Abstract
Minor head injuries in childhood are common and usually resolve without complications. Nevertheless, it may be followed by concussion, hemorrhage, and even ischemic infarcts. Such infarcts are rarely reported but data suggests, less than 2% of ischemic strokes in children are the result of head injury.1,2
Here, we report a case of a five-year-old boy presented with deficit movement of the right-side of the body and total cessation of speech after a fall from bed three days prior to the hospital visit. The diffusion-weighted MRI image showed an infarct in the left basal ganglia region. Other causes of stroke in the child were ruled out. The child was managed conservatively. Basal ganglia infarct secondary to minor trauma is a rare event. Possible mechanisms leading to such an event and management are discussed in this report. 1–4
Keywords: Infarct, trivial trauma, basal ganglia, stroke, aphasia.
Introduction
Stroke is one of the top 10 common causes of childhood deaths. However, mild head bumps not unusual during childhood very seldom progress to infarction but if it does then the most common sites of infarcts are striatocapsular and the basal ganglia region.4 The traumas are unlikely to cause basal ganglia bleeding5 but can lead to acute ischemic stroke. The lenticulostriate arteries supplying the basal ganglia are the small perforating arteries arising from the anterior part of the circle of Willis (Fig-1).6 These are end arteries, often lacking collateral supply and mineralizing angiopathy of the lenticulostriate arteries is the main factor of the infarction as they are susceptible to occlusion following minor trauma.6
Fig-1: Circle of Willis7
Affected children present with hemiparesis and facial paresis soon after trauma, and there is a typical finding of infarcts and calcification in basal ganglia on neuroimaging.4
Most children have a complete recovery of their symptoms with no recurrence following a six-month follow-up.6
Case Report
A five-year-old boy came to Square Hospital in August 2020 with deficiency of movement of the right side of his body and loss of speech. This was preceded by a fall from bed three days back. There was no associated history of seizures. On examination the child was sick-looking and pale with no sign of meningeal irritation. There was café au lait spots on his skin, reduced tone on the left side, he could not close his left eye completely and had mild drawing of angle of mouth towards left. His prothrombin time was 10.2 seconds (normal range is 12-17 seconds), control 11.4 seconds and INR 0.89, activated partial thromboplastin time was 19.8 seconds (normal range is 26-36 seconds) and control 27.5 seconds. The complete blood count report showed hemoglobin level as 11.6gm/dl, lower neutrophils (39.3%) and elevated eosinophils (25%). The homocysteine level was normal, and the ECHO cardiography report also showed no abnormality. A diffusion weighted MRI image (Techniques-T2 FLAIR Axial, DWI Axial, ADC Axial and 2D TOF MIP Axial & 3D MIP) of brain showed a well-defined area of restricted diffusion in left capsuloganglionic region involving jenu and posterior limb of internal capsule which was dark on ADC (Fig-2). The Magnetic Resonance Angiography (MRA) findings of intracranial arteries and branches were unremarkable (Fig-3). After ruling out other possibilities of the diagnosis it was established that the child suffered acute stroke syndrome with right sided hemiplegia and aphasia as a result of the fall as per the history. The child was treated conservatively with Aspirin 75 mg once daily and multivitamin. The patient was evaluated 12 months after the episode, and he had recovered fully. A recent follow up of the patient confirmed the child is doing well and there was no recurrence. Basal ganglia infarcts secondary to minor trauma is a rare event. Possible mechanisms leading to such an event and management are discussed in this report.3,8
Fig- 2: Diffusion Weighted Image MRI of brain- DWI Axial on the left and ADC Axial image on the right shows acute left capsuloganglionic infarct.
Fig-3: MRA shows no remarkable changes of intracranial arteries.
