Delayed Progressive Intracranial Bleeding in Pediatric Acute Epidural Hemorrhage Treated Expectantly

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International Journal of Surgery Case Reports 105 (2023) 108005

Available online 20 March 2023

2210-2612/© 2023 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

Case report

Delayed progressive intracranial bleeding in pediatric acute epidural hemorrhage treated expectantly

Andre Marolop Pangihutan Siahaan

^a^,^*

, Martin Susanto

^a

, Donny Luis

^b

, Muhammad Chairul

^c

, Sonny Giat Raja Saragih

^d

, Ade Ricky Harahap

^e

aDepartment of Neurosurgery, Faculty of Medicine, Universitas Sumatera Utara, Medan, Indonesia

bDivision of Neurosurgery, Murni Teguh Memorial Hospital, Medan, Indonesia

cDivision of Neurosurgery, Mitra Sejati General Hospital, Medan, Indonesia

dDepartment of Surgery, Faculty of Medicine, Universitas Tanjungpura, Pontianak, Indonesia

eDivision of Neurosurgery, Dr. M. Djamil General Hospital, Padang, Indonesia

A R T I C L E I N F O Keywords:

Progressive epidural hematoma Case report

Pediatric

A B S T R A C T

Introduction and importance: Progressive epidural hematoma (PEDH) after traumatic brain injury is usually found in the first 24 h after accident. However, EDH enlargement on day six after admission is rarely observed.

Presentation of case: We present the case of a 14-year-old boy who presented to the emergency room after a car accident with only a headache without any neurological deficit. The computed tomography (CT) scan revealed a slight epidural hematoma, which then treated expectantly. On day 6, the patient developed severe headache. CT- Scan showed enlarged epidural hematoma with significant mass effect. The emergency clot evacuation was completed successfully.

Clinical discussion: Progressive intracranial hemorrhage is any increase in pre-existing intracranial bleeding or the presence of a new hematoma on a CT scan. Young age and cranial fracture have been identified as risk factors for PEDH morbidity and mortality. Coagulation parameters may be a predictor of progressive intracranial bleeding, but their accuracy remains unclear. Still, the decision to conduct a CT scan as a follow-up is debatable, but it should be performed when neurological deterioration occurs.

Conclusion: Although rare, PEDH could still be occurred six days after trauma. Linear fracture and young age are among the risk factors. A thorough routine neurological examination is crucial in treating this condition.

1. Introduction

Traumatic Brain Injury (TBI) is an acquired injury to the brain caused by an external mechanical force that can result in temporary or per- manent impairment. Aside from subdural hematoma (SDH), epidural hematoma (EDH) is a common pathological finding following a TBI accident [1]. There are two primary approaches to management, namely expectant management, and surgical management. Initial CT findings and patient condition are primarily used to determine the optimal treatment approach [2]. In some patients, however, a progressive epidural hematoma (PEDH) may develop.

PEDH is defined primarily by a radiologic characteristic: absence or small size of an EDH on the initial CT scan after trauma with notable

expansion of the hemorrhage size on follow up CT scans during patient evolution [3]. Even though the reported incidence of PEDH is relatively low (between 5.6 % and 13.3 %), it tends to occur rapidly and dramatically [4]. Moreover, the extent of a growing hemorrhage and the onset of hematoma advancement are uncertain.

The majority of PEDH occur within the first 24 h of admission [5].

PEDH occurring more than three days after admission is a rare occur- rence. Here, we describe a case of PEDH that developed in a pediatric patient who was treated expectantly six days after an accident. This work is reported according to the SCARE criteria and the revised 2020 SCARE guidelines [6].

Abbreviations: TBI, Traumatic brain injury; EDH, Epidural hematoma; SDH, Subdural hematoma; PEDH, Progressive epidural hematoma; CT, Computed tomography; GCS, Glasgow Coma Scale.

* Corresponding author at: Department of Neurosurgery, Faculty of Medicine, Universitas Sumatera Utara, Jl. Dr. Mansyur No. 5, Medan 2015, Indonesia.

E-mail address: [email protected] (A.M.P. Siahaan).

