- Email: [email protected]
Transient ischemic attack post craniectomy: A case report and review of sinking skin flap syndrome
Interdisciplinary Neurosurgery 20 (2020) 100640
Contents lists available at ScienceDirect
Interdisciplinary Neurosurgery journal homepage: www.elsevier.com/locate/inat
Case Reports & Case Series
Transient ischemic attack post craniectomy: A case report and review of sinking skin flap syndrome
T
⁎
Zhihui Denga,b, , John Davisb a b
Division of Physical Medicine and Rehabilitation, Department of Medicine, McMaster University, Hamilton, Canada Hamilton Health Sciences Regional Rehabilitation Centre, Hamilton, Canada
A R T I C LE I N FO
A B S T R A C T
Keywords: Decompressive craniectomy Sinking skin flap syndrome Transient ischemia attack Cranioplasty
There is evidence of disrupted cerebral blood flow in patients with a skull defect due to decompressive craniectomy though the pathophysiologic mechanism is yet to be established. Here we report on a patient who suffered one episode of transient ischemic attack (TIA) when developing early stage sinking skin flap syndrome (SSFS) post decompressive craniectomy due to a severe traumatic brain injury. This case report of TIA as a clinical presentation of SSFS indicates a potentially increased risk of stroke in patients with this condition. The theories of the pathophysiology underlining neurological manifestations post craniectomy are reviewed. Early cranioplasty is recommended, particularly for those with other known stroke risk factors.
1. Introduction Decompressive craniectomy (DC) is performed for neurological emergency to release malignant intracranial hypertension that is refractory of medical management. Its substantial effects of reducing mortality have been well demonstrated in the setting of traumatic brain injury [1], ischemic stroke [2], subarachnoid hemorrhage [3], cerebral venous sinus thrombosis [4], and severe intracranial infectious and/or inflammatory diseases [5]. However, the complications of DC in some patients during their later stage of recovery have been identified, especially sinking skin flap syndrome (SSFS), which adversely affects their functional rehabilitation. Here we report one patient who developed early stage SSFS after DC and presented with one transient ischemic attack (TIA) episode 3 weeks following his initial traumatic brain injury. The theories of pathophysiology of SSFS are reviewed. 2. Case report One 51-year old man sustained a traumatic brain injury (TBI) due to an assault, then underwent a surgical procedure of right frontotemporal parietal craniectomy, with evacuation of acute subdural hematoma (SDH). Postoperative head CT scan showed interval improvement in the mass effect of SDH on brain parenchyma with leftward midline shift. 18 days post his injury, he was transferred to our Acquired Brain Injury (ABI) Rehabilitation Unit when he was medically stable. The skin flap at his craniectomy site was somewhat concave, and he presented with
⁎
some higher-level cognitive impairments and weakness in his left arm (MRC 4/5), while ambulating and performing ADLs independently. Previous medical history was significant for type 2 diabetes mellitus requiring insulin Lantus 20 units, metformin 1000 mg BID, and Linagliptin 5 mg daily. During his inpatient treatment on our unit, there was some fluctuation of blood glucose due to his inconsistent compliance with diet. He also had dyslipidemia, for which he took rosuvastatin 10 mg daily. Because of his history of cigarette smoking, he used a nicotine patch 7 mg daily. Due to his recent brain injury, Dalteparin 5000 units was given for VTE prophylaxis. He did not have any pre-TBI history of stroke or TIA. 2 days after admission to the ABI unit, he experienced a TIA-like episode after walking for 15–20 min off unit with his wife. At that time, he developed left-sided facial droop and slurred speech lasting for less than 5 min, and worsened left arm weakness lasting for about 1 h. Meanwhile, he experienced headache and fatigue lasting for a few hours. Urgent head CT scan was performed which, however, did not reveal new pathology, but only demonstrated findings of early stage sinking skin flap syndrome (Fig. 1A–D). The subsequent neurological workup for TIA, including normal Duplex carotid vertebral ultrasound, was unremarkable. The neurosurgery service subsequently replaced his bone flap. Within two days post cranioplasty, his left-side weakness and cognitive impairment started to improve significantly compared to his baseline functioning prior to the surgery. His head CT scan 3 weeks post-cranioplasty showed overall improvement in mass effect on the frontal lobe with near complete resolution of midline shift (Fig. 1E–H).
