Endovascular management of spontaneous inferior phrenic artery pseudoaneurysm presenting as massive hemothorax.

Sir,

Spontaneous massive hemothorax secondary to the inferior phrenic artery pseudoaneurysm (IPAP) is very rare. Few case reports of hemothorax caused by IPAP secondary to pancreatitis, trauma, tuberculosis, or iatrogenic injury have been described in the literature.[12] Delayed hemothorax secondary to IPAP has also been described in the literature.[3] Superselective catheterization and endovascular angioembolization of these pseudoaneurysms is an effective and minimally invasive procedure for the treatment of IPAP.

We report a case of a 38-year-old male who was brought to our hospital emergency department with complaints of massive hemoptysis (around 350 ml) and breathlessness. The patient had a history of blood-stained sputum for 3 days for which he was admitted at an outside hospital and was transferred to our institute after a bout of massive hemoptysis. On admission, his oxygen saturation was 94%; hemoglobin was 9.8 g/dL; pH was 7.33; PaO2 was 120 mmHg; and PaCO2 was 29.0 mmHg. He was hypotensive with mild tachycardia (blood pressure [BP]: 101/61 mmHg and heart rate: 110 bpm). Coagulation profiles (platelets – 1.1 lacs, prothrombin time – 13 s, and international normalized ratio – 1.1) were normal. No history of any trauma, surgery, or medication for any chronic illness was present. Vasculitis workup was also negative.

Chest radiography showed massive right hemothorax with mediastinal shift. A computed tomography (CT) scan done outside showed pseudoaneurysm in the right lower thoracic cavity. We repeated CT pulmonary angiography and aortogram to confirm the parent vessel of a pseudoaneurysm. A pseudoaneurysm measuring 3.2 cm × 3 cm was seen in the right lower hemithorax arising from a branch of dilated and tortuous inferior phrenic artery [Figure 1]. In view of falling BP and oxygen saturation levels, the patient was referred for urgent endovascular angioembolization of the pseudoaneurysm. Using Sim 1 catheter and Terumo J-shaped guidewire, selective cannulation of the right inferior phrenic artery was done at D12-L1 level. Angiography confirmed the presence of pseudoaneurysm from the intrathoracic branch of the inferior phrenic artery [Figure 2]. Superselective catheterization of that branch was done using progreat microcatheter (Terumo Interventional Systems, NJ, USA), and embolization was performed using two 2 mm × 2 mm pushable microcoils. Post-embolization angiogram confirmed complete occlusion of pseudoaneurysm [Figure 2]. The patient's postoperative course was uneventful. He was hemodynamically stable (BP: 122/80 mmHg and heart rate around 78 bpm) and was discharged after 2 days with improving blood parameters (hematocrit 36% and hemoglobin 9.5 g/dL).

Figure 1

Upper row images show hypertrophied inferior phrenic artery from the aorta (white arrow) reaching up to intrathoracic pseudoaneurysm (black arrow). Associated right hemithoracic hemothorax seen (white arrow).Lower row images show intrathoracic pseudoaneurysm (black arrow) with hemothorax (white arrow)

Figure 2

Right side image shows pseudoaneurysm opacifying through hypertrophied right phrenic artery. Left side image shows coil occlusion of the feeding branch with nonopacification of the pseudoaneurysm

DISCUSSION

Spontaneous IPAP is rare. After an extensive literature search, we could not find even a single case report of spontaneous IPAP causing massive hemothorax. Blunt thoracic trauma and pulmonary tuberculosis with nonbronchial systemic circulation are the most common causes described in the literature.[1] Since the inferior phrenic artery arises directly from the aorta or celiac artery, high systemic pressure might lead to massive hemothorax in cases of IPAP. Depending on the site of injury of the inferior phrenic artery, the presentation could be pericardial tamponade,[4] intraperitoneal hemorrhage,[5] subcapsular hematoma,[5] or hemothorax.[1]

Endovascular angioembolization is reliable and effective management as compared to thoracotomy for controlling intrathoracic arterial hemorrhage.[6] Carrillo et al. described high morbidity and high failure rates to find the source of hemorrhage associated with thoracotomy, making subtraction angiography followed by transcatheter superselective embolization more accurate, and reliable method for treatment.[7] However, in cases of associated injuries such as diaphragmatic injury thoracotomy might be effective single-stage management.[1] In our case, the bleeding point was identified on CT angiogram, and endovascular treatment was performed. For the management of hemothorax, chest tube insertion should be done only after the management of pseudoaneurysm to prevent rebleeding.[3] We did ultrasonography-guided pigtail drainage of the hemothorax on the second day after which our patient was discharged with improving hemodynamic parameters.

CONCLUSION

Spontaneous IPAP is rare. Noninvasive modalities such as CT angiography can suggest the diagnosis. Endovascular angioembolization is a minimally invasive procedure and is the treatment of choice for the management of such pseudoaneurysms.

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Conflicts of interest

There are no conflicts of interest.