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DOI: 10.1055/s-0045-1809639
Cranioplasty as a Catalyst for Neurofunctional and Emotional Restoration in Severe Brain Injury: A Case Report Analysis
Cranioplastia como catalisador para restauração neurofuncional e emocional em lesão cerebral grave: Análise de relato de casoFunding The authors received no financial support for this article's research, authorship, or publication.
Abstract
Cranioplasty, frequently utilizing autologous bone grafts, is a critical surgical intervention targeting the management of “syndrome of the trephined” in patients who have previously undergone craniectomy. This syndrome manifests as a spectrum of clinical symptoms, including persistent headaches, cognitive impairments, motor deficits, and diminished neurological function, which are primarily associated with compromised cranial vault integrity and resultant disturbances in intracranial pressure homeostasis and cerebrospinal fluid dynamics. Cranioplasty restores skull protection and intracranial pressure balance, which has been shown to contribute significantly to neurological improvements, including the reduction of neuropsychological deficits, better control of seizure activity, and partial mitigation of cerebral atrophy. This procedure thus plays a crucial role in enhancing cognitive and functional recovery in post-craniectomy patients. This report details the neuropsychological and functional assessments conducted pre- and post-operatively on a 27-year-old male patient who underwent cranioplasty after a prior craniectomy, which was necessitated by a subdural hemorrhage caused by an explosive incident, resulting in significant shrapnel wounds.
Resumo
A cranioplastia, frequentemente utilizando enxertos ósseos autólogos, é uma intervenção cirúrgica crítica que visa o tratamento da "síndrome do trefinado" em pacientes previamente submetidos a craniectomia. Essa síndrome se manifesta como um espectro de sintomas clínicos, incluindo cefaleias persistentes, comprometimentos cognitivos, déficits motores e diminuição da função neurológica, que estão principalmente associados ao comprometimento da integridade da calota craniana e aos consequentes distúrbios na homeostase da pressão intracraniana e na dinâmica do líquido cefalorraquidiano. A cranioplastia restaura a proteção craniana e o equilíbrio da pressão intracraniana, o que demonstrou contribuir significativamente para melhorias neurológicas, incluindo a redução de déficits neuropsicológicos, melhor controle da atividade convulsiva e mitigação parcial da atrofia cerebral. Portanto, esse procedimento desempenha um papel crucial no aprimoramento da recuperação cognitiva e funcional em pacientes pós-craniectomia. Este relatório detalha as avaliações neuropsicológicas e funcionais realizadas pré e pós-operatórias em um paciente do sexo masculino de 27 anos, submetido a cranioplastia após uma craniectomia prévia, necessária devido a uma hemorragia subdural causada por um incidente explosivo, resultando em ferimentos significativos por estilhaços.
Keywords
cranioplasty - syndrome of the trephined - neuropsychological recovery - intracranial pressure - craniectomy - brain injuryPalavras-chave
cranioplastia - síndrome do trepanado - recuperação neuropsicológica - pressão intracraniana - craniectomia - lesão cerebralIntroduction
Decompressive Craniectomy is a surgical procedure that has developed significantly over the centuries and still holds an important place in contemporary neurosurgery. It is considered a life-saving option for addressing elevated intracranial pressure (ICP),[1] The primary objective of craniectomy is to excise a section of the cranial bone (“bone flap”) and open the dura mater to create space, thereby preventing secondary parenchymal damage and brain herniation. This intervention aims to reduce ischemic complications while enhancing cerebral blood flow, perfusion, and compliance. Indications for craniectomy include Traumatic Brain Injury (TBI), Middle Cerebral Artery (MCA) infarction, and Acute Subdural Hematoma (ASDH).[2] Following decompressive craniectomy, cranioplasty is typically performed either for aesthetic enhancement or to provide protection against the “syndrome of the trephined,” which refers to neurological decline that may occur after the initial procedure.[3] Syndrome of the trephined (SoT) is a severe complication following decompressive craniectomy resulting in neurological decline which can progress to aphasia and catatonia.[4] To differentiate neurologically between the periods of craniectomy and cranioplasty, neuropsychological assessment serves as the most effective method for evaluating the nature, severity, and characteristics of cognitive complaints.[5] Neuropsychological assessments are frequently conducted to provide additional information about a variety of developmental disorders.[6]
Case Report
Patient Overview: A 27-year-old male was admitted to the Intensive Care Unit (ICU) after sustaining multiple injuries due to an airstrike directed toward his home by the occupation military, resulting in extensive shrapnel wounds. The most severe injury involved head trauma from the shrapnel, leading to a critical brain injury with left-side subarachnoid hemorrhage.
Initial Treatment: Upon admission, the medical team stabilized the patient and performed an emergency left-sided craniectomy to alleviate intracranial pressure.
Postoperatively: Patient regained full consciousness and orientation. A neurological assessment was conducted, indicating that the patient experienced a slight loss of his senses of smell and taste, as well as emotional blunting. Furthermore, it was notable that, despite knowing of the deaths of his two younger brothers in the same crisis, he exhibited no visible signs of sadness or emotional reaction.
Follow-up Procedure: Forty days after the initial surgery, the patient returned to the hospital for a scheduled cranioplasty, which was completed on the same day without complications. A subsequent neurological assessment performed after the second surgery showed that his sensory and emotional functions returned to normal. After the cranioplasty, the patient's emotional state changed significantly. He became visibly affected by the loss of his brothers, expressing his grief through tears and verbalizing regret over their deaths. Notably, his sense of smell and taste also returned to equilibrium.
Discussion
In this case, we observed that the complications following the craniectomy procedure were alleviated through the subsequent cranioplasty. This provides substantial evidence that sensory and emotional deficits arising from head trauma may be temporary and that these issues are amenable to treatment and improvement.
Traumatic brain injury (TBI) is the leading cause of subarachnoid hemorrhage (SAH), a condition recognized as a negative prognostic factor associated with progressive neurological decline, heightened morbidity, and increased mortality rates,[7] Subarachnoid hemorrhage (SAH) leads to sensory and emotional deficits through a cascade of events beginning with the presence of blood in the subarachnoid space, which triggers chemical meningitis and increases intracranial pressure (ICP) for days to weeks.[8] This elevated ICP compresses brain structures and disrupts cerebral blood flow, affecting areas that regulate sensory perception and emotional control. Patients may experience a sudden, severe headache, followed by impaired consciousness and rapid neurological decline, with symptoms such as confusion, restlessness, and mood disturbances. Furthermore, secondary complications like hydrocephalus can worsen symptoms, resulting in prolonged headaches, cognitive impairment, and motor deficits. The cumulative impact of these processes often leads to lasting sensory and emotional deficits, affecting quality of life and recovery.[9] Craniectomy, a critical neurosurgical intervention for managing intracranial hypertension in brain-injured patients, offers substantial preventive benefits against complications arising from elevated ICP. By strategically removing a significant portion of the skull on one or both sides and opening the dura, this procedure allows cerebral edema to expand beyond the cranial vault, thus alleviating ICP and reducing the risk of cerebral herniation.[10] [11] This decompressive effect not only prevents ischemic damage but also improves cerebral blood flow, supporting neurological stability and recovery in cases of severe brain injury.[12] Even though craniectomy is intended to mitigate complications of elevated ICP and trauma, it can paradoxically lead to specific complications, notably the Syndrome of the Trephined (SoT). SoT is characterized by neurological deterioration when the bone flap is not replaced with post-craniectomy. Though its incidence varies and is often underdiagnosed, SoT is identifiable by symptom reversal upon bone replacement, the only definitive treatment.[13] According to[14] Syndrome of the trephined (SoT) is a rare, but significant complication of a craniectomy characterized by neurological dysfunction that improves with cranioplasty. Decompressive craniectomy (DC) effectively lowers intracranial pressure (ICP) by removing a section of the skull, though it can lead to disruptions in cerebrospinal fluid (CSF) dynamics and decrease cerebral blood flow.[15] A “sinking” skin flap often signifies a pressure imbalance between intracranial and extracranial spaces (elimination of the effects of atmospheric pressure on the brain), with CSF pressure typically lower before cranioplasty and restored afterward.[16] Advanced imaging, including MRI, has demonstrated that cranioplasty not only reestablishes CSF flow but also restores normal postural ICP variations and improves cerebral blood flow on both the affected and opposite sides of the brain.[15] [17] [18] [19] Replacing the skull bone is essential for alleviating symptoms of Syndrome of the Trephined (SoT) by reinstating normal intracranial physiology, with symptom improvement reported within a mean of four days after cranioplasty, often leading to complete symptom resolution.[14]
In this case, the patient experienced emotional and sensory dysfunctions because of complications from the Syndrome of the Trephined (SoT), with symptoms persisting for 40 days despite pharmacological treatment and physiotherapy. Notably, all neurological complications are resolved immediately following cranioplasty. From our perspective, restoring the removed bone flap is the main effective cure for post-craniectomy complications, demonstrating a significant correlation between bone restoration and symptom resolution. However, further studies with larger sample sizes are necessary to validate these promising findings.
Conflicts of Interest
The authors declare that they have no competing interests.