Corresponding author at: Division of Physical Medicine and Rehabilitation, Department of Medicine, McMaster University, Hamilton, Canada. E-mail address: [email protected] (Z. Deng).
https://doi.org/10.1016/j.inat.2019.100640 Received 16 June 2019; Received in revised form 25 November 2019; Accepted 29 November 2019 2214-7519/ © 2019 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Interdisciplinary Neurosurgery 20 (2020) 100640
Z. Deng and J. Davis
Fig. 1. Head CT scan before (A-D) and after (E-H) cranioplasty A-D: Head CT scan right during/after TIA-like episode. Evidence of early sunken flap syndrome: concavity of the right bone flap with mass effect on the subjacent brain parenchyma with right subfalcine, uncal, descending transtentorial herniation, and leftward midline shift measuring 11 mm E-H: Head CT scan 3 weeks post cranioplasty. Overall improvement in mass effect on the frontal lobe, with near complete resolution of midline shift and right lateral ventricular effacement.
3. Discussion
cause is intrinsic cerebral arterial changes, such as atherosclerosis, lipohyalinosis, inflammation and arterial dissection. Moreover, embolus originating remotely from heart or extracranial vessels may travel to cerebral vessels leading to ischemia. In addition, inadequate perfusion pressure or elevated blood viscosity can reduce cerebral blood flow causing TIA and stroke. The alteration of cerebral blood flow and metabolic changes following DC has been demonstrated by imaging studies, including xenon CT and MR spectroscopy [13], transcranial Doppler (TCD) ultrasonography, positron emission tomography [12] and CT perfusion [14]. The mechanism underlying these changes is yet to be clarified. However, it appears likely that changes in blood flow with a skull defect contributed, at least partially, to the transient ischemic episode in this patient. Other contributing factors may have included vascular and circulation changes related to his history of diabetes, dyslipidemia and smoking, though post-TIA workup was unremarkable. This case demonstrates an increased risk of TIA and stroke in patients with SSFS, especially in those with other stroke risk factors.
As a lifesaving measure, a large craniectomy is often required in the setting of intractable intracranial hypertension. Following this surgical procedure, the skin flap over the craniectomy site gradually relaxes and may begin to sink as cerebral edema resolves. A subpopulation of patients with this type of skull defect experience worse than expected neurological dysfunction and impairment hindering later course of rehabilitation. For example, Cooper and colleagues reported that DC for severe TBI is associated with unfavorable outcome compared with severe TBI patients managed conservatively [6]. The term “syndrome of the trephined” was coined by Grant in 1939 to describe a number of neurological symptoms attributable to craniectomy, such as headaches, vertigo, seizures, memory and behavioral disturbance [7]. In 1977 the term “sinking skin flap syndrome” was used to describe a sinking skin flap over the craniectomy site with secondary neurological deterioration developing weeks or months following craniectomy. The diagnosis can be confirmed by clinical improvement following cranioplasty [8]. There are totally less than 100 reported SSFS cases in literature [9]. Consistent with previously described SSFS, the patient in this case report developed concavity of the skin flap approximately two weeks post DC, with an upright position precipitating symptoms characteristic of SSFS [10]. His head CT scan demonstrated the features of early SSFS. Within a couple days post cranioplasty, he experienced significant improvement of his multiple neurological deficits including headache, left arm weakness and cognitive impairment, confirming the diagnosis of SSFS [8,10,11]. Different theories have been proposed for the pathophysiology of SSFS, though the exact mechanism is yet to be established. One hypothesis is that the direct effect of atmospheric pressure causes compression of brain tissue via the skin flap due to the lack of skull protection [8], leading in turn to neurological symptoms. Another theory is that DC causes a change in CSF hydrodynamics, which has a deleterious effect [11]. In addition, some studies demonstrate disturbance of cerebral blood flow regulation with associated cerebral metabolism change following DC [12]. Any pathophysiological processes leading to a disturbance of blood supply to brain tissue can cause stroke and TIA. The most common
4. Conclusion The patient in this study developed one episode of TIA associated with SSFS post decompressive craniectomy. In addition to the existing evidence of an unfavorable outcome post craniectomy in some patients, this case serves as an evidence of potentially increased risk of TIA and stroke in the setting of SSFS due to a skull defect. For the sake of stroke prevention, early cranioplasty should be taken into consideration when patients have developed signs of SSFS with clinical deterioration, especially for those having other known stroke risk factors. Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper. Acknowledgement No funding was received for this study or the publication of this 2
Interdisciplinary Neurosurgery 20 (2020) 100640
Z. Deng and J. Davis
report. All named authors meet the International Committee of Medical Journal Editors (ICMJE) criteria for authorship of this manuscript, take responsibility for the integrity of the work as a whole, and have given their final approval of the version to be published. We thank the patient in the study and all the team members who provided care for him on the Acquired Brain Injury Rehabilitation Unit.
