Case report:
A 16-year-old boy presented with sudden onset, rapid progressive weakness of the left side extremities. In the exam, the patient had right side hypoesthesia to pain and light touch (positive brown squared syndrome). No urinary or fecal incontinency was detected in the physical exam. The patient had no positive history for any recent trauma, and his past medical history was negative for any hematologic disease. Laboratory exams did not show any coagulopathy or blood dyscrasia.
Preoperative MRI was done, showing a heterogeneous extra- medullary lesion posterolateral to the cord, spanning from C2 to C6 (Fig. 1) with hyper-intensity in T1-weighted images, hypo-intensity in T2-weighted images, and without significant enhancement after gadolinium injection. Significant compression was made on the cord roots in the left side.
The patient was transferred to the operation room and underwent emergency decompression by C3-C7 conservative laminectomy. The dura was opened and subdural hemorrhage was evacuated. During the operation, a hematoma was visualized upon the opening of the thecal sac and evacuated with gentle suction (Figure 2). No evidence of abnormal vasculature or masses was observed. Hematoma fragments were collected and sent for histo-pathologic evaluation. Pathologic report of the lesion confirmed the diagnosis of hematoma.The patient underwent magnetic resonant angiography (MRA) post-op for detection of vascular disorders, but it did not show any abnormality.
In the post-operation examination, the cervical pain was completely healed and two days post-operation the weakness in the extremities was gradually improved. Left side extremity strength improved to consistent grade 2/5 throughout with reported rare ability to move his leg against gravity. Four weeks after surgery in outpatient follow up, motor power showed a remarkable improvement, and he could walk on his own feet.
Review of the Literature :
A review of the English literature was conducted by searching Medline and PubMed from 2010 to August 2021. The terms “spinal subdural hematoma”, “acute spinal subdural hematoma”, “spontaneous spinal subdural hematoma” and “idiopathic spinal subdural hematoma” were used. In a search of Medline, totally 42 articles were found. The papers were reviewed; if the history of trauma, coagulopathy, vascular abnormality or iatrogenic cause was found, the papers were excluded. The studied factors were age, gender, sign and symptoms, level of involvement, type of treatment (surgical intervention or conservative therapy), and patient outcome. After exclusion of unrelated articles, 20 papers were included in our study containing the present case(1, 6-20) (Table 1).
To the best of our knowledge, this is the first case of acute non-traumatic idiopathic SSDH in pediatric age patients which presented with acute paresthesia and weakness. Previously, 20 cases were reported in the literature on spontaneous spinal SDH from 2010. Of these cases, 12 were male and 7 were female. The patients’ age ranged from 27 to 79 years with a mean of 49 years; only 8% were under 30, and one patient was less than 27. In most of the cases (80%), thoracic and cervicothoracic areas were involved; also, the lumbosacral area was involved in 14% of cases and in only 1 case (present case) cervical segment was involved. The most common presentation was acute paresthesia and back-pain. Other common symptoms consist of bladder, bowl dysfunction, and paraplegia. Laminectomy was done as the main surgical intervention in 8 cases and conservative therapy in 13 cases. 17 cases experienced good recovery, 3 of whom had partial recovery and only 1 case had poor recovery. The percentage of good recovery in those who underwent surgery was 62.5%, while this percentage was 84.7 in the conservative treatment group.
Our case was the first report of SSDH in pediatric and the 3rd case of cervical subdural hematoma. The outcome of spontaneous subdural hematoma in the spine seems to be favorable. About 90% of cases had partial recovery or better. The presented case also had a significant recovery after operation and was discharged with normal limbs strength from hospital and could walk normally without a stick in outpatient fallow up.