Acute myocardial infarction and left ventricular thrombus in a young male with thromboangiitis obliterans: a case report and literature review

Abstract

Thromboangiitis obliterans (TAO) is an inflammatory vascular disease that primarily affects the small- and medium-sized arteries of the extremities. Coronary artery involvement in TAO is rarely reported, and we present the first case of TAO involving the coronary arteries with concomitant left ventricular thrombus. In addition, our case report includes a literature review on coronary artery involvement in TAO and draws comparisons with systemic vasculitis with coronary artery involvement. The case report and literature review aims to offer physicians valuable insights for diagnosis and treatment of TAO with coronary arteries involvement, while also advocating for increased research efforts to explore therapeutic strategies for this rare condition, including both pharmacological treatment and interventional therapy.

Introduction

Thromboangiitis obliterans (TAO), also known as Buerger’s disease, is a segmental, non-atherosclerotic inflammatory condition that predominantly affects the small and medium-sized arteries, veins, and nerves of the limbs [1]. Reports of TAO involving the coronary arteries are exceedingly rare, and there is currently no consensus on the optimal revascularization strategies or specific antithrombotic regimens for these patients [2]. Herein, we present a case of a patient with established TAO who developed acute anterior myocardial infarction accompanied by a left ventricular thrombus. This case report also provides a comprehensive review of the literature on coronary involvement in TAO and compares it with systemic vasculitis, aiming to summarize current knowledge in this field. We call upon cardiovascular specialists to focus research efforts on this unique patient population, addressing both diagnostic and therapeutic approaches, including interventional and pharmacological strategies.

Case report

A 28-year-old male came into our emergency room with chief complaints of acute chest pain and sweating. The electrocardiogram showed ST segment elevation in V1-V4 and frequent ventricular bigeminy (Fig. 1). Serological test disclosed a significant increase in myoglobin (196.20 ng/ml) and cardiac troponin T (>10000.0 ng/ml). He was diagnosed with acute anterior myocardial infarction and hospitalized in cardiac care unit. Emergent coronary angiography was performed and demonstrated the total occlusion of middle and distal segments of right coronary artery with the formation of multiple corkscrew-collaterals and reverse perfusion of left anterior descending coronary artery, while no stenosis was observed in left coronary artery (Fig. 2, Supplementary Material). Although there was no identified involvement of the left coronary artery by the coronary angiography, we considered that a transient thrombus could have been there.

Further medical history was taken. This young patient has been smoking one package of cigarettes per day for more than 10 years. He has no family history or other risk factors for coronary atherosclerotic heart disease. No evidences were found for hyperlipemia or diabetes mellitus. He has been diagnosed with TAO 5 years ago in our hospital, with the clinical manifestations of Raynaud’s phenomenon of right hand and dry gangrene of right ring finger (Fig. 3). He has been taking aspirin, Sarpogrelate hydrochloride, Beraprost sodium and cilostazol after discharge. However, he didn’t follow the instruction of smoking cessation. Ulceration of the distal portion of his left foot was found 2 years ago, and it was basically healed after carful nursing. Physical examination showed the absence of right ring finger and ischemia over the distal portion of left foot.

Fig. 1

Electrocardiogram. ST segment elevation in V1-V4 and frequent ventricular bigeminy

Fig. 2

Coronary angiography. Total occlusion of middle and distal segments of right coronary artery with the formation of multiple lateral branches (arrow, left). Angiography image of left coronary artery (right)