[6] [7] [8]
[9]
References
[10] [1] R.S. Polin, et al., Decompressive bifrontal craniectomy in the treatment of severe refractory posttraumatic cerebral edema, Neurosurgery 41 (1) (1997) 84–92 discussion 92-4. [2] K. Vahedi, et al., Early decompressive surgery in malignant infarction of the middle cerebral artery: a pooled analysis of three randomised controlled trials, Lancet Neurol. 6 (3) (2007) 215–222. [3] E. Güresir, et al., Decompressive craniectomy in subarachnoid hemorrhage, Neurosurg Focus 26 (6) (2009) E4. [4] J.M. Ferro, et al., Second International Study on Cerebral Vein and Dural Sinus Thrombosis (ISCVT 2) Investigators. Decompressive surgery in cerebrovenous thrombosis: a multicenter registry and a systematic review of individual patient data, Stroke 42 (10) (2011) 2825–2831. [5] A. Di Rienzo, et al., Decompressive craniectomy for medically refractory
[11]
[12]
[13]
[14]
3
intracranial hypertension due to meningoencephalitis: report of three patients, Acta Neurochir. (Wien) 150 (10) (2008) 1057–1065 discussion 1065. D.J. Cooper, et al., Decompressive craniectomy in diffuse traumatic brain injury, N. Engl. J. Med. 364 (16) (2011) 1493–1502. F.C. Grant, N.C. Norcross, Repair of cranial defects by cranioplasty, Ann. Surg. 110 (4) (1939) 488–512. A. Yamaura, H. Makino, Neurological deficits in the presence of the sinking skin flap following decompressive craniectomy, Neurol. Med. Chir. (Tokyo) 17 (1 Pt 1) (1977) 43–53. M. Annan, et al., Sinking skin flap syndrome (or Syndrome of the trephined): a review, Br. J. Neurosurg. 29 (3) (2015) 314–318. M. Schorl, Sinking skin flap syndrome (SSFS) – clinical spectrum and impact on rehabilitation, Cent. Eur. Neurosurg. 70 (2) (2009) 68–72. H. Fodstad, et al., Effect of cranioplasty on cerebrospinal fluid hydrodynamics in patients with the syndrome of the trephined, Acta Neurochir. (Wien). 70 (1–2) (1984) 21–30. P.A. Winkler, et al., Influence of cranioplasty on postural blood flow regulation, cerebrovascular reserve capacity, and cerebral glucose metabolism, J. Neurosurg. 93 (1) (2000) 53–61. K. Yoshida, et al., Dynamics of cerebral blood flow and metabolism in patients with cranioplasty as evaluated by 133Xe CT and 31P magnetic resonance spectroscopy, J. Neurol. Neurosurg. Psychiatry 61 (2) (1996) 166–171. S. Sakamoto, et al., CT perfusion imaging in the syndrome of the sinking skin flap before and after cranioplasty, Clin. Neurol. Neurosurg. 108 (6) (2006) 583–585.