Discussion
A study by Yang FH of 16 infants less than 18 months of age showed that about 62% of infants developed symptoms of hemiparesis, facial paresis, and convulsions within 72 hours of minor trauma and on scanning showed basal ganglia calcification. Another study by Lingappa L et al. reported 22 cases of infants presenting with stroke and basal ganglia calcification after minor fall with similar symptoms. Gowda VK published a paper of 38 children not only showing similar presentation but two were siblings, indicating an underlying genetic predisposition. Some cases may also remain asymptomatic and may present later with more severe trauma. According to the National Institute of Health, CT scan is the investigation of choice to visualize the punctate calcification in basal ganglia. Most of the children recover completely without recurrences but the management with aspirin and duration of therapy remains unclear.4
Study of a total of 94 pediatric patients in a span of five years having ischemic arterial stroke in North India were analyzed where 48 patients were diagnosed with basal ganglia stroke following minor head trauma. The onset of symptoms occurred 60 minutes after the trauma; 45 out of the 48 patients had hemiparesis, 13 patients had transient hemi dystonia on the side of the hemiparesis four days after the trauma. CT scan showed basal ganglia calcification and MRI showed infarcts in areas supplied by lenticulostriate arteries of basal ganglia. The majority of patients showed complete recovery at 18 months of follow-up. 5 Another study published in 2016 in India described a case of a 10-month-old girl presenting with right sided hemiparesis affecting her right-sided arm and leg along with right sided facial palsy with a history of fall while trying to walk on her own, 12 hours prior to the onset of the symptoms. There was no vomiting or loss of consciousness. The child could not sit or stand. MRI showed an infarct involving the left capsuloganglionic region. The child was managed with osmotic diuretics and low molecular weight heparin was given for five days then stopped. The child improved 12 hours after the treatment and in 10 days there was resolution of weakness. This case (Fig-4) and the presenting case at Square hospital have similar MRI images.2
Fig-4: MRI Brain restricted diffusion on DWI on the left and ADC image on the right are both suggestive of left capsuloganglionic acute infarct2
A case study of a 2-year-old-boy brought in a hospital with right-sided hemiparesis also had a history of minor fall while playing on the ground. The child did not lose consciousness, had no history of vomiting, seizure, or bleeding from ear, nose, or throat. The weakness of the limb was noticed 16 hours post trauma. CT scan showed calcification in bilateral basal ganglia region and MRI showed well-defined area of restricted diffusion in left thalamus and adjoining basal ganglia suggestive of infarct. MRA showed normal blood flow through internal carotids, anterior cerebral, middle cerebral, both vertebral and basilar arteries. The child was managed with aspirin and antiepileptic drugs. A follow-up after three months showed improved muscle tone of the limbs.1
A relevant case published from Karnataka, India described an 11-year-old previously healthy girl presenting with right upper and lower limb hemiparesis, deviation of face and slurring of speech after a fall from stairs three days prior to the symptoms. CT scan was normal, MRI of brain showed left gangliocapsular region, left corona radiate and left frontotemporal lobe which are areas supplied by left middle cerebral artery (Fig-5). The MRA confirmed the diagnosis showing absent blood flow of the left middle cerebral artery suggesting occlusion of the artery (Fig-6). The child was managed with low molecular weight heparin for 10 days and within 7 days she showed improvement. She was discharged with aspirin and atorvastatin for 6 months. Upon follow up she showed complete recovery.9
Fig-5: MRI brain DWI and ADC showing diffusion restriction suggestive of left sided infarct supplied by middle cerebral artery9
Fig- 6: MRA showing blood flow disruption in left middle cerebral artery suggestive of occlusion9
Pathogenesis
There are several hypotheses regarding the pathogenesis of ischemic stroke after minor head injury in children. These are based on the structural features of intracranial blood vessels and brain of children that make them more vulnerable to mechanical injury.10 The broad understanding is, trivial trauma mechanically results in brain shift which may stretch the arteries, causing vasospasm that disrupts the flow between the fixed intracerebral and mobile extracerebral parts of the deep perforating vessels. The spasm leads to intimal lesion and thrombus formation. Another explanation is, the trauma can cause spasm of the middle cerebral artery, and if the child continues to cry for a while, there will be decrease pCO2 which will further aggravate the vessel spasm. Infectious agents like echovirus, cytomegalovirus, Epstein Barr virus, and mycoplasma may also secondarily cause mineralizing microangiopathies. 5,11
Fig-7: Lenticulostriate arteries from Middle Cerebral Artery 12
The lateral lenticulostriate arteries, anterior choroidal artery, posterior communicating artery from the middle cerebral artery along with the medial lenticulostriate artery and recurrent artery of Heubner from anterior cerebral artery form part of deep perforators which are also end arteries (Fig-7). In children the lateral lenticulostriate arteries have a more acute angle with middle cerebral artery and are shorter in length; these specific anatomical features and the relatively unmyelinated brain in children makes them more susceptible to injury. As explained above, the sudden trauma displaces the brain, stretches the arteries that specifically injures the mobile segment of the lenticulostriate arteries that lies between the fixed intracerebral and extracerebral parts. This jerk may also cause vasospasm and induce ischemia. Both the ischemia and endothelial injury of lenticulostriate arteries lead to thrombus formation and infarction.1,10
Although angiopathy means disease of blood vessels, the term is commonly used for damages of small blood vessels.13
The mineralizing angiopathy of lenticulostriate arteries that is mainly being discussed in this study is visualized as punctate calcification of basal ganglia in CT scans. The specific aetiology of mineralization in this type of case remains unknown.1,3
If mineralizing angiopathy results in infarction of brain tissue and is manifested in unilateral symptoms of hemiparesis as was discussed in the cases above, then further scanning is required because CT scan may miss the infarct.
