Journal of Cardiology Cases最新文献

Giant cell myocarditis (GCM) is a potentially lethal subtype of myocarditis. Herein, we report a case of a 22-year-old woman with GCM who was successfully treated with prednisolone monotherapy. The patient had a fever and shortness of breath and was referred to our hospital. Laboratory test results revealed elevated troponin I levels. Cardiac magnetic resonance (CMR) showed high intensity in the inferoseptal segment of the left ventricle on T2-weighted short tau inversion recovery imaging without late gadolinium enhancement (LGE), suggesting predominant edema rather than necrosis. The patient was diagnosed with GCM based on an endomyocardial biopsy, which revealed lymphocyte infiltration and multinucleated giant cells in the absence of granuloma formation. Subsequently, the patient received intravenous methylprednisolone at 1000 mg/day for 3 days followed by oral prednisolone at 30 mg/day, which normalized troponin levels. Follow-up CMR revealed improved cardiac inflammation; therefore, the patient was discharged without prescribing another immunosuppressive agent. Prednisolone was tapered and terminated three years after discharge. The patient went one year without medication and had no recurrence of GCM on follow-up. This case highlights the presence of mild GCM, successfully treated by steroid monotherapy, in which the mismatch between high-intensity T2 areas and LGE suggests mild inflammation.

Learning objective

Giant cell myocarditis (GCM) is potentially lethal and usually requires multiple immunosuppressive agents. Here, we report a patient with GCM with preserved left ventricular ejection fraction. Cardiac magnetic resonance revealed focal high T2 signal intensity areas without late gadolinium enhancement, indicating myocardial edema without necrosis. The patient remained in remission with prednisolone monotherapy for 2 years. Our report indicates that “mild” GCM may be treated with prednisolone monotherapy.

A 34-year-old man with a history of Kawasaki disease had been experiencing chest pain at rest since middle school. Multidetector-row computed tomography showed no aneurysm formation; however, the right coronary artery had an anomalous origin with moderate stenosis. Invasive coronary angiography revealed moderate right coronary artery stenosis with a fractional flow reserve of 0.97. Finally, with a positive acetylcholine provocation test and elevated index of microvascular resistance, the patient was diagnosed with microvascular and epicardial vasospastic angina in the endotypes of ischemia with nonobstructive coronary arteries. This is the first reported case of both microvascular and epicardial vasospastic angina after Kawasaki disease. In patients with a history of Kawasaki disease, even those without cardiac sequelae, coronary endothelial and microvascular dysfunctions should be taken into consideration.

Learning objective

We report the first case of both microvascular and epicardial vasospastic angina in the endotypes of ischemia with nonobstructive coronary arteries after Kawasaki disease.

Coronary endothelial and microvascular dysfunctions should be taken into consideration in patients with a history of Kawasaki disease, even those without cardiac sequelae.

Spontaneous coronary artery dissection (SCAD) is a rare cause of acute coronary syndrome (ACS), which typically occurs in women at low risk of atherosclerosis. We herein report a case of SCAD in a 57-year-old man who later developed Takayasu arteritis. The patient presented to our hospital complaining of chest pain and was diagnosed with unstable angina. Emergent coronary angiography was performed, and optical coherence tomography revealed that ACS was caused by SCAD. The patient was treated medically without further ballooning or stenting. Because there was a bilateral difference in blood pressure, the systemic artery was screened by contrast-enhanced computed tomography, which showed left subclavian artery occlusion, proximal stenosis of the superior mesenteric artery, right common iliac artery dissection, and left external iliac artery dissection. Based on these results and ¹⁸F-fluorodeoxyglucose positron emission tomography findings, we diagnosed Takayasu arteritis. Prednisolone and tocilizumab were selected for medical treatment, and the patient was in a good condition at one year after the diagnosis. Takayasu arteritis can cause dissection of various arteries and should be suspected when atypical SCAD or multiple dissections are present. Early initiation of immunosuppressive therapy can control disease activity.

Learning objective

Spontaneous coronary artery dissection (SCAD) is an important cause of acute coronary syndrome. In this case, we experienced a case of SCAD which turned out to be the first symptom of Takayasu arteritis. Immunosuppressive therapy was effective for both coronary lesion and systemic vasculitis. Not only fibromuscular dysplasia, but also various types of vasculitis should therefore be considered in the differential diagnosis when encountering atypical SCAD cases.

