Introduction:
Major hemoptysisis defined as one of the following: bleeding of more than or equal to 200 ml per 24 hours; bleeding resulting in hemodynamic instability; or respiratory compromise or blood loss resulting in a reduction in hematocrit to less than 30%
(1). Massive hemoptysis constitutes 1-1.5% of all hemoptysis. They can be life-threatening due to compromised gas exchange or sudden circulatory collapse, with 50-100% mortality (2). With the advanced CT scanner, the cause for hemoptysis could often be easily diagnosed for prompt treatment.
Bronchial artery embolization is a safer nonsurgical option along with medical management and can also be considered a safer option in patients who are not fit for pneumonectomy, or as a bridge to definitive surgical management(3). The failure to recognize the presence of hypertrophied non-bronchial systemic arteries results in immediate recurrent life-threatening hemoptysis and therefore, it is imperative to identify the presence of these vessels in the angiogram and the embolization of the same (4). The purpose of this article is to describe a case of massive hemoptysis due to hypertrophied non—bronchial/bronchial arteries with embolization of the same and the ways to predict these non-bronchial branches for an effective embolization.
Case Report:
A 60-year-old woman a known case of tuberculosis involving the left lung had developed a left main bronchus- esophageal fistula 15 years ago. It was diagnosed with a clinical history of recurrent cough along with other relevant laboratory investigations. She then underwent left main bronchus stenting. During the subsequent follow-up clinical admissions, she developed multiple episodes of pseudomonal infections involving the left upper lobe and eventually underwent a left upper lobectomy in 2017.
She had multiple episodes of mild hemoptysis, which were transient, and recovered without major hospital admissions. She had a massive hemotpysis of more than 200 ml in 12 hours and was admitted to the ER of our hospital. After initial resuscitation by the ER team, clinical examination and blood investigations revealed no definite hemodynamic compromise, normal hemoglobin and coagulation parameters.
A CT angiogram of the thorax showed the presence of hypertrophied bronchial and non-bronchial systemic arteries with a flow-related saccular aneurysm from the left subclavian artery (figure 1). Given multiple hypertrophied vessels, massive hemoptysis and higher mortality associated with massive hemoptysis, the patient was immediately taken up for an embolization procedure.
Embolization was done extensively involving the left internal mammary artery, the lateral thoracic branch of the left axillary artery, 2 left bronchial arteries, and 2 intercostal arteries. Embolization was achieved using polyvinyl alcohol particles of about 300-500 microns, 500-700 microns (Contour device, Boston Scientific, Massachusetts, USA), pushable platinum coils (5×7, 4×4 and 3×3 Nester coils, Cook medical system, Bloomington, USA) and Spoingostan standard gelfoam particle (1x1x1 mm, Johnson & Johnson medical limited).
Post the embolization procedure, the patient experienced mild chest pain for 2 days which subsequently reduced. Hemoptysis stopped and there was no further recurrence during the hospital stay. She had a few low-grade fever spikes which were managed with antipyretics and antibiotics.
Discussion:
The sources of massive hemoptysisare commonly the bronchial arteries (more than 90%), 5% through the aorta or systemic arterial branches and the remaining 5% due to the pulmonary circulation (5). In the scenario of acute or chronic lung diseases, pulmonary arterioles remain occluded due to hypoxic vasoconstriction, thrombosis, and vasculitis. So bronchial arteries tend to dilate and enlarge to replace pulmonary circulation. The enlarged bronchial arteries tend to get eroded by a bacterial agent or due to elevated blood pressure (6).
A diagnosis can be achieved by conventional radiograph, flexible bronchoscopy (FOB) and/or computed tomography of the chest. Radiographs help in the diagnosis of pneumonia, tuberculous infection, cancer or lung abscess which are the usual suspects of massive hemoptysis. But the sensitivity is too low and Hirschberg et.al. found radiographs only to be diagnostic in 50% of cases (7).
FOB is diagnostic and therapeutic in cases of a central bronchial lesion. If chest radiographs are normal or non-localizing, then the localizing potential of FOB is also limited and ranges from 0 to 31% (7). Also, the excess blood in bronchi can limit visualization through FOB.
CT angiogram of the thorax is a proven diagnostic modality in arriving at a specific diagnosis, and localizing the site of bleeding. CT and FOB are complementary tools and the combined use produces the best results in the evaluation of hemoptysis. CT also helps in identifying the bronchial and non-bronchial systemic feeders, thus help in achieving targeted embolization. The imaging finding of pleural thickening of more than 3 mm is very suggestive of the presence of non-bronchial systemic feeders and these feeders should specifically be targeted for embolization (8).
Angiography is done in the catheterization laboratory after a femoral puncture. After the advent of advanced CT scanners, the role of angiography for diagnosis is obsolete and it is predominantly done for embolization. A descending thoracic aortogram is done using a 5F pigtail to identify the number of bronchial arteries, feeders and abnormal findings. Selective catheterization of the vessels involved will help in characterizing the abnormal findings. The abnormal findings in angiogram include a) hypertrophied and tortuous bronchial arteries, b) focal hypervascularity, c)contrast extravasation into lung parenchymla/cavity, d)shunting into the pulmonary artery or vein, and e) bronchial artery aneurysms. The angiograms should include subclavian, internal mammary, inferior phrenic and intercostal arteries (as these arteries tend to supply these areas through the pleura or chest wall). Lack of selective cannulation and embolization of these non-bronchial systemic feeders can result in an immediate recurrence after successful bronchial artery embolization.
Embolization can be performed using various embolic agents which include gelfoams, polyvinyl alcohol particles, coils and glue (n-butyl cyanacrylate- NBCA). Gelfoam, being a relatively inexpensive temporary embolic agent, can result in recanalization of the embolized vessels and cause recurrence. PVA particles had good mid-term results with a reduced risk of recurrence (9).
The complications of bronchial artery embolization include recurrence and it is reported to be between 9.8% to as high as 57% (10). In an Indian study, the recurrence rate was reported at 18.2% over a 6-month follow-up period (10). The failure to embolize the hypertrophied non-bronchial systemic arteries is the common cause for recurrence. The common adverse effect is chest pain. Our patient experienced mild chest pain which subsided after 2 days. Post embolization syndrome (fever, leukocytosis, and pain) can be seen in a few patients and it will generally subside with antibiotics. Dysphagia can occur if esophageal vessels are embolized in addition to bronchial vessels. The most dreaded complication is spinal cord ischemia due to inadvertent embolization of radiculomedullary vessels which can arise from the bronchial or intercostal vessels. In a case series, this complication was reported in 0.6-4.4% of cases (9). The presence of radicular branches is not a contraindication for embolization, but the presence of artery of Adamkewich is an absolute contraindication for embolization. Other less common complications include transient ischemic attack, stroke and blindness due to embolizing material crossing the bronchopulmonary anastomosis. In an invitro study, the size of bronchopulmonary anastomosis was found to be 325 nm and hence PVA particles should be at least 350 nm for the bronchial artery embolization (8).
Conclusion:
Thus, bronchial artery embolization is a safe and effective minimally invasive strategy for massive hemoptysis. The search for non-bronchial systemic feeders like subclavian, axillary and intercostal, inferior phrenic vessels are imperative to control hemoptysis and prevent a recurrence. The pleural thickening of more than 3 mm in CT is an important predictor of the presence of hypertrophied non-bronchial systemic feeders.
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