The computed tomography angiography further confirmed the severe segmental stenoses of the left popliteal artery, left peroneal artery, left anterior tibial artery and left posterior tibial artery with smooth vessels in non-involved region (Fig. 4). Ultrasonic cardiogram (UCG) revealed an abnormal motion of segmental left ventricular wall and the left ventricular ejection fraction was 41%. On the 27th day of admission, a left ventricular apical thrombus was noted by UCG, cardiovascular magnetic resonance imaging confirmed the myocardial lesion and thrombus (Fig. 5). Embolism-related indicators were tested. The results of anti-nuclear antibody, anti-phospholipid antibody, anti-neutrophil cytoplasmic antibody, anti-SCL-70 antibody, lupus anti-coagulant complex and tumor markers were all negative by laboratory studies. There were no mutations in coagulation factor II, coagulation factor V or JAK2 V617F. Furthermore, protein C and protein S were at the normal level. He received aspirin, warfarin, benazepril hydrochloride, β-blocker and Sarpogrelate hydrochloride and was discharged after symptom remission. Three years after discharge, a telephone follow-up was conducted, during which the patient reported a satisfactory recovery with no recurrence of chest pain or other symptoms. Additionally, significant improvement was observed in the distal limb lesions. Due to the complete resolution of symptoms, the patient expressed a reluctance to undergo repeat coronary angiography.

Fig. 3

The clinical manifestations of the patient’s digits

Fig. 4

Computed tomography angiography of lower limbs. Severe stenoses of left popliteal artery and left anterior tibial artery, and severe segmental stenoses in left peroneal artery and left posterior tibial artery (arrow)

Fig. 5

Thrombus in left ventricular apex. A left ventricular apical thrombus demonstrated by ultrasonic cardiogram (arrow, left) and cardiovascular magnetic resonance (arrow, right)

Discussion

TAO is a segmental non-arteriosclerotic vasculitis disease of unknown origin that typically affects the small and medium-sized vessels of upper and lower extremities [3]. It is uncommon for TAO to involve large vessels, although there have been rare cases reported involving coronary arteries in patients with Buerger’s disease (Table 1) [4,5,6,7,8,9,10,11,12,13,14]. Current published cases show no clear pattern of coronary artery involvement in TAO, with all three major vessels potentially affected and presenting primarily as acute myocardial infarction, particularly STEMI, but also as CTO lesions. It is worth noting that TAO patients may present with acute myocardial infarction and seek care from the cardiology department, which may possibly lead to misdiagnosis [2]. Therefore, it is crucial for cardiologists to be aware of this possibility. In this case report, we first present a unique case of a young TAO patient who developed acute myocardial infarction with left ventricular thrombus and aims to increase awareness and understanding of such presentations.

Table 1 Summary of the main reported cases of Buerger’s disease with coronary involvement

Despite being recognized for over a century, the precise pathological mechanism of TAO remains unclear [15]. Nevertheless, thrombosis and inflammation are widely acknowledged as pivotal components in the disease’s pathological process [1, 16, 17]. Multiple studies have demonstrated the presence of inflammatory thrombi in the affected vessels of patients during the acute phase of TAO [18]. These thrombi are typically occlusive, accompanied by infiltration of inflammatory cells such as neutrophils and multinucleated giant cells [1]. This pattern of thrombosis distinguishes TAO from other systemic vasculitides that involve coronary arteries [19, 20]. The distinguishing features between TAO and systemic vasculitides involving the coronary arteries are summarized in Table 2. Systemic vasculitis is characterized by the presence of inflammatory leukocytes in vessel walls, leading to reactive damage to mural structures. In contrast, TAO is distinguished by intraluminal thrombosis without involvement of the vessel wall. Therefore, a thorough patient history and comprehensive auxiliary examinations are crucial for accurately determining the underlying cause of myocardial infarction.

Thrombus formation in TAO is primarily driven by endothelial dysfunction, chronic inflammation, immune responses, and oxidative stress [21]. Persistent inflammation damages endothelial cells, exposing collagen and tissue factor, which promote platelet adhesion and activation [22]. Elevated pro-inflammatory cytokines, such as interleukins and TNF-α, exacerbate endothelial injury and recruit leukocytes, while neutrophils release extracellular traps (NETs) that stabilize thrombi [23]. Immune complexes and autoantibodies further inhibit natural anticoagulants, leading to a hypercoagulable state. Oxidative stress, often induced by smoking, generates reactive oxygen species that impair endothelial function and activate the coagulation cascade [24]. Genetic predispositions may also increase thrombosis susceptibility [25]. This complex interplay fosters an environment conducive to thrombus development in TAO, suggesting potential therapeutic targets, including anti-inflammatory agents, antioxidants, and anticoagulants, to mitigate thrombogenesis and enhance patient outcomes.

