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Lee, Kim, Kim, Lim, and Park: Decompressive Hemicraniectomy and Duroplasty in Toddlers and Preschool Children with Refractory Intracranial Hypertension after Unilateral Hemispheric Stroke

Abstract

Objective

Life-threatening hemispheric stroke is associated with a high mortality and morbidity. Decompressive hemicraniectomy has been regarded as an effective treatment option for refractory intracranial hypertension. Here, we reported the clinical course of 5 children with decompressive craniectomy and duroplasty after non-traumatic refractory intracranial hypertension.

Methods

Four toddlers and one preschool-girl were included in this study; there were 3 boys and 2 girls with a mean age of 34.6 months (range 17-80). Decompressive craniectomy including duroplasty was performed in cases of dilatation of pupil size after intensified standard medical therapy had proven insufficient. All children had a Pediatric Glasgow Coma Scale score <8 at pre-operation state. The mean time-point of craniectomy after stroke attack was 12 hours (range 4-19).

Results

During the long-term follow-up period (mean 47.6 months), no children died. One year later, when we checked their Glasgow Outcome Scale scores, only one toddler received a score of 4 (moderate disability). But the others had good recoveries although they had minor physical or mental deficits. According to the Pediatric Cerebral Performance Category Scale, 4 children received a score of 2 (mild disability).

Conclusion

Despite our small cases, we suggest that decompressive hemicraniectomy and duroplasty is an acceptable and life-saving treatment for refractory intracranial hypertension after unilateral hemispheric stroke in toddlers and preschool children.

INTRODUCTION

Stroke is rare in children, however, a common cause of neurological disease, and it is a major cause of death ranks in the top ten in pediatric period11,15). Documented incidence has been reported as 2.5-8/100000 children/year12,13).
Medical treatment includes maintaining cerebral perfusion pressure with hyperventilation and osmotherapy, barbiturate coma and in case of thrombotic ischemia, thrombolytic procedure, and anticoagulanttherapy. However, if all medical treatment is fail to drop the intracranial pressure (ICP) and deterioration of patient progressed, alternative treatments, such as surgical decompression could be mandatory.
The common rationale of decompressive craniectomy with or without duroplasty is to let the volume expansion of the swelling brain to extracranial space via removed skull flap and prevent cerebral herniation and secondary damage of brain parenchyme.
In case of traumatic brain injury, decompressive craniotomy for pediatric patient has been reported20). However, there has not been a definite evidence or standard guideline for decompressive craniectomy for pediatric patient with non-traumatic acute stroke with uncontrolled refractory high intracranial pressure.
We will introduce our series about the functional and clinical outcome after decompressive craniectomy to control refractory high ICP due to non-traumatic acute stroke in pediatric patient.

MATERIALS AND METHODS

Between March 2004 and December 2006, decompressive-hemicraniectomy and duroplasty was performed in 5 toddlers and preschool children with non-traumatic, malignant refractory high ICP. Patient characteristics and outcome were summarized in Table 1.
Any patient have no trauma history, and refractory high ICP was diagnosed as clinical evidence of acute stroke, massive infarction or hemorrhage with midline shift, compression of basal cisterns in conventional radiologic examination such as computed tomography (CT) and magnetic resonance image (MRI) and, neurological deterioration consisting of decreased level of consciousness or increasing levels of sedation, or somnolence or stupor compared with baseline status at admission17). And we regard sustained refractory high ICP as a failure of intensified standard medical therapy.
All operations were performed by one pediatric neurosurgeon and all patients received treatment in pediatric intensive care unit of one medical center.

Medical treatment

In all patients, CT or MRI was obtained immediately, after stabilization of ventilation and hemodynamicsat first. Blood sugar, body temperature, ventilation/oxygenation, blood volume and trans-cutaneous oxygen saturation, arterial blood pressure was continuously monitored and controlled in pediatric intensive care unit. Clinical and neurologic status was evaluated with the Glasgow Coma Scale (GCS). To reduce ICP, hyperosmolar therapy is typically begun with use of osmotic diuretics such as mannitol as needed.
Decompressive craniectomy was performed in cases of failure of intensified standard medical therapy.

Surgical protocol

Craniectomy involved the removal of a largebone flap ipsilateral to the involved hemisphere includingfrontal, parietal, and temporal bone.
A large skin flap was lifted from the skull with meticulous blood control, multipleburr holes were made, large bone flap removed. In the temporalregion, the craniectomies were extended toward the floor of the middlefossa to maximize decompression. Multiple openings in the dura were made; a dural patchwas placed and sutured. In every case, intra-cerebral pressure monitor probe was positioned epidural space of bone margin.
The bone flaps were stored in a bone bank and then, following improvement of clinical and neurologic status, cranioplasty was performed 3 to 6 months later.
Neurological outcomes were evaluated using neurological-examinations. Glasgow Outcome Scale (GOS)5), and Pediatric Cerebral Performance Category Scale (PCPCS)4) (Table 2) calculated every 6 months after discharge.

RESULTS

Five pediatric patients were treated with decompressive hemicraniectomy with duroplasty for non-traumatic and refractory intracranial hypertension after unilateral hemispheric stroke. Four patients were toddler and one patient was pre-school girl. Three were boy and two were girl. The mean age was 34.6 months old (range 17-80 months).
Four cases were caused by ischemic stroke, and another one case was hemorrhagic stroke. One case of hemorrhagic stroke, patient had been diagnosed by thrombocytopenia. One case of ischemic stroke, patient had atrial septal defect (ASD). And including this patient three cases of ischemic stroke patients were taken angiography; however there was no abnormal findings in angiography. In all patients, conventional laboratory result and special serum anti-body test, including anti-phospholipid antibody were within normal rage. At the time of operation, all patients had a GCS score <8 (median 7, range 6-8) and all patients hadunilateral mydriasis. Surgical decompressive hemicraniectomies were performed at a mean of 12 hours (range 4-19 hours). In all cases we had performed supratentorial hemicraniectomies. There was no bilateral or occipital craniectomy case. The mean peak ICP was 28.6 mm Hg (range 25-30 mm Hg). And the ICP of all patients were dropped to normal range within one and half days after operation.
There were no surgical complications, such as cerebrospinal fluid leak or wound infection, even after cranioplasty. One of ischemic stroke patient had got a ventriculoperitoneal shunt operation because of post-stroke hydrocephalus.
The mean follow-up period was 47.6 months (36-68 months).GOS and PCPCS for 5 patients had been measured to evaluate postoperative neurological outcome. Based on the regular follow up GOS scores, 5 patients had shown satisfied recoveries : 4 had good recoveries (GOS score of 5), and 1 had moderate disabilities (GOS score of 4). And there was no patient shown severe disability or death. The PCPCS scores also had revealed contend results : 4 patients received scores of 2, and only one patient scored of 3.

Patient presentation

A 17-month-old girl was transferred to our emergency department with decreased mentality. Neurological examination revealed a stuporous child without occasional spontaneous eye opening. Her right pupil was 6 mm in size and was not response to light. And her left pupil was 3 mm and was sluggishly reactive to light. Hemiplegia, hyper-reflexia, and Babinski's sign were observed on the left side. Preoperative GCS score was 7. In general physical examination, she had an ejection systolic murmur. By pediatriccardiologist consultation, patient was diagnosis with ASD.
Initial brain CT scan was suggestive of acute infarction and hemorrhagic transformation atright temporal area. And emergency MRI sturdy was performed with MR angiography and diffusion-weighted image. There was no arterial filling defect on cerebral MR angiography. But there was appropriate for acute cerebral infarction with hemorrhagic transformation with significant mid-line shift. She was given high doses of mannitol and hypertonic saline. However, there was no neurologic improvement.
Decompressive surgery was performed 5 hours following her loss of consciousness. After surgery, she was treated in pediatric intensive care unit with be kept intubated for 7 days.
A brain CT at the time of post-operative 7 days revealed sustained brain swelling, but midline shift did not noted anymore. The immediate post-operative ICP was 28 mm Hg. The ICP dropped to normal gradually within one day after surgery. At the time of postoperative 3-month, she returned for an elective cranioplasty. The bone flap was secured without any complications.
At the end of follow-up, 41 months after surgery, this patient maintained a PCPCS of 2, and GOS of 5, and she had showed mild left side hemiparesis, mild behavioral disorder and strabismus.

