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Life-Threatening Hemothorax Caused by Spontaneous Extracranial Vertebral Aneurysm Rupture in Neurofibromatosis Type 1
Case Report
Life-Threatening Hemothorax Caused by Spontaneous Extracranial Vertebral Aneurysm Rupture in Neurofibromatosis Type 1 Kwang Seok Han1, Kyung Mi Lee2, Bum Joon Kim3, Byung Duk Kwun1, Seok Keun Choi1, Sung Ho Lee4
Key words Aneurysm - Coil embolization - Hemothorax - Neurofibromatosis - Vertebral artery -
Abbreviations and Acronyms NF1: Neurofibromatosis type 1 VA: Vertebral artery From the Departments of 1Neurosurgery, 2Radiology, and 3 Neurology, College of Medicine, Kyung Hee University, Seoul; and 4Department of Neurosurgery, Seoul National University Hospital, Seoul National University College of Medicine, Seoul, Korea To whom correspondence should be addressed: Sung Ho Lee, M.D., Ph.D. [E-mail: [email protected]; [email protected]] Citation: World Neurosurg. (2019) 130:157-159. https://doi.org/10.1016/j.wneu.2019.07.007 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
- BACKGROUND:
Although vascular abnormality is an uncommon comorbidity of neurofibromatosis type 1 (NF1), it is potentially fatal. We present spontaneous hemothorax caused by rupture of a vertebral artery (VA) aneurysm in a patient with NF1.
- CASE
DESCRIPTION: A 36-year-old man with a history of NF1 was transferred to the emergency department with dyspnea. Chest computed tomography scan revealed hemothorax in the left lung field with mediastinal shifting and aneurysmal dilatation of the left VA at the C6 vertebra level. Immediate drainage of the hematoma by chest tube insertion was performed. Diagnostic angiogram showed a 3- to 4-cm fusiform aneurysm of the VA. After the angiogram, cardiopulmonary arrest occurred after a rebleed of the VA aneurysm. The aneurysmal segment of the VA was urgently occluded with detachable coils. Postoperatively, the patient was in intensive care for 1 month because of fulminant pneumonia. After the patient regained consciousness, he was found to have right hemiparesis from a small infarction at the pons. The patient’s function improved to near normal after 1 year of recovery.
- CONCLUSIONS:
Hemothorax caused by VA rupture in a patient with NF1 is an extremely rare condition that can be fatal. Careful examination with suspicion for early detection and treatment is required for this urgent condition. Endovascular coiling was safe even for an unstable patient with massive bleeding.
INTRODUCTION Neurofibromatosis type 1 (NF1), also known as von Recklinghausen disease, is an autosomal dominant disorder characterized by café-au-lait macules, neurofibromas, axillary freckling, Lisch nodule, optic pathway glioma, and bony dysplasia.1 Vascular abnormalities, including aneurysm, stenosis, and fistula, are less common features found in NF1.2 Hemothorax in NF1 is a rare but fatal condition. Here, we report a patient with NF1 who suffered a life-threatening hemothorax caused by rupture of an extracranial vertebral artery (VA) aneurysm. CASE DESCRIPTION A 36-year-old man was transferred to the emergency department complaining of chest discomfort and dyspnea. He had a history of NF1, and several café-au-lait spots and numerous subcutaneous nodules were observed on examination. Chest
radiograph showed a massive hemothorax and contralateral mediastinal shifting. He had no recent trauma history. His blood pressure was 110/70 mm Hg and arterial oxygen saturation level 94% on 2 L/min of oxygen supply with nasal cannula, but hemoglobin count was 12.4 g/dL, decreased from 16.7 g/dL at a previous hospital 8 hours earlier. Enhanced chest computed tomography scan revealed hemothorax, hemomediastinum and extrapleural hematoma, and an aneurysmal sac on the left VA at the C6 vertebra level (Figure 1). A chest tube was inserted, and 2 L of blood was extracted. Diagnostic angiography showed an aneurysmal dilatation measuring 3e4 cm in diameter on the left VA at the extraosseous portion. Immediate occlusion of the lesion was postponed because the right VA was not visible, and only the distal interosseous part of the right VA was supplied from muscular branches of the thyrocervical trunk. Both posterior
WORLD NEUROSURGERY 130: 157-159, OCTOBER 2019
communicating arteries were the fetal type, but the basilar and vertebral arteries were not shown on the internal carotid artery angiogram. Therefore, we planned a balloon test occlusion of the left VA for endovascular trapping of the dissected segment for the following morning. If the test failed, bypass surgery would have been required. However, immediately after the angiography, the patient became dyspneic and, shortly afterward, asystolic, which seemed to have resulted from rebleeding of the VA aneurysm. Immediate chest tube opening and cardiac compressions were performed. The patient’s vital signs returned, but hemoglobin count had decreased to 9.9 g/dL. Because of the urgent situation and despite the risk of ischemia, we decided to occlude the aneurysmal segment. A guiding catheter (Fubuki, Asahi, Japan) was inserted into the proximal portion of the left VA, and then double
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CASE REPORT KWANG SEOK HAN ET AL.
