CASE REPORT Year : 2023  |  Volume : 12  |  Issue : 1  |  Page : 39-42

Pituitary apoplexy secondary to dengue fever-induced-thrombocytopenia: A case report and review of literature

Rishi Agarwal1, Hardeva Ram Nehara2, Babulal Meena1, RP Agrawal1
1 Department of Medicine and SP Medical College, Bikaner, Rajasthan, India
2 Department of Endocrinology, SP Medical College, Bikaner, Rajasthan, India

Date of Submission10-Nov-2022Date of Decision19-Dec-2022Date of Acceptance16-Jan-2023Date of Web Publication08-Feb-2023

Correspondence Address:
Hardeva Ram Nehara
Department of Endocrinology, SP Medical College, Bikaner, Rajasthan
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/2221-6189.369078

Rationale: Pituitary apoplexy (PA) is a rare endocrine emergency that requires prompt diagnosis and management. Dengue fever-induced-thrombocytopenia may rarely predispose to PA.
Patient's Concern: A 58-year-old male patient having known pituitary macroadenoma presented to the emergency department with fever, a sudden onset severe headache, and altered sensorium.
Diagnosis: Pituitary apoplexy caused by dengue fever-induced-thrombocytopenia.
Interventions: Conservative management with fluids, mannitol, dexamethasone and symptomatic treatment.
Outcomes: The patient responded well to the treatment and was discharged uneventfully.
Lessons: Although dengue hemorrhagic fever is a rare cause of pituitary apoplexy, it should be considered if a patient presents with headache and altered sensorium, and prompt initiation of treatment is crucial to prevent fatality and neuro-ophthalmic deficits.

Keywords: Dengue hemorrhagic fever; Pituitary adenoma; Apoplexy; Thrombocytopenia; Endocrine emergency

How to cite this article:
Agarwal R, Nehara HR, Meena B, Agrawal R P. Pituitary apoplexy secondary to dengue fever-induced-thrombocytopenia: A case report and review of literature. J Acute Dis 2023;12:39-42
How to cite this URL:
Agarwal R, Nehara HR, Meena B, Agrawal R P. Pituitary apoplexy secondary to dengue fever-induced-thrombocytopenia: A case report and review of literature. J Acute Dis [serial online] 2023 [cited 2023 Feb 9];12:39-42. Available from: https://www.jadweb.org/text.asp?2023/12/1/39/369078   1. Introduction 

Pituitary apoplexy (PA) is an acute catastrophic event caused by infarction or hemorrhage in the pituitary gland with or without pre-existing tumor that may lead to severe headache, vomiting, altered sensorium, diminished vision, and ophthalmoplegia[1]. PA may be induced by multiple precipitating factors including thrombocytopenia or may occur spontaneously[2]. Thrombocytopