Childs Nerv Syst DOI 10.1007/s00381-015-2732-4
CASE REPORT
Embolisation of pulmonary vasculature during endovascular therapy—a case report Shweta S. Naik 1 & V. Sudhir 1 & H. R. Arvinda 2 & M. Radhakrishnan 1 & G. S. Umamaheswara Rao 1
Received: 23 January 2015 / Accepted: 20 April 2015 # Springer-Verlag Berlin Heidelberg 2015
Abstract Systemic complications following liquid glue embolisation of intracranial pial arteriovenous fistulae (AVF) are uncommonly reported. We report a patient who had a pulmonary embolism of a liquid glue during endovascular treatment of a pial AVF. The patient had haemodynamic instability, pulmonary hypertension, increased alveolar dead space and increased brain natriuretic peptide levels. In addition to other supportive measures, her pulmonary hypertension was controlled with sildenafil. Ten months after the event, the patient had a considerable improvement of the clinical and laboratory variables and a significant radiographic resolution of the glue from the pulmonary circulation.
Keywords Pulmonary embolism . Arteriovenous fistula . Glue . Endovascular . Sildenafil
* G. S. Umamaheswara Rao [email protected] Shweta S. Naik [email protected] V. Sudhir [email protected] H. R. Arvinda [email protected] M. Radhakrishnan [email protected] 1
2
Department of Neuroanaesthesia, National Institute of Mental Health and NeuroSciences, Hosur Road, Bengaluru, Karnataka PIN 560029, India Department of Neuroimaging and Interventional Radiology, National Institute of Mental Health and NeuroSciences, Hosur Road, Bengaluru, Karnataka PIN 560029, India
Introduction Intracranial pial arteriovenous fistulae (AVF) are high-flow vascular lesions with abnormal communications between the arterial and venous system. Pial AVFs account for 1.6 % of all intracranial vascular malformations [1]. Therapeutic embolisation is one of the strategies for the treatment of intracranial pial AVF, apart from microneurosurgery. In the majority of patients, embolisation could lead to a total and permanent cure and in some patients it will be an adjunctive therapy to microneurosurgery. Many embolic agents have been used to treat brain AVFs. These include various types of microspheres, silk thread, polyvinyl alcohol, avitene and the liquid cyanoacrylates including isobutyl 2-cyanoacrylate (IBCA) and n-butyl 2-cyanoacrylate (NBCA). Most of the complications of these treatments relate to the central nervous system. Complications involving other organ systems are uncommonly observed and rarely reported. Pulmonary complications have been reported after embolisation of cerebral vascular lesions in adults. However, reports of non-thrombotic pulmonary embolism (PE) following endovascular therapy of cerebral pial AVF in children and the management of this complication have been limited in number. We report the critical care management and post-discharge follow-up of a patient who had pulmonary embolism of a liquid glue during endovascular treatment of a cerebral pial AVF. Informed written consent and assent were obtained from the patient’s mother and the patient, respectively, for the publication of this case report.
Case description A 9-year-old girl presented to the neurosurgery clinic with a 2year history of headache and swelling of the left cheek. The swelling was insidious in onset, gradually increasing in size
Childs Nerv Syst
with time and was associated with pain on movement of the jaw. Her clinical examination revealed a swelling over the left maxillary region which was soft in consistency, had a smooth surface and was associated with pain on palpation. Other organ functions were normal. Her 4-vessel cerebral digital subtraction angiography (DSA) revealed a high flow pial AVF supplied by the middle cerebral artery and draining into the basal vein of Rosenthal (Fig. 1). Endovascular embolisation of the AVF under general anaesthesia was planned. Since deployment of Guglielmi detachable coils (GDC) into the fistulous sac was considered to entail the risk of distal migration into the vasculature, a decision was made to inject the glue into the fistulous sac with a proximal balloon inflation to achieve flow arrest (Fig. 2). About 14 ml of 17 % NBCA was injected into the sac of the fistulae which resulted in complete occlusion of the sac with a good filling of the right middle cerebral artery. A few minutes later, the patient had tachycardia (164 beats/min), hypertension (160/100 mmHg) and ventricular ectopics. It was observed that the balloon used to arrest the flow had ruptured and resulted in embolisation of the glue distally into pulmonary circulation causing sudden haemodynamic instability. Pulmonary angiography revealed dense radio-opaque material occluding the pulmonary vasculature bilaterally. The patient was shifted to the intensive care unit (ICU) for further management of this non-thrombotic pulmonary embolism. In the ICU, the patient had sinus tachycardia with a heart rate of 140–150 beats/min and a respiratory rate of 30– 40 breaths/min. Her end-tidal carbon dioxide pressure (PetCO2) was 18 mmHg with a corresponding PaCO2 of 46.5 mmHg indicating an increase in the alveolar dead space. Her PaO2 was 60.3 mmHg at an FiO2 of 50 %. On neurological examination, the patient had a Glasgow coma scale of E3 VT M5, no motor deficits and symmetrical pupils that were reacting to light. Mechanical ventilation was continued under intravenous sedation. Additionally, she was treated with anticoagulants, steroids and antibiotics. Anticoagulation was achieved with
unfractionated heparin (15 U/kg/h) administered intravenously for 48 h, which was later changed to subcutaneous enoxaparin (40 mg/24 h) and finally to oral warfarin (2.5 mg/24 h). A transthoracic echocardiographic examination at 24 h following the pulmonary embolism revealed a mild tricuspid regurgitation and pulmonary artery hypertension with a pulmonary artery systolic pressure (PASP) of 51 mmHg. Sildenafil citrate in a dose of 10 mg was administered three times daily to treat this pulmonary hypertension. The patient’s haemodynamic instability began to improve on the second day. The heart rate improved to about 110– 120 beats/min while the blood pressure remained consistently around 110/70 mmHg. An improvement in the PetCO2 was also observed from the initial 18 to 24–28 mmHg with a corresponding PaCO2 of 40 mmHg over the next 2–3 days. A 2D echocardiogram, repeated at 72 h, after the event demonstrated an improvement in pulmonary hypertension with a PASP of 38 mmHg. On the fourth day, the patient developed polyuria with a urine output of 3 ml/kg/h. Serum and urine electrolytes and osmolality were, however, within normal limits. Her serum brain natriuretic peptide (BNP) was 155 pg/ml (normal,
CASE REPORT
Embolisation of pulmonary vasculature during endovascular therapy—a case report Shweta S. Naik 1 & V. Sudhir 1 & H. R. Arvinda 2 & M. Radhakrishnan 1 & G. S. Umamaheswara Rao 1
Received: 23 January 2015 / Accepted: 20 April 2015 # Springer-Verlag Berlin Heidelberg 2015
Abstract Systemic complications following liquid glue embolisation of intracranial pial arteriovenous fistulae (AVF) are uncommonly reported. We report a patient who had a pulmonary embolism of a liquid glue during endovascular treatment of a pial AVF. The patient had haemodynamic instability, pulmonary hypertension, increased alveolar dead space and increased brain natriuretic peptide levels. In addition to other supportive measures, her pulmonary hypertension was controlled with sildenafil. Ten months after the event, the patient had a considerable improvement of the clinical and laboratory variables and a significant radiographic resolution of the glue from the pulmonary circulation.
Keywords Pulmonary embolism . Arteriovenous fistula . Glue . Endovascular . Sildenafil
* G. S. Umamaheswara Rao [email protected] Shweta S. Naik [email protected] V. Sudhir [email protected] H. R. Arvinda [email protected] M. Radhakrishnan [email protected] 1
2
Department of Neuroanaesthesia, National Institute of Mental Health and NeuroSciences, Hosur Road, Bengaluru, Karnataka PIN 560029, India Department of Neuroimaging and Interventional Radiology, National Institute of Mental Health and NeuroSciences, Hosur Road, Bengaluru, Karnataka PIN 560029, India
Introduction Intracranial pial arteriovenous fistulae (AVF) are high-flow vascular lesions with abnormal communications between the arterial and venous system. Pial AVFs account for 1.6 % of all intracranial vascular malformations [1]. Therapeutic embolisation is one of the strategies for the treatment of intracranial pial AVF, apart from microneurosurgery. In the majority of patients, embolisation could lead to a total and permanent cure and in some patients it will be an adjunctive therapy to microneurosurgery. Many embolic agents have been used to treat brain AVFs. These include various types of microspheres, silk thread, polyvinyl alcohol, avitene and the liquid cyanoacrylates including isobutyl 2-cyanoacrylate (IBCA) and n-butyl 2-cyanoacrylate (NBCA). Most of the complications of these treatments relate to the central nervous system. Complications involving other organ systems are uncommonly observed and rarely reported. Pulmonary complications have been reported after embolisation of cerebral vascular lesions in adults. However, reports of non-thrombotic pulmonary embolism (PE) following endovascular therapy of cerebral pial AVF in children and the management of this complication have been limited in number. We report the critical care management and post-discharge follow-up of a patient who had pulmonary embolism of a liquid glue during endovascular treatment of a cerebral pial AVF. Informed written consent and assent were obtained from the patient’s mother and the patient, respectively, for the publication of this case report.