Contents lists available at ScienceDirect
Interdisciplinary Neurosurgery journal homepage: www.elsevier.com/locate/inat
Case Reports & Case Series
Transient ischemic attack post craniectomy: A case report and review of sinking skin flap syndrome
T
⁎
Zhihui Denga,b, , John Davisb a b
Division of Physical Medicine and Rehabilitation, Department of Medicine, McMaster University, Hamilton, Canada Hamilton Health Sciences Regional Rehabilitation Centre, Hamilton, Canada
A R T I C LE I N FO
A B S T R A C T
Keywords: Decompressive craniectomy Sinking skin flap syndrome Transient ischemia attack Cranioplasty
There is evidence of disrupted cerebral blood flow in patients with a skull defect due to decompressive craniectomy though the pathophysiologic mechanism is yet to be established. Here we report on a patient who suffered one episode of transient ischemic attack (TIA) when developing early stage sinking skin flap syndrome (SSFS) post decompressive craniectomy due to a severe traumatic brain injury. This case report of TIA as a clinical presentation of SSFS indicates a potentially increased risk of stroke in patients with this condition. The theories of the pathophysiology underlining neurological manifestations post craniectomy are reviewed. Early cranioplasty is recommended, particularly for those with other known stroke risk factors.
1. Introduction Decompressive craniectomy (DC) is performed for neurological emergency to release malignant intracranial hypertension that is refractory of medical management. Its substantial effects of reducing mortality have been well demonstrated in the setting of traumatic brain injury [1], ischemic stroke [2], subarachnoid hemorrhage [3], cerebral venous sinus thrombosis [4], and severe intracranial infectious and/or inflammatory diseases [5]. However, the complications of DC in some patients during their later stage of recovery have been identified, especially sinking skin flap syndrome (SSFS), which adversely affects their functional rehabilitation. Here we report one patient who developed early stage SSFS after DC and presented with one transient ischemic attack (TIA) episode 3 weeks following his initial traumatic brain injury. The theories of pathophysiology of SSFS are reviewed. 2. Case report One 51-year old man sustained a traumatic brain injury (TBI) due to an assault, then underwent a surgical procedure of right frontotemporal parietal craniectomy, with evacuation of acute subdural hematoma (SDH). Postoperative head CT scan showed interval improvement in the mass effect of SDH on brain parenchyma with leftward midline shift. 18 days post his injury, he was transferred to our Acquired Brain Injury (ABI) Rehabilitation Unit when he was medically stable. The skin flap at his craniectomy site was somewhat concave, and he presented with
⁎
some higher-level cognitive impairments and weakness in his left arm (MRC 4/5), while ambulating and performing ADLs independently. Previous medical history was significant for type 2 diabetes mellitus requiring insulin Lantus 20 units, metformin 1000 mg BID, and Linagliptin 5 mg daily. During his inpatient treatment on our unit, there was some fluctuation of blood glucose due to his inconsistent compliance with diet. He also had dyslipidemia, for which he took rosuvastatin 10 mg daily. Because of his history of cigarette smoking, he used a nicotine patch 7 mg daily. Due to his recent brain injury, Dalteparin 5000 units was given for VTE prophylaxis. He did not have any pre-TBI history of stroke or TIA. 2 days after admission to the ABI unit, he experienced a TIA-like episode after walking for 15–20 min off unit with his wife. At that time, he developed left-sided facial droop and slurred speech lasting for less than 5 min, and worsened left arm weakness lasting for about 1 h. Meanwhile, he experienced headache and fatigue lasting for a few hours. Urgent head CT scan was performed which, however, did not reveal new pathology, but only demonstrated findings of early stage sinking skin flap syndrome (Fig. 1A–D). The subsequent neurological workup for TIA, including normal Duplex carotid vertebral ultrasound, was unremarkable. The neurosurgery service subsequently replaced his bone flap. Within two days post cranioplasty, his left-side weakness and cognitive impairment started to improve significantly compared to his baseline functioning prior to the surgery. His head CT scan 3 weeks post-cranioplasty showed overall improvement in mass effect on the frontal lobe with near complete resolution of midline shift (Fig. 1E–H).
