Systematic Review of Syndrome of the Trephined and Reconstructive Implications

Abstract

Syndrome of the trephined (SoT) is a severe complication following decompressive craniectomy resulting in neurological decline which can progress to aphasia, catatonia, and even death. While cranioplasty can reverse neurological symptoms of SoT, awareness of SoT is poor outside of the neurosurgery community. The authors performed a systematic review of the literature on SoT with a focus on reconstructive implications. Search terms “syndrome of the trephined” and “sunken flap syndrome” were applied to PubMed to identify primary studies through October 2021. Full-text review yielded 11 articles discussing SoT and reconstructive techniques or implications with 56 patients undergoing cranial reconstruction. Average age of the patients was 41.8±9.5 years. Sixty-three percent of the patients were male. The most common indication for craniectomy was traumatic brain injury (43%), followed by tumor resection (23%), intracerebral hemorrhage (11%), and aneurysmal subarachnoid hemorrhage (2%). Patients most commonly suffered from motor deficits (52%), decreased wakefulness (30%), depression or anxiety (21%), speech deficits (16%), headache (16%), and cognitive difficulties (2%). Time until presentation of symptoms following decompression was 4.4±8.9 months. Patients typically underwent cranioplasty with polyetheretherketone (48%), titanium mesh (21%), split thickness calvarial bone (16%), full thickness calvarial bone (14%), or split thickness rib graft (4%). Eight percent of patients required free tissue transfer for soft tissue coverage. Traumatic Brain Injury (TBI) was a risk factor for development of SoT when adjusting for age and sex (odds ratio: 8.2, 95% confidence interval: 1.2–8.9). No difference significant difference was observed between length until initial improvement of neurological symptoms following autologous versus allograft reconstruction (P=0.47). SoT can be a neurologically devastating complication of decompressive craniectomy which can resolve following urgent cranioplasty. Familiarity with this syndrome and its reconstructive implications is critical for the plastic surgery provider, who may be called upon to assist with these urgent cases.

Mccune Albright Syndorme/Craniofacial Fibrous Dysplasia And Postcraniectomy Syndrome: Case Report And Literature Review

“…Its diagnosis supposes the precedent of craniectomy accompanied by neurological symptoms such as motor weakness, cognitive deficit, language deficit, altered level of consciousness, headache, seizures, cranial nerve alterations, psycho-emotional symptoms, in a patient who has not undergone cranioplasty. The exacerbation of these symptoms seems to be related to postural changes, especially those that involve standing vertically, as well as the progressive sinking of the skin flap [15][16][17][18].…”

“…On the other hand, considering the established relationship between the pressure and volume of cerebrospinal fluid and its pathophysiology, there could be a greater risk of developing alterations in patients with pressure changes in the third ventricle [14]. Its treatment is cranioplasty, before which the symptoms improve, as well as elements such as cerebrospinal fluid pressure at the lumbar level and cerebral perfusion pressure return to normal levels, suggesting a causal relationship to the defect and its change [15][16][17][18][19]. In the presence of hydrocephalus after craniectomy, the shunt does not seem to represent an improvement in patients, instead leading to greater neurological deterioration that is based on the understanding of the patho-physiology of the syndrome that we have so far [16].…”

Can we do better at measuring patient-reported outcomes after cranioplasty? A systematic review

“…Cranioplasty is a common procedure to repair a skull defect resulting from a previous operation or injury [ 1 , 2 ]. While the procedure is primarily performed to protect the brain from outside forces, cranioplasty can improve neurological function, enhance glymphatic fluid circulation, and restore intracranial pressure adaptations and cerebrospinal fluid circulation [ 3 , 4 ].…”

“…Recent studies have also highlighted the impact of cranioplasty on syndrome of the trephined or “sinking flap syndrome.” This syndrome refers to the neurological deterioration that occurs after a large craniectomy, with various symptoms including headaches, motor weakness, worsened hemisyndrome, language deficits, and cognitive disorders with or without an orthostatic component. These symptoms can improve or resolve entirely as early as 3–4 days after a cranioplasty [ 1 , 2 , 5 – 7 ].…”

Sunken Skin Flap Syndrome: Neurological Dysfunction After Decompressive Craniectomy

“…However, awareness of SSFS is poor outside of the neurosurgery community. 2 One systematic review of the literature found that the most common indication for craniectomy is traumatic brain injury (43%), followed by tumor resection (23%), intracerebral hemorrhage (11%), and aneurysmal subarachnoid hemorrhage (2%). 2 Following craniectomy, SSFS can occur within a few days or up to a year after.…”