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.