Authors' Contributions
JQ, AE, RK, MA, NS: Literature search, manuscript preparation, revised manuscript, submission, and corresponding author. Literature search, preparation of the manuscript, revision of the manuscript; initiated, conceptualized, and organized the research; supervised and critically reviewed the manuscript; and finalized the manuscript. All authors read and approved of the final manuscript.
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References
- 1 Agrawal D, Hussain N. Decompressive craniectomy in cerebral toxoplasmosis. Eur J Clin Microbiol Infect Dis 2005; 24 (11) 772-773
- 2 Vitali M, Marasco S, Romenskaya T. et al. Decompressive craniectomy in severe traumatic brain injury: the intensivist's point of view. Diseases 2023; 11 (01) 22
- 3 Schuss P, Vatter H, Marquardt G. et al. Cranioplasty after decompressive craniectomy: the effect of timing on postoperative complications. J Neurotrauma 2012; 29 (06) 1090-1095
- 4 Mustroph CM, Stewart CM, Mann LM, Saberian S, Deibert CP, Thompson PW. Systematic review of syndrome of the trephined and reconstructive implications. J Craniofac Surg 2022; 33 (06) e647-e652
- 5 Kessels RPC, Hendriks MPH. Neuropsychological Assessment, in Encyclopedia of Mental Health (Second Edition), H.S. Friedman, Editor. 2016. Academic Press: Oxford; 197-201
- 6 Semrud-Clikeman M, Swaiman KF. 10 - Neuropsychological Assessment, in Swaiman's Pediatric Neurology (Sixth Edition), K.F. Swaiman, et al., Editors. 2017. Elsevier; 65-72
- 7 Griswold DP, Fernandez L, Rubiano AM. Traumatic subarachnoid hemorrhage: a scoping review. J Neurotrauma 2022; 39 (1–2): 35-48
- 8 Lesley Foulkes TS, Davis S, Woodburnm J, Halls A. Subarachnoid Haemorrhage: A guide for patients and carers. 2019: 49
- 9 Alexandrov AV, Krishnaiah B. Subarachnoid Hemorrhage (SAH). 2023 2023/07 [cited 2023; Available from: https://www.msdmanuals.com/professional/neurologic-disorders/stroke/subarachnoid-hemorrhage-sah
- 10 Bergman KS, Beekmans V, Stromswold J. Considerations for neuroprotection in the traumatic brain injury population. Crit Care Nurs Clin North Am 2015; 27 (02) 225-233
- 11 Patel K, Kolias AG, Hutchinson PJ. What's new in the surgical management of traumatic brain injury?. J Neurol 2015; 262 (01) 235-238
- 12 Bor-Seng-Shu E, Figueiredo EG, Fonoff ET, Fujimoto Y, Panerai RB, Teixeira MJ. Decompressive craniectomy and head injury: brain morphometry, ICP, cerebral hemodynamics, cerebral microvascular reactivity, and neurochemistry. Neurosurg Rev 2013; 36 (03) 361-370
- 13 Martínez-Viñuela I, Paredes-Borrachero I, Rubio-Mellado M. et al. [Syndrome of the trephined]. Rehabilitacion (Madr) 2021; 55 (04) 325-328
- 14 Ashayeri K, M Jackson E, Huang J, Brem H, Gordon CR. Syndrome of the Trephined: A Systematic Review. Neurosurgery 2016; 79 (04) 525-534
- 15 Lilja-Cyron A, Andresen M, Kelsen J, Andreasen TH, Fugleholm K, Juhler M. Long-Term Effect of Decompressive Craniectomy on Intracranial Pressure and Possible Implications for Intracranial Fluid Movements. Neurosurgery 2020; 86 (02) 231-240
- 16 Yamaura A, Makino H. Neurological deficits in the presence of the sinking skin flap following decompressive craniectomy. Neurol Med Chir (Tokyo) 1977; 17 (1 Pt 1): 43-53
- 17 Halani SH, Chu JK, Malcolm JG. et al. Effects of Cranioplasty on Cerebral Blood Flow Following Decompressive Craniectomy: A Systematic Review of the Literature. Neurosurgery 2017; 81 (02) 204-216
- 18 Songara A, Gupta R, Jain N, Rege S, Masand R. Early Cranioplasty in Patients With Posttraumatic Decompressive Craniectomy and Its Correlation with Changes in Cerebral Perfusion Parameters and Neurocognitive Outcome. World Neurosurg 2016; 94: 303-308
- 19 Dujovny M, Fernandez P, Alperin N, Betz W, Misra M, Mafee M. Post-cranioplasty cerebrospinal fluid hydrodynamic changes: magnetic resonance imaging quantitative analysis. Neurol Res 1997; 19 (03) 311-316
