Multimodal Approach of Isolated Pulmonary Vasculitis: A Single-Institution Experience

Yanartaş, Mehmed; Karakoç, Ayşe Zehra; Zengin, Ahmet; Taş, Serpil; Yıldızeli, Şehnaz Olgun; Mutlu, B.; Atas, Halil; Alıbaz-Öner, Fatma; İnanç, Nevsun; Direşkeneli, Haner; Bozkurtlar, Emine; Erkılınç, Atakan; Çimşit, Çağatay; Bekiroglu, G Nural; Yıldızeli, Bedrettin

doi:10.1016/j.athoracsur.2021.08.009

Cited by 4 publications

(9 citation statements)

References 26 publications

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“…It is important to note that characteristic manifestations commonly associated with vasculitis, such as multiple organ involvement, aneurysms, ulcers, fever, and specific autoantibodies, were absent, rendering them inadequate to fulfill the diagnostic criteria for vasculitis like polyarteritis and Behcet's disease. The diagnosis of IPV is usually uncertain until confirmed through biopsy, which aligns with findings reported in the majority of published studies on IPV to date [ 2 – 4 ].…”

Section: Discussionsupporting

confidence: 76%

“…Studies on IPV are limited; only a few studies, particularly those that focus on the use of PEA in patients with IPV, have been conducted [ 2 – 4 , 7 ]. However, because the PEA techniques for IPV are similar to those for CTEPH, previous results in patients with CTEPH were helpful as a point of comparison.…”

Section: Discussionmentioning

confidence: 99%

“…The incidence and underlying cause of IPV remain unclear because it often mimics chronic thromboembolic pulmonary hypertension (CTEPH). The true diagnosis is based on surgical specimen biopsy and the exclusion of systemic involvement, rather than preoperative assessments based on clinical manifestations, laboratory tests, and imaging [ 2 ]. This infers a high likelihood of long-term underdiagnosis.…”

Section: Introductionmentioning

confidence: 99%

“…Pulmonary endarterectomy (PEA), a surgical procedure that has undergone revisions over the past several decades, is the cornerstone of curative therapy for CTEPH, an obstructive illness that leads to precapillary pulmonary hypertension (PH) [ 5 ]. Although both IPV and CTEPH are categorized as the cause of group 4 PH with certain similarities in pathophysiological features, microstructures point to important differences [ 2 ]. However, experience with PEA in occlusive IPV is limited to isolated instances in different centers [ 6 ].…”

Section: Introductionmentioning

confidence: 99%

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Pulmonary endarterectomy in patients with occlusive isolated pulmonary vasculitis

Ma,

Song,

et al. 2023

Eur J Med Res

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Background Isolated pulmonary vasculitis (IPV) is a rare, insidious, and localized inflammatory disease affecting the pulmonary arteries, often leading to severe luminal obstruction. The prognosis for patients with occlusive IPV is poor, and there is currently a lack of effective treatments. The objective of this study was to evaluate the performance of pulmonary endarterectomy (PEA) as a treatment for occlusive IPV. Methods This single-center retrospective analysis included patients who received PEA for occlusive IPV between January 2018 and June 2022. Clinical characteristics and hemodynamic parameters were evaluated at baseline and follow-up. Results Among 114 consecutive patients who underwent PEA, occlusive IPV was identified in 7 patients. Two patients underwent bilateral PEA for the involvement of both pulmonary arteries. Patch angioplasty was performed to treat four severe constrictions. One patient died from residual pulmonary hypertension after limited PEA of a transmural vascular lesion. In addition, no obvious surgical complications were observed. Three months after PEA, a substantial relief in symptoms was achieved. Also, there is a decrease in the mean pulmonary artery pressure (median 33 [20–48] mmHg before versus median 21 [16–26] mmHg after; P < 0.018) and pulmonary arterial resistance (median 234 [131–843] dyn.s.cm−5 versus median 180 [150–372] dyn.s.cm−5; P = 0.310). Three patients experienced a relapse of restenosis of the treated arteries within a 6-month follow-up period, despite daily oral prednisolone administration. They were treated with balloon pulmonary angioplasty of both the main pulmonary arteries and branches. Conclusions PEA is a valuable choice for treating occlusive IPV, with notable hemodynamic and clinical advantages. To increase long-term vascular patency, complete management should be optimized.

