- Email: [email protected]
Arterial spasm following perimesencephalic nonaneurysmal subarachnoid hemorrhage in a pediatric patient
Arterial Spasm Following Perimesencephalic Nonaneurysmal Subarachnoid Hemorrhage in a Pediatric Patient Ahmet Sert, MD*, Kursad Aydin, MD*, Ozgur Pirgon, MD*, Dilek Emlik, MD†, and Mehmet Erkan Ustun, MD‡ Perimesencephalic nonaneurysmal hemorrhage is a benign form of subarachnoid hemorrhages. This entity is well recognized as a distinct type of subarachnoid hemorrhage in adults. However, perimesencephalic nonaneurysmal subarachnoid hemorrhage in pediatric patients is not well recognized. Angiographic changes such as vasospasm are uncommon in patients, especially in pediatric patients suffering from this type of hemorrhage. This case study reports a 12-year-old male who suffered from perimesencephalic nonaneurysmal subarachnoid hemorrhage. Cerebral carotid angiography performed on the tenth day of the posthemorrhagic period revealed severe vasospasm affecting the basilar artery. The patient, treated symptomatically, was discharged after improvement. One year later, magnetic resonance angiography revealed completely normal features. © 2005 by Elsevier Inc. All rights reserved. Sert A, Aydın K, Pirgon O, Emlik D, Ustun ME. Arterial spasm following perimesencephalic nonaneurysmal subarachnoid hemorrhage in a pediatric patient. Pediatr Neurol 2005;32:275-277.
From Departments of *Pediatrics, †Radiology, and ‡Neurosurgery, Selcuk University Meram Medical Faculty, Konya, Turkey.
© 2005 by Elsevier Inc. All rights reserved. doi:10.1016/j.pediatrneurol.2004.09.011 ● 0887-8994/05/$—see front matter
Introduction Atraumatic subarachnoid hemorrhage is an uncommon entity in the pediatric population. In large series, the occurrence in children is within the range of 0.6% to 4.6% [1,2], suggesting an incidence of 1-3 per million [3]. Among 167 children with documented subarachnoid hemorrhage, Hourihan et al. [4] found that 52% were due to ruptured aneurysms, 26% from bleeding arteriovenous malformations, and 19% were idiopathic. If no aneurysm can be identified in a child with a confirmed case of atraumatic subarachnoid hemorrhage, several other intracranial and systemic factors are to be considered [5]. Perimesencephalic nonaneurysmal hemorrhage, defined as blood in the cisterns around the midbrain with a negative four-vessel angiographic study, is uniformly associated with an excellent outcome without associated rebleeding or symptomatic delayed arterial spasm [6]. One of the distinguishing features of perimesencephalic nonaneurysmal hemorrhage is the uncommon occurrence of cerebral vasospasm [7-9]. The occurrence of diffuse and severe vasospasm in pediatric patients has not been reported in the pediatric neurology literature. This report describes a pediatric patient with perimesencephalic nonaneurysmal subarachnoid hemorrhage who developed severe vasospasm.
Case Report A healthy 12-year-old male was admitted to the hospital complaining about intermittent headache that did not respond to paracetamol for the last 2 days. He awoke from sleep on the second day with acute worsening of the headache and the onset of vomiting and neck pain. He was taken to the emergency room. Physical examination revealed lethargy and nuchal rigidity. He responded to voices and performed simple commands. His cranial nerve, sensory, and motor examinations were within the normal limits. Laboratory examinations, including complete blood count, serum electrolytes, liver function tests, lipid profiles, and coagulation tests, were all within normal limits. Cerebral computed tomography disclosed localized acute subarachnoid hemorrhage in the perimesencephalic area (Fig 1). The lesion displayed on cranial computed tomography was hyperintense on axial T1- and T2-weighted magnetic resonance imaging of the brain. Cerebral angiography performed on the tenth day of the posthemorrhagic period indicated severe vasospasm affecting a proximal part of the basilar artery (Fig 2). The patient was treated symptomatically. He improved clinically during his stay at the hospital, and was discharged after 22 days. One year later, the magnetic resonance angiography was completely normal. At the 1-year follow up, he was doing well without any headache and his examination was completely normal.
