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| ANALYTICAL REPORT |
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| Year : 2012 | Volume
: 8
| Issue : 1 | Page : 145-147 |
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Meningioma with hemorrhagic onset: Two case reports
Mukul Vij1, Sushila Jaiswal2, Awadhesh Kumar Jaiswal3, Sheo Kumar4, Sanjay Behari3
1 Department of Pathology, Global Hospital and Health City, Chennai, India
2 Department of Pathology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India
3 Department of Neurosurgery, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India
4 Department of Radio Diagnosis, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India
| Date of Web Publication | 19-Apr-2012 |
Correspondence Address:
Mukul Vij
Department of Pathology, Global Hospital and Health City, Chennai
India
DOI: 10.4103/0973-1482.95198
Haemorrhage is a rare complication of meningiomas that can occur spontaneously, after embolization, stereotactic radiation and perioperatively. Our first case was a 16 year old male, admitted with spastic quadriparesis, and retention of urine. Magnetic Resonance Imaging (MRI) revealed anteriorly placed cervical intradural extramedullary mass. Patient underwent emergency surgery following sudden worsening of neurological symptoms and intratumoral bleed was noted peroperatively. Tumor was labeled as angiomatous meningioma with hemorrhage. The second case was of a 45 year female who presented with history of sudden onset weakness in right upper and lower limb followed by unconsciousness. MRI revealed heterogeneous lesion in left parasagittal area with intratumoral bleed. Left frontal craniotomy with tumour decompression was performed. Tumour was labelled as meningothelial meningioma with haemorrhage. Meningiomas with hemorrhagic onset remain rare, and pathophysiology is still incompletely understood. Prevention and outcome of intratumoral haemorrhage highly depends on early diagnosis and adequate treatment.
Keywords: Meningioma, intracranial, spinal, haemorrhage
How to cite this article:
Vij M, Jaiswal S, Jaiswal AK, Kumar S, Behari S. Meningioma with hemorrhagic onset: Two case reports. J Can Res Ther 2012;8:145-7 |
| > Introduction | |  |
Intracranial tumours presenting with haemorrhage constitute 1.5 to 5.4% of all tumours, more in malignant pathology such as metastatic tumours or gliomas. [1] Haemorrhage is a an unusual presentation of benign tumours except in cases of pituitary adenoma. [2] Meningiomas are benign, slow-growing, and highly vascularised tumours; it is extremely rare that their onset is apoplectiform, mimicking cerebrovascular incidents and thus affecting diagnostic workup, treatment, and outcome. [3] Intratumoral haemorrhage (ITH) can occur spontaneously, after embolization, stereotactic radiation and perioperatively. [4],[5] Extratumoural haemorrhage, e.g. subdural, subarachnoid, intraparenchymal and intraventricular, have also been described. [3],[5] Haemorrhage is more common in intracranial meningioma. Only four case reports of spinal meningioma, all with extratumoral subarachnoid haemorrhage have been published. [6] We present two cases of intratumoral bleed in meningioma including first case report of spinal meningioma with ITH.
| > Case Reports | | |
Case 1
The first case is of a 16 year old male admitted with spastic quadriparesis and numbness for 2 months, straining at micturition for 1 month and retention of urine 20 days back. On examination, motor power of right upper limb, left upper, left lower and right lower limb was 2/5, 1/5, 3/5, and 4/5 respectively. All deep tendon reflexes were exaggerated. Bilateral planters were extensor. Sensory loss was 50-70% for all modalities below C5. The clinical possibility of neurofibroma or meningioma was kept. MRI showed an anteriorly placed dural based lesion from C2 to C5 levels which was isointense lesion on T1 and T2 weighted images with enhancement on contrast administration [Figure 1]A-C. The lesion was compressing the cord. Radiological impression was anteriorly placed intradural extramedullary meningioma. After admission, he had sudden onset worsening of neurological status with tachypnoea. He was taken for emergency surgery. Via posterior midline approach, C2-C5 laminectomy was done. Gentle internal decompression was done and entire tumor was removed in piece meal under operating microscope. Tumor was pinkish, firm, partially suckable and vascular [Figure 2]. Evidence of intratumoral bleed was present. Histopathology revealed a cellular tumor with prominent congested vessels and areas of hemorrhage [Figure 3]. The tumor cells were arranged in whorls with oval nuclei having pseudoinclusions and variable eosinophilic cytoplasm [Figure 4]. Tumour was labeled as angiomatous meningioma with hemorrhage. Patient expired following refractory ventricular fibrillation following hypokalemia on 3 rd post operative day.  | Figure 1: Magnetic resonance imaging images of cervical spinal meningioma: T1 image showing isointense (arrow) lesion (A), T2 image showing isointense (arrow) lesion (B), enhancement (arrow) following contrast administration (C)
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 | Figure 2: Intraoperative image showing spinal meningioma with areas of bleed
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 | Figure 3: Histopathological examination revealing a cellular tumor of spinal meningioma with prominent congested vessels and areas of hemorrhage