Table 2 Comparison of characteristics between TAO and systemic vasculitis

Indeed, there is a lack of consensus regarding the optimal revascularization strategy for patients with coronary artery involvement in TAO and systemic vasculitis. In the case of STEMI patients, primary PCI is recommended to enhance patient outcomes [26]. On the other hand, it is essential for individuals with different types of coronary artery issues to focus on treating the primary disease because regression of coronary lesions may happen at the same time as alleviating the primary condition. For TAO patients, smoking cessation stands as the foremost treatment strategy, while vasodilators like prostaglandin analogs and phosphodiesterase inhibitors may alleviate symptoms [3]. Similarly, for the majority of systemic vasculitis patients, a treatment regimen involving glucocorticoids and immunosuppressants can ameliorate their condition [27]. Consequently, the coronary artery involvement in many patients may ameliorate with primary disease treatment. Hence, the necessity of immediate intervention therapy during the acute phase remains a topic of debate. Moreover, for patients necessitating delayed revascularization, the decision between PCI and CABG remains uncertain [28, 29]. In this case, after comprehensive discussion among the team, we ultimately opted for a conservative treatment strategy combining smoking cessation with medication, rather than proceeding with PCI or CABG. The favorable outcomes observed during follow-up further underscore the critical importance of smoking cessation as a therapeutic strategy.

Research indicates that patients with systemic vasculitis exhibit a heightened likelihood of stent restenosis post-PCI and graft vessel stenosis or occlusion following CABG [29,30,31]. This increased rate of restenosis is attributed to the inflammatory state of the arteries. Some studies even propose that individuals with coronary artery vasculitis should steer clear of coronary revascularization during active inflammation, as the latter elevates the risk of MACE by approximately tenfold [32]. Wang et al. also posit that if revascularization becomes imperative during active inflammation, CABG surpasses PCI, with their study revealing a notably higher incidence of MACE events in the PCI cohort compared to the CABG cohort among patients with Takayasu’s arteritis [29]. In a study encompassing 806 patients with Takayasu’s arteritis, Huang et al. observed that while cardiac mortality rates were akin in both CABG and PCI groups, the restenosis rate was markedly higher in the PCI cohort than in the CABG cohort [28]. Nonetheless, these conclusions predominantly stem from Takayasu’s arteritis with a heightened likelihood of coronary artery involvement, rather than TAO. However, TAO can affect internal mammary arteries, which may be problematic for patients requiring coronary artery bypass grafting surgery [33].

Given the substantial pathophysiological variances between TAO and Takayasu’s arteritis, these findings may not be universally applicable to TAO. The vascular wall inflammation in TAO is not as pronounced as in Takayasu’s arteritis, suggesting that PCI might present a viable option for these patients.

In the present case report, the patient exhibited both acute myocardial infarction and left ventricular thrombus. While it is challenging to definitively ascertain whether the left ventricular thrombus originated from the TAO-related pathological process or from left ventricular dysfunction post-AMI, patients with TAO have a higher thrombotic burden compared to those with systemic vasculitis. Although the optimal antithrombotic regimen for such patients remains undetermined, it is likely that a more aggressive antithrombotic strategy may be required compared to that for systemic vasculitis.

Conclusion

TAO, an inflammatory vascular disease typically affecting the extremities’ small- and medium-sized arteries, is rarely associated with coronary artery involvement. Our case report presents the first case of TAO with concurrent coronary artery involvement and left ventricular thrombus. We further reviewed the literature on coronary artery involvement in TAO and comparing it with systemic vasculitis affecting the coronary arteries. This case report and literature review aims to provide clinicians with critical insights into diagnosing and treating TAO with coronary artery involvement, while also championing for intensified research into therapeutic strategies for this uncommon condition, encompassing pharmacological and interventional approaches.