HEMOTHORAX AND VERTEBRAL ARTERY ANEURYSM
Figure 1. (A) Contrast-enhanced chest computed tomography scan showing massive hemothorax, hemomediastinum and mediastinal contralateral shifting, and aneurysmal dilatation of the left vertebral artery (arrow). (B) Left subclavian arteriogram revealing aneurysmal dilatation
microcatheters (Excelsior SL-10, straight and 45 preshaped [Boston Scientific, Natick, Massachusetts, USA]) were placed. Morphology of the aneurysm was not grossly changed. The affected segment was almost completely occluded after insertion of all stocked 25 detachable coils. After the procedure, carotid angiography showed increased flow in both posterior communicating arteries, and both intracranial VAs were visualized by retrograde flow (Figure 2). Perfusion
computed tomography scan after endovascular surgery showed normal perfusion throughout the brain, and there was no deterioration on neurologic examination. The patient was stable after the procedure, but he suffered from fulminant pneumonia, and an extracorporeal membrane oxygenation device was applied at the seventh postoperative day. When he regained consciousness, he demonstrated rightsided hemiparesis. Magnetic resonance
Figure 2. (A) The basilar artery (arrow) and anterior inferior cerebellar artery were revealed on the internal cerebral angiogram after occlusion (not shown) of the aneurysmal
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(3e4 cm in diameter) of the proximal vertebral artery. (C) The posterior cerebral and superior cerebellar arteries were mainly supplied by the internal cerebral artery via fetal-type posterior communicating arteries.
imaging revealed a subacute infarction at the left side of the pons. After 29 days of intensive medical care, he was transferred to the department of rehabilitation. His hemiparesis improved with time, and he was discharged home at the 70th postoperative day. A follow-up angiography at 6 months showed complete occlusion of the dissected segment and development of collateral flow from the muscular branches to the left VA. The final modified Rankin Scale score was 1 at the 1-year follow-up visit.
segment of the left vertebral artery. (B) Magnetic resonance T2 image on the 27th postoperative day shows an infarction in the subacute stage on the left side of the pons.
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.07.007
CASE REPORT KWANG SEOK HAN ET AL.