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 | Figure 4: Meningioma displaying whorls of tumor cells with areas of hemorrhage
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Case 2
The second case was of 45 year female who presented with history of sudden onset weakness in right upper and lower limb followed by unconsciousness for 4 days. On examination, patient was conscious, however not verbalising. Pupils were reacting to light. Her Glasgow coma scale was E1, V1, M5. Her deep tendon reflexes were exaggerated on right side. Computed tomography (CT) scan axial section showed hyperdense lesion in the left frontal parasagittal region [Figure 5]A. The lesion was heterogeneously enhancing on contrast. MRI axial section T1 image showed iso to hypointense lesion [Figure 5]B, T2 image showing hypointesne lesion [Figure 5]C and enhancement of lesion following contrast [Figure 5]D. She was undertaken for emergency surgery and left frontal craniotomy with tumour decompression was performed. Intraoperatively, the tumor was dirty grey soft, vascular with areas of bleed. Histopathology revealed tumour cells as polygonal to oblong and spindle shaped cells with oval nuclei, displaying nuclear pseudoinclusions and moderate eosinophilic cytoplasm areas of haemorrhage [Figure 6]A. Prominent foci of haemorrhage along with bits of tumour tissue and glial tissue were noted [Figure 6]B. Psammoma bodies were noted. No necrosis, mitosis and atypia were noted. Tumour was labelled meningothelial meningioma with organising haemorrhage. Postoperatively patient improved. She became conscious and was following simple commands, however right hemiplegia persisted.  | Figure 5: Radiological images of parasagittal meningioma: computed tomography scan axial section showing hyperdense lesion in the left frontal parasagittal region (A), Magnetic resonance imaging axial section T1 image showing iso to hypointense lesion (B), T2 image showing hypointesne lesion (C) and enhancement of lesion following contrast (D)
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 | Figure 6: (A) Parasagital meningioma with foci of hemorrhage. (B) islands of meningothelial cells with extensive hemorrhage
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| > Discussion | | |
Most cases of haemorrhage associated with meningiomas are extratumoural and subarachnoid, whereas subdural, intracerebral and ITH are reported less frequently. [3],[5] Some reviews suggest that location of meningiomas are unrelated to the risk of bleeding. Other reports suggests that convexity meningioma tend to bleed more. [5] Bosnjak et al., on review of literature identified that the mortality rate associated with bleeding from meningiomas has significantly decreased since the advent of CT scanning from 21.1 to 13.9% [3] They attributed this decrease to earlier diagnosis, better surgical techniques, and advancement in monitoring of patients. The proportions of angiomatous (16%) and malignant (5.7%) hemorrhagic variants of meningiomas were found to be higher in the study by Bosnjak et al., than in meningiomas overall. [3] Bruno et al., also suggested that angiomatous and malignant meningioma tends to bleed more frequently. [5] Pitkethly et al., however did not document any cases of bleeding in 81 patients with angiomatous meningiomas. [7]
The mechanisms of spontaneous ITH in meningiomas are not yet understood, and several hypotheses have been proposed. The most common hypothesis is the rupture of the abnormal vasculature of tumor. This is based on findings such as weak thin-walled vessels on morphology or peritumoral vascular erosion by the tumor directly. [3] In our first case, multiple thin walled congested vessels were identified in histopathology. Gruskiewicz et al., mentioned that necrosis of the tumor can cause direct breakdown of the tumor vessels and subsequent haemorrhage. [8] Jones reported noted granulation tissue around a central area of necrosis and hypothesized that bleeding is derived from neovasculature. [9] Another hypothesis suggests that enlarged, tortuous feeding arteries are less resistant to blood pressure changes and susceptible to rupture. Rupture of the bridging veins secondary to their stretch is also a probable cause of haemorrhage. Kim et al., identified infarction of the meningioma in cases with haemorrhage. [10] Blood dyscrasias, anticoagulation therapy, seizures, trauma, malignant transformation, have also been considered as possible causes of haemorrhage. [3] Minor head trauma may tear stretched subdural bridging veins overlying slow-expanding meningiomas. This may be true in some parasagittal or convexity meningiomas in which pure subdural hemorrahge occur. In cases with pure intratumoral bleeding, head injury is unlikely to be causative. Concomitant vascular malformation or aneurysm may influence the hemodynamics of the meningioma.
| > Conclusion | | |
Meningiomas with hemorrhagic onset remain rare, and pathophysiology is still incompletely understood. Prevention and outcome of intratumoral bleeding highly depend on early diagnosis and adequate treatment. As spinal meningiomas with haemorrhage are extremely rare, neurosurgeons should be aware of this complication to reduce mortality.
| > References | | |
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| 2. | Wakai S, Fukushima T, Teramoto A, Sano K. Pituitary apoplexy: Its incidence and clinical significance. J Neurosurg 1981;55:187-93.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
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