Microsoft Word - 13SinghAbhay_Pituitary Romanian Neurosurgery (2018) XXXII 2: 297 - 302 | 297             DOI: 10.2478/romneu-2018-0036 Pituitary apoplexy in setting of Dengue Hemorrhagic Fever with thrombocytopenia: case report and review of literature Abhay Singh1, Rahul Gupta2, Gangesh Gunjan3, Harjinder Singh Bhatoe4, Dhawal Sharma4 1General Surgery, Neurosurgery, Fortis Hospital, Noida, U.P., INDIA 2Neurosurgery, Additional Director Neurosurgery, Fortis Hospital, Noida, U.P., INDIA 3Consultant Neurosurgery, Fortis Hospital, Noida, U.P., INDIA 4Neurosurgery, Fortis Hospital, Noida, U.P., INDIA Abstract: Pituitary apoplexy is an acute clinical syndrome. It may occur spontaneously or as a result of several precipitating factors; one such factor being thrombocytopenia. Acute febrile illness accompanying with bleeding tendency is the main clinical feature of dengue. If the diagnosis is made in time, urgent treatment in the form of decompression of optic nerves may help to save vision. According to literature, only seven cases have been reported with pituitary apoplexy in setting of Dengue hemorrhagic fever. We report eighth case of Pituitary apoplexy in patient having Dengue hemorrhagic fever with its management and review of literature. Key words: Dengue Hemorrhagic Fever, Pituitary apoplexy, Thrombocytopenia Introduction Pituitary apoplexy is an acute clinical syndrome characterized by sudden headache, vomiting, visual disturbances, ophthalmoplegia, altered consciousness, secondary to infarction or hemorrhage within a pituitary tumor or nontumorous pituitary gland. [1,2] The diagnosis is often missed in the early stages. CT and MRI of the brain are helpful in making the diagnosis of pituitary apoplexy. Pituitary apoplexy may occur spontaneously or as a result of several precipitating factors; one such factor being thrombocytopenia. Dengue is an infectious disease which can be seen worldwide. Acute febrile illness accompanying with bleeding tendency is the main clinical feature of dengue. [3] Dengue hemorrhagic fever (DHF) is characterized by fever, hemorrhagic tendencies, thrombocytopenia, and increased vascular permeability. [4] According to literature, only seven cases have been reported with pituitary apoplexy in setting of Dengue hemorrhagic 298 | Singh et al - Pituitary apoplexy in Dengue Hemorrhagic fever             fever. We report eighth case of Pituitary apoplexy in the setting of Dengue hemorrhagic fever with its management and review of literature. Case report A 48 year old female presented with history of high grade fever 3 days. She was evaluated at local hospital where serology for Dengue infection was positive and she had low platelet count. Patient was referred to our hospital for further management. There was no history of petechial rash and spontaneous bleeding from mucosal surfaces. On third day of admission she complaint of sudden diminution of vision and severe headache. The platelet count decreased to 17,000. (Table 1) Table 1 - Daily Platelet count Platelets /mm3) On Admission Day 1 Day 2 Day 3 Day 4 Day 5 Platelet count 72,000 50,000 22,000 17,000 33,000 131,000 NCCT Head was done which was suggestive of presence of sellar/suprasellar well defined heterogeneously hyperdense mass (25mm x 20mm x 18mm) with hyperdense area of bleed. (Figure 1) Figure 1- NCCT Head Axial sections: Sellar/suprasellar well defined heterogeneously hyperdense mass with hyperdense area of bleed MRI brain showed well defined in homogeneously enhancing sellar- suprasellar mass with internal area of bleed. (Figure 2, 3 &4) Figure 2 - MRI T2 WI Coronal Sections: Heterogeneously hyperintense signal with presence of areas of bleed Figure 3- MRI FLAIR Axial sections: Well defined extraaxial mass in sellar / suprasellar region measuring 2.6 x 2.3 x 2 cm in size Figure 4 - MRI contrast enhanced Coronal sections: Mild to moderate inhomogenous post contrast enhancement with hemorrhagic area The optic chiasm was draped on its superior surface with no visualized area of in Romanian Neurosurgery (2018) XXXII 2: 297 - 302 | 299             between planes. On examination patient had Bitemporal hemianopia by confrontation method. Patient’s hormone profile revealed decreased serum thyroid and prolactin hormone levels [Growth Hormone (GH) = 2.30 ng/ mL; Luteinizing Hormone (LH) =2.64 mIU/mL; Prolactin (PRL) =2.23 ng/mL; Cortisol = 6.53 μ/dL; Follicle Stimulating Hormone = 5.85 mIU/mL; T3 = 47.87 ng/dL; Thyroxine (T4) = 4.82 μg/dL; Thyroid Stimulating Hormone (TSH) = 1.220 μIU/mL)] Patient underwent several platelet units transfusion and was operated after platelet count reached above 100,000. Urgent Transnasal transsphenoidal decompression of the pituitary macroadenoma was performed. Intraoperatively, there was evidence of bleed inside the tumor Post operative period was uneventful and patient was discharged subsequently. Histopathology was consistent with Pituitary apoplexy with bleed. On three month follow up, patient had hypocortisolemia with no visual defect. Discussion Pituitary apoplexy may occur spontaneously or as a result of several precipitating factors such as head trauma, dynamic tests to evaluate pituitary function, surgery (mainly cardiac surgery), coagulation disorders, medications including aspirin, estrogens, heparin, and dopamine agonists and some conditions such as chronic systemic hypertension, diabetes mellitus, and radiotherapy. [1, 2, 5, 6] The precipitants of pituitary apoplexy can be classified into 4 categories: (1) reduced blood flow into the pituitary adenoma, resulting in infarction; (2) acute increase in blood flow to the pituitary gland from the hypothalamus portal system because it may increase the intra sellar pressure; (3) stimulation of the pituitary gland, as occurs in any stress inducing states; and (4) thrombocytopenia because of increased risk of bleeding. [5, 7] Dengue fever is endemic in tropical countries and can range from a nonspecific febrile illness to DHF, which is characterized by high grade fever, marked thrombocytopenia (<100,000/μL), and increased vascular permeability. [4] In DHF, patients may have episodes of hypotension and hypovolemia, leading to oscillations in blood pressure. [4] Because it is an acute systemic illness, there is an increased demand on the pituitary gland and pituitary stimulation. In addition, DHF is characterized by severe thrombocytopenia and increased hemorrhagic tendencies. All these factors combined may have a compounding effect on the risk of pituitary apoplexy in a pre existing pituitary adenoma. However, the association of DHF and PA is exceedingly rare and only few case reports document their co occurrence. [6, 8, 9, 10, 11, 12] Other causes of visual deterioration in patients with dengue fever are optic neuropathy, maculopathy, retinal capillary occlusion, foveolitis, and retinal hemorrhage. [8, 13] The review of literature regarding the incidence of pituitary apoplexy in the setting of DHF revealed seven previously reported cases. The summary of these cases along with their comparative features has been described in Table 2. (Table 2) 300 | Singh et al - Pituitary apoplexy in Dengue Hemorrhagic fever             Table 2 - Summary of the reported cases of pituitary apoplexy in setting of Dengue Hemorrhagic Fever Reference Age/ Sex Clinical Feature Platelet counts Hormonal Profile MRI findings Management Follow up Kumar et al (2011) 31/F Vision