Corresponding author at: Division of Physical Medicine and Rehabilitation, Department of Medicine, McMaster University, Hamilton, Canada. E-mail address: [email protected] (Z. Deng).
https://doi.org/10.1016/j.inat.2019.100640 Received 16 June 2019; Received in revised form 25 November 2019; Accepted 29 November 2019 2214-7519/ © 2019 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Interdisciplinary Neurosurgery 20 (2020) 100640
Z. Deng and J. Davis
Fig. 1. Head CT scan before (A-D) and after (E-H) cranioplasty A-D: Head CT scan right during/after TIA-like episode. Evidence of early sunken flap syndrome: concavity of the right bone flap with mass effect on the subjacent brain parenchyma with right subfalcine, uncal, descending transtentorial herniation, and leftward midline shift measuring 11 mm E-H: Head CT scan 3 weeks post cranioplasty. Overall improvement in mass effect on the frontal lobe, with near complete resolution of midline shift and right lateral ventricular effacement.
3. Discussion
cause is intrinsic cerebral arterial changes, such as atherosclerosis, lipohyalinosis, inflammation and arterial dissection. Moreover, embolus originating remotely from heart or extracranial vessels may travel to cerebral vessels leading to ischemia. In addition, inadequate perfusion pressure or elevated blood viscosity can reduce cerebral blood flow causing TIA and stroke. The alteration of cerebral blood flow and metabolic changes following DC has been demonstrated by imaging studies, including xenon CT and MR spectroscopy [13], transcranial Doppler (TCD) ultrasonography, positron emission tomography [12] and CT perfusion [14]. The mechanism underlying these changes is yet to be clarified. However, it appears likely that changes in blood flow with a skull defect contributed, at least partially, to the transient ischemic episode in this patient. Other contributing factors may have included vascular and circulation changes related to his history of diabetes, dyslipidemia and smoking, though post-TIA workup was unremarkable. This case demonstrates an increased risk of TIA and stroke in patients with SSFS, especially in those with other stroke risk factors.
As a lifesaving measure, a large craniectomy is often required in the setting of intractable intracranial hypertension. Following this surgical procedure, the skin flap over the craniectomy site gradually relaxes and may begin to sink as cerebral edema resolves. A subpopulation of patients with this type of skull defect experience worse than expected neurological dysfunction and impairment hindering later course of rehabilitation. For example, Cooper and colleagues reported that DC for severe TBI is associated with unfavorable outcome compared with severe TBI patients managed conservatively [6]. The term “syndrome of the trephined” was coined by Grant in 1939 to describe a number of neurological symptoms attributable to craniectomy, such as headaches, vertigo, seizures, memory and behavioral disturbance [7]. In 1977 the term “sinking skin flap syndrome” was used to describe a sinking skin flap over the craniectomy site with secondary neurological deterioration developing weeks or months following craniectomy. The diagnosis can be confirmed by clinical improvement following cranioplasty [8]. There are totally less than 100 reported SSFS cases in literature [9]. Consistent with previously described SSFS, the patient in this case report developed concavity of the skin flap approximately two weeks post DC, with an upright position precipitating symptoms characteristic of SSFS [10]. His head CT scan demonstrated the features of early SSFS. Within a couple days post cranioplasty, he experienced significant improvement of his multiple neurological deficits including headache, left arm weakness and cognitive impairment, confirming the diagnosis of SSFS [8,10,11]. Different theories have been proposed for the pathophysiology of SSFS, though the exact mechanism is yet to be established. One hypothesis is that the direct effect of atmospheric pressure causes compression of brain tissue via the skin flap due to the lack of skull protection [8], leading in turn to neurological symptoms. Another theory is that DC causes a change in CSF hydrodynamics, which has a deleterious effect [11]. In addition, some studies demonstrate disturbance of cerebral blood flow regulation with associated cerebral metabolism change following DC [12]. Any pathophysiological processes leading to a disturbance of blood supply to brain tissue can cause stroke and TIA. The most common
4. Conclusion The patient in this study developed one episode of TIA associated with SSFS post decompressive craniectomy. In addition to the existing evidence of an unfavorable outcome post craniectomy in some patients, this case serves as an evidence of potentially increased risk of TIA and stroke in the setting of SSFS due to a skull defect. For the sake of stroke prevention, early cranioplasty should be taken into consideration when patients have developed signs of SSFS with clinical deterioration, especially for those having other known stroke risk factors. Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper. Acknowledgement No funding was received for this study or the publication of this 2
Interdisciplinary Neurosurgery 20 (2020) 100640
Z. Deng and J. Davis
report. All named authors meet the International Committee of Medical Journal Editors (ICMJE) criteria for authorship of this manuscript, take responsibility for the integrity of the work as a whole, and have given their final approval of the version to be published. We thank the patient in the study and all the team members who provided care for him on the Acquired Brain Injury Rehabilitation Unit.
