Korean Journal of Cerebrovascular Surgery 2008;10(2):383-386.
Published online June 1, 2008.
Ruptured Intracranial Aneurysm Successfully Treated by Clipping in a Patient with Idiopathic Thrombocytopenic Purpura: A Case Report.
Choi, Jae Eun , Joo, Sung Pil , Seo, Bo ra , Kim, Tae Sun
Department of Neurosurgery, Chonnam National University Hospital & Medical School, Gwangju, Korea. nsjsp@chonnam.ac.kr
Abstract
We report here on a case of a ruptured left posterior communicating artery (P-com) aneurysm that was treated by clipping in a patient with idiopathic thrombocytopenic purpura (ITP) and steroids were used to control the platelet count during the perioperative period. A 34-year-old female who had been suffering from ITP for four years experienced the sudden onset of a headache and vomiting while showering. She was referred to our hospital as a case of subarachnoid hemorrhage (SAH) due to a ruptured P-com aneurysm. Aneurysmal neck clipping was performed via the left pterional approach 6 hours after the ictus. The aneurysmal clipping was successful without an increased bleeding tendency during the operation. Intravenous steroid injection was given after aneurysmal clipping for 7 days and then it was tapered off. It is important to maintain an adequate platelet count in SAH patients with chronic ITP in order to avoid hemorrhagic diathesis during surgery. Intravenous steroid injection is a helpful method for maintaining an adequate platelet count in these patients during surgery.
Key Words: Aneurysm, idiopathic thrombocytopenic purpura, steroid

Introduction 


  
Idiopathic thrombocytopenic purpura (ITP) was first described by P.G Werlhof in 1735.4) The disease is characterized by the premature destruction of autoantibodycoated platelets, causing thrombocytopenia and subsequent bleeding. Terada et al.9) reported a case of subarachnoid hemorrhage (SAH) with ITP but SAH in patients with ITP is very rare on literature, and neurosurgical intervention in patients with ITP is also rare because intraoperative bleeding is difficult to control.3)4)8) Intraoperative bleeding control in ITP patients is very important in order to achieve a good postoperative result.8) 
   We report a case of successful clipping of the ruptured left P-com aneurysm associated with ITP after perioperative steroid therapy. 

Case Report 

   A 34-year-old female who had been suffering from ITP for 4 years experienced the sudden onset of a severe headache and vomiting while taking a shower. She was referred to our hospital and computed tomographic (CT) scans revealed SAH (Fig. 1A), Her mental status became aggravated after brain CT angiogram (CTA), thus the CT scan was immediately re-evaluated, and rebleeding was confirmed after follow-up CT (Fig. 1B). The patient's final preoperative platelet count was 43×103/mm3, and her platelet count on admission was 155×103/mm3 (Fig. 2, Table 1). Calcortr
® (the methyloxazolin derivative of Prednisolone) 24 mg was administered in order to maintain the platelet count before operation. Brain CTA showed an aneurysm in the left P-com artery (Fig. 1A). Craniotomy was performed under operating microsopic guidance via left pterional approach. Sylvian dissection was done, hematoma was observed during dissection and some of them were removed. Then aneurysmal neck clipping was secessfully performed. Postoperative CT scan showed that the aneurysmal clipping was successfully performed with an estimated blood loss of 500 ml, but the hematoma remained (Fig. 1C). 
   A neurological exam was immediately performed after the operation using the Glasgow Coma Scale, on which the patient showed eye opening to voice, verbal response to intubated, and motor response to localized to pain with right hemiparesis grade IV-. Extubation was performed on the first postoperative day. Follow-up brain CT scan was performed on the third postoperative day and revealed that the hemorrhage had increased in size, with an 8-mm midline shift to the right. However, we decided to maintain conservative treatment, which included mannitolization and intravenous steroid injection, because mental deterioration did not occur. The patient's platelet count remained greater than 85×103/mm3 during the postoperative period (Fig. 3). Her mentality gradually improved, and she was subsequently referred to the Department of Rehabilitation Medicine for general rehabilitation treatment. She continued to experience intermittent headaches, and her right hemiparesis showed improvement to grade IV-/IV+ at the final follow-up performed in the outpatient department 11 months after the operation. 

Discussion 

   The platelet counts in ITP patients can be increased by steroids, immunosuppressant agents, splenectomy, platelet transfusion, and high-dose gamma globulin therapy.3)4) However, the risk of severe bleeding is low in chronic ITP patients, and treatment is generally not indicated until platelet levels drop below 30×103/mm3.4) Steroids and immunosuppressant agents cannot achieve an increase of platelet counts that is sufficient to control intraoperative bleeding.8) Therefore, uncontrolled intraoperative bleeding is most fearful to neurosurgeons during surgery. Splenectomy is still the most effective treatment option for patients who are refractory or have a relapse after steroid administration.4) Humphreys et al.2) performed an emergency splenectomy followed by platelet transfusion in four ITP cases with intracerebral hematoma and achieved good postoperative results. However, splenectomy is only 50
~85% effective.7) Platelet transfusion will increase the platelet counts, but the effect is temporary and may lead to the formation of antiplatelet antibodies. Imbach et a1.3) introduced high-dose gamma globulin therapy in 1981. The resultant increase in platelet count was adequate to tolerate surgery within a few days and remained effective for about a week.3) Gamma globulin plays a role in reducing platelet destruction by the reticuloendothelial system and contributes to the inhibition of anti-platelet antibodies binding to platelets.8) 
   Cerebral aneurysm does not always require immediate surgery, unlike intracranial hemorrhage with cerebral herniation,5) and the operation can usually be delayed until the possibility of hemorrhagic diathesis can be controlled. However, we experienced rebleeding and hematoma formation in our patient, and therefore decided to perform an emergency operation. 
   Craniotomy and aneurysmal clipping or endovascular coil embolization are the treatment of choice in SAH patients. Endovascular coil embolization is hard to control when bleeding occurs during intervention. Therefore, We decided craniotomy and aneurysmal clipping were performed in our case because we figured it would be easier to control than endovascular treatment if intraoperative bleeding were to occur. 
   We assumed that this chronic ITP patient might be at an increased risk for the possibility of aneurysmal rupture due to the natural course of ITP, such as the concern of vasculitis. 
   Our patient's neurological condition was not good due to complications of SAH, and the bleeding was not caused by a diathesis, but was rather due to the natural course of ruptured intracranial aneurysms. The patient had been given oral steroids before the SAH attack in an attempt to maintain the platelet counts. Therefore, we changed the medication to intravenous steroid injection (the steroid was tapered off) during the perioperative periods, and the platelet counts did not fall to the level at which hemorrhagic diathesis may occur during perioperative periods. 
   Very rare cases of cerebral aneurysm associated with ITP have previously been reported.8) We believed that this case was an incidentally detected case of SAH with chronic ITP. Though management of ITP patients with ruptured cerebral aneurysms has rarely been discussed it is known to be important to maintain an adequate serum platelet count in order to control bleeding. We recommend perioperative steroid therapy with or without transfusion followed by after the hemorrhagic diathesis is completely controlled. 

Conclusion 

   We successfully treated an ITP patient with a ruptured intracranial aneurysm by clipping. We also emphasize that it is important to maintain adequate platelet counts in SAH patients with chronic ITP in order to avoid hemorrhagic diathesis during surgery. Intravenous steroid injection is a helpful method for maintaining platelet counts during surgery in these patients, as noted. 


REFERENCES


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