Communications should be addressed to: ¨ niversitesi Meram Tıp Fakültesi; Pediatrik Dr. Aydin; Selçuk U Nöroloji Bilim Dalı; 42080 Konya, Turkey. Received June 23, 2004; accepted September 27, 2004.
Sert et al: Nonaneurysmal Subarachnoid Hemorrhage in a Child 275
Discussion Perimesencephalic nonaneurysmal hemorrhage is well recognized as a distinct type of subarachnoid hemorrhage [10,11]. Patients with a typical clinical picture of this entity are adults with acute onset of headache without loss of consciousness or focal neurologic symptoms. Neurologic examination may be normal, and often only meningeal irritation can be determined. This entity is initially diagnosed on the basis of computed tomographic examination, demonstrating a localized area of hemorrhage, centered within the perimesencephalic or prepontine cisterns without intracerebral or intraventricular extension, and diagnosis is usually confirmed with a subsequent four-vessel cerebral angiogram [10,12]. Perimesencephalic nonaneurysmal hemorrhage in pediatric patients has been reported rarely. There are only two children, a 3-year-old male and a 4-year-old male, with perimesencephalic nonaneurysmal hemorrhage in the literature [11]. Cerebral vasospasm develops in approximately one half to two thirds of patients with aneurysmal subarachnoid hemorrhage. Angiographic changes compatible with cerebral vasospasm are uncommon, especially in pediatric patients [6]. Diffuse vasospasm, as observed in our patient, has not been previously reported in pediatric patients. Symptomatic cerebral vasospasm is extremely rare, but it is complicated when it occurs [2,12]. The cause of perimesencephalic nonaneurysmal subarachnoid hemorrhage has not been defined, yet a venous or capillary source, a ruptured perforating artery, a pontine capillary telangiectasia, a primary intramural hematoma of the basilar artery, and low-flow vascular malformation have all been proposed [6,13,14]. The clinical and radiographic characteristics of the patient with subarachnoid
Figure 2. Cerebral angiography of patient reveals severe vasospasm of the basilar artery.
hemorrhage described here are consistent with perimesencephalic nonaneurysmal subarachnoid hemorrhage. Cerebral computed tomography demonstrated localized acute subarachnoid hemorrhage in the perimesencephalic area. His cerebral angiography demonstrated severe vasospasm affecting a proximal part of the basilar artery. Furthermore, other intracranial and systemic causes of subarachnoid hemorrhage in the pediatric age group were excluded. Because cerebral angiography carries a 0.4-1.1% risk of neurologic complication in the pediatric population, a second angiogram was not performed in this case. Magnetic resonance angiography was normal. In conclusion, the cause and incidence of perimesencephalic nonaneurysmal subarachnoid hemorrhage in pediatric patients are yet unknown. This report presents the first case of arterial spasm following perimesencephalic nonaneurysmal subarachnoid hemorrhage in a pediatric patient. Due to its benign clinical outcome, perimesencephalic hemorrhage should be considered when a child presents with a subarachnoid hemorrhage in a perimesencephalic distribution on computed tomography and a negative four-vessel cerebral angiogram. When severe vasospasm is observed, a diagnosis of perimesencephalic nonaneurysmal subarachnoid hemorrhage should not be excluded. References
Figure 1. Cerebral axial computed tomographic scan of patient reveals subarachnoid hemorrhage limited within the perimesencephalic area.