Case description A 9-year-old girl presented to the neurosurgery clinic with a 2year history of headache and swelling of the left cheek. The swelling was insidious in onset, gradually increasing in size
Childs Nerv Syst
with time and was associated with pain on movement of the jaw. Her clinical examination revealed a swelling over the left maxillary region which was soft in consistency, had a smooth surface and was associated with pain on palpation. Other organ functions were normal. Her 4-vessel cerebral digital subtraction angiography (DSA) revealed a high flow pial AVF supplied by the middle cerebral artery and draining into the basal vein of Rosenthal (Fig. 1). Endovascular embolisation of the AVF under general anaesthesia was planned. Since deployment of Guglielmi detachable coils (GDC) into the fistulous sac was considered to entail the risk of distal migration into the vasculature, a decision was made to inject the glue into the fistulous sac with a proximal balloon inflation to achieve flow arrest (Fig. 2). About 14 ml of 17 % NBCA was injected into the sac of the fistulae which resulted in complete occlusion of the sac with a good filling of the right middle cerebral artery. A few minutes later, the patient had tachycardia (164 beats/min), hypertension (160/100 mmHg) and ventricular ectopics. It was observed that the balloon used to arrest the flow had ruptured and resulted in embolisation of the glue distally into pulmonary circulation causing sudden haemodynamic instability. Pulmonary angiography revealed dense radio-opaque material occluding the pulmonary vasculature bilaterally. The patient was shifted to the intensive care unit (ICU) for further management of this non-thrombotic pulmonary embolism. In the ICU, the patient had sinus tachycardia with a heart rate of 140–150 beats/min and a respiratory rate of 30– 40 breaths/min. Her end-tidal carbon dioxide pressure (PetCO2) was 18 mmHg with a corresponding PaCO2 of 46.5 mmHg indicating an increase in the alveolar dead space. Her PaO2 was 60.3 mmHg at an FiO2 of 50 %. On neurological examination, the patient had a Glasgow coma scale of E3 VT M5, no motor deficits and symmetrical pupils that were reacting to light. Mechanical ventilation was continued under intravenous sedation. Additionally, she was treated with anticoagulants, steroids and antibiotics. Anticoagulation was achieved with
unfractionated heparin (15 U/kg/h) administered intravenously for 48 h, which was later changed to subcutaneous enoxaparin (40 mg/24 h) and finally to oral warfarin (2.5 mg/24 h). A transthoracic echocardiographic examination at 24 h following the pulmonary embolism revealed a mild tricuspid regurgitation and pulmonary artery hypertension with a pulmonary artery systolic pressure (PASP) of 51 mmHg. Sildenafil citrate in a dose of 10 mg was administered three times daily to treat this pulmonary hypertension. The patient’s haemodynamic instability began to improve on the second day. The heart rate improved to about 110– 120 beats/min while the blood pressure remained consistently around 110/70 mmHg. An improvement in the PetCO2 was also observed from the initial 18 to 24–28 mmHg with a corresponding PaCO2 of 40 mmHg over the next 2–3 days. A 2D echocardiogram, repeated at 72 h, after the event demonstrated an improvement in pulmonary hypertension with a PASP of 38 mmHg. On the fourth day, the patient developed polyuria with a urine output of 3 ml/kg/h. Serum and urine electrolytes and osmolality were, however, within normal limits. Her serum brain natriuretic peptide (BNP) was 155 pg/ml (normal,
