Abstract：
OBJECTIVE: To determine the prevalence of hemoptysis secondary to post-embolization systemic collaterals and review the recurrence rate and treatment outcomes.
METHODS: The records of 930 patients with PAVM (801 with known or possible HHT), from a single HHT center between July 2, 1996 and July 22, 2021, were searched for a single lifetime episode of hemoptysis secondary to post-embolization systemic collaterals. Embolization was performed with permanent particles or gelatin slurry. Clinical features and treatment outcomes of identified patients were reviewed.
RESULTS: Twenty-eight embolization procedures have been performed in 9 patients with post-PAVM embolization systemic artery collateral reperfusion. This included 8 patients with known HHT. Permanent particles were used in 5 cases and gelatine slurry was used in 19 cases. Due to the recurrence of hemoptysis, four patients required four embolizations each, two patients required three embolizations and two patients required two embolizations. Chronic unresolving hemoptysis was the presentation in 5 patients and massive hemoptysis requiring ICU admission in 4. The lifetime prevalence and incidence of hemoptysis secondary to systemic artery reperfusion in HHT patients was estimated to be 1.0% and 0.05%, respectively. Bronchial artery origin was most common (8 patients). In the first patient treated at this center, a major adverse event resulting in myocardial infarct and stroke occurred with the use of 300-500-micron permanent particles. This was presumed to be due to left-to-right shunting and subsequent systemic embolization. Subsequent patients were treated with gelatin sponge slurry without adverse events. This patient ultimately expired due to large volume hemoptysis, in the setting of bilateral diffuse PAVMs. A second patient, with history of childhood bronchial artery coil embolization, expired from large volume hemoptysis while awaiting lobectomy. In two cases, patients underwent surgery, including one lobectomy and one pneumonectomy, for recurrent hemoptysis (requiring at least five hospital admissions). The remaining five patients achieved prolonged resolution of hemoptysis with endovascular treatment alone.
CONCLUSIONS: Lifetime prevalence of hemoptysis secondary to PAVM post-embolization systemic collaterals is rare, but recurrence is high. In this limited series, embolization with gelatin sponge slurry appeared safe, although surgical resection may ultimately be required in refractory and multifocal disease.
METHODS: The records of 930 patients with PAVM (801 with known or possible HHT), from a single HHT center between July 2, 1996 and July 22, 2021, were searched for a single lifetime episode of hemoptysis secondary to post-embolization systemic collaterals. Embolization was performed with permanent particles or gelatin slurry. Clinical features and treatment outcomes of identified patients were reviewed.
RESULTS: Twenty-eight embolization procedures have been performed in 9 patients with post-PAVM embolization systemic artery collateral reperfusion. This included 8 patients with known HHT. Permanent particles were used in 5 cases and gelatine slurry was used in 19 cases. Due to the recurrence of hemoptysis, four patients required four embolizations each, two patients required three embolizations and two patients required two embolizations. Chronic unresolving hemoptysis was the presentation in 5 patients and massive hemoptysis requiring ICU admission in 4. The lifetime prevalence and incidence of hemoptysis secondary to systemic artery reperfusion in HHT patients was estimated to be 1.0% and 0.05%, respectively. Bronchial artery origin was most common (8 patients). In the first patient treated at this center, a major adverse event resulting in myocardial infarct and stroke occurred with the use of 300-500-micron permanent particles. This was presumed to be due to left-to-right shunting and subsequent systemic embolization. Subsequent patients were treated with gelatin sponge slurry without adverse events. This patient ultimately expired due to large volume hemoptysis, in the setting of bilateral diffuse PAVMs. A second patient, with history of childhood bronchial artery coil embolization, expired from large volume hemoptysis while awaiting lobectomy. In two cases, patients underwent surgery, including one lobectomy and one pneumonectomy, for recurrent hemoptysis (requiring at least five hospital admissions). The remaining five patients achieved prolonged resolution of hemoptysis with endovascular treatment alone.
