Stentless left main revascularisation for takayasu arteritis using directional coronary atherectomy and a drug-coated balloon: a case report

Abstract

Background

Coronary involvement in Takayasu arteritis is characterized by chronic fibrotic thickening of the arterial intima and is associated with a high risk of restenosis after coronary intervention. In young women, coronary artery bypass grafting and coronary stenting are often undesirable, owing to long-term procedural implications and considerations regarding future pregnancy.

Case summary

A 21-year-old woman presented with severe left main coronary artery stenosis caused by Takayasu arteritis. After inflammation had been adequately controlled with glucocorticoid therapy and a monoclonal antibody directed against the interleukin-6 receptor, stentless revascularization was undertaken. Directional removal of fibrotic tissue was followed by inflation of a drug-coated coronary balloon. This approach led to a complete relief of the patient’s symptoms, and imaging over 5 years demonstrated sustained patency of the left main coronary artery without restenosis. The patient gave birth to two healthy children without experiencing cardiovascular complications.

Discussion

This case illustrates the potential value of a stentless strategy that integrates systemic control of vascular inflammation with targeted removal of fibrotic coronary tissue. The favourable long-term outcome emphasizes the importance of deferring coronary intervention until inflammatory activity has been adequately suppressed. It further suggests that a personalized revascularization approach without stent implantation may be a viable alternative in patients for whom conventional bypass surgery or coronary stenting is undesirable.

Learning points.

• We report a case of severe left main coronary artery (LMCA) stenosis secondary to Takayasu arteritis.

• We show the potential of stentless percutaneous coronary intervention after adequate immunosuppressive control as a promising treatment option.

• At the 5-year follow-up, the LMCA remained patent; the patient successfully delivered two healthy children, indicating an excellent long-term outcome.

Introduction

Takayasu arteritis (TA) is a chronic, idiopathic large-vessel vasculitis affecting the aorta and its major branches, predominantly in young women.¹ Immunosuppressive therapy is the mainstay of treatment, with glucocorticoids (GCs) as first-line agents.² In GC-refractory disease, tocilizumab (TCZ), an anti-interleukin-6 receptor monoclonal antibody, has proven efficacious.^3,4

Up to 60% of patients with TA have coronary artery involvement, although only 5%–20% exhibit clinical symptoms.^5,6 Such lesions are typically located in the ostial or proximal segments and are characterized by fibrotic intimal thickening secondary to chronic inflammation,⁷ distinguishing them from conventional atherosclerotic lesions. Strategies for coronary involvement in TA include percutaneous coronary intervention (PCI) and coronary artery bypass grafting (CABG); however, the optimal revascularization strategy remains controversial. Furthermore, as the majority of patients are young women, careful consideration is required when invasive procedures are selected.

Here, we report a case of severe left main coronary artery (LMCA) stenosis secondary to TA. After adequate control of vascular inflammation was achieved with GCs and TCZ, the patient underwent stentless PCI comprising directional coronary atherectomy (DCA) and drug-coated balloon (DCB) angioplasty, with favourable long-term outcomes.

Summary figure

The timeline for the summary figure is provided as a separate figure.

Time Point	Event
Day 0	Syncope and exertional chest pain led to hospital admission
Day 2	Left main coronary artery (LMCA) stenosis identified upon coronary computed tomography (CT) angiography and coronary angiography (CAG)
Day 5	Takayasu arteritis was diagnosed based on vascular imaging and positron emission tomography, which showed active arterial inflammation
Day 6	Immunosuppressive therapy initiated with prednisolone and tocilizumab
Day 40	Inflammatory markers had decreased (erythrocyte sedimentation rate: 23 → 8 mm/h)
Day 65	Persistent severe LMCA stenosis upon repeat CAG
Day 75	Stentless PCI performed: directional coronary atherectomy followed by paclitaxel-coated balloon angioplasty
Post-procedure	Symptoms resolved and exercise tolerance improved
5 years later	No restenosis confirmed on follow-up coronary CT angiography; the patient had two healthy deliveries

Case presentation

A 21-year-old woman presented with syncope and a 1-month history of exertional chest pain. Exercise stress electrocardiography revealed ST-segment depression, and coronary computed tomography angiography (CCTA) demonstrated severe stenosis of the LMCA, prompting hospitalization. Coronary angiography (CAG) confirmed 90% stenosis of the LMCA, with well-developed collateral circulation from the right coronary artery (Figure 1A and 1B). Her left radial artery pulse was weak, and contrast-enhanced CT revealed additional stenosis of the left subclavian artery (Figure 1C). After exclusion of alternative diagnoses, the patient was diagnosed with TA based on the clinical and imaging findings. Positron emission tomography (PET) revealed arterial wall inflammation (Figure 1D).