276
PEDIATRIC NEUROLOGY
Vol. 32 No. 4
[1] Locksley HB. Natural history of subarachnoid hemorrhage, intracranial aneurysms and arteriovenous malformations. Based on 6368 cases in the cooperative study. J Neurosurg 1966;25:219-39. [2] Pasqualin A, Mazza C, Cavazzani P, Scienza R, DaPian R. Intracranial aneurysms and subarachnoid hemorrhage in children and adolescents. Child Nerv Syst 1986;2:185-90. [3] van Gijn J, Van Dongen KJ, Vermeulen M, Hijdra A. Perimesencephalic hemorrhage: A nonaneurysmal and benign form of subarachnoid hemorrhage. Neurology 1985;35:493-7.
[4] Hourihan MD, Gates PC, McAllister VL. Subarachnoid hemorrhage in childhood and adolescence. J Neurosurg 1984;60:1163-6. [5] Shucart WA, Wolpert SM. Intracranial arterial aneurysms in childhood. Am J Dis Child 1974;127:288-93. [6] Schievink WI, Wijdicks EFM, Spetzler RF. Diffuse vasospasm after pretruncal nonaneurysmal subarachnoid hemorrhage. AJNR Am J Neuroradiol 2000;21:521-3. [7] Wijdicks EFM, Schievink WI. Perimesencephalic nonaneurysmal subarachnoid hemorrhage: First hint of a cause? Neurology 1997; 49:634-6. [8] Schwartz TH, Solomon RA. Perimesencephalic nonaneurysmal subarachnoid hemorrhage: Review of literature. Neurosurgery 1996;39: 433-40. [9] Schievink WI, Wijdicks EFM, Nichols DA, Ebersold MJ. Perimesencephalic subarachnoid hemorrhage: Additional perspectives from four cases. Stroke 1994;25:1507-11.
[10] Rinkel GJ, Wijdicks EFM, Hasan D, et al. Outcome in patients with subarachnoid haemorrhage and negative angiography according to pattern of haemorrhage on computed tomography. Lancet 1991;338: 964-8. [11] Anderson RC, Baskin J, Feldstein NA. Perimesencephalic nonaneurysmal subarachnoid hemorrhage: Case report and review of the literature. Pediatr Neurosurg 2002;37:258-61. [12] Rinkel GJ, Wijdicks EF, Hasan D, Brouwers PJ, van Gijn J. The clinical course of perimesencephalic nonaneurysmal subarachnoid hemorrhage. Ann Neurol 1991;29:463-8. [13] Schievink WI, Wijdicks EFM. Pretruncal subarachnoid hemorrhage: An anatomically correct description of the perimesencephalic subarachnoid hemorrhage. Stroke 1997;28:2572. [14] Pinto AN, Ferro JM, Canhao P, Campos J. How often is a perimesencephalic subarachnoid haemorrhage CT pattern caused by ruptured aneurysms? Acta Neurochir 1993;124:79-81.
Sert et al: Nonaneurysmal Subarachnoid Hemorrhage in a Child 277
From Departments of *Pediatrics, †Radiology, and ‡Neurosurgery, Selcuk University Meram Medical Faculty, Konya, Turkey.
© 2005 by Elsevier Inc. All rights reserved. doi:10.1016/j.pediatrneurol.2004.09.011 ● 0887-8994/05/$—see front matter
Introduction Atraumatic subarachnoid hemorrhage is an uncommon entity in the pediatric population. In large series, the occurrence in children is within the range of 0.6% to 4.6% [1,2], suggesting an incidence of 1-3 per million [3]. Among 167 children with documented subarachnoid hemorrhage, Hourihan et al. [4] found that 52% were due to ruptured aneurysms, 26% from bleeding arteriovenous malformations, and 19% were idiopathic. If no aneurysm can be identified in a child with a confirmed case of atraumatic subarachnoid hemorrhage, several other intracranial and systemic factors are to be considered [5]. Perimesencephalic nonaneurysmal hemorrhage, defined as blood in the cisterns around the midbrain with a negative four-vessel angiographic study, is uniformly associated with an excellent outcome without associated rebleeding or symptomatic delayed arterial spasm [6]. One of the distinguishing features of perimesencephalic nonaneurysmal hemorrhage is the uncommon occurrence of cerebral vasospasm [7-9]. The occurrence of diffuse and severe vasospasm in pediatric patients has not been reported in the pediatric neurology literature. This report describes a pediatric patient with perimesencephalic nonaneurysmal subarachnoid hemorrhage who developed severe vasospasm.