[6] [7] [8]
[9]
References
[10] [1] R.S. Polin, et al., Decompressive bifrontal craniectomy in the treatment of severe refractory posttraumatic cerebral edema, Neurosurgery 41 (1) (1997) 84–92 discussion 92-4. [2] K. Vahedi, et al., Early decompressive surgery in malignant infarction of the middle cerebral artery: a pooled analysis of three randomised controlled trials, Lancet Neurol. 6 (3) (2007) 215–222. [3] E. Güresir, et al., Decompressive craniectomy in subarachnoid hemorrhage, Neurosurg Focus 26 (6) (2009) E4. [4] J.M. Ferro, et al., Second International Study on Cerebral Vein and Dural Sinus Thrombosis (ISCVT 2) Investigators. Decompressive surgery in cerebrovenous thrombosis: a multicenter registry and a systematic review of individual patient data, Stroke 42 (10) (2011) 2825–2831. [5] A. Di Rienzo, et al., Decompressive craniectomy for medically refractory
[11]
[12]
[13]
[14]
3
intracranial hypertension due to meningoencephalitis: report of three patients, Acta Neurochir. (Wien) 150 (10) (2008) 1057–1065 discussion 1065. D.J. Cooper, et al., Decompressive craniectomy in diffuse traumatic brain injury, N. Engl. J. Med. 364 (16) (2011) 1493–1502. F.C. Grant, N.C. Norcross, Repair of cranial defects by cranioplasty, Ann. Surg. 110 (4) (1939) 488–512. A. Yamaura, H. Makino, Neurological deficits in the presence of the sinking skin flap following decompressive craniectomy, Neurol. Med. Chir. (Tokyo) 17 (1 Pt 1) (1977) 43–53. M. Annan, et al., Sinking skin flap syndrome (or Syndrome of the trephined): a review, Br. J. Neurosurg. 29 (3) (2015) 314–318. M. Schorl, Sinking skin flap syndrome (SSFS) – clinical spectrum and impact on rehabilitation, Cent. Eur. Neurosurg. 70 (2) (2009) 68–72. H. Fodstad, et al., Effect of cranioplasty on cerebrospinal fluid hydrodynamics in patients with the syndrome of the trephined, Acta Neurochir. (Wien). 70 (1–2) (1984) 21–30. P.A. Winkler, et al., Influence of cranioplasty on postural blood flow regulation, cerebrovascular reserve capacity, and cerebral glucose metabolism, J. Neurosurg. 93 (1) (2000) 53–61. K. Yoshida, et al., Dynamics of cerebral blood flow and metabolism in patients with cranioplasty as evaluated by 133Xe CT and 31P magnetic resonance spectroscopy, J. Neurol. Neurosurg. Psychiatry 61 (2) (1996) 166–171. S. Sakamoto, et al., CT perfusion imaging in the syndrome of the sinking skin flap before and after cranioplasty, Clin. Neurol. Neurosurg. 108 (6) (2006) 583–585.