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poorly understood, current consensus appears to be alteration of cerebral vascular tone secondary to interaction between sympathetic overactivity and endothelial dysfunction, resulting in dysautoregulation.[5] Endothelin, endothelium-derived peptide, has been considered to participate in the vasospasm of preeclampsia.[6] Factors implicated in antepartum cases without pre-eclampsia/ eclampsia are not yet clearly defined. The major complications[7] are nonaneurysmal SAH, PRES, intracerebral hemorrhage, transient ischemic attacks and cerebral infarction. Til now, there are no specific guidelines for treatment, it is mainly based on expert opinion and case reports. The precipitating factors must be tackled aggressively and offending drugs if any, should be discontinued. Depending on the severity of vasospasm drugs such as nimodipine, verapamil, magnesium sulfate and milrinone can be tried.[7] RCVS often has a good prognosis. Our patient was unusual in that RCVS occurring in antepartum period in a normal pregnancy without any precipitating factors. Is it just a co-occurrence or antepartum period can also be a risk factor is not certain, hence further study of large series of cases is warranted. RCVS though benign can be fatal at times. It can occur during antepartum period also. Clinicians should be aware of such a rare association so that early intervention will reduce the mortality and morbidity.

Amita Bhargava, Basavaraj F. Banakar, Guruprasad S. Pujar, K. Shubhkaran, Hemant Jangid1 Department of Neurology, 1Satyam MRI Centre, Dr. S. N. Medical College, Jodhpur, Rajasthan, India E-mail: [email protected]

References 1. 2. 3. 4. 5. 6. 7.

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Calabrese LH, Dodick DW, Schwedt TJ, Singhal AB. Narrative review: Reversible cerebral vasoconstriction syndromes. Ann Intern Med 2007;146:34-44. Singhal AB. Cerebral vasoconstriction syndromes. Top Stroke Rehabil 2004;11:1-6. Call GK, Fleming MC, Sealfon S, Levine H, Kistler JP, Fisher CM. Reversible cerebral segmental vasoconstriction. Stroke 1988;19:1159-70. Ducros A, Bousser MG. Reversible cerebral vasoconstriction syndrome. Pract Neurol 2009;9:256-67. Tan LH, Flower O. Reversible cerebral vasoconstriction syndrome: An important cause of acute severe headache. Emerg Med Int 2012;2012:303152. Ito T, Sakai T, Inagawa S, Utsu M, Bun T. MR angiography of cerebral vasospasm in preeclampsia. AJNR Am J Neuroradiol 1995;16:1344-6. Sattar A, Manousakis G, Jensen MB. Systematic review of reversible cerebral vasoconstriction syndrome. Expert Rev Cardiovasc Ther 2010;8:1417-21.

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Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.128349

Received: 04-01-2014 Review completed: 06-01-2014 Accepted: 30-01-2014

Dengue hemorrhagic fever: A rare cause of pituitary apoplexy Sir, Dengue hemorrhagic fever (DHF) is associated with low platelet counts, but a case leading to apoplexy in pituitary adenoma is extremely rare, only few cases have been reported. We report one such an interesting case of pituitary apoplexy. A 43-year-old man presented with fever of 103°F of 5 days duration. There was no history of petechial rash and spontaneous bleeding from mucosal surfaces. He had not taken any medication or had bleeding disorder prior to this presentation. Complete blood count showed low platelet counts (47,000/cumm) and positive dengue serology both immunoglobulin G and M. 1 day after admission, he had sudden diminution of vision in both eyes with headache and vomiting. Examination revealed decreased visual acuity, hand movement and finger counting at 1 m in the right and left eye, respectively and bitemporal hemianopsia. Optic disc/retinal examination of both eyes was normal. Magnetic resonance imaging (MRI) brain revealed a homogenously enhancing T1 isointense, T2 subtle hyperintense sella and suprasellar mass (23 mm × 21 mm × 20 mm) with evidence of acute hemorrhage [Figure 1a]. Serum prolactin was 14.33 ng/ml (normal 4-30 ng/ml) and serum thyroid stimulating hormone was 1.46 μIU/ml (normal 0.3-3.0 μIU/ml). A diagnosis of pituitary apoplexy was made and urgent transnasal transsphenoidal decompression of the macroadenoma was done, after platelet transfusion (>100,000/cumm). Intraoperatively, there was evidence of bleed inside the tumor. Histopathologic examination revealed pituitary adenoma diffusely infiltrated by red blood cells [Figure 1b]. Post-operative computed tomography showed near complete tumor resection [Figure 1c]. Post-operative period was uneventful with progressive visual improvement from the second Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1

