The Neuroradiology Journal 21: 721-724, 2008
www. centauro. it
Can a Warning Leak in a Patient with Unruptured Aneurysm Mask an Underlying Gastrointestinal Pathology? A Case Report W.L. POON*, D.C. SUH, J.W. CHOI, B.S. CHOI, E.Y. CHA, J.Y. BANG**, K.D. HAHM**, H.J. JANG***, K-H. DO, M.C. LEE**** * Department of Diagnostic Radiology, Tuen Mun Hospital; Tuen Mun, Hong Kong Department of Radiology and Research Institute of Radiology; ** Department of Anesthesiology and Pain Medicine; *** Respiratory and Critical Care Medicine; **** Neurology, University of Ulsan, College of Medicine, Asan Medical Center; Seoul, Korea
Key words: respiratory distress syndrome, adult (ARDS), brain, hemorrhage, anesthesia, achalasia, aneurysm, angiography
SUMMARY – Aggravating headache accompanied by nausea and epigastric discomfort suggesting a warning leak in a 39-year-old woman with a giant thrombosed intracranial aneurysm prompted us to undertake coiling of the aneurysm. After uneventful coil embolization of the aneurysm, collapse of the lung related to bronchospam developed, and was found to have a gastrointestinal pathology which had gone undetected before the procedure. Despite its rarity, gastrointestinal pathology mimicking warning leak should have been considered in a patient with a warning leak sign.
Introduction Warning leak refers to the sudden onset of a headache days to weeks preceding the rupture of an intracranial aneurysm resulting in acute subarachnoid haemorrhage (SAH). It is reported in 15% to 60% of all patients admitted with an SAH 1,2. In addition to headache, nausea and vomiting are the second most common presenting symptoms 3,4. In rare clinical situations, they can be either mimicked or masked by underlying gastrointestinal problems. We report a rare anaesthetic complication caused by bronchspasm leading to pulmonary edema and lung collapse in a patient with previously undiagnosed achalasia while undergoing endovascular aneurysmal coiling under general anaesthesia. Case Report A 39-year-old woman was admitted for investigation of a severe headache accompanied by nausea and epigastric discomfort. Her chest
postero-anterior and lateral views were initially regarded as normal (figure 1A,B). On physical examination she was found to have left homonymous hemianopia. Subsequent computed tomography and cerebral angiography demonstrated a giant aneurysm at the junction between the right posterior communicating artery and the right posterior cerebral artery (figure 1C). She fasted overnight before the procedure. At the end of successful embolization with GDC coils (figure 1D,E), the oxygen saturation of the patient gradually decreased and ventilation became difficult (figure 1F). A post-procedural DynaCT scan of the patient’s head showed no evidence of subarachnoid hemorrhage. Her consciousness and vital signs were intact and she remained on mechanical ventilation for a week without further neurological deficit. The residual pulmonary edema and collapse gradually subsided (figure 1G). A barium swallow study showed a dilated oesophagus with smooth tapering of the lower oesophagus, showing the ‘bird’s beak’ appearance typical of achalasia (figure 1H) which was treated by balloon dilatation. 721
Can a Warning Leak in a Patient with Unruptured Aneurysm Mask an Underlying Gastrointestinal Pathology?
A
B
C
722
D
W.L. Poon
www. centauro. it
The Neuroradiology Journal 21: 721-724, 2008
F
E
G
H
Figure 1 A) Chest PA shows the suspected air density (an arrow) of esophagus in the mediastium. B) Lateral view shows narrowing of the trachea with anterior deviation caused by the dilated esophagus (arrow). C) Left vertebral angiogram demonstrating a giant aneurysm at the junction of the right proximal P2 segment. D) Complete embolization of the aneurysm and parent artery was successfully performed using coils. E) Late phase of the right internal carotid arteriogram shows a good collateral filling of the right PCA cortical branches (arrow). F) Chest radiograph taken in the angiosuite immediately after endovascular coiling of the aneurysm shows near total collapse of the left lung and the presence of pulmonary edema in the right lung. G) Endotracheal suction and ventilator support led to resolution of the pulmonary edema and collapse, as seen on a chest radiograph taken five hours after the procedure. H) Barium swallow study demonstrated a grossly dilated oesophagus with a tapering lower end, giving rise to a ‘bird’s beak’ appearance, both typical findings in achalasia. After balloon dilatation of the esophageal gastric junction, and her chest radiograph returned to normal (not shown).
