Positional aphasia as a manifestation of syndrome of the trephined: illustrative case.

Erica C Johnson,William C Broaddus,Patrick F O’Brien

doi:10.3171/case21629

Erica C Johnson, William C Broaddus + Show 1 more

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https://doi.org/10.3171/case21629

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Abstract

BACKGROUNDThe authors report a case of a 66-year-old male who presented acutely with a subdural hematoma who was managed operatively with craniotomy. His course was complicated by a postoperative epidural hematoma, which, on the basis of intraoperative findings at the second surgery, was managed with evacuation of the hematoma and removal of the bone flap.OBSERVATIONSThe patient’s subsequent recovery was remarkable for a reproducible positional aphasia in the early postoperative period with an ultimate diagnosis of syndrome of the trephined. The patient’s cerebral edema permitted early autologous cranioplasty, which resulted in resolution of the patient’s symptoms.LESSONSThe authors believe this case to be the first described of isolated positional aphasia as a manifestation of syndrome of the trephined. Recognition and treatment of the syndrome resulted in a positive patient outcome.

Highlights

The authors report a case of a 66-year-old male who presented acutely with a subdural hematoma who was managed operatively with craniotomy
The “syndrome of the trephined” or “sinking skin flap syndrome” is a rare complication of a craniectomy characterized by postoperative neurological deterioration caused by cortical dysfunction of the area below the craniotomy that improves after cranioplasty.[1]
Observations The syndrome of the trephined is a rare phenomenon whose etiology and manifestations have yet to be fully described in the literature

Summary

BACKGROUND

The authors report a case of a 66-year-old male who presented acutely with a subdural hematoma who was managed operatively with craniotomy. The “syndrome of the trephined” or “sinking skin flap syndrome” is a rare complication of a craniectomy characterized by postoperative neurological deterioration caused by cortical dysfunction of the area below the craniotomy that improves after cranioplasty.[1] Ashayeri et al.[2] published a review in 2016 based on 54 cases that found this syndrome can occur in adults at any age, with an average reported age of 49 years and a male predominance (60%) This phenomenon was most often found after craniectomy for treatment of neurotrauma, and the time to onset of symptoms after surgery was 5.1 ± 10.8 months after surgery.[2] This review described presenting symptoms in order of frequency. We present a case of positional aphasia as the solitary manifestation of the syndrome of the trephined that became apparent on postoperative day 3 after decompressive craniectomy for an epidural hematoma

Illustrative Case

Discussion