DISCUSSION Most aneurysms of the extracranial VAs are arterial dissections caused by penetrating or blunt trauma.3 Spontaneous extracranial VA aneurysm is very rare and usually associated with hereditary connective tissue disorders, including Ehler-Danlos syndrome, Marfan disease, and neurofibromatosis.4 Vascular lesions in NF1 are less common causes of comorbidity, and their prevalence has been reported to be 2%e3.6%.5,6 The aorta and renal artery are the most commonly involved sites, whereas the VA is much less frequently affected.6 Less than 40 cases of hemothorax in patients with NF1 have been reported in the literature.7 It is twice more common in women than in men, and most patients are 40e60 years of age.8 The most recent report identified the intercostal artery and small branches of the subclavian artery as the most common bleeding sources. Rarely, venous aneurysm, thoracic mass, and meningocele are also reported as a source of hemothorax.9-11 A case of VA aneurysm in a patient with systemic atherosclerosis resulting in extrapleural hematoma has been reported.12 Depending on the individual situation, the reported treatment options vary and include open surgery, video-assisted thoracic surgery, and endovascular intervention including coil embolization or stent placement.13-15 Despite treatment, VA aneurysm rupture was lethal when the bleeding point was not identified and/or the initial vital signs were unstable, especially in publications before 2000.5 However, outcomes have much improved to excellent in recent reports with prompt management.11,14 Several reports have suggested the risk of an aneurysm or dissection in a patient with NF1; therefore, early obliteration should be considered. However, those rare lesions are hard to be detected during routine follow-up. In the present case, the aneurysm was properly obliterated; however, there was a risk of hypoperfusion to the brainstem and upper spinal cord after coil embolization. Fortunately, retrograde flow via a fetal type of both posterior communicating arteries and antegrade flow via muscular branches
HEMOTHORAX AND VERTEBRAL ARTERY ANEURYSM
from both VAs were present. However, the patient did suffer a small cerebral infarction at the pons. Because the infarction was found in the subacute to chronic stage at 27 days after the occlusion, the exact timing and cause were not known. A possible cause of the infarction was hemodynamic insufficiency and instability. Delayed follow-up angiography showed late development of collateral vessels from muscular branches to the distal part of the left VA. Although the patient was neurologically intact just after the procedure, his poor medical condition, especially during fulminant pneumonia, may have caused hemodynamic insufficiency and thrombogenic condition. The second possible cause was a stump embolization that was a thrombosed embolus from the obliterated segment. The aneurysmal segment was not completely obliterated after embolization, and slight antegrade flow remained because of limited stocked coils. The final possible cause was an embolus during follow-up diagnostic angiography. We report an extremely rare case of hemothorax from VA aneurysm rupture in a patient with NF1 who was treated with coil embolization. Hemothorax in NF1 is a rare but potentially fatal condition, and a careful examination with suspicion is required for early detection. The treatment strategy is not yet established, but prompt and aggressive management in a tailored manner has shown positive results. REFERENCES 1. Neurofibromatosis. Conference statement. National Institutes of Health Consensus Development Conference. Arch Neurol. 1988;45:575-578. 2. Arai K, Sanada J, Kurozumi A, Watanabe T, Matsui O. Spontaneous hemothorax in neurofibromatosis treated with percutaneous embolization. Cardiovasc Intervent Radiol. 2007;30:477-479. 3. Fusco MR, Harrigan MR. Cerebrovascular dissections: a review. Part II: blunt cerebrovascular injury. Neurosurgery. 2011;68:517-530 [discussion: 530]. 4. Morasch MD, Phade SV, Naughton P, GarciaToca M, Escobar G, Berguer R. Primary extracranial vertebral artery aneurysms. Ann Vasc Surg. 2013;27:418-423. 5. Miura H, Taira O, Uchida O, Usuda J, Hirai S, Kato H. Spontaneous haemothorax associated with von Recklinghausen’s disease: review of occurrence in Japan. Thorax. 1997;52:577-578 [discussion: 575-576].