Contents lists available at ScienceDirect

International Journal of Surgery Case Reports

journal homepage: www.elsevier.com/locate/ijscr

https://doi.org/10.1016/j.ijscr.2023.108005

Received 17 October 2022; Received in revised form 7 March 2023; Accepted 16 March 2023

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International Journal of Surgery Case Reports 105 (2023) 108005

2 2. Presentation of case

A 14 year-old right handed boy was presented to the emergency unit 2 h after a motor vehicle accident. He was riding a motorcycle with moderate speed (around 40–50 km per hour) that was suddenly fell down after hitting a hole on the road. He was not wearing helmet during the accident. According to the emergency medical technician accom- panying the patient, the patient was unresponsive around five to 10 min after the accident, but regained his consciousness back on the way to the hospital.

On examination, he was fully alert with GCS 15/15 with clear airway. Blood pressure was 130/70, heart rate was 92 times per minute, respiratory rate was 20 times per minute, and SaO2 was 98 %. He only complained headache (Visual Analogue Scale 6/10). Neurological examination revealed symmetric pupils with equal reflex to light and no deficit in motoric or sensory examination. Reflexes were also normal.

According to the patient's mother, the patient was healthy prior to the accident with no history of medication and no known allergies.

Laboratory examination revealed hemoglobin was 13.2 g/dL (reference [13–18)], leukocyte was 12,200/μL (reference 4000–11,000/μL), thrombocyte was 350.000/mm³(reference 140.000–450.000/mm³).

Serum electrolyte, renal function test, liver function test, and routine hemorrhagic screening test (PT, APTT, and TT) were in normal range.

X-ray of the chest and cervical was normal. Head CT Scan revealed fracture on the right temporal (Fig. 1A) with suggestive slight epidural hematoma (Fig. 1B). Patient was managed expectantly using analgetic, tranexamic acid, and diuretic osmotic. Bolus of mannitol was used as diuretic osmotic agent with loading dose at 1 g/kg, following by 0.5 g/

kg divided into four doses. The patient reported decrease in headache intensity, and this approach of treatment was continued.

On day six, patient complained that the headache was progressively worsened. Beside of the progressive headache, there was no other complain. He was fully alert with normal motoric and sensory examination. A control CT Scan was conducted and revealed a significant progressive epidural hematoma on the right temporoparietal region compared to the initial CT with midline shift of 1 cm (Fig. 1C). Labo- ratory examination showed normal hemorrhagic screening test with minimal increase of D-dimer. An emergency craniotomy to evacuate the epidural hematoma was performed by general neurosurgeon. We found slack brain with good pulsation after removing blood clot. Following operation, headache was reduced significantly and the patient was discharge on day 5 after operation with no neurological deficit.

3. Discussion

Progressive intracranial bleeding, described as any increment in pre- existing intracranial bleeding or the presence of a new hematoma on a CT scan, is linked to poorer outcomes in pediatrics, such as increased mortality rates and long-term neurological morbidities, delayed recovery, and poor long-term results. Since there is variation in the definition, true incidence of progressive intracranial bleeding is still unknown [7].

We reported a case of delayed PEDH that was happened on day six after admission in pediatric with normal coagulation status. To our knowl- edge, it was an uncommon yet interesting finding, especially since most PEDH were reported in the first 24 h after impact [5].

Theoretically, when the brain injury occurs, it may cause a local inflammatory response that leads to diffuse injury via the neurotrans- mitter flood. And the brain will respond by forcing CSF from cranial subarachnoid spaces and lateral ventricles into the spinal subarachnoid space to increase brain volume. Thus, as ICP gradually rises, the compensatory mechanism may cause blood vessel constriction and decreased cerebral blood flow [8]. This explains the signs and symptoms of brain injury. Initial loss of consciousness following trauma is a typical symptom, followed by a complete transient recovery or “lucid interval”

and a rapid progression of neurological deterioration. A CT scan of the head is the primary imaging technique used to evaluate intracranial pathology. Additionally, Magnetic Resonance Imaging (MRI) could be utilized to diagnose traumatic brain injury. Current update of diagnostic biomarker is Brain Trauma Indicator™ (BTI™), which is the first blood- based biomarker for TBI, which has been approved by the Food and Drug Administration of the United States (FDA). BTI™ measures assesses two brain-specific protein biomarkers: ubiquitin C-terminal hydrolase-L1 and glial fibrillary acidic protein (GFAP) [9].