Confirmatory Diagnostic Imaging
When children present with neurological deficit after a head trauma, it is mandatory to perform brain scans. CT scan is mainly done to exclude the presence of hematoma.
If hematoma is absent then other causes of cerebral ischemia like embolism from acquired or congenital cardiac diseases, arterial dissections, dehydration, meningitis, homocystinuria, Down’s syndrome, Williams syndrome, sickle cell disease, etc must be ruled out before establishing a case of ischemia due to the head trauma.1,2
To detect the linear calcification (mineralizing angiopathy) of lenticulostriate vessels, CT scan with sliced multi-planar reconstruction is the investigation of choice. However, it will not necessarily show the infarct.9,10
Since CT scan is not reliable in showing parenchymal infarcts from ischemic stroke within 24 hours of the stroke, MRI is mandatory in such cases.9 The Diffusion Weighted Imaging (DWI) and Apparent Diffusion Coefficient (ADC) map in MRI are considered the gold standard and is crucial to detect infarcts from ischemic stroke in infants and children.9,14 MRA is also recommended to scan the blood flow within the intracranial blood vessels and the neck vessels for detecting occlusion of arteries. 1,9
All these protocols were followed in the case at Square Hospital to finally establish the diagnosis of an infarct as a result of trivial trauma.
Conclusion
Minor head injuries are common in children which may result in brain concussion but mostly subside without any complication. The above case presented, and the relative studies prove that mineralizing lenticulostriate vessels’ angiopathy is a well-recognized cause of ischemic stroke in healthy children post minor trauma or fall. It is usually represented with neurological deficit in the form of hemiparesis a few hours after the injury and is not associated with seizures. There is a distinct clinic-radiological entity that has favorable prognosis with antithrombotic therapy.10,15
Basal Ganglia
Basal ganglia are part of the brain mainly responsible for motor control. It is known as the gatekeeper of initiating voluntary movements as it effectively chooses which action to approve and what to reject. It is also associated with reward, cognitive, decision-making functions, and refines the movements further from the signals received from sensory stimuli.16–18
Anatomically, it is a group of subcortical nuclei situated at the base of the brain which includes the caudate nucleus, globus pallidus, putamen, substantia nigra pars reticulata, subthalamic nucleus and ventral pallidum (Fig-8). They are like an electrical circuit board connected to different parts of the brain sending signals back and forth.16
Fig-8: Basal Ganglia Structures19
Basal ganglia pathology can affect balance and coordination, muscle weakness, cause tremors and shakiness, slurred speech, vision problems, and may lead to Parkinson and Huntington disease.16,18
The cases mentioned above are serious concerns as it involves children who have a long way to develop physically, mentally, emotionally, and spiritually. The brain is one organ of the human body that continues to develop till the mid to late twenties.20.
Stroke remains a life threatening and debilitating condition. Although rare, stroke in children is not totally uncommon as is evident. It can progress to a recurrent condition and arterial ischemic stroke can further develop into a hemorrhagic condition as a complication of ischemic injury itself or from the drugs used to treat it.21 Thus, any neurological deficit in children must be addressed at once, without delay. If an infarction can be diagnosed immediately with diffusion weighted MRI along with an MRA to scan the intracranial blood vessels to detect the presence or absence of occlusion, then in most instances, basal ganglia infarct following trivial trauma showed good outcome with conservative management. More studies on the pathogenesis, genetic cause and follow up of cases are needed to come to a consolidated conclusion.
Authors of this article:
1. Prof. Brig. Gen (Retd) Jahangir Alam, MBBS, MCPS, FCPS (Radiology), Fellow MRI (USA), Head-Radiology & Imaging Operation, Ex-Head, Department of Radiology & Imaging, Armed Forces Medical College & CMH Dhaka. Ex-Senior Consultant & Coordinator, Diagnostic & Interventional Radiology, Apollo Hospitals Dhaka.Visiting Professor-Paediatric Neuroradiology, Dhaka Shishu Hospital.
2. Dr. Maliha Mannan Ahmed, MBBS (BMC), MBA (ULAB), Masters in Healthcare Leadership (Brown University, USA) and Level 1 Certification on Precision Nutrition.
The Executive Editor of The Coronal.
3. Dr. Major (Retd) Zeena Salwa, MBBS, DCH, FCPS (Paed) Clinical Training on Paed Neurology Training on EEG and Epilepsy in USA Consultant, Paediatrics & Child Development Centre (CDC).
4. Dr. Jannatul Ferdaus, MBBS, MD (Radiology) Gold Medalist Specialist-Radiology & IMAGIING.
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