723
Can a Warning Leak in a Patient with Unruptured Aneurysm Mask an Underlying Gastrointestinal Pathology?
Discussion Intermittent severe headache associated with nausea in patients with an unruptured giant aneurysm in the posterior circulation strongly refers to a warning leak sign because a cumulative five year rupture rate of a giant aneurysm in the posterior circulation with a size more than 25 mm reaches 50 percent 5,6. Even though the warning leak has been emphasized in order not to be missed 7, to the best of our knowledge, gastrointestinal pathology masking warning leak has not been described. Achalasia is an idiopathic motility disorder of the oesophagus characterized by an imbalance of the excitatory and inhibitory neuronal activities in the lower oesophageal sphincter 8 as the neurons containing inhibitory neurotransmitters such as nitric oxide and vasoac-
W.L. Poon
tive intestinal peptide are absent in the region of the lower sphincter 9,10. The achalasia in our patient was finally successfully treated by balloon dilatation under fluoroscopic guidance. In conclusion, warning leak can rarely mask an underlying gastrointestinal disorder which can impose a high risk of acute respiratory distress for patients receiving general anesthesia for interventional radiological procedures as well as other types of surgery. Acknowlegment This study was supported by a grant from Korea Healthcare technology R&D Project, Ministry of Health & Welfare, Republic of Korea. (A080201)
References 1 Leblanc R: The minor leak preceding subarachnoid hemorrhage. J Neurosurg 66: 35-39, 1987. 2 Zikk D, Rapoport Y et Al: Acute airway obstruction and achalasia of the esophagus. Ann Otol Rhinol Laryngol 98: 641-643, 1989. 3 Waga S, Otsubo K et Al: Warning signs in intracranial aneurysms. Surg Neurol 3: 15-20, 1975. 4 Torner JC, Kassell NF et Al: Preoperative prognostic factors for rebleeding and survival in aneurysm patients receiving antifibrinolytic therapy: Report of the cooperative aneurysm study. Neurosurgery 9: 506-513, 1981. 5 Wiebers DO: Unruptured intracranial aneurysms: Natural history and clinical management. Update on the international study of unruptured intracranial aneurysms. Neuroimaging Clin N Am 16: 383-390, vii, 2006. 6 Wiebers DO, Whisnant JP et Al: Unruptured intracranial aneurysms: Natural history, clinical outcome, and risks of surgical and endovascular treatment. Lancet 362: 103-110, 2003. 7 Ostergaard JR: Warning leak in subarachnoid haemorrhage. BMJ 301: 190-191, 1990. 8 Vantrappen G, Janssens J et Al: Achalasia, diffuse esophageal spasm, and related motility disorders. Gastroenterology 76: 450-457, 1979.
724
9 Aggestrup S, Uddman R et Al: Lack of vasoactive intestinal polypeptide nerves in esophageal achalasia. Gastroenterology 84: 924-927, 1983. 10 Tottrup A, Svane D et Al: Nitric oxide mediating nanc inhibition in opossum lower esophageal sphincter. Am J Physiol 260: 385-389, 1991.
