Clinical Imaging xxx (2014) xxx–xxx
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Case Report
A new identified complication of intracystic hemorrhage in a large pineal gland cyst☆ Raman Mehrzad a,⁎, Suprav Mishra a, Alexander Feinstein b, Michael G. Ho c a b c
Department of Internal Medicine, Steward Carney Hospital, Tufts University School of Medicine, Boston, MA, USA Department of Radiology, Steward Carney Hospital, Tufts University School of Medicine, Boston, MA, USA Department of Neurology, Steward Carney Hospital, Tufts University School of Medicine, Boston, MA, USA
a r t i c l e
i n f o
Article history: Received 3 December 2013 Received in revised form 9 March 2014 Accepted 13 March 2014 Available online xxxx
a b s t r a c t Pineal gland cysts are typically asymptomatic, benign cysts most commonly found incidentally in adults. In rare cases, a large pineal gland cyst can be complicated by intracystic hemorrhage, which could then manifest with neurological symptoms. We report a new complication of intracystic hemorrhage in a large pineal gland cyst in a 40-year-old man with new onset seizures.
Keywords: Pineal apoplexy Seizures Hemorrhage Complication
1. Introduction Pineal gland cysts generally are found incidentally on brain imaging studies in the adult population. Typically, pineal gland cysts are asymptomatic and benign, which rarely require additional investigations. In rare cases, a large pineal gland cyst can be complicated by intracystic hemorrhage, which could then manifest with neurological symptoms. Reported symptoms include headaches, extraocular gaze palsy, papilledema, nausea, vomiting, syncope, ataxia, and rarely communicating hydrocephalus. Seizures as a manifestation of pineal gland intracystic hemorrhage have not been well described in the literature. We report a new complication of intracystic hemorrhage in a large pineal gland cyst in a 40-year-old man with new onset seizures.
1.1. Case report A 40-year-old Vietnamese man was admitted to hospital after new onset seizure. Patient’s seizure was described as bilateral arm flexion with leg extension, followed by rhythmic convulsions lasting for 1 min, which was then followed by post-ictal confusion, headaches, ☆ Conflict of interest: The authors declare that they have no conflict of interest. ⁎ Corresponding author. Steward Carney Hospital, 2100 Dorchester Avenue, Boston, MA 02124, USA. Tel.: +1 774 240 0060. E-mail addresses:
[email protected] (R. Mehrzad),
[email protected] (S. Mishra),
[email protected] (A. Feinstein),
[email protected] (M.G. Ho).
© 2014 Elsevier Inc. All rights reserved.
and muscle soreness for about 5 min. While in the hospital emergency room, he had a second seizure similar to the first one, which was witnessed again by his wife and medical staff. His past medical history consisted of migraine headaches and gastroesophageal reflux disease. His home medications were sucralfate, pantopropazole, and calcium carbonate. The patient had no history of head trauma, developmental delay, central nervous system infections, or family history of seizures. He denied smoking, alcohol abuse, or recreational drug use. On arrival to the hospital, the patient’s vital signs were as follows: temperature, 36.4°C (97.5°F); pulse, 81 beats/min; blood pressure, 158/84 mmHg; respiration, 16 breaths/min; and oxygen saturation, 99%. He was initially somnolent and confused, likely due to his post-ictal state, but was easily arousable with mildly dysarthric speech, following one step commands, and complaining of severe headache and dizziness. A fundoscopic exam did not reveal any signs of papilledema. His pupillary reflexes were symmetric and equally reactive to light (4 mm→2 mm bilaterally). His extraocular eye movements were full range of motion without any limitations in upgaze or nystagmus. The rest of his physical and neurological examination was normal including his cranial nerve, motor, sensory, coordination, and gait examination. A complete blood count and complete metabolic profile were normal except for a mildly elevated white blood count at 11,100/μl and mildly decreased potassium at 3.2 mmol/l. A full toxicology screen was normal. The patient’s initial brain computed tomography (CT) showed a hyperdense fluid level, with a sharply demarcated smooth cyst in the pineal gland, consistent with acute blood products (Fig. 1A). An electroencephalogram was performed and showed mild diffuse
http://dx.doi.org/10.1016/j.clinimag.2014.03.009 0899-7071/© 2014 Elsevier Inc. All rights reserved.