WORLD NEUROSURGERY 130: 157-159, OCTOBER 2019
6. Oderich GS, Sullivan TM, Bower TC, et al. Vascular abnormalities in patients with neurofibromatosis syndrome type I: clinical spectrum, management, and results. J Vasc Surg. 2007;46: 475-484. 7. Miura T, Kawano Y, Chujo M, Miyawaki M, Mori H, Kawahara K. Spontaneous hemothorax in patients with von Recklinghausen’s disease. Jpn J Thorac Cardiovasc Surg. 2005;53:649-652. 8. Degbelo FDA, Cito G, Guendil B, Christodoulou M, Abbassi Z. Spontaneous hemothorax in a patient with von Recklinghausen’s disease: a case report and review of the literature. Am J Case Rep. 2019;20:674-678. 9. Nopajaroonsri C, Lurie AA. Venous aneurysm, arterial dysplasia, and near-fatal hemorrhages in neurofibromatosis type 1. Hum Pathol. 1996;27: 982-985. 10. Fohrding LZ, Sellmann T, Angenendt S, et al. A case of lethal spontaneous massive hemothorax in a patient with neurofibromatosis 1. J Cardiothorac Surg. 2014;9:172. 11. Jeong SC, Kim JJ, Choi SY, Kim YH, Kim IS. Successful surgical treatment of massive spontaneous hemothorax due to intrathoracic secondary degeneration of a neurofibroma from mediastinal involvement of type 1 neurofibromatosis. J Thorac Dis. 2018;10:E203-E206. 12. Nenadic D, Balevic M, Milojevic M, Tanskovic S. Extracranial vertebral artery aneurysm rupture complicated by extrapleural haematoma. EJVES Short Rep. 2018;38:12-14. 13. Miyazaki T, Tsuchiya T, Tagawa T, Yamasaki N, Nagayasu T. Spontaneous hemothorax associated with von Recklinghausen’s disease: report of a case. Ann Thorac Cardiovasc Surg. 2011;17:301-303. 14. Mydin MI, Sharma A, Zia Z, Hawari M, Jadoon M, Majewski A. A novel approach in managing rightsided haemothorax in neurofibromatosis type 1. Asian Cardiovasc Thorac Ann. 2015;23:573-575. 15. Misao T, Yoshikawa T, Aoe M, Ueda Y, Yodoya M, Sakurai J. Recurrent rupture of intercostal artery aneurysms in neurofibromatosis type 1. Gen Thorac Cardiovasc Surg. 2012;60:179-182.
Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 7 June 2019; accepted 1 July 2019 Citation: World Neurosurg. (2019) 130:157-159. https://doi.org/10.1016/j.wneu.2019.07.007 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
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Life-Threatening Hemothorax Caused by Spontaneous Extracranial Vertebral Aneurysm Rupture in Neurofibromatosis Type 1 Kwang Seok Han1, Kyung Mi Lee2, Bum Joon Kim3, Byung Duk Kwun1, Seok Keun Choi1, Sung Ho Lee4
Key words Aneurysm - Coil embolization - Hemothorax - Neurofibromatosis - Vertebral artery -
Abbreviations and Acronyms NF1: Neurofibromatosis type 1 VA: Vertebral artery From the Departments of 1Neurosurgery, 2Radiology, and 3 Neurology, College of Medicine, Kyung Hee University, Seoul; and 4Department of Neurosurgery, Seoul National University Hospital, Seoul National University College of Medicine, Seoul, Korea To whom correspondence should be addressed: Sung Ho Lee, M.D., Ph.D. [E-mail: [email protected]; [email protected]] Citation: World Neurosurg. (2019) 130:157-159. https://doi.org/10.1016/j.wneu.2019.07.007 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
- BACKGROUND:
Although vascular abnormality is an uncommon comorbidity of neurofibromatosis type 1 (NF1), it is potentially fatal. We present spontaneous hemothorax caused by rupture of a vertebral artery (VA) aneurysm in a patient with NF1.
- CASE
DESCRIPTION: A 36-year-old man with a history of NF1 was transferred to the emergency department with dyspnea. Chest computed tomography scan revealed hemothorax in the left lung field with mediastinal shifting and aneurysmal dilatation of the left VA at the C6 vertebra level. Immediate drainage of the hematoma by chest tube insertion was performed. Diagnostic angiogram showed a 3- to 4-cm fusiform aneurysm of the VA. After the angiogram, cardiopulmonary arrest occurred after a rebleed of the VA aneurysm. The aneurysmal segment of the VA was urgently occluded with detachable coils. Postoperatively, the patient was in intensive care for 1 month because of fulminant pneumonia. After the patient regained consciousness, he was found to have right hemiparesis from a small infarction at the pons. The patient’s function improved to near normal after 1 year of recovery.
- CONCLUSIONS:
Hemothorax caused by VA rupture in a patient with NF1 is an extremely rare condition that can be fatal. Careful examination with suspicion for early detection and treatment is required for this urgent condition. Endovascular coiling was safe even for an unstable patient with massive bleeding.
INTRODUCTION Neurofibromatosis type 1 (NF1), also known as von Recklinghausen disease, is an autosomal dominant disorder characterized by café-au-lait macules, neurofibromas, axillary freckling, Lisch nodule, optic pathway glioma, and bony dysplasia.1 Vascular abnormalities, including aneurysm, stenosis, and fistula, are less common features found in NF1.2 Hemothorax in NF1 is a rare but fatal condition. Here, we report a patient with NF1 who suffered a life-threatening hemothorax caused by rupture of an extracranial vertebral artery (VA) aneurysm. CASE DESCRIPTION A 36-year-old man was transferred to the emergency department complaining of chest discomfort and dyspnea. He had a history of NF1, and several café-au-lait spots and numerous subcutaneous nodules were observed on examination. Chest
radiograph showed a massive hemothorax and contralateral mediastinal shifting. He had no recent trauma history. His blood pressure was 110/70 mm Hg and arterial oxygen saturation level 94% on 2 L/min of oxygen supply with nasal cannula, but hemoglobin count was 12.4 g/dL, decreased from 16.7 g/dL at a previous hospital 8 hours earlier. Enhanced chest computed tomography scan revealed hemothorax, hemomediastinum and extrapleural hematoma, and an aneurysmal sac on the left VA at the C6 vertebra level (Figure 1). A chest tube was inserted, and 2 L of blood was extracted. Diagnostic angiography showed an aneurysmal dilatation measuring 3e4 cm in diameter on the left VA at the extraosseous portion. Immediate occlusion of the lesion was postponed because the right VA was not visible, and only the distal interosseous part of the right VA was supplied from muscular branches of the thyrocervical trunk. Both posterior
WORLD NEUROSURGERY 130: 157-159, OCTOBER 2019
communicating arteries were the fetal type, but the basilar and vertebral arteries were not shown on the internal carotid artery angiogram. Therefore, we planned a balloon test occlusion of the left VA for endovascular trapping of the dissected segment for the following morning. If the test failed, bypass surgery would have been required. However, immediately after the angiography, the patient became dyspneic and, shortly afterward, asystolic, which seemed to have resulted from rebleeding of the VA aneurysm. Immediate chest tube opening and cardiac compressions were performed. The patient’s vital signs returned, but hemoglobin count had decreased to 9.9 g/dL. Because of the urgent situation and despite the risk of ischemia, we decided to occlude the aneurysmal segment. A guiding catheter (Fubuki, Asahi, Japan) was inserted into the proximal portion of the left VA, and then double
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CASE REPORT KWANG SEOK HAN ET AL.
HEMOTHORAX AND VERTEBRAL ARTERY ANEURYSM
Figure 1. (A) Contrast-enhanced chest computed tomography scan showing massive hemothorax, hemomediastinum and mediastinal contralateral shifting, and aneurysmal dilatation of the left vertebral artery (arrow). (B) Left subclavian arteriogram revealing aneurysmal dilatation
microcatheters (Excelsior SL-10, straight and 45 preshaped [Boston Scientific, Natick, Massachusetts, USA]) were placed. Morphology of the aneurysm was not grossly changed. The affected segment was almost completely occluded after insertion of all stocked 25 detachable coils. After the procedure, carotid angiography showed increased flow in both posterior communicating arteries, and both intracranial VAs were visualized by retrograde flow (Figure 2). Perfusion
computed tomography scan after endovascular surgery showed normal perfusion throughout the brain, and there was no deterioration on neurologic examination. The patient was stable after the procedure, but he suffered from fulminant pneumonia, and an extracorporeal membrane oxygenation device was applied at the seventh postoperative day. When he regained consciousness, he demonstrated rightsided hemiparesis. Magnetic resonance
Figure 2. (A) The basilar artery (arrow) and anterior inferior cerebellar artery were revealed on the internal cerebral angiogram after occlusion (not shown) of the aneurysmal
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(3e4 cm in diameter) of the proximal vertebral artery. (C) The posterior cerebral and superior cerebellar arteries were mainly supplied by the internal cerebral artery via fetal-type posterior communicating arteries.