Dae Chul Suh, MD Department of Radiology and Research Institute of Radiology University of Ulsan, College of Medicine Asan Medical Center 388-1 Pungnap2-dong, Songpa-gu Seoul, 138-736, Korea Tel.: 822-3010-4366 Fax: 822-476-0090 E-mail: [email protected]
www. centauro. it
Can a Warning Leak in a Patient with Unruptured Aneurysm Mask an Underlying Gastrointestinal Pathology? A Case Report W.L. POON*, D.C. SUH, J.W. CHOI, B.S. CHOI, E.Y. CHA, J.Y. BANG**, K.D. HAHM**, H.J. JANG***, K-H. DO, M.C. LEE**** * Department of Diagnostic Radiology, Tuen Mun Hospital; Tuen Mun, Hong Kong Department of Radiology and Research Institute of Radiology; ** Department of Anesthesiology and Pain Medicine; *** Respiratory and Critical Care Medicine; **** Neurology, University of Ulsan, College of Medicine, Asan Medical Center; Seoul, Korea
Key words: respiratory distress syndrome, adult (ARDS), brain, hemorrhage, anesthesia, achalasia, aneurysm, angiography
SUMMARY – Aggravating headache accompanied by nausea and epigastric discomfort suggesting a warning leak in a 39-year-old woman with a giant thrombosed intracranial aneurysm prompted us to undertake coiling of the aneurysm. After uneventful coil embolization of the aneurysm, collapse of the lung related to bronchospam developed, and was found to have a gastrointestinal pathology which had gone undetected before the procedure. Despite its rarity, gastrointestinal pathology mimicking warning leak should have been considered in a patient with a warning leak sign.
Introduction Warning leak refers to the sudden onset of a headache days to weeks preceding the rupture of an intracranial aneurysm resulting in acute subarachnoid haemorrhage (SAH). It is reported in 15% to 60% of all patients admitted with an SAH 1,2. In addition to headache, nausea and vomiting are the second most common presenting symptoms 3,4. In rare clinical situations, they can be either mimicked or masked by underlying gastrointestinal problems. We report a rare anaesthetic complication caused by bronchspasm leading to pulmonary edema and lung collapse in a patient with previously undiagnosed achalasia while undergoing endovascular aneurysmal coiling under general anaesthesia. Case Report A 39-year-old woman was admitted for investigation of a severe headache accompanied by nausea and epigastric discomfort. Her chest
postero-anterior and lateral views were initially regarded as normal (figure 1A,B). On physical examination she was found to have left homonymous hemianopia. Subsequent computed tomography and cerebral angiography demonstrated a giant aneurysm at the junction between the right posterior communicating artery and the right posterior cerebral artery (figure 1C). She fasted overnight before the procedure. At the end of successful embolization with GDC coils (figure 1D,E), the oxygen saturation of the patient gradually decreased and ventilation became difficult (figure 1F). A post-procedural DynaCT scan of the patient’s head showed no evidence of subarachnoid hemorrhage. Her consciousness and vital signs were intact and she remained on mechanical ventilation for a week without further neurological deficit. The residual pulmonary edema and collapse gradually subsided (figure 1G). A barium swallow study showed a dilated oesophagus with smooth tapering of the lower oesophagus, showing the ‘bird’s beak’ appearance typical of achalasia (figure 1H) which was treated by balloon dilatation. 721
Can a Warning Leak in a Patient with Unruptured Aneurysm Mask an Underlying Gastrointestinal Pathology?
A
B
C
722
D
W.L. Poon
www. centauro. it
The Neuroradiology Journal 21: 721-724, 2008
F
E
G
H
Figure 1 A) Chest PA shows the suspected air density (an arrow) of esophagus in the mediastium. B) Lateral view shows narrowing of the trachea with anterior deviation caused by the dilated esophagus (arrow). C) Left vertebral angiogram demonstrating a giant aneurysm at the junction of the right proximal P2 segment. D) Complete embolization of the aneurysm and parent artery was successfully performed using coils. E) Late phase of the right internal carotid arteriogram shows a good collateral filling of the right PCA cortical branches (arrow). F) Chest radiograph taken in the angiosuite immediately after endovascular coiling of the aneurysm shows near total collapse of the left lung and the presence of pulmonary edema in the right lung. G) Endotracheal suction and ventilator support led to resolution of the pulmonary edema and collapse, as seen on a chest radiograph taken five hours after the procedure. H) Barium swallow study demonstrated a grossly dilated oesophagus with a tapering lower end, giving rise to a ‘bird’s beak’ appearance, both typical findings in achalasia. After balloon dilatation of the esophageal gastric junction, and her chest radiograph returned to normal (not shown).