Address for correspondence
Publication History
Received: 08 February 2025
Accepted: 22 May 2025
Article published online:
16 July 2025
© 2025. Sociedade Brasileira de Neurocirurgia. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/)
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References
- 1 Agrawal D, Hussain N. Decompressive craniectomy in cerebral toxoplasmosis. Eur J Clin Microbiol Infect Dis 2005; 24 (11) 772-773
- 2 Vitali M, Marasco S, Romenskaya T. et al. Decompressive craniectomy in severe traumatic brain injury: the intensivist's point of view. Diseases 2023; 11 (01) 22
- 3 Schuss P, Vatter H, Marquardt G. et al. Cranioplasty after decompressive craniectomy: the effect of timing on postoperative complications. J Neurotrauma 2012; 29 (06) 1090-1095
- 4 Mustroph CM, Stewart CM, Mann LM, Saberian S, Deibert CP, Thompson PW. Systematic review of syndrome of the trephined and reconstructive implications. J Craniofac Surg 2022; 33 (06) e647-e652
- 5 Kessels RPC, Hendriks MPH. Neuropsychological Assessment, in Encyclopedia of Mental Health (Second Edition), H.S. Friedman, Editor. 2016. Academic Press: Oxford; 197-201
- 6 Semrud-Clikeman M, Swaiman KF. 10 - Neuropsychological Assessment, in Swaiman's Pediatric Neurology (Sixth Edition), K.F. Swaiman, et al., Editors. 2017. Elsevier; 65-72
- 7 Griswold DP, Fernandez L, Rubiano AM. Traumatic subarachnoid hemorrhage: a scoping review. J Neurotrauma 2022; 39 (1–2): 35-48
- 8 Lesley Foulkes TS, Davis S, Woodburnm J, Halls A. Subarachnoid Haemorrhage: A guide for patients and carers. 2019: 49
- 9 Alexandrov AV, Krishnaiah B. Subarachnoid Hemorrhage (SAH). 2023 2023/07 [cited 2023; Available from: https://www.msdmanuals.com/professional/neurologic-disorders/stroke/subarachnoid-hemorrhage-sah
- 10 Bergman KS, Beekmans V, Stromswold J. Considerations for neuroprotection in the traumatic brain injury population. Crit Care Nurs Clin North Am 2015; 27 (02) 225-233
- 11 Patel K, Kolias AG, Hutchinson PJ. What's new in the surgical management of traumatic brain injury?. J Neurol 2015; 262 (01) 235-238
- 12 Bor-Seng-Shu E, Figueiredo EG, Fonoff ET, Fujimoto Y, Panerai RB, Teixeira MJ. Decompressive craniectomy and head injury: brain morphometry, ICP, cerebral hemodynamics, cerebral microvascular reactivity, and neurochemistry. Neurosurg Rev 2013; 36 (03) 361-370
- 13 Martínez-Viñuela I, Paredes-Borrachero I, Rubio-Mellado M. et al. [Syndrome of the trephined]. Rehabilitacion (Madr) 2021; 55 (04) 325-328
- 14 Ashayeri K, M Jackson E, Huang J, Brem H, Gordon CR. Syndrome of the Trephined: A Systematic Review. Neurosurgery 2016; 79 (04) 525-534
- 15 Lilja-Cyron A, Andresen M, Kelsen J, Andreasen TH, Fugleholm K, Juhler M. Long-Term Effect of Decompressive Craniectomy on Intracranial Pressure and Possible Implications for Intracranial Fluid Movements. Neurosurgery 2020; 86 (02) 231-240
- 16 Yamaura A, Makino H. Neurological deficits in the presence of the sinking skin flap following decompressive craniectomy. Neurol Med Chir (Tokyo) 1977; 17 (1 Pt 1): 43-53
- 17 Halani SH, Chu JK, Malcolm JG. et al. Effects of Cranioplasty on Cerebral Blood Flow Following Decompressive Craniectomy: A Systematic Review of the Literature. Neurosurgery 2017; 81 (02) 204-216
- 18 Songara A, Gupta R, Jain N, Rege S, Masand R. Early Cranioplasty in Patients With Posttraumatic Decompressive Craniectomy and Its Correlation with Changes in Cerebral Perfusion Parameters and Neurocognitive Outcome. World Neurosurg 2016; 94: 303-308
- 19 Dujovny M, Fernandez P, Alperin N, Betz W, Misra M, Mafee M. Post-cranioplasty cerebrospinal fluid hydrodynamic changes: magnetic resonance imaging quantitative analysis. Neurol Res 1997; 19 (03) 311-316