Delayed recurrence of acute subdural hematoma in a patient with plasminogen activator inhibitor mutation
- Department of Neurosurgery, University of Louisville, Louisville, Kentucky, United States.
DOI:10.25259/SNI_180_2020Copyright: © 2020 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.
How to cite this article: Zaid Aljuboori, Mohammed Nuru, Alexandria Schaber, Haring Nauta, Emily Sieg. Delayed recurrence of acute subdural hematoma in a patient with plasminogen activator inhibitor mutation. 18-Sep-2020;11:292
How to cite this URL: Zaid Aljuboori, Mohammed Nuru, Alexandria Schaber, Haring Nauta, Emily Sieg. Delayed recurrence of acute subdural hematoma in a patient with plasminogen activator inhibitor mutation. 18-Sep-2020;11:292. Available from: https://surgicalneurologyint.com/surgicalint-articles/10270/
Background: Plasminogen activator inhibitor type I (PAI-1) is important for balancing the fibrinolytic effect of plasmin, and deficiency can result in increased risk of bleeding. We report a case of a patient with PAI-1 deficiency who presented with delayed spontaneous recurrence of an acute subdural hematoma (aSDH) after evacuation.
Case Description: A 29-year-old male presented with altered mental status (AMS) after a fall at a construction site with Glasgow Coma Scale (GCS 4T). His coagulation profile was normal, and brain computed tomography (CT) showed a left-sided aSDH. He underwent emergent evacuation of the hematoma. On postoperative day 2, he was started on heparin for venous thromboembolism (VTE) prophylaxis. His neurological examination improved and was discharged with no focal deficits. Three days later, he presented with sudden AMS (GCS 7T); CT head showed a large hematoma at the site of original surgery. The hematoma was evacuated emergently. On readmission, the family informed providers that the patient had a history of PAI-1 deficiency. Postoperatively, only mechanical VTE prophylaxis was used and the patient was started on oral TXA per hematology recommendation. The patient improved and was discharged with no focal deficit. On follow-up, he remained neurologically stable.
Conclusion: PAI-1 deficiency should be suspected in patients with delayed posttraumatic/surgical bleeding and a normal coagulation profile. If PAI-1 deficiency is evident or suspected, then a trial of antifibrinolytic agent should be used to treat and prevent recurrence of bleeding. Furthermore, chemical VTE prophylaxis should be avoided as it increases the risk for bleeding.
Keywords: Fibrinolysis, Plasminogen activator inhibitor, Subdural, Tranexamic acid, Trauma
Plasminogen and its activation system are an important part of the normal coagulation cascade as they promote fibrinolysis of mature thrombi.[
The patient is a 29-year-old Amish male who presented with altered mental status (AMS) after a fall from height. His initial Glasgow Coma Scale (GCS) was 4T with a dilated nonreactive left pupil. His computed tomography (CT) of the brain showed an acute left-sided aSDH with a 15 mm rightward shift [
The patient presented 3 days later with sudden AMS that was preceded by a sudden headache. His GCS was 7T and CT head showed large aSDH at the site of previous surgery [
Plasmin is the primary enzyme responsible for fibrinolysis and is formed by the action of tissue plasminogen activator on plasminogen.[
The management of patients with PAI-1 deficiency requires a multistep approach, and it is important to coordinate with the hemoncology team as they have the required expertise to guide the process [
For the treatment of acute hemorrhage (e.g., intracranial hemorrhage with or without evacuation), the administration of intravenous antifibrinolytics (e.g., TXA) is effective in controlling the bleeding in PAI-1 patients.[
The patient education regarding bleeding manifestations and when to seek treatment is important. Furthermore, patients should be offered genetic counseling, since PAI-1 deficiency is inherited in an autosomal recessive manner.[
PAI-1 deficiency is a rare hereditary bleeding disorder that was recently discovered. It should be considered in patients with delayed posttraumatic or postsurgical bleeding, especially in a population known to harbor the genetic mutation (e.g., Amish). If PAI-1 deficiency is established or highly suspected, then a trial of antifibrinolytic agents (e.g., TXA) should be considered. They can also be used as prophylaxis to prevent bleeding in patients with PAI-1 deficiency before any surgical intervention. Finally, chemical VTE prophylaxis puts the patients at risk of bleeding.
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