Please cite this article as: Mehrzad R, et al, A new identified complication of intracystic hemorrhage in a large pineal gland cyst, Clin Imaging (2014), http://dx.doi.org/10.1016/j.clinimag.2014.03.009
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Fig. 1. (A) Brain CT. Hyperdense fluid level, with a sharply demarcated smooth cyst in the pineal gland consistent with acute hemorrhage. (B) Axial T1-weighted image demonstrates a very well defined, 14×13×11 mm, thin-walled, cystic structure in the pineal gland. There is a fluid–fluid level within this mass, with intermediate–high signal contents in the dependent portion, compatible with hemorrhagic or proteinaceous products, and low signal contents in the non-dependent portion, compatible with cystic fluid. The signal of the fluid in the non-dependent portion, however, is higher than that of cerebrospinal fluid (CSF). (C) Axial fat-suppressed post-gadolinium T1-weighted image demonstrates no enhancement within the mass. (D) Axial T2-weighted image redemonstrates a fluid–fluid level within the pineal gland cystic mass, with the low signal contents in the dependent portion, compatible with subacute or chronic hemorrhagic products, and high signal contents in the non-dependent portion, compatible with more simple fluid. A thin rim surrounding the mass is of low signal suggests recurring hemorrhage. (E) Axial FLAIR image redemonstrates a fluid–fluid level within the pineal gland cystic mass, with the low– intermediate signal contents in the dependent portion, compatible with subacute or chronic hemorrhagic products, and high signal contents in the non-dependent portion, suggesting proteinaceous components rather than simple CSF. (F) Sagittal T1-weighted image redemonstrates a cystic mass with fluid–fluid level in the pineal gland. (G) Sagittal fatsuppressed post-gadolinium T1-weighted image demonstrates no enhancement within the mass.
background slowing, consistent with his post-ictal state, but without epileptiform discharges that would be suggestive of an underlying seizure disorder. Brain magnetic resonance imaging (MRI) revealed a 13×14×11 mm pineal gland cyst with subacute hemorrhage/blood
product within the cyst, as manifested by a fluid–fluid level best seen in the T2/FLAIR axial images (Fig. 1B–G). The patient was admitted to our hospital and was started on Levetiracetam for seizure prevention. He did not have any more
Please cite this article as: Mehrzad R, et al, A new identified complication of intracystic hemorrhage in a large pineal gland cyst, Clin Imaging (2014), http://dx.doi.org/10.1016/j.clinimag.2014.03.009
R. Mehrzad et al. / Clinical Imaging xxx (2014) xxx–xxx
seizures afterwards. Upon follow-up of patient in clinic, the patient remains seizure free for over 6 months after tapering off of Levetiracetam. 2. Discussion We present a case of intracystic hemorrhage in an unusually large pineal gland cyst. Pineal gland cysts are usually found incidentally, less than 10 mm in size [1]. The most common age of presentation is between 40 and 49 years old, but they can occur in all age groups [1,2]. The incidence can be fairly high: with up to 23% found in healthy patients volunteering in imaging studies and up to 40% of cases in autopsy series [3]. There is a female predominance [1,2]. A degenerative process in the gland has been suggested to be origin of these cysts [4]. Cysts without neurological symptoms are often discovered as incidental findings on MRI images [5]. Headache and visual changes are the most common symptoms in cysts that exceed N10 mm [2,6]; however, these lesions are typically asymptomatic, and studies indicate that these lesions usually remain stable in size over time [7]. For symptomatic pineal gland cysts, neurological symptoms such as paroxysmal and chronic headaches, gaze palsy, papilledema, occlusive hydrocephalus, and Parinaud syndrome have been reported in the literature [8]. Other neurological manifestations that have been observed are nausea, vomiting, syncope, and ataxia [9–12]. Our patient had chronic headaches and dizziness, but also seizures, which, to our knowledge, has never been reported with details of clinical history in a patient with intracystic hemorrhage in a pineal gland cyst. Two previous studies have mentioned seizures associated with pineal cyst glands [13,14]. In the first article, Barboriak et al. [13] reviewed 32 cases of patients with various underlying neurological disorders, two of which presented with seizures, requiring brain CT/MRI followup in which pineal gland cysts were incidentally found. In this population, Barboriak et al. state that many of the patients in their study had other intracerebral abnormalities also present. The authors do not explain whether the patients who presented with seizures were a result of the other intracerebral abnormalities present in this population (i.e., gliomas, medulloblastomas, neurofibromatosis, demyelinating disease, etc.) or from the pineal gland cyst that was incidentally discovered. In the second article, Fleege et al. [14] retrospectively reviewed MRI and CT scans of 19 patients with large symptomatic pineal gland cysts and found 3 patients with presenting symptoms of seizures. The authors describe that the seizure patients with the pineal gland cysts in the article developed Parinaud
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syndrome and hydrocephalus, which could be a likely cause of seizure, rather than from pineal gland intracystic hemorrhage, as in our case. Our case is unique because we provide a clear case with detailed history and imaging correlating pineal gland intracystic hemorrhage with seizures. None of the prior studies described cases of pineal gland intracystic hemorrhage correlating with seizures. 3. Conclusion Pineal gland cysts are typically benign lesions. Larger cysts are very rare but can present with intracystic hemorrhage (“pineal apoplexy”) and may cause dangerous outcomes [5]. Seizure is a new identified manifestation that should be considered as a possible consequence of intracystic hemorrhage in a pineal gland cyst. References [1] Pastel DA, Mamourian AC, Duhaime AC. Internal structure in pineal cysts on highresolution magnetic resonance imaging: not a sign of malignancy. J Neurosurg Pediatr 2009;4(1):81–4. [2] Di Costanzo A, Tedeschi G, Di Salle F, Golia F, Morrone R, Bonavita V. Pineal cysts: an incidental MRI finding? J Neurol Neurosurg Psychiatry 1993;56(2):207–8. [3] Pu Y, Mahankali S, Hou J, Li J, Lancaster JL, Gao JH, Appelbaum DE, Fox PT. High prevalence of pineal cysts in healthy adults demonstrated by high-resolution, noncontrast brain MR imaging. AJNR Am J Neuroradiol 2007;28(9):1706–9. [4] Patel AJ, Fuller GN, Wildrick DM, Sawaya R. Pineal cyst apoplexy: case report and review of the literature. Neurosurgery 2005;57(5):E1066 [discussion E1066]. [5] Sarikaya-Seiwert S, Turowski B, Hänggi D, Janssen G, Steiger HJ, Stummer W. Symptomatic intracystic hemorrhage in pineal cysts. J Neurosurg Pediatr 2009;4:130–6. [6] Fain JS, Tomlinson FH, Scheithauer BW, Parisi JE, Fletcher GP, Kelly PJ, Miller GM. Symptomatic glial cysts of the pineal gland. J Neurosurg 1994;80(3):454–60. [7] Cauley KA, Linnell GJ, Braff SP, Filippi CG. Serial follow-up MRI of indeterminate cystic lesions of the pineal region: experience at a rural tertiary care referral center. AJR Am J Roentgenol 2009;193(2):533–7. [8] Wisoff JH, Epstein F. Surgical management of symptomatic pineal cysts. J Neurosurg 1992;77:896–900. [9] Apuzzo ML, Davey LM, Manuelidis EE. Pineal apoplexy as sociated with anticoagulant therapy. Case Rep J Neurosurg 1976;45:223–6. [10] Koenigsberg RA, Faro S, Marino R, Turz A, Goldman W. Imaging of pineal apoplexy. Clin Imaging 1996;20:91–4. [11] Swaroop GR, Whittle IR. Pineal apoplexy: an occurrence with no diagnostic clinicopathological features. Br J Neurosurg 1998;12:274–6. [12] Turtz AR, Hughes WB, Goldman HW. Endoscopic treatment of a symptomatic pineal cyst: technical case report. Neurosurgery 1995;37:1013–4. [13] Barboriak DP, Lee L, Provenzale JM. Serial MR imaging of pineal cysts: Implications for natural history and follow-up. ARJ Am J Roentgenol Mar 2001;176(3):737–43. [14] Fleege MA, Miller GM, Fletcher GP, Fain JS, Scheithauer BW. Benign glial cysts of the pineal gland: unusual imaging characteristics with histologic correlation. AJNR AM J Neuroradiol Jan 1994;15(1):161–6.
Please cite this article as: Mehrzad R, et al, A new identified complication of intracystic hemorrhage in a large pineal gland cyst, Clin Imaging (2014), http://dx.doi.org/10.1016/j.clinimag.2014.03.009