loss, H/A, vomiting Hypothyroid, rest of the hormonal profile normal Low (45,000/m m3) Pituitary macroadeno ma (16×22 mm) with acute bleed Endoscopic endonasal decompressio n of the tumor Residual bitemporal field defects at 3-month F/U Wildenberg et al – Case 1 ( 2012) 40/M H/A, vomiting, acromegali c features Acromegaly; Hypogonadotro pic hypogonadism Mildly decreased (98,000/m m3) Intrasellar lesion s/o – pituitary apoplexy Transsphenoi dal surgery 20 days after apoplexy Acromegaly persists; other hormones normal Wildenberg et al – Case 2 ( 2012) 38/M Prolactino ma on medical Mx, presents with visual field deficits Hypogonadism; Prolactin decreased on medical management Mildly decreased (79,000/m m3) Pituitary mass with areas of bleed Urgent transsphenoi dal surgery Visual field defects improved. Hormonal deficits persist Tan et al. (2014) 53/M Acute-onse t Lt third, sixth nerve palsy, Rt temporal hemianopi a Hyperprolactino ma; thyrotropin and gonadotropin deficiency Low (16,000/m m3) 37×24×31 mm hemorrhagic pituitary adenoma with B/L cavernous sinus extension Underwent transsphenoi dal surgery for decompressio n (two surgeries) Prolactin normal, anterior pituitary deficits and right eye visual defects persist Mishra et al (2015) 43/M Decreased vision, H/A, vomiting Normal hormonal profile Low (47,000/m m3) 23×21×20 mm sellar and suprasellar mass with bleed Urgent transsphenoi dal decompressio n of tumor Visual acuity improved. Residual visual field deficits persist Balaparames wara Rao et al (2016) 45/M Severe H/A, vomiting and LOC Normal hormonal profile Low (27,000/m m3) Pituitary apoplexy (sellar and suprasellar lesion) and obstructive hydrocephal EVDs to manage HCP; followed by endoscopic endonasal decompressio n Vision normal. Anterior pituitary deficits present Romanian Neurosurgery (2018) XXXII 2: 297 - 302 | 301             us Varma et al (2016) 39 /F Fever, melena, vision loss ,altered sensorium Hypothyroid, Hypogonadotro phic hypogonadism Low (11,000/m m3) Pituitary adenoma with apoplexy Urgent Decompresio n of Tumor Symptoms Subsided Our Case 48/F Fever, H/A, Vision Loss Hypothyroidism & Hypoprolactino ma Low (17,000) Pituitary apoplexy (sellar and suprasellar lesion) Urgent transsphenoi dal decompressio n of tumor Visual acuity improved, hypocortisole mia In our case we had a smooth outcome but as reported by Balaparameswara Rao et al, it can be strormy i.e. Fluid & electrolyte imbalance which can occur in pituitary surgery. In all cases Decompresion of the tumour by transpenoidal technique was done and was successful. Vision improved in half of the cases. Conclusions Pituitary apoplexy can occur in setting of Dengue hemorrhagic fever. Dengue hemorrhagic fever should be considered as a cause of pituitary apoplexy. Transsphenoidal decompression of the tumor has good results in these cases. If the diagnosis is made in time, urgent treatment in the form of decompression of optic nerves may help to save vision. Correspondence Dr. Abhay Singh – Fortis Hospital, Sec-62, Gautambudh Nagar, Noida, U.P. India - 201301 E mail- abhaysingh291183@gmail.com Mobile No. 8130503451 References 1. Möller Goede DL, Brändle M, Landau K, Bernays RL, Schmid C. Pituitary apoplexy: Re evaluation of risk factors for bleeding into pituitary adenomas and impact on outcome. Eur J Endocrinol 2011; 164:37 43. 2. Semple PL, Webb MK, de Villiers JC, Laws ER Jr. Pituitary apoplexy. Neurosurgery 2005; 56: 65 72. 3. Wiwanitkit V. Dengue fever: Diagnosis and treatment. Expert Rev Anti Infect Ther 2010; 8:841‑5. 4. Simmons CP, Farrar JJ, Nguyen VV, Wills B. Dengue. N Engl J Med 2012; 366:1423 32. 5. Biousse V, Newman NJ, Oyesiku NM. 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