imaging revealed a subacute infarction at the left side of the pons. After 29 days of intensive medical care, he was transferred to the department of rehabilitation. His hemiparesis improved with time, and he was discharged home at the 70th postoperative day. A follow-up angiography at 6 months showed complete occlusion of the dissected segment and development of collateral flow from the muscular branches to the left VA. The final modified Rankin Scale score was 1 at the 1-year follow-up visit.
segment of the left vertebral artery. (B) Magnetic resonance T2 image on the 27th postoperative day shows an infarction in the subacute stage on the left side of the pons.
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.07.007
CASE REPORT KWANG SEOK HAN ET AL.
DISCUSSION Most aneurysms of the extracranial VAs are arterial dissections caused by penetrating or blunt trauma.3 Spontaneous extracranial VA aneurysm is very rare and usually associated with hereditary connective tissue disorders, including Ehler-Danlos syndrome, Marfan disease, and neurofibromatosis.4 Vascular lesions in NF1 are less common causes of comorbidity, and their prevalence has been reported to be 2%e3.6%.5,6 The aorta and renal artery are the most commonly involved sites, whereas the VA is much less frequently affected.6 Less than 40 cases of hemothorax in patients with NF1 have been reported in the literature.7 It is twice more common in women than in men, and most patients are 40e60 years of age.8 The most recent report identified the intercostal artery and small branches of the subclavian artery as the most common bleeding sources. Rarely, venous aneurysm, thoracic mass, and meningocele are also reported as a source of hemothorax.9-11 A case of VA aneurysm in a patient with systemic atherosclerosis resulting in extrapleural hematoma has been reported.12 Depending on the individual situation, the reported treatment options vary and include open surgery, video-assisted thoracic surgery, and endovascular intervention including coil embolization or stent placement.13-15 Despite treatment, VA aneurysm rupture was lethal when the bleeding point was not identified and/or the initial vital signs were unstable, especially in publications before 2000.5 However, outcomes have much improved to excellent in recent reports with prompt management.11,14 Several reports have suggested the risk of an aneurysm or dissection in a patient with NF1; therefore, early obliteration should be considered. However, those rare lesions are hard to be detected during routine follow-up. In the present case, the aneurysm was properly obliterated; however, there was a risk of hypoperfusion to the brainstem and upper spinal cord after coil embolization. Fortunately, retrograde flow via a fetal type of both posterior communicating arteries and antegrade flow via muscular branches
HEMOTHORAX AND VERTEBRAL ARTERY ANEURYSM
from both VAs were present. However, the patient did suffer a small cerebral infarction at the pons. Because the infarction was found in the subacute to chronic stage at 27 days after the occlusion, the exact timing and cause were not known. A possible cause of the infarction was hemodynamic insufficiency and instability. Delayed follow-up angiography showed late development of collateral vessels from muscular branches to the distal part of the left VA. Although the patient was neurologically intact just after the procedure, his poor medical condition, especially during fulminant pneumonia, may have caused hemodynamic insufficiency and thrombogenic condition. The second possible cause was a stump embolization that was a thrombosed embolus from the obliterated segment. The aneurysmal