There are several well-known risk factors of PEDH. The first is age.

Though it is not significant statistically, pediatrics is more susceptible to develop PEDH. It is presumed that blood vessels based on duramater may be more easily splitted or avulsed in younger patients due to skull deformation. Duramater will become more adherent to the cranium along with age, thereby reducing the likelihood of epidural hematoma in adult [5]. On contrary, Oertel et al. (2002) reported older age as risk factor of PEDH, because of the tendency of vascular fragility in older population due to higher incident of several blood vessel fragility risk factors, such as hypertension, diabetes mellitus, and amyloidosis [4].

Time interval between impact and CT scan is also associated with PEDH. It is not surprising that the earlier the initial CT scan is conducted after impact, the higher the risk of progressive haemorrhagic on follow- up CT scans. A two to three hours interval between impact and initial

A B C

Fig. 1.Non-contrast head CT-scan of the patient. A linear fracture was seen on left temporal (A) on initial CT, along with slight epidural hematoma on temporoparietal region (B). Follow op CT on day 6 (C) showed a significant increase of EDH volume, along with midline shift.

A.M.P. Siahaan et al.

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3 imaging is associated with PEDH [4,5]. If the time between the impact and the initial CT scan is faster, it is probable that the epidural hematoma does not reach its maximum volume and still may evolve rapidly.

The association among cranial fracture and a higher likelihood of epidural hematoma or PEDH has been identified in earlier studies, as well as in the present study. In addition, some authors assume that the appearance of a cranial fracture, which has been recognized as a common trait of reported cases of PEDH, should be considered a risk factor for the development of this condition. A skull fracture is found in most of the epidural bleeding, and the diploic vein represents one of the hemorrhagic sources. Because of the lower pressure, epidural bleeding caused by diploic vein rupture extended more gradually than those induced by arterial sources (such as the middle meningeal artery) [10].

This, alongside the fact that the duramater is not firmly attached to the skull in children, may explain the association between delayed progressive epidural bleeding and skull fracture. Furthermore, in our case, the bleeding progressed in the temporal region, where the dura mater becomes less attached to the cranial than in comparison with other cranial regions [11].

The coagulation parameters (PLT, PT, and APTT) used to identify coagulation disorder in this report are widely reachable in most healthcare facilities; however, their accuracy in estimating progressive intracranial bleeding is debatable. Engstrom et al. found that a rise in PT and APTT as well as a reduction in thrombocytes were predictors of progressive intracranial bleeding. Other studies, however, imply that progressive bleeding following a head trauma is linked with diffuse coagulation intravascular, as characterized by a high proportion of fibrin degradation products or D-dimer as well as a decreased level of fibrinogen [12]. These criteria could be an indicator of coagulation disorder at an early stage after trauma.

EDH is treated based on several variables, including neurological status (Glasgow Coma Scale (GCS)), hematoma size, patient age, and CT findings. Numerous studies have been reporting a successful conservative treatment in managing epidural hematoma [13]. Patients with a GCS of 13–15, a volume <40 mm, and a midline shift <6 mm are candidates for conservative management. It has been increasingly evident that many slight epidural haemorrhages resolve without com- plications after expectant treatment. In some reports, a few expectantly treated cases required surgical intervention due to deteriorating neurological status. Minor EDH can often extend along the brain's con- vexity and do not produce a midline shift or mass effect, as initial finding in our case. Observation is an effective and safe treatment for this type of EDH, particularly in pediatric patients. In pediatric population, the ne- cessity for surgical approach should be determined by serial neurological exam and radiology evidence.

As stated previously, increased ICP can cause blood vessel constriction and decrease cerebral blood flow, so decreasing ICP can be a goal of acute TBI management. In initial presentation, we found significant headache in our patient, consistent with increased ICP, that was confirmed with image finding. Mannitol is a medication that is commonly used in patients with high intracranial pressure (ICP) and has been demonstrated the effectiveness of mannitol in reducing ICP levels.

In some instances, repeated and prolonged administration of mannitol may paradoxically compensate the brain by producing idiogenic os- moles. It may occur due to mannitol's ability to penetrate cells [14].

According to a study, mannitol may leak through the altered blood-brain barrier in the vicinity of brain gliomas, reversing the initial plasma-to- blood osmotic gradient and possibly promoting ICP rebound. Any disruption of the BBB caused by an injury can also result in the accu- mulation of osmotically active molecules, which can exacerbate cerebral edema. As a result of this negative effect, other solutions were sought to replace mannitol. Other solutions, such as hypertonic saline (HTS), were found to be superior to mannitol in the treatment of TBI-related elevated intracranial pressure (ICP) [15]. In our case, the hemorrhage continued to bleed after expectant management using mannitol and tranexamic acid, along with analgetic was conducted. We performed emergency

evacuation craniotomy due to PEDH, and the patient recovered gradually.

We included tranexamic acid as part of our expectant management.

Tranexamic acid is a plasmin inhibitor that reduces fibrin clot break- down. Tranexamic acid was shown to be effective in reducing progressive intracranial bleeding in adults with TBI, though the effect was not statistically significant [16]. Despite the limited evidence, it is suggested that tranexamic acid administration be considered if there is a significant risk of ongoing bleeding. In our case, progressive bleeding occurred despite the administration of tranexamic acid. However, we found no seizure in our patient, which was said to be significantly increased in children treated with tranexamic acid [17].

One of the main concerns in our case was repeated CT-Scan. There is a well-known correlation between repeated CT scan and increase risk of malignancy on pediatric and young adult population [18]. Even progression of epidural hematoma is reported low, younger age is a known predictor of progression risk. Conversion of treatment approach from expectant to surgical in epidural hematoma was reported significantly increased if initial CT scan was conducted in the first 6 h after accident.

Kim et al. discovered that children with epidural bleeding >10 cc on initial CT were at risk for delayed progressive bleeding, and that follow- up imaging should be taken into consideration in this population.

Nevertheless, the timing and frequency of reimaging are not explicitly described, making it difficult to implement these findings into thera- peutic practice [19]. However, Flaherty et al. (2018) reported that reimaging in pediatric CT scan is common, but rarely changes approach of management, so that reimaging should be conducted only in neurological deterioration setting [20]. A lack of strong evidence hinders the development of guideline on expectant management of TBI in pediatric population. Further research in this area is needed.

4. Conclusion

In conclusion, we reported a delayed progressive bleeding, on six days after admission, in pediatric epidural hematoma treated expectantly. Among the known risk factors, we discovered cranial fracture and young age. We also found that the best timing to consider reimaging was when neurological deterioration happened.

Sources of funding None.

Ethical approval

Ethical approval for case report was exempted in our institution.

Parental consent for minors

Since the patient is a minor, written informed consent was obtained from the patient's parents for publication of this case report and associated images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Author contribution

Andre Marolop Pangihutan Siahaan, Martin Susanto = Study concept, Data collection, and surgical therapy for the patient.

Martin Susanto and Andre Marolop Pangihutan Siahaan =Writing- original draft preparation.

Ade Ricky Harahap, Muhammad Chairul =Editing and writing.

Andre Marolop Pangihutan Siahaan, Donny Luis, and Sonny Giat Raja Saragih =senior author and manuscript reviewer.

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4 Registration of research studies

Not applicable.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Guarantor

Andre Marolop Pangihutan Siahaan.

E-mail address: [email protected] (Andre Marolop Pan- gihutan Siahaan).

Full Postal Address:

Department of Neurosurgery, Faculty of Medicine, Universitas Sumatera Utara, Jl. Dr. Mansyur No. 5, Medan 2015, Indonesia.

Conflict of interest None declared.

Acknowledgment None.

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