week, she began to experience adverse effects, including catheter-related complications, infections, and hospital admissions. When lomitapide treatment was initiated, the frequency of apheresis reduced, the LDL-C levels improved and she has not had any further hospital admissions since. Initially, she suffered from gastrointestinal disturbances. However, after 3 years of treatment with lomitapide 20 mg/day, the patient has not experienced any adverse effects.DiscussionIn this female with homozygous FH adding lomitapide treatment to LDL apheresis has contributed to improved LDL-C levels, a reduction in LDL apheresis sessions and enhanced quality of life. No adverse effects have been reported. These findings suggest that lomitapide can be a drug of choice in patients with homozygous FH.
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- Papageorgiou, Joanna-Maria, et al.
(författare)
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Fulminant myocarditis in a COVID-19 positive patient treated with mechanical circulatory support : - a case report
- 2021
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Ingår i: European Heart Journal. Case Reports. - : Oxford University Press. - 2514-2119. ; 5:2
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Tidskriftsartikel (refereegranskat)abstract
- Background Coronavirus disease 2019 (COVID-19) spreading from Wuhan, Hubei province in China, is an expanding global pandemic with significant morbidity and mortality. Even though respiratory failure is the cardinal form of severe COVID-19, concomitant cardiac involvement is common. Myocarditis is a challenging diagnosis due to heterogeneity of clinical presentation, ranging from mild symptoms to fatal arrhythmia and cardiogenic shock (CS). The aetiology is often viral and endomyocardial biopsy (EMB) is the gold standard for definite myocarditis. However, the diagnosis is often made on medical history, clinical presentation, magnetic resonance imaging, and blood tests. Case summary We present a 43-year-old man with mixed connective tissue disease treated with hydroxychloroquine who rapidly developed CS 4 days from symptom onset with fever and cough, showing positive polymerase chain reaction nasopharyngeal swab for severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) RNA. While computed tomography of the thorax was normal, high-sensitivity troponin T was elevated and electrocardiogram showed diffuse ST elevation and low voltage as signs of myocardial oedema. Echocardiography showed severe depression of left ventricular function. The myocardium recovered completely after a week with mechanical circulatory support (MCS). EMB was performed but could neither identify the virus in the cardiomyocytes, nor signs of inflammation. Still the most probable aetiology of CS in this case is myocarditis as a sole symptom of COVID-19. Discussion COVID-19 patients in need of hospitalization present commonly with respiratory manifestations. We present the first case of fulminant myocarditis rapidly progressing to CS in a COVID-19 patient without respiratory failure, successfully treated with inotropes and MCS.
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- Pettersson, Staffan, et al.
(författare)
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An uncommon case of congenital thoracic venous anomaly and extracardiac sinus venosus defect in an asymptomatic adult first presenting with brain abscesses : a case report
- 2022
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Ingår i: European Heart Journal. Case Reports. - : Oxford University Press. - 2514-2119. ; 6:2
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Tidskriftsartikel (refereegranskat)abstract
- Background Congenital thoracic venous anomalies (CTVAs) with right-to-left shunt constitute an uncommon source of paradoxical embolization in adults. We present a case of a healthy and physically fit individual with a rare asymptomatic anomaly first presenting with brain abscesses after a visit to the dental office; persistent left superior vena cavae (PLSVC) without bridging vein, over-riding right-sided superior vena cavae (RSVC) connected to the left atrium (LA), and an extracardiac sinus venosus defect. Case summary A 29-year-old male presented to the neurosurgical unit due to intracranial abscesses requiring intervention following a visit to his dentist. The abscess cultures isolated bacteria commonly found in the normal oral flora. Transthoracic echocardiography revealed an enlarged coronary sinus consistent with PLSVC. An agitated saline study was performed and raised suspicion of simultaneous extra- and intracardiac shunting. Magnetic resonance angiography confirmed the presence of a PLSVC and revealed an RSVC connected to the LA; however, no intracardiac shunt was evident. Electrocardiogram-gated computed tomography was therefore conducted and discovered the rudimentary remains of the physiologic RSVC forming a connection to the right atrium, explaining the bilateral contrast loading seen on the agitated saline study and diagnosing an extracardiac sinus venosus defect (SVD). The patient recovered and has been referred for surgery. Discussion This case illustrates a CTVA and a forme fruste type SVD resulting in a severe complication in a healthy adult. We highlight the diagnostic challenges posed, suggest early usage of agitated saline studies, and discuss the rationale for surgical correction of this patient.
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