segment was not completely obliterated after embolization, and slight antegrade flow remained because of limited stocked coils. The final possible cause was an embolus during follow-up diagnostic angiography. We report an extremely rare case of hemothorax from VA aneurysm rupture in a patient with NF1 who was treated with coil embolization. Hemothorax in NF1 is a rare but potentially fatal condition, and a careful examination with suspicion is required for early detection. The treatment strategy is not yet established, but prompt and aggressive management in a tailored manner has shown positive results. REFERENCES 1. Neurofibromatosis. Conference statement. National Institutes of Health Consensus Development Conference. Arch Neurol. 1988;45:575-578. 2. Arai K, Sanada J, Kurozumi A, Watanabe T, Matsui O. Spontaneous hemothorax in neurofibromatosis treated with percutaneous embolization. Cardiovasc Intervent Radiol. 2007;30:477-479. 3. Fusco MR, Harrigan MR. Cerebrovascular dissections: a review. Part II: blunt cerebrovascular injury. Neurosurgery. 2011;68:517-530 [discussion: 530]. 4. Morasch MD, Phade SV, Naughton P, GarciaToca M, Escobar G, Berguer R. Primary extracranial vertebral artery aneurysms. Ann Vasc Surg. 2013;27:418-423. 5. Miura H, Taira O, Uchida O, Usuda J, Hirai S, Kato H. Spontaneous haemothorax associated with von Recklinghausen’s disease: review of occurrence in Japan. Thorax. 1997;52:577-578 [discussion: 575-576].
WORLD NEUROSURGERY 130: 157-159, OCTOBER 2019
6. Oderich GS, Sullivan TM, Bower TC, et al. Vascular abnormalities in patients with neurofibromatosis syndrome type I: clinical spectrum, management, and results. J Vasc Surg. 2007;46: 475-484. 7. Miura T, Kawano Y, Chujo M, Miyawaki M, Mori H, Kawahara K. Spontaneous hemothorax in patients with von Recklinghausen’s disease. Jpn J Thorac Cardiovasc Surg. 2005;53:649-652. 8. Degbelo FDA, Cito G, Guendil B, Christodoulou M, Abbassi Z. Spontaneous hemothorax in a patient with von Recklinghausen’s disease: a case report and review of the literature. Am J Case Rep. 2019;20:674-678. 9. Nopajaroonsri C, Lurie AA. Venous aneurysm, arterial dysplasia, and near-fatal hemorrhages in neurofibromatosis type 1. Hum Pathol. 1996;27: 982-985. 10. Fohrding LZ, Sellmann T, Angenendt S, et al. A case of lethal spontaneous massive hemothorax in a patient with neurofibromatosis 1. J Cardiothorac Surg. 2014;9:172. 11. Jeong SC, Kim JJ, Choi SY, Kim YH, Kim IS. Successful surgical treatment of massive spontaneous hemothorax due to intrathoracic secondary degeneration of a neurofibroma from mediastinal involvement of type 1 neurofibromatosis. J Thorac Dis. 2018;10:E203-E206. 12. Nenadic D, Balevic M, Milojevic M, Tanskovic S. Extracranial vertebral artery aneurysm rupture complicated by extrapleural haematoma. EJVES Short Rep. 2018;38:12-14. 13. Miyazaki T, Tsuchiya T, Tagawa T, Yamasaki N, Nagayasu T. Spontaneous hemothorax associated with von Recklinghausen’s disease: report of a case. Ann Thorac Cardiovasc Surg. 2011;17:301-303. 14. Mydin MI, Sharma A, Zia Z, Hawari M, Jadoon M, Majewski A. A novel approach in managing rightsided haemothorax in neurofibromatosis type 1. Asian Cardiovasc Thorac Ann. 2015;23:573-575. 15. Misao T, Yoshikawa T, Aoe M, Ueda Y, Yodoya M, Sakurai J. Recurrent rupture of intercostal artery aneurysms in neurofibromatosis type 1. Gen Thorac Cardiovasc Surg. 2012;60:179-182.
Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 7 June 2019; accepted 1 July 2019 Citation: World Neurosurg. (2019) 130:157-159. https://doi.org/10.1016/j.wneu.2019.07.007 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
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