723
Can a Warning Leak in a Patient with Unruptured Aneurysm Mask an Underlying Gastrointestinal Pathology?
Discussion Intermittent severe headache associated with nausea in patients with an unruptured giant aneurysm in the posterior circulation strongly refers to a warning leak sign because a cumulative five year rupture rate of a giant aneurysm in the posterior circulation with a size more than 25 mm reaches 50 percent 5,6. Even though the warning leak has been emphasized in order not to be missed 7, to the best of our knowledge, gastrointestinal pathology masking warning leak has not been described. Achalasia is an idiopathic motility disorder of the oesophagus characterized by an imbalance of the excitatory and inhibitory neuronal activities in the lower oesophageal sphincter 8 as the neurons containing inhibitory neurotransmitters such as nitric oxide and vasoac-
W.L. Poon
tive intestinal peptide are absent in the region of the lower sphincter 9,10. The achalasia in our patient was finally successfully treated by balloon dilatation under fluoroscopic guidance. In conclusion, warning leak can rarely mask an underlying gastrointestinal disorder which can impose a high risk of acute respiratory distress for patients receiving general anesthesia for interventional radiological procedures as well as other types of surgery. Acknowlegment This study was supported by a grant from Korea Healthcare technology R&D Project, Ministry of Health & Welfare, Republic of Korea. (A080201)
References 1 Leblanc R: The minor leak preceding subarachnoid hemorrhage. J Neurosurg 66: 35-39, 1987. 2 Zikk D, Rapoport Y et Al: Acute airway obstruction and achalasia of the esophagus. Ann Otol Rhinol Laryngol 98: 641-643, 1989. 3 Waga S, Otsubo K et Al: Warning signs in intracranial aneurysms. Surg Neurol 3: 15-20, 1975. 4 Torner JC, Kassell NF et Al: Preoperative prognostic factors for rebleeding and survival in aneurysm patients receiving antifibrinolytic therapy: Report of the cooperative aneurysm study. Neurosurgery 9: 506-513, 1981. 5 Wiebers DO: Unruptured intracranial aneurysms: Natural history and clinical management. Update on the international study of unruptured intracranial aneurysms. Neuroimaging Clin N Am 16: 383-390, vii, 2006. 6 Wiebers DO, Whisnant JP et Al: Unruptured intracranial aneurysms: Natural history, clinical outcome, and risks of surgical and endovascular treatment. Lancet 362: 103-110, 2003. 7 Ostergaard JR: Warning leak in subarachnoid haemorrhage. BMJ 301: 190-191, 1990. 8 Vantrappen G, Janssens J et Al: Achalasia, diffuse esophageal spasm, and related motility disorders. Gastroenterology 76: 450-457, 1979.
724
9 Aggestrup S, Uddman R et Al: Lack of vasoactive intestinal polypeptide nerves in esophageal achalasia. Gastroenterology 84: 924-927, 1983. 10 Tottrup A, Svane D et Al: Nitric oxide mediating nanc inhibition in opossum lower esophageal sphincter. Am J Physiol 260: 385-389, 1991.
Dae Chul Suh, MD Department of Radiology and Research Institute of Radiology University of Ulsan, College of Medicine Asan Medical Center 388-1 Pungnap2-dong, Songpa-gu Seoul, 138-736, Korea Tel.: 822-3010-4366 Fax: 822-476-0090 E-mail: [email protected]