Case Report A healthy 12-year-old male was admitted to the hospital complaining about intermittent headache that did not respond to paracetamol for the last 2 days. He awoke from sleep on the second day with acute worsening of the headache and the onset of vomiting and neck pain. He was taken to the emergency room. Physical examination revealed lethargy and nuchal rigidity. He responded to voices and performed simple commands. His cranial nerve, sensory, and motor examinations were within the normal limits. Laboratory examinations, including complete blood count, serum electrolytes, liver function tests, lipid profiles, and coagulation tests, were all within normal limits. Cerebral computed tomography disclosed localized acute subarachnoid hemorrhage in the perimesencephalic area (Fig 1). The lesion displayed on cranial computed tomography was hyperintense on axial T1- and T2-weighted magnetic resonance imaging of the brain. Cerebral angiography performed on the tenth day of the posthemorrhagic period indicated severe vasospasm affecting a proximal part of the basilar artery (Fig 2). The patient was treated symptomatically. He improved clinically during his stay at the hospital, and was discharged after 22 days. One year later, the magnetic resonance angiography was completely normal. At the 1-year follow up, he was doing well without any headache and his examination was completely normal.
Communications should be addressed to: ¨ niversitesi Meram Tıp Fakültesi; Pediatrik Dr. Aydin; Selçuk U Nöroloji Bilim Dalı; 42080 Konya, Turkey. Received June 23, 2004; accepted September 27, 2004.
Sert et al: Nonaneurysmal Subarachnoid Hemorrhage in a Child 275
Discussion Perimesencephalic nonaneurysmal hemorrhage is well recognized as a distinct type of subarachnoid hemorrhage [10,11]. Patients with a typical clinical picture of this entity are adults with acute onset of headache without loss of consciousness or focal neurologic symptoms. Neurologic examination may be normal, and often only meningeal irritation can be determined. This entity is initially diagnosed on the basis of computed tomographic examination, demonstrating a localized area of hemorrhage, centered within the perimesencephalic or prepontine cisterns without intracerebral or intraventricular extension, and diagnosis is usually confirmed with a subsequent four-vessel cerebral angiogram [10,12]. Perimesencephalic nonaneurysmal hemorrhage in pediatric patients has been reported rarely. There are only two children, a 3-year-old male and a 4-year-old male, with perimesencephalic nonaneurysmal hemorrhage in the literature [11]. Cerebral vasospasm develops in approximately one half to two thirds of patients with aneurysmal subarachnoid hemorrhage. Angiographic changes compatible with cerebral vasospasm are uncommon, especially in pediatric patients [6]. Diffuse vasospasm, as observed in our patient, has not been previously reported in pediatric patients. Symptomatic cerebral vasospasm is extremely rare, but it is complicated when it occurs [2,12]. The cause of perimesencephalic nonaneurysmal subarachnoid hemorrhage has not been defined, yet a venous or capillary source, a ruptured perforating artery, a pontine capillary telangiectasia, a primary intramural hematoma of the basilar artery, and low-flow vascular malformation have all been proposed [6,13,14]. The clinical and radiographic characteristics of the patient with subarachnoid
Figure 2. Cerebral angiography of patient reveals severe vasospasm of the basilar artery.