Figure 1.

Three-dimensional computed tomography angiography, positron emission tomography, and coronary angiography in this case; (A) 99% stenosis of the ostial lesion in the left main coronary artery. (B) Rentrop grade 3 collaterals to the left coronary artery. (C) Severe stenosis of the left subclavian artery (white arrow) upon three-dimensional computed tomography angiography. (D) Positron emission tomography identified active inflammatory uptake in the aorta and its branches (red arrow).

Immunosuppressive therapy was initiated with oral prednisolone (50 mg/day, tapered by 10 mg every 2–3 weeks) and subcutaneous TCZ (162 mg, once weekly). Inflammatory activity was monitored mainly via the erythrocyte sedimentation rate (ESR) because the patient’s C-reactive protein levels remained normal throughout hospitalization. By day 40, the prednisolone had been tapered to 20 mg, and her ESR had decreased from 23 to 8 mm/h (Figure 2). As no clinical flare was observed and her inflammation was considered adequately controlled, PET was not repeated.

Figure 2.

Treatment regimens and temporal changes in erythrocyte sedimentation rate. The patient received prednisolone (50 mg/day) and tocilizumab (162 mg/week). Prednisolone was tapered by 10 mg every 2–3 weeks. After 40 days, her erythrocyte sedimentation rate had decreased from 23 to 8 mm/h. PSL, prednisolone.

Nevertheless, repeat CAG on day 65 revealed that the LMCA stenosis persisted. Cardiopulmonary exercise testing revealed a reduction in the patient’s exercise tolerance of three metabolic equivalents, substantially affecting her daily activities. Her SYNTAX score I was 14, and her SYNTAX score II recommended CABG (estimated 4-year mortality: PCI, 2.0% vs. CABG, 0.3%). However, as she was young and the lesion had an inflammatory nature, we hesitated to perform CABG. Pre-procedural intravascular ultrasound (IVUS) showed no prominent calcification; therefore, DCA followed by DCB angioplasty was selected.

On day 75, DCA was performed with the Atherocut device (Nipro Corporation, Osaka, Japan) for the LMCA lesion (Figure 3A and 3B). IVUS via a 60 MHz catheter (NIRS Dualpro; Nipro Corporation) demonstrated fibrotic intimal thickening without plaque attenuation. The three-layer structure of the arterial wall was indistinct (Figure 3C–E). Subsequently, angioplasty was performed via scoring (NSE Alpha; Nipro Corporation) and paclitaxel-coated (SeQuent Please; B. Braun Melsungen AG, Melsungen, Germany) balloons (Figure 3F). The DCA had debulked the lesion, reducing the stenosis (Figure 3G). The patient’s haemodynamic status remained stable throughout. Histopathological examination of specimens obtained via DCA revealed disruption and multilayering of the internal elastic lamina but no lipid core formation, atheromatous changes, or active inflammatory cell infiltration (Figure 4).

Figure 3.

Coronary angiography and IVUS findings before and after stentless PCI via DCA and DCB; (A) 99% stenosis of the left main coronary artery (LMCA) before percutaneous coronary intervention (PCI). (B) PCI was performed via the right femoral artery by using an 8-Fr guiding catheter (Roadmaster TH 8Fr CL3.5; Nipro Corporation, Osaka, Japan) and a Sion blue guidewire (ASAHI Intecc, Seto, Japan). Directional coronary atherectomy (DCA) was performed using the Atherocut catheter. (C) Intravascular ultrasound (IVUS) before PCI demonstrated fibrotic intimal thickening without attenuated plaque. The three-layered vessel wall structure (intima, media, and adventitia) was indistinct. (*) (D) IVUS after DCA showed expansion of the minimum lumen area (MLA) to 2.87 mm². Because the tissue had been resected close to the medial layer (arrowhead), further debulking was considered difficult. (E) IVUS following scoring balloon and paclitaxel-coated balloon angioplasty demonstrated the presence of a crack and further expansion of the MLA to 6.97 mm². (F) Angioplasty was subsequently performed with a scoring balloon (NSE Alpha 3.0 × 13 mm) and a paclitaxel-coated balloon (SeQuent Please 3.5 × 15 mm). (G) Postprocedural angiography showed a satisfactory result.

Figure 4.

Histopathological findings of the left main coronary artery lesion. Elastica van Gieson-stained sections demonstrated the intima, internal elastic lamina, and medial smooth muscle layer, with no granulomatous inflammation or lymphocytic infiltration. The internal elastic lamina exhibited multilayering and fragmentation.

Post-interventional cardiopulmonary exercise testing revealed that the patient’s exercise capacity exceeded the age-predicted reference. Her chest pain had resolved, and she was discharged home in stable condition, subsequently returning to normal daily and social activities.