CONCLUSIONS: Lifetime prevalence of hemoptysis secondary to PAVM post-embolization systemic collaterals is rare, but recurrence is high. In this limited series, embolization with gelatin sponge slurry appeared safe, although surgical resection may ultimately be required in refractory and multifocal disease.
摘要：
目的：确定栓塞后全身经络继发咯血的患病率，并评估复发率和治疗结果。
方法：930名PAVM患者（801名已知或可能患有HHT）的记录，在1996年7月2日至2021年7月22日期间,我们从一个HHT中心搜索了一次因栓塞后全身侧支继发的终生咯血.用永久性颗粒或明胶浆液进行栓塞。对已确定患者的临床特征和治疗结果进行了回顾。
结果：对9例PAVM栓塞后全身动脉侧支再灌注患者进行了28次栓塞手术。这包括8名已知HHT的患者。5例使用永久性颗粒，19例使用明胶浆液。由于咯血复发,四名患者每人需要四次栓塞,2例患者需要3次栓塞,2例患者需要2次栓塞.5例患者出现慢性无法解决的咯血，4例需要入住ICU的大咯血。HHT患者全身动脉再灌注继发咯血的终生患病率和发生率估计为1.0%和0.05%,分别。支气管动脉起源最常见（8例）。在这个中心治疗的第一个病人，导致心肌梗死和卒中的主要不良事件发生在使用300-500微米永久性颗粒时.据推测，这是由于左向右分流和随后的全身性栓塞所致。随后患者接受明胶海绵浆液治疗，无不良事件发生。该患者最终因大量咯血而死亡，在双侧弥漫性PAVM的情况下。第二例患者，有儿童支气管动脉弹簧圈栓塞史，在等待肺叶切除术期间因大量咯血而过期。在两种情况下，患者接受了手术，包括一次肺叶切除术和一次全肺切除术,用于复发性咯血（需要至少5次住院）。其余5例患者仅通过血管内治疗即可长期缓解咯血。
结论：栓塞后全身络脉继发于PAVM的咯血的终生患病率很少，但复发率很高。在这个有限的系列中，用明胶海绵浆液栓塞似乎是安全的，尽管难治性和多灶性疾病最终可能需要手术切除。
方法：930名PAVM患者（801名已知或可能患有HHT）的记录，在1996年7月2日至2021年7月22日期间,我们从一个HHT中心搜索了一次因栓塞后全身侧支继发的终生咯血.用永久性颗粒或明胶浆液进行栓塞。对已确定患者的临床特征和治疗结果进行了回顾。
结果：对9例PAVM栓塞后全身动脉侧支再灌注患者进行了28次栓塞手术。这包括8名已知HHT的患者。5例使用永久性颗粒，19例使用明胶浆液。由于咯血复发,四名患者每人需要四次栓塞,2例患者需要3次栓塞,2例患者需要2次栓塞.5例患者出现慢性无法解决的咯血，4例需要入住ICU的大咯血。HHT患者全身动脉再灌注继发咯血的终生患病率和发生率估计为1.0%和0.05%,分别。支气管动脉起源最常见（8例）。在这个中心治疗的第一个病人，导致心肌梗死和卒中的主要不良事件发生在使用300-500微米永久性颗粒时.据推测，这是由于左向右分流和随后的全身性栓塞所致。随后患者接受明胶海绵浆液治疗，无不良事件发生。该患者最终因大量咯血而死亡，在双侧弥漫性PAVM的情况下。第二例患者，有儿童支气管动脉弹簧圈栓塞史，在等待肺叶切除术期间因大量咯血而过期。在两种情况下，患者接受了手术，包括一次肺叶切除术和一次全肺切除术,用于复发性咯血（需要至少5次住院）。其余5例患者仅通过血管内治疗即可长期缓解咯血。
结论：栓塞后全身络脉继发于PAVM的咯血的终生患病率很少，但复发率很高。在这个有限的系列中，用明胶海绵浆液栓塞似乎是安全的，尽管难治性和多灶性疾病最终可能需要手术切除。
