DOI: 10.5137/1019-5149.JTN.10618-14.2

Kronik Bilinç Bozuklukları Olan Hastalarda Kraniyoplasti
Yapılması Kognitif ve Motor İşlevi Arttırmakta Etkili Olabilir mi?
Bir Olgu Raporu
Francesco Corallo, Rocco Salvatore Calabrò, Antonino Leo, Placido Bramantı
IRCCS Centro Neurolesi “Bonino - Pulejo”, Messina, Italy
Corresponding Author: Rocco Salvatore Calabrò / E-mail: [email-masked]

ABSTRACT
The “trephined” syndrome is a relatively rare complication after a large skull bone defect, consisting of a sunken skin above the bone defect
with neurological symptoms such as severe headaches, mental changes, focal deficits, or seizures. Emerging data show improvements in
the patients’ neurological status after cranioplasty, i.e. the surgical intervention to repair cranial defects. Herein we report a 55-year-old man
who attended our Neurorehabilitation Research Institute for a chronic disorder of consciousness (DOC) due to a severe right nucleo-capsular
hemorrhage. His motor and neuropsychological status greatly improved after cranioplasty. Our results confirmed previous reports that
cranioplasty may significantly improve neuropsychological and motor function, as evaluated by specific tests, in patients with skull bone
defects, and even in patients with DOC.
Keywords: Cranioplasty, Minimally conscious state, Cognitive rehabilitation, Disorder of consciousness

ÖZ
“Trephined” sendromu büyük bir kafatası kemik defektinin nispeten nadir bir komplikasyonudur ve kemik defektinin üzerinde içeri çökmüş
deri görünümü ve şiddetli baş ağrıları, zihinsel değişiklikler, fokal defisitler veya havaleler gibi nörolojik belirtilerden oluşur. Elimizdeki veriler,
hastaların nörolojik durumunun kraniyal defektlerin onarımı için kullanılan bir cerrahi girişim olan kraniyoplasti sonrasında düzeldiğini
göstermektedir. Burada şiddetli sağ nükleo-kapsüler kanama nedeniyle kronik bilinç bozukluğu olan ve Nörorehabilitasyon Araştırma
Enstitümüze gelen 55 yaşında bir hastayı sunuyoruz. Hastanın motor ve nöropsikolojik durumu kraniyoplasti sonrasında büyük iyileşme
gösterdi. Sonuçlarımız kraniyoplastinin kafatası kemiği defektleri ve hatta bilinç bozukluğu durumu olan hastalarda spesifik testlerle belirlendiği
şekilde nöropsikolojik ve motor işlevi önemli ölçüde düzeltebileceğini bildiren önceki raporları doğrulamıştır.
ANAHTAR SÖZCÜKLER: Kraniyoplasti, Minimal bilinçli durum, Kognitif rehabilitasyon, Bilinç bozukluğu

Abbrevıatıons: CRS-R: coma recovery scale-revised, GCS: Glasgow coma scale, ADL: activities of daily living, IADL: instrumental
activities of daily living, FIM: functional independence measure, MMSE: mini mental state examination

Introduction
Cranioplasty is the major intervention to repair cranial defects,
and it gives relief to psychological drawbacks and increases
the social performance in addition to improving motor performance. Although several reconstruction materials are used
in cranioplasty, there are no studies able to prove that one is
more suitable than another. Therefore, many research topics
are focused on developing of both biological and non-biological materials. Cranioplasty is performed mostly after traumatic brain injuries (10). Cancer surgery and the decompressive craniectomies are the leading causes of cranial defects.
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On the other hand, contraindications for cranioplasty are the
presence of hydrocephalus, infection, and brain swelling.
Before performing a reconstructive cranioplasty, it is recommended to wait from 3 to 6 months in order to prevent the
development of devitalized autograft or allograft infections
(10). The “trephined” syndrome (or sinking skin flap, SSF) is
due to a defect in the skull bone, and it is characterized by
neurological symptoms such as seizures, severe headache,
focal deficits and cognitive and behavioral changes. Unfortunately, SSF may lead to paradoxical herniation with consequent coma or death (8).

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Case Report

Can Cranioplasty be Effective in Improving
Cognitive and Motor Function in Patients with
Chronic Disorders of Consciousness? A Case Report

Corallo F. et al: Usefulness of Cranioplasty in DOC

Patients undergoing cranioplasty may have important and
unexpected improvement in cognitive and motor function,
although the pathomechanism remains unclear. However,
it has been suggested that the decrease of local cerebral
compression may improve cerebrospinal fluid hydrodynamics,
global cerebral hemodynamics, and thus the metabolism to
globally improve performance (1).
There are few studies that demonstrate an improvement in
the consciousness of patients in the vegetative or minimally
conscious state following cranioplasty (5-9). The possible
effects of this breach on the patients’ neurological recovery
are poorly understood and have been scarcely evaluated
until now. The effect of cranioplasty on cognitive and motor
functions in severely brain-injured individuals remains
controversial (3). Herein we describe the case of a patient in
vegetative state (VS) with recovery of consciousness after
repositioning of the skull.
Case Presentation
In October 2011, a 55-year-old man experienced sudden
onset of motor deficits and language difficulties, with

consciousness impairment. After he was admitted to the
emergency room, a CT scan of the skull showed a massive
right-side nucleo-capsular hemorrhage with perilesional
cerebral edema. As the clinical conditions worsened, the
patient underwent neurosurgery intervention, consisting
in decompressive craniectomy, hematoma evacuation and
positioning of an intracranial pressure monitoring device.
A subsequent brain CT scan showed a significant reduction
of the parenchymal herniation through the bony crypt and
a near complete reabsorption of the intraventricular blood,
while the Angio-CT did not reveal any vascular abnormalities.
The patient who was in a VS was then admitted to our
Research Institute for an intensive neurological rehabilitation
(consisting of motor and cognitive rehabilitation performed
twice/day), with a progressive improvement in general
and neurological conditions. During the hospitalization he
suffered some critical episodes characterized by complex
partial seizures, helpfully treated with lamotrigine (200 mg/
day). On June 2012, the patient (who was in a Minimally
Conscious State, MCS) was transferred to a Neurosurgery Unit
to undergo cranioplasty. Prior to placement of the case the
patient has performed a CT scan 3D (Figure 1).
In July 2012, the patient returned to our institution to continue
his rehabilitation treatment (Figure 2). The neurological
examination and the scores obtained by proper clinical scales
and neuropsychological tests before and after the intervention
of cranioplasty showed a significant improvement in
consciousness and global cognitive performances. We also
found that the ADL and IADL index score, which is a daily
activities functional score, significantly improved (Figure 3).
Moreover, a partial autonomy of everyday-life activity was
reached at discharge (December 2012), beside a further
improvement in motor and cognitive function.
Discussion

Figure 1: 3D CT reconstruction of the skull defect after
craniectomy.

This case demonstrates a clear evidence of a possible recovery
of cognitive, behavioral and motor function after a severe
acquired brain injury, partially related to a proper cranioplasty.
Moreover, we observed a slow and only partial recovery
in the activities of daily living, as well as in some residual
motor and cognitive impairment. The neurological basis

Figure 2: The patient’s
skull before and after
cranioplasty.

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Corallo F. et al: Usefulness of Cranioplasty in DOC

Admission (T0)

Before cranioplasty (T1)

After cranioplasty (T2)

for late recoveries remains unknown, although unexpected
VS improvements have been observed in patients with the
use of dopaminergic or GABAergic drugs or with intrathecal
baclofen. In this case, no specific drug was administered
and only two procedures, in the opinion of the authors,
represents the potential candidates influencing the recovery
of consciousness, i.e. early and long-term rehabilitation and
cranioplasty (9).
The potential improvements of neuropsychological function
after cranioplasty have been shown in several studies,
although the mechanism remains unclear, even though some
authors have attributed neurological and cognitive deficits
to the atmospheric pressure transmitted through the cranial
cavity (2). This hypothesis is partly confirmed by the findings
that cranioplasty improves the neuropsychological state and
motor function in patients with bone defects of the skull,
maybe thanks to the improvement of the cerebral blood flow
(CBF) (5,7).
To date, only another case of a patient in VS with recovery of
consciousness following cranioplasty has been described,
confirming our hypothesis of a positive effect of this method
on the cognitive performances, although many other
variables must be taken into account (9). It is known that
early and long-term rehabilitation can affect the outcome in
severely brain injury, even in VS and MCS patients. In this case,
multidisciplinary programs and intensive rehabilitation were
started early, and then continued uninterruptedly throughout
the period of unconsciousness and impaired response.
Indeed, it is possible that the long-term rehabilitation helped
to recover the function until the unexpected success, but
it is very doubtful that a more appropriate model of motor
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Figure 3: Clinical scales and
neuropsychological tests,
performed at admission
(TO), before (T1) and after
craniplasty (T2).

and cognitive stimulation, as expected by multidisciplinary
rehabilitation programmers, may also influence the recovery
of consciousness inducing mechanisms of neuronal plasticity.
Unfortunately, so far no clinical hypotheses can be invoked
to better support our findings. Beyond these two speculative
cues (cranioplasty and rehabilitation), no better explanation
can be considered to better understand the amazing recovery
of consciousness described either by Sancisi et al. (9) or by our
group.
A deeper understanding of the factors influencing the
outcomes in VS/MCS individuals is urgent, and any possible
method leading to the patient’s recovery must be used before
discharge.
The repair of the cranial defect provides a clear benefit to
the patients in terms of neurological improvement in many
cognitive domains as well as in quality of life. Thus, larger
and multicenter studies should be fostered to confirm these
interesting and promising findings.
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