Acute limb ischemia (ALI) related to total knee arthroplasty (TKA) is rare. Most occlusions are caused by thrombus formation in the popliteal artery (PA). Currently such cases are revascularized using less invasive approaches such as endovascular therapy or Fogarty thrombectomy. We report a case of ALI in a 65-year-old woman with complete rupture of the PA due to a TKA procedure. She had resting pain and motor paralysis in her right lower extremity after TKA. Contrast-enhanced computed tomography showed occlusion of the right femoropopliteal artery. Subsequently, she was referred to our hospital with a diagnosis of ALI. Initially, a less invasive revascularization procedure was unsuccessfully attempted. Therefore, we performed an emergency distal bypass and succeeded in revascularization. Intraoperative examination revealed a complete rupture of the PA. Postoperatively, the patient exhibited no signs of myonephropathic metabolic syndrome. Although there was significant motor impairment, the affected limbs were successfully salvaged. ALI with complete rupture of the PA associated with TKA has not been reported previously. In cases of iatrogenic ALI after TKA, it would be essential to consider diagnostic and revascularization methods that account for the possibility of severe injury to the PA.

Learning objective

Acute limb ischemia after total knee arthroplasty is a rare and life- and limb-threatening condition. The underlying pathological mechanism is often thrombus occlusion due to mechanical stimuli of the popliteal artery (PA). There are no established treatments for this condition, and less invasive approaches such as endovascular procedures and Fogarty thrombectomy are often used. However, in cases involving severe damage to the PA, bypass surgery may be necessary, and revascularization procedures should be considered accordingly.

We present three cases of hypoplastic left heart syndrome (HLHS) complicated by congenital esophageal atresia and trachea-esophageal fistula (EA/TEF). The standard treatment for HLHS involves a staged surgical approach, eventually reaching Fontan completion. There is no report of patients with both HLHS and EA/TEF, and no established treatment strategy exists for such cases. Given the significant risk of simultaneously operating on HLHS and EA/TEF, we elected to pursue staged repair for each condition separately. Initially, soon after birth, we performed gastrostomy to secure the nutritional pathway for EA/TEF and stabilize breathing. Subsequently, we conducted bilateral pulmonary artery banding (bil-PAB) and ductal stenting for HLHS, as the Norwood operation carried an unacceptably high risk in these patients. Two of these patients were able to transition to home care, while the other patient died during hospitalization due to complications after EA repair. A combination of bil-PAB with ductal stenting for HLHS and staged repair for EA/TEF may provide effective management for patients with both conditions.

Learning objective

Hypoplastic left heart syndrome (HLHS) and congenital esophageal atresia (EA) are both life-threatening conditions that require early intervention after birth. There are few reports of patients with both conditions, and no treatment strategy is established. Although the procedure carries a high risk, we successfully performed ductal stenting with bilateral pulmonary artery banding for HLHS, as well as staged repair procedures for EA. Our approach may be a viable strategy for these conditions.

Inflammatory bowel disease (IBD) is a complex chronic inflammatory intestinal disease. The development of de novo IBD after solid organ transplantation with immunosuppressive agents has been rarely reported. We present the case of a 65-year-old man with repeated colitis after heart transplantation (HTx) who was diagnosed with Crohn's disease (CD). The patient underwent HTx due to non-ischemic dilated cardiomyopathy. Six months after HTx, he developed serious diarrhea and a transient fever, which persisted for about 6 months. Valganciclovir or any antibiotic agents were not effective for his symptoms and longitudinal ulcers in colonoscopy aggravated during the course, so that we made a diagnosis of CD. We started 5-aminosalicylic acid and found improvement in his symptoms and colonoscopic findings. However, 7 months after improvement, CD worsened. We started ustekinumab by which his condition successfully went into remission again. While oral immunosuppressive drugs are thought to suppress autoimmune diseases in general, IBD should be included in the differential diagnoses for recurring enterocolitis after HTx. Poorly controlled CD can lead to serious and potentially fatal complications, but in this case, ustekinumab has been used safely and effectively for the treatment of CD.