DISCUSSION

In stroke patient, most common cause of death is due to uncontrolled ICP associated with large hemispheric infarctions10,12). When it is present, it is associated with a dramatic increase in mortality. In addition, early ICP elevation in patients with large hemispheric infarctions is highly concluded with high mortality16). It means that the one of most important goal of acute phase stroke treatment is control the ICP and prevent a secondary damage due to brain swelling.
In spite of many clinical and experimental trials on medical therapeutic method, most of themhave not been shown to benefit pediatric stroke patient. There has been a widely accepted definite guideline for oxygen saturation, temperature, serum glucose level, blood pressure for adult stroke patient1). However, for pediatric stroke patient, even in many of mostly basic and elementary medical guideline has not been established yet14).
To prevent and control ICP, thrombolytic therapy such as clot lysis has an accepted role in acute stroke treatment. In adult stroke patient group, there are antithrombotic management of adult ischemic stroke protocol and guideline published by the American Heart Association1), such as intravenous tissue plasminogen activator, intra-arterial thrombolysis and oral administration of aspirin. But, for pediatric stroke patient, there is no guideline of this useful treatment method either14).
To the best of our knowledge, the first report about the benefits of decompressive hemicraniectomy in pediatric patient was described by Carter et al.3) at 1997. However, from this time, only a few report deals with decompressive craniectomy for pediatric patient were published.
In adult acute stroke patient group, the effect of decompressive craniectomy also has been established. Vahedi et al.21) reported that in a meta-analysis combining the data of 93 subjects from three small, randomized, controlled trials, mortality for conservative management in adult ischemic stroke patients with early brain edema was 71% as compared with 22% for decompressive surgical intervention.
Nowadays, some case reports and review has been published favorable outcome after decompressive surgery in childhood acute stroke2,17). It is not often however in some article, decompressive craniotomy have been regarded as emerging procedure can save life, for pediatric stroke patient6,11), even guideline for craniectomy was introduced11). In point of fact, there is some debate indications and optimal timing of decompressive craniectomy12,19). Widely accepted proper operation timing is immediately after signs of herniation and a midline shift which was defined as a more than equals 5-mm contralateral shift of the midline structures on the preoperative cranial CT scan on septum pellucidum level, compressed basal cisterns, or uncal herniation--had to be present on cranial CT had been noted. Recent studies in adult stroke group have shown that this procedure not only reduces mortality but also improves neurological functional outcome. Especially when operation is performed in the early hours8,12,18). This procedure may be life-saving if done early in cases of impending herniation, is associated with good outcomes in pediatric stroke patients9,11). Maybe if operation was performed earlier, it may minimize the vicious circle of brain swelling, increased ICP, ischemia, and infarction12).
In our series, four toddlers and one pre-school girl with refractory high ICP due to non-traumatic, ischemic and hemorrhagic stroke were treated with decompressive craniectomy. All had GCS scores <8 at operation. All of them were survived and have a relatively satisfied recovery. Four patient received PCPCS score of 2 (mild disability) and only one received that of 3 (moderate disability) after 3 years later post operation. These results showed that decompressive craniectomy can not only play a rescuer therapeutic role but also guaranty acceptable clinical outcome in the treatment regimen in non-traumatic, refractory high ICP in pediatric field.
In some articles, decompressive craniotomy has been tried to apply more alternative pediatric field. Meningitis and encephalitis could be not very rare diseases of pediatric period. And these diseases could make a malignant intracranial hypertension. Even in adult, effectiveness of decompressivecraniectomy has been not established firmly. Aghakhani et al.2) report that decompressive craniectomy was performed in pediatric patient with malignant intracranial hypertension due to infectious encephalitis. In adult patient group, there had been not infrequently reported that malignant intracranial hypertension due to encephalitis treated with decompressive craniectomy. Now, even in case of pediatric patient with infectious disease in central nerve system, decompressive craniectomy could be considered as life-saving-treatment options.
Gordon et al.7) report that pediatric stroke can affect not only patient itself, but also his or her whole family. Over half of stroke survivors have suffer from decrease in quality of life and affects the entire family, and relates to both neurological deficits and psychosocial factors11). Hence, if all medical treatment is fail to drop the ICP in non-traumatic pediatric acute stroke patient, without hesitation, a decompressive craniectomy could be regarded as next step treatment option.

CONCLUSION

In this study, we describes the young pediatric patients have undergone decompressivehemicraniectomy for a non-traumatic acute hemispheric stroke. It also shows that decompressive hemicraniectomy can be lifesaving and can besafely performed in toddler and pre-school children. And young children can get a survival and relatively independence and family satisfaction.Decompressive hemicraniectomy should be considered as an alternative therapy for patients with life-threatening brains welling refractory to medical management.

References

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Fig. 1
A : Non enhanced brain CT revealed an acute right-sided middle cerebral artery territory infarct with mass effect manifested as right lateral ventricular compression, attenuation of the basal cisterns and right convexity sulci effacement. Because of midline shift due to severe brain parenchymal swelling, posterior horn of lateral ventricle was not seen. And hemorrhagic transformation was also noted at same side area. B and C : DWI MRI scan of his brain revealed that lesion was appropriate for acute cerebral infarction with hemorrhagic transformation with significant mid-line shift. CT : computed tomography, DWI : diffusion weighted image, MRI : magnetic resonance imaging.
jkns-51-86-g001.jpg
Fig. 2
Brain CT at the time of post-operative 7 days revealed sustained brain swelling with decompressive craniectomy, but midline shift did not noted anymore.
jkns-51-86-g002.jpg
Fig. 3
Three years later, there was encephalomalasic change in right-sided middle cerebral artery territory. The bone flap was secured.
jkns-51-86-g003.jpg
Table 1
Patient characteristics and outcome
jkns-51-86-i001.jpg

GCS : Glasgow Coma Scale, PCPCS : Pediatric Cerebral Performance Category Scale, GOS : Glasgow Outcome Scale, MCA : middle cerebral artery

Table 2
Pediatric Cerebral Performance Category Scale (PCPCS)
jkns-51-86-i002.jpg
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