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post-operative day till 3 months. Vision was 6/60 and 6/36 in the right and left eye respectively, with residual bilateral temporal field defects after 3 month follow-up. Pituitary apoplexy is a condition where spontaneous hemorrhage occur into the pituitary adenoma with

a

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c

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Figure 1a: Magnetic resonance imaging of brain in sagittal plane showing an intrasellar mass that is isointense in T1-weighted (a) and subtle hyperintense in T2-weighted (b) images. Areas of a high-intensity signal on both T1- and T2-weighted images suggest intratumoral bleeding

development of sudden deterioration of vision, headache and vomiting. DHF is one of the causes of low platelet count leading to petechial rash and spontaneous bleeding from mucosal surfaces. Our patent was a diagnosed case of DHF with a low platelet count without any rash or systemic bleeding. MRI of the brain revealed pituitary macroadenoma with apoplexy. This catastrophe had arisen due to acute bleeding into the pituitary adenoma, probably predisposed by the thrombocytopenia and pituitary stimulation due to the acute systemic illness associated with DHF. Only few cases with this association have been reported in medical literature. [1,2] This rare association of DHF with pituitary apoplexy indicates that a low platelet count due to DHF may cause pituitary apoplexy. Our patients suggests that sudden onset headache with visual disturbances in patients with DHF warrants MR-imaging to exclude pituitary apoplexy. Other causes of visual deterioration in patients with dengue fever are optic neuropathy, maculopathy, retinal capillary occlusion, foveolitis, and retinal hemorrhage.[3-5] If the diagnosis is made in time, urgent treatment in the form of decompression of optic nerves may help to save vision as in the presented case.

Sudhansu Sekhar Mishra, Souvagya Panigrahi, Srikanta Das Department of Neurosurgery, S.C.B. Medical College and Hospital, Cuttack, Odisha, India E-mail: [email protected]

References 1. 2. 3. 4. Figure 1b: Photomicrograph shows pituitary adenoma infiltrated by blood cells indicating recent bleeding (H and E, original magnification ×200)

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Kumar V, Kataria R, Mehta VS. Dengue hemorrhagic fever: A rare cause of pituitary tumor hemorrhage and reversible vision loss. Indian J Ophthalmol 2011;59:311-2. Wildemberg LE, Neto LV, Niemeyer P, Gasparetto EL, Chimelli L, Gadelha MR. Association of dengue hemorrhagic fever with multiple risk factors for pituitary apoplexy. Endocr Pract 2012;18:e97-101. Sanjay S, Wagle AM, Au Eong KG. Optic neuropathy associated with dengue fever. Eye (Lond) 2008;22:722-4. Siqueira RC, Vitral NP, Campos WR, Oréfice F, de Moraes Figueiredo LT. Ocular manifestations in Dengue fever. Ocul Immunol Inflamm 2004;12:323-7. Su DH, Bacsal K, Chee SP, Flores JV, Lim WK, Cheng BC, et al. Prevalence of dengue maculopathy in patients hospitalized for dengue fever. Ophthalmology 2007;114:1743-7. Access this article online Quick Response Code:

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b

Figure 1c: Post-operative computed tomography axial (a) and sagittal (b) view demonstrates near complete sellar decompression

Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1

DOI: 10.4103/0028-3886.128350

Received: 05-01-2014 Review completed: 26-01-2014 Accepted: 26-01-2014 93

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Dengue hemorrhagic fever: a rare cause of pituitary apoplexy.

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