Learning objective

Colitis is a common complication after heart transplantation (HTx). Although cytomegalovirus colitis or posttransplant lymphoproliferative disorder are observed commonly, de novo inflammatory bowel disease (IBD) should be considered when serious refractory colitis occurs. Not only 5-aminosalicylic acid but also ustekinumab, which is a monoclonal antibody to the p40 subunit of interleukin (IL)-12 and IL-23, may be a safe and effective treatment for de novo IBD after HTx.

An 89-year-old man with symptomatic severe aortic stenosis underwent transcatheter aortic valve implantation due to old age and a history of coronary artery bypass grafting. Computed tomography showed a tricuspid aortic valve and severe calcification at the aortic valve annulus, with a perimeter of 88.7 mm. The 34-mm Evolut PRO+ (Medtronic Inc., Minneapolis, MN, USA) was selected. After balloon aortic valvuloplasty, deployment of the Evolut PRO+ was attempted, but significant expansion failure was observed. Upon retraction and removal of the Evolut PRO+ from the body, frame deformation was observed. A new Evolut PRO+ was tried again, but a similar finding was noted as a magatama-like infolding on transesophageal echocardiography. Fortunately, the patient's hemodynamics were relatively stable. Post-dilation was performed using a 25 mm Z-MED II (NuMED, Inc., Montreal, Canada) for reshaping.

Learning objective

In self-expanding transcatheter aortic valves (TAVs), bending of the TAV frame is widely known as one of the key problems. However, this is rare and infrequently encountered. In this case, TAV frame infolding occurred repeatedly, and the morphology of the infolding was evaluated in vitro and in vivo. Furthermore, we report that some TAVs can be reshaped by post-dilation.

Non-reentrant fascicular tachycardia (NRFT) developed in a 6-year-old Japanese boy. Because of drug-resistant recurrences, he received catheter mapping and ablation at age 10 years. An electrocardiogram exhibited a superior left-axis deviation, a right bundle branch block-type configuration, and relatively narrow QRS with sharp R wave. It suggested verapamil-sensitive ventricular tachycardia (VT), but showed no sensitivity to verapamil or reentrant characteristics in the electrophysiological study. Detailed VT mapping determined the earliest presystolic Purkinje potential on the left posterior fascicle at the mid-ventricular septum. Radiofrequency current applications to the lesion led to his NRFT-free life without restriction.

Learning objectives

Purkinje-related idiopathic ventricular tachycardias (VTs) are commonly due to reentrant mechanisms, and non-reentrant fascicular tachycardia (NRFT) is a rare form of idiopathic VT in adults. Although it is crucial to distinguish NRFT from reentrant VTs, there is no information about the electrophysiological studies and the treatment effect in pediatric-onset NRFT.

We present a case of a man with ischemic cardiomyopathy and single chamber implantable cardioverter-defibrillator who developed sinus arrest creating sudden dependence on right ventricular (RV) pacing. He presented with cardiogenic shock secondary to abrupt onset ventricular dyssynchrony from RV pacing, which required emergent stabilization and completely resolved with atrial pacing.

Learning objective

To establish a basic understanding of cardiogenic shock management. To reinforce the adverse effects associated with right ventricular pacing.

A man in his 70s with a history of mitral valve replacement (MVR) and long-standing persistent atrial fibrillation (AF) presented with effort angina. Coronary angiography revealed severe stenosis of the left main coronary artery (LMCA). As it was an emergent case, PCI (percutaneous coronary intervention) was selected for treatment. Intravascular ultrasonography revealed no atherosclerotic lesions in the LMCA. The LMCA was effectively dilated by the drug-eluting stent. No elevation in intracardiac pressure was observed in cardiac catheterization after PCI. Computed tomography scan indicated potential compression of the LMCA by the surrounding structures. In cases of long-standing persistent AF and an enlarged atrium after MVR, the possibility of LMCA stenosis due to anatomical changes should be considered.

Learning Objectives

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  Peri-valvular regurgitation and long-standing persistent atrial fibrillation can potentially cause atrial enlargement.

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  Coronary artery stenosis without atherosclerosis can occur due to compression from surrounding structures or shifting of the coronary artery.

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  Stent therapy provides a temporary solution and coronary artery bypass grafting or switching should be considered if re-stenosis occurs.