Dual antiplatelet therapy with aspirin (100 mg) and prasugrel (3.75 mg) was continued for 6 months and de-escalated to aspirin alone after CCTA confirmed no restenosis. The patient became pregnant, and the aspirin was stopped at 8 months’ gestation; she delivered two healthy children. The 5-year follow-up CCTA revealed no LMCA restenosis (Figure 5).

Figure 5.

Coronary computed tomography angiography at the 5-year follow-up. No restenosis was observed in the left main coronary artery (arrow).

Discussion

Although coronary involvement is less common in TA than aortic involvement, it can result in life-threatening ischaemia and therefore warrants meticulous clinical management. Severe LMCA stenosis, as seen in this case, presents a considerable therapeutic challenge. The pathogenesis of coronary artery stenosis in TA differs markedly from that of atherosclerosis and is typically characterized by fibrotic intimal thickening attributable to chronic inflammation. In this case, IVUS revealed hallmark features of TA, including loss of the normal three-layered arterial structure, the absence of lipid-rich plaques, and marked luminal narrowing due to fibrotic proliferation.⁸ DCA-based debulking followed by DCB angioplasty was selected to maximize acute luminal gain. Histopathological examination showed disruption and multilayering of the internal elastic lamina. Notably, the specimens obtained via DCA lacked inflammatory cell infiltration, which is consistent with effective pre-procedural inflammatory control achieved with GCs and TCZ.

While stenting remains the standard of care for atherosclerotic lesions, it carries a high risk of in-stent restenosis in inflammatory vasculopathies such as TA.^9,10 Moreover, in women of childbearing potential, stent implantation should be avoided if possible. Although CABG may be recommended for complex lesions,¹¹ patients with TA typically require ongoing immunosuppression, which complicates perioperative management and increases the risk of infection and disease flare. Indeed, Arita et al. reported on TA flares triggered by perioperative GC withdrawal.¹² In our case, concomitant left subclavian artery stenosis precluded the use of the left internal thoracic artery as a conduit, as per the 2014 European Society of Cardiology guidelines,¹³ further limiting the long-term utility of CABG. In this case, stentless DCA followed by DCB angioplasty yielded 5-year patency and favourable pregnancy outcomes.

Although the combination of DCA and DCB seems a promising strategy—particularly for LMCA lesions—it requires high technical expertise and meticulous intravascular imaging guidance. In this case, the procedure was performed by an interventional cardiologist with extensive experience in DCA. Mechanical circulatory support was readily available in case of haemodynamic deterioration. Liang et al. conducted a systematic review of PCI for TA-related coronary disease, noting that the evidence supporting the effectiveness of DCB is limited owing to the small number of reported cases,¹⁴ and no consensus has been reached on the degree of inflammatory control required before intervention or on the optimal biomarkers for its assessment. The favourable outcome in this case likely resulted from the two-tier inflammation-control strategy: (i) sustained suppression of chronic vasculitis with GCs plus TCZ, and (ii) paclitaxel-coated balloon angioplasty after DCA-based debulking performed to reduce acute focal inflammation and inhibit smooth-muscle proliferation at the lesion site. These observations underscore the importance of integrating systemic inflammation control with lesion-tailored revascularization in the management of TA-related coronary disease. Our patient had clinically stable TA managed via immunosuppression, and the generalizability of her outcomes to patients with active or refractory disease remains uncertain.

In conclusion, this report highlights the potential of stentless PCI after adequate immunosuppressive control as a promising treatment option for coronary involvement in TA. Further prospective studies are needed to validate the long-term safety and efficacy of this strategy, define the optimal timing of intervention, and standardize procedural aspects of DCA and DCB use in TA-related coronary lesions.

Lead author biography

Yota Koyabu,MD,PhD is a cardiologist and Lecturer in the Department of Cardiovascular Medicine at Dokkyo Medical University Hospital, Tochigi, Japan. His clinical expertise is in cardiac catheter-based interventions. He is actively involved in patient care as a member of the hospital’s Heart Center.

Author contributions

Yota Koyabu (Conceptualization, Data curation, Investigation, Visualization, Writing—original draft, Writing—review & editing [lead]), Suguru Hirose (Conceptualization, Project administration, Visualization, Writing—review & editing [lead], Data curation, Writing—original draft [supporting]), Mayo Wada (Data curation [supporting]), Masashi Sakuma (Supervision [lead]), and Shigeru Toyoda (Supervision [supporting])

Consent: The authors confirm that written consent for submission and publication of this case report, including images and associated text, has been obtained from the patient in line with COPE guidance.

Funding

None.

Data availability

The data underlying this article are available within the article.