hemorrhage described here are consistent with perimesencephalic nonaneurysmal subarachnoid hemorrhage. Cerebral computed tomography demonstrated localized acute subarachnoid hemorrhage in the perimesencephalic area. His cerebral angiography demonstrated severe vasospasm affecting a proximal part of the basilar artery. Furthermore, other intracranial and systemic causes of subarachnoid hemorrhage in the pediatric age group were excluded. Because cerebral angiography carries a 0.4-1.1% risk of neurologic complication in the pediatric population, a second angiogram was not performed in this case. Magnetic resonance angiography was normal. In conclusion, the cause and incidence of perimesencephalic nonaneurysmal subarachnoid hemorrhage in pediatric patients are yet unknown. This report presents the first case of arterial spasm following perimesencephalic nonaneurysmal subarachnoid hemorrhage in a pediatric patient. Due to its benign clinical outcome, perimesencephalic hemorrhage should be considered when a child presents with a subarachnoid hemorrhage in a perimesencephalic distribution on computed tomography and a negative four-vessel cerebral angiogram. When severe vasospasm is observed, a diagnosis of perimesencephalic nonaneurysmal subarachnoid hemorrhage should not be excluded. References
Figure 1. Cerebral axial computed tomographic scan of patient reveals subarachnoid hemorrhage limited within the perimesencephalic area.
276
PEDIATRIC NEUROLOGY
Vol. 32 No. 4
[1] Locksley HB. Natural history of subarachnoid hemorrhage, intracranial aneurysms and arteriovenous malformations. Based on 6368 cases in the cooperative study. J Neurosurg 1966;25:219-39. [2] Pasqualin A, Mazza C, Cavazzani P, Scienza R, DaPian R. Intracranial aneurysms and subarachnoid hemorrhage in children and adolescents. Child Nerv Syst 1986;2:185-90. [3] van Gijn J, Van Dongen KJ, Vermeulen M, Hijdra A. Perimesencephalic hemorrhage: A nonaneurysmal and benign form of subarachnoid hemorrhage. Neurology 1985;35:493-7.
[4] Hourihan MD, Gates PC, McAllister VL. Subarachnoid hemorrhage in childhood and adolescence. J Neurosurg 1984;60:1163-6. [5] Shucart WA, Wolpert SM. Intracranial arterial aneurysms in childhood. Am J Dis Child 1974;127:288-93. [6] Schievink WI, Wijdicks EFM, Spetzler RF. Diffuse vasospasm after pretruncal nonaneurysmal subarachnoid hemorrhage. AJNR Am J Neuroradiol 2000;21:521-3. [7] Wijdicks EFM, Schievink WI. Perimesencephalic nonaneurysmal subarachnoid hemorrhage: First hint of a cause? Neurology 1997; 49:634-6. [8] Schwartz TH, Solomon RA. Perimesencephalic nonaneurysmal subarachnoid hemorrhage: Review of literature. Neurosurgery 1996;39: 433-40. [9] Schievink WI, Wijdicks EFM, Nichols DA, Ebersold MJ. Perimesencephalic subarachnoid hemorrhage: Additional perspectives from four cases. Stroke 1994;25:1507-11.
[10] Rinkel GJ, Wijdicks EFM, Hasan D, et al. Outcome in patients with subarachnoid haemorrhage and negative angiography according to pattern of haemorrhage on computed tomography. Lancet 1991;338: 964-8. [11] Anderson RC, Baskin J, Feldstein NA. Perimesencephalic nonaneurysmal subarachnoid hemorrhage: Case report and review of the literature. Pediatr Neurosurg 2002;37:258-61. [12] Rinkel GJ, Wijdicks EF, Hasan D, Brouwers PJ, van Gijn J. The clinical course of perimesencephalic nonaneurysmal subarachnoid hemorrhage. Ann Neurol 1991;29:463-8. [13] Schievink WI, Wijdicks EFM. Pretruncal subarachnoid hemorrhage: An anatomically correct description of the perimesencephalic subarachnoid hemorrhage. Stroke 1997;28:2572. [14] Pinto AN, Ferro JM, Canhao P, Campos J. How often is a perimesencephalic subarachnoid haemorrhage CT pattern caused by ruptured aneurysms? Acta Neurochir 1993;124:79-81.
Sert et al: Nonaneurysmal Subarachnoid Hemorrhage in a Child 277