Pituitary Apoplexy with Subarachnoid Hemorrhage and Intraventricular Hemorrhage: a Rare Case Report

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1 DOI / Case Report Pituitary Apoplexy with Subarachnoid Hemorrhage and Intraventricular Hemorrhage: a Rare Case Report Hsin-Hung Wu 1, Jiann-Her Lin 2,3, Cheng-Ta Hsieh 1,4,* ABSTRACT Pituitary apoplexy is an uncommon but well-known condition that leads to acute deteriorated neurological and endocrinal deficits. However, subarachnoid hemorrhage and intraventricular hemorrhage resulting from pituitary apoplexy are rarely reported in the literature. We reported a 75-year-old female presenting with severe headache and visual impairment. Magnetic resonance images of the brain revealed a pituitary macroadenoma. Acute deteriorated neurological deficits developed after admission during the correction of hyponatremia. The computed tomographic scan of the brain revealed pituitary apoplexy with diffuse subarachnoid hemorrhage and intraventricular hemorrhage. The relevant literature is reviewed. Keywords: pituitary apoplexy, subarachnoid hemorrhage, intraventricular hemorrhage, pituitary adenoma INTRODUCTION Pituitary apoplexy is an uncommon but potentially life-threatening condition caused by either hemorrhage or infarction of the pituitary adenoma.[1, 2] Hemorrhage within the tumor is the most common type of pituitary apoplexy.[3] Subarachnoid hemorrhage and intraventricular hemorrhage are rare manifestations of pituitary apoplexy and the actual mechanism remains unclear.[3-5] Here, we report the case of a patient with pituitary apoplexy with subarachnoid hemorrhage and intraventricular hemorrhage and review the relevant literature. CASE REPORT A 75-year-old woman with a history of hypertension began to experience headaches 4 days 1 Division of Neurosurgery, Department of Surgery, Cathay General Hospital, Taipei, Taiwan 2 Department of Neurosurgery, Taipei Medical University Hospital, Taipei, Taiwan 3 College of Medical Science and Technology, Taipei Medical University, Taipei, Taiwan 4 Department of Medicine, School of Medicine, Fu-Jen Catholic University, New Taipei City, Taiwan Submitted October, 24, 2016; final version accepeted February, 06, * Corresponding author: Cheng-Ta Hsieh (nogor@mail2000.com.tw) 輔仁醫學期刊第 15 卷第 2 期

2 Hsin-Hung Wu Jiann-Her Lin Cheng-Ta Hsieh prior to her initial presentation. Her headache was described as stabbing in nature, located in the bilateral frontal area, with radiation to the top of the head. She also reported generalized weakness, nausea, vomiting, and blurred vision associated with the onset of headache. She was initially managed as an outpatient, but soon after presented to the emergency department with persistent headache, nausea and vomiting. She was conscious, with a Glascow coma scale (GCS) of 14, and her neurological exam was non-focal, with extra-ocular movements and visual fields intact. The laboratory examination revealed a decreased sodium level of 105mEq/L. The hormone study revealed the cortisol level was >60 ug/dl, thyroid-stimulating hormone was 0.58 miu/ml, Free T4 was 0.48 ng/ dl, T3 was 33 ng/dl, insulin-like growth factor-1 was 42.7 ng/ml, prolactin was 7.82 ng/ml. The computed tomography (CT) scan of the brain revealed a large tumor occupying the seller region with suprasellar extension, but showed no evidence of hemorrhage within the tumor or the intracranial space (Figure 1). The magnetic resonance (MR) images of the brain showed a pituitary macroadenoma with suprasellar extension (Figure 2). No evidence of hemorrhage within the tumor or intracranial space was found. Hyponatremia was treated with 3% saline and hydrocortisone, with a dosage of 200mg per six hours. However, the subject became unconscious with a GCS of 8 about eight hours later. The laboratory examination revealed the serum sodium was 115 meq/l. The CT of the brain showed hemorrhage within the pituitary macroadenoma (Figure 3A), diffuse subarachnoid hemorrhage, intraventricular hemorrhage, and dilated ventricles (Figure 3B). The reconstruction of the CT angiogram showed no evidence of intracranial aneurysm (Figure 4). Extra-ventricular drainage was put in place and removal of the pituitary macroadenoma using an endoscopic trans-sphenoidal approach was urgently performed. During the operation, a hematoma within the tumor was identified. After removal of the hematoma, a large amount of cerebrospinal fluid flowed out from the right side of the tumor. The tumor was partially removed and the dura mater was sealed with tissue sealant. The pathologic examination confirmed the diagnosis of non-functional pituitary tumor with hemorrhage. Post-operation, she remained comatose with a GCS of 5 and the intracranial pressure was within normal range (less than 20mmHg). Diabetes insipidus developed subsequently and she expired two weeks after surgery due to brainstem dysfuncion. DISCUSSION With advances in neuroimaging, pituitary apoplexy is now more frequently identified. Incidence has been estimated to be from 1.5% to 27.7% [1, 2]. Most patients with pituitary apoplexy characteristically present with headache, visual impairment, ophthalmoplegia, deteriorated state of consciousness, and variable endocrine deficits. These symptoms may develop within hours to days, depending on the extension of intratumoral hemorrhage.[6] Although pituitary apoplexy with subarachnoid hemorrhage is rarely reported, a previous episode of headache is considered a warning headache, and visual deterioration is a cardinal sign of pituitary apoplexy. These symptoms occurred in 80% of 38 cases of major pituitary apoplexy in a review by Wakai et al [2]. Pituitary 92 Fu-Jen Journal of Medicine Vol.15 No