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Section: Discussionsupporting

confidence: 76%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Pulmonary endarterectomy in patients with occlusive isolated pulmonary vasculitis

Ma,

Song,

et al. 2023

Eur J Med Res

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“…On the contrary, CT imaging revealed partially recanalized pulmonary arteries and there were multiple perfusion defects in her V/Q scan. Although successfully treated cases with primary pulmonary vasculitis were reported by PEA, 6,7 data about TAK patients with Group IV PH is scarce. There are two case reports in the literature; one died shortly after the operation due to heart failure, 8 and the other improved symptomatically and functionally, although this report lacks hemodynamic and V/Q scan evaluation after PEA.…”

mentioning

confidence: 99%

Case report: Pulmonary endarterectomy in Takayasu arteritis patient with group IV pulmonary hypertension: A rare clinical scenario

Unaldi,

Firlatan,

Duzgun

et al. 2024

Int J of Rheum Dis

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an unfavorable prognosis, and patients with PH exhibit a mortality risk seven times higher compared to those without PH. 3 Pulmonary endarterectomy (PEA) is a preferred therapeutic intervention for patients with accessible pulmonary artery lesions in chronic thromboembolic pulmonary hypertension (CTEPH), the predominant etiology within Group IV PH. 5 However, the investigations regarding the efficacy of PEA in the treatment of Group IV PH associated with TAK are notably scarce. In this context, we aim to describe a case of TAK characterized by pulmonary artery involvement and Group IV PH and its management through pulmonary endarterectomy.A 28-year-old female patient was diagnosed with Type V Takayasu arteritis concomitant with Group IV pulmonary hypertension in 2017 [Mean pulmonary artery pressure (PAP): 30 mmHg, Table 1].For induction therapy, the patient received high-dose methylprednisolone (1000 mg/day for 3 days) and cyclophosphamide (500 mg/ week for 3 weeks, 500 mg/10-day for three cycles, and 500 mg/ month for 3 months). Additionally, she was put on tadalafil and vitamin K antagonist therapy. The prednisolone was gradually tapered from an initial 40 mg/day to a maintenance dose of 5 mg/day. Following approximately 6 months of cyclophosphamide therapy, the patient received subcutaneous tocilizumab 162 mg/week. In the course of her echocardiographic assessments, systolic PAP measurements were consistently lower than 40 mmHg. Therefore, we decided to follow up her with serial right heart catheterization (RHC) (Table 1). During follow-up, in 2018, ambrisentan was incorporated into her treatment regimen. In 2019, upon admission for exertional dyspnea, computed tomography angiography (CTA) revealed findings consistent with a TAK flare. Consequently, tocilizumab was discontinued, and infliximab, in conjunction with methotrexate, was initiated. During stable follow-up under treatment, the patient was admitted to our clinic in November 2022 with a 3-month history of worsening dyspnea. At her admission, the patient presented with World Health Organization Functional Capacity (WHO FC) IV, and

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Ex Vivo Comparison of the Elastic Properties of Vascular Substitutes Used for Pulmonary Artery Replacement

Hireche,

Canaud,

Peyron

et al. 2024

Journal of Surgical Research

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Multimodal Approach of Isolated Pulmonary Vasculitis: A Single-Institution Experience

Cited by 4 publications

References 26 publications

Pulmonary endarterectomy in patients with occlusive isolated pulmonary vasculitis

Pulmonary endarterectomy in patients with occlusive isolated pulmonary vasculitis

Case report: Pulmonary endarterectomy in Takayasu arteritis patient with group IV pulmonary hypertension: A rare clinical scenario

Ex Vivo Comparison of the Elastic Properties of Vascular Substitutes Used for Pulmonary Artery Replacement

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