"Rabbit Ear" scalp deformity caused by massive subdural effusion in infant following bilateral burr-hole drainage

Abstract

Subdural effusion (SDE) in an infant is a rare clinical scenario, which may be secondary to a variety of etiologies. Massive SDE is an extremely rare complication of head injury. It usually runs a self-limiting course. Though neurosurgical intervention is occasionally needed, different methods of surgical procedure for management includes burr-hole alone, burr-holes with subdural drain placement, twist drill craniotomy with drain and even craniotomy. The authors report a rare case of progressive massive SDE, which despite bilateral burr-hole placement and drainage failed and presented with visual deterioration and massive bulge of scalp at burr-hole sites producing rabbit ear sign in a 10 month old infant. Ultimately cystoperitoneal shunt was carried out in a desperate attempt to prevent impending rupture of scalp sutures at sites of previous burr-hole placement. Astonishingly not only complete resolution of hygroma, but visual recovery also took place. Patient is doing well at 6 months following shunt with regaining normal vision and appropriate developmental milestones. A magnetic resonance imaging scan of brain at last follow-up revealed mild ventriculomegaly with subduro-peritoneal shunt in situ and rest of brain was unremarkable. Such cases have not been reported in literature until date.

Commentary

“…Persistent accumulation of fluid in the subdural space may be either hygroma or hematoma or effusion. [1] Subdural effusion (SDE) following decompressive craniectomy for traumatic brain injury constitutes one of the common late complication. [2] SDE may show spontaneous resolution or enlarge with passage of time.…”

“…[6] Treatment modalities including pediatric cases are tapping of anterior frontanelle, single stage burr-hole, burr-holes with drains placed in subdural space kept for few days, drain placement following simple twist drill, and even simple craniotomy. [1,[4][5][6] Although every therapeutic surgical option has some limitation, frontanelle tapping can be used in infants for all stages of SDE and being a minimal invasive and massive hemorrhage is very rare occurrence provided punctures are carried out using small needle. However, continuous external drainage following burr-hole or twist drill potentially carry a risk of meningitis and usually advocated to be kept for less than 5 days.…”

“…[7] Satyarthee et al also reported a rare interesting case of progressive massive SDE in a 10-month-old male infant, who despite bilateral burr-hole placement and drainage, after discharge from hospital presented with progressive diminution of visual acuity and massive scalp bulge at placed burr-hole sites, mimicking rabbit ear sign, necessitated subduro-peritonreal shunt desperately in a prophylactic attempt to prevent impending scalp swellings rupture. [1] So timely detection of such progressive cases of SDE, although very rare incidence, is crucial and appropriate measure can avoid catastrophic neurological sequlae.…”

Is subduro-peritoneal shunt surgery the first or last resort in managing subdural effusion developing after supratentorial tumor surgery in infancy?

“…Further infantile desmoplastic ganglioglioma carries a favorable prognosis after gross total resection despite being aggressive and infiltrating in nature. [ 5 ] VandenBerg et al . recommended against requirement of adjuvant therapy, even after incomplete resection.…”

“…Was drastic shunt surgery warranted or was a trial of minimally invasive procedure like anterior frontanelle tapping, twist drill or burr hole drainage prudent. [ 3 4 5 6 7 ]…”