3 pituitary apoplexy with subarachnoid hemorrhage and intraventricular hemorrhage Figure 1. Computed tomographic scan of the brain revealed a mass located in the sellae turcica with suprasellar extension. There was no evidence of hemorrhage within the subarachnoid or intraventricular spce. Figure 2. T1-weighted magnetic resonance images of the brain revealed a pituitary tumor with suprasellar and parasellar extenson. Figue 3A Figure 3B Figure 3. Subsequent computed tomographic scan of the brain revealed diffuse subarachnoid hemorrhage around the tumor (A), with extension into the intraventricular space(b). 輔仁醫學期刊第 15 卷第 2 期

4 Hsin-Hung Wu Jiann-Her Lin Cheng-Ta Hsieh Figure 4. Reconstruction of the computed tomographic scan with angiogram revealed no obvious vascular lesion. apoplexy occurs in 65-95% of patients with pre-existing macroadenoma. Several risk factors have been implicated in the development of pituitary apoplexy, including head trauma, dynamic hormone test, elevated level of estrogen, bromocriptine therapy for existing adenoma, anticoagulant therapy, angiography, and radiation [3]. Pituitary apoplexy may result from hemorrhage, infarction or both in pituitary adenoma. The pathophysiology remains unclear [7]. Rapid tumor growth that outstrips the artery supply is hypothesized, but even the microadenoma can bleed [8]. Another theory is that a tumor growing inside the narrow space between the pituitary stalk and the diaphragm sellae leads to constriction of the thin rich vascular network and ischemia, necrosis and hemorrhage in the anterior lobe and tumor tissue [9]. In our patient, only the correction of hyponatremia was prescribed and an increase of sodium with 10 meq/l was achieved when the patient became comatose. No other predisposing factor was found. We considered the pituitary apoplexy complicated with subarachnoid hemorrhage and intraventricular hemorrhage to be caused by ruptured vessels in the tumor becoming distorted with growth or by the hemodynamic changes [3]. To the best of our knowledge, this is the first reported case to present a series of images of pituitary macroadenoma from non-hemorrhage to acute hemorrhage within a short period. In general, the hemorrhage is frequently enclosed within the encapsulated adenoma and the confined space of the sellae turcica. Intra-tumoral bleeding is the most common type of pituitary apoplexy [3]. Therefore, subarachnoid hemorrhage is rarely seen in patients with pituitary apoplexy [3-5]. In an anatomical study of cadaver headache, Qi et al. reported the pituitary stalk was enveloped by the arachnoid sleeve that was the direct upward extension of the basal arachnoid membrane covering the diaphragma sellae.[10] Depending on the relationship with the arachnoid sleeve, the pituitary stalk can be divided into three parts, including [1]subarachnoid, [2] intra-arachnoid, and [3] extra-arachnoid. When pituitary apoplexy occurs, the hemorrhage may disrupt the arachnoid sleeve 94 Fu-Jen Journal of Medicine Vol.15 No

5 pituitary apoplexy with subarachnoid hemorrhage and intraventricular hemorrhage or penetrate upwards into the subarachnoid part, resulting in subarachnoid hemorrhage and intraventricular hemorrhage within the third ventricle. MR imaging is the standard tool used to investigate the features of pituitary adenoma, such as hemorrhage, infarction, suprasellar extension, compression of the optic chiasm, or cavernous sinus destruction. The signal intensity of pituitary apoplexy varies, based on the different stages of hemorrhage[11]. On T1-weighted MR images, in the early acute stage, an enlarged gland that is isoor hypointense to the brain can be seen, while in the late acute or sub-acute stage, a hyperintense gland is seen. In the chronic stage, a hyperintense empty sella filled with cerebrospinal fluid can be seen on T2-weighted MR images. Restricted diffusion within an adenoma may be an early sign of apoplexy. Contrast administration often reveals rim-enhancement. However, because subarachnoid hemorrhage is a rare manifestation of pituitary apoplexy, it may be indistinguishable from aneurysmal subarachnoid hemorrhage. Reconstruction of the CT angiogram is the primary method to distinguish the bleeding from pituitary adenoma and aneurysm. The management of pituitary apoplexy includes immediate administration of high-dose corticosteroids to combat adrenal insufficiency [12]. Emergency surgery in the form of trans-sphenoidal decompression is recommended if the patient presents with acute onset of vision loss, blindness, or a diminished level of consciousness. Conservative treatment is justified if the patient is stable without progressive neurological deficits. In conclusion, although pituitary apoplexy with subarachnoid hemorrhage and intraventricular hemorrhage is rare, our case offered series images to confirm the possible causes of the acute bleeding. REFERENCES [1]. Moller-Goede DL, Brandle M, Landau K, et al. Pituitary apoplexy: re-evaluation of risk factors for bleeding into pituitary adenomas and impact on outcome. European journal of endocrinology / European Federation of Endocrine Societies 2011;164: [2]. Wakai S, Fukushima T, Teramoto A, et al. Pituitary apoplexy: its incidence and clinical significance. Journal of neurosurgery 1981;55: [3]. Nakahara K, Oka H, Utsuki S, et al. Pituitary apoplexy manifesting as diffuse subarachnoid hemorrhage. Neurologia medico-chirurgica 2006;46: [4]. Sergides IG, Minhas PS, Anotun N, et al. Pituitary apoplexy can mimic subarachnoid haemorrhage clinically and radiologically. Emergency medicine journal : EMJ 2007;24: 308. [5]. Wohaibi MA, Russell NA, Ferayan AA, et al. Pituitary apoplexy presenting as massive subarachnoid hemorrhage. Journal of neurology, neurosurgery, and psychiatry 2000;69: [6]. Cardoso ER, Peterson EW. Pituitary apoplexy: a review. Neurosurgery 1984;14: [7]. Ranabir S, Baruah MP. Pituitary apoplexy. Indian journal of endocrinology and metabolism 2011;15 Suppl 3: S [8]. Epstein S, Pimstone BL, De Villiers JC, et al. Pituitary apoplexy in five patients with pituitary tumours. British medical journal 1971;2: 輔仁醫學期刊第 15 卷第 2 期

6 Hsin-Hung Wu Jiann-Her Lin Cheng-Ta Hsieh [9]. Rovit RL, Fein JM. Pituitary apoplexy: a review and reappraisal. Journal of neurosurgery 1972;37: [10]. Song-tao Q, Xi-an Z, Hao L, et al. The arachnoid sleeve enveloping the pituitary stalk: anatomical and histologic study. Neurosurgery 2010;66: [11]. Semple PL, Jane JA, Lopes MB, et al. Pituitary apoplexy: correlation between magnetic resonance imaging and histopathological results. Journal of neurosurgery 2008;108: [12]. Satyarthee GD, Mahapatra AK. Pituitary apoplexy in a child presenting with massive subarachnoid and intraventricular hemorrhage. Journal of clinical neuroscience : official journal of the Neurosurgical Society of Australasia 2005;12: Fu-Jen Journal of Medicine Vol.15 No

7 pituitary apoplexy with subarachnoid hemorrhage and intraventricular hemorrhage 腦垂體中風合併蛛網膜下腔出血及腦室出血 : 罕見病例報告 吳鑫鴻 1, 林建和 2,3, 謝政達 1,4* 中文摘要 腦垂體中風是導致急性神經和內分泌障礙, 不常見卻是己知的疾病 然而, 由腦垂體中風引起蛛網膜下腔出血及腦室內出血的個案, 在文獻中很少被報導 我們報導一位 75 歲的女性呈現嚴重頭痛和視力障礙, 其腦部磁振照影檢查顯示有腦垂體大腺瘤, 但住院期間在嬌正病患低血鈉時, 病患發生急生神經功能缺損, 追蹤的腦部電腦斷層檢查, 發現有腦垂體中風合併瀰漫性蛛網膜下腔出血及腦室內出血 關鍵字 : 腦垂體中風 ; 蛛網膜下腔出血 ; 腦室內出血 ; 腦垂體腺瘤 國泰綜合醫院神經外科台北醫學大學附設醫院神經外科台北醫學大學醫學科技學院輔仁大學醫學系投稿日期 :2016 年 10 月 24 日 接受日期 :2017 年 02 月 06 日 * 通訊作者 : 謝政達電子信箱 :nogor@mail2000.com.tw 輔仁醫學期刊第 15 卷第 2 期

8 Hsin-Hung Wu Jiann-Her Lin Cheng-Ta Hsieh 98 Fu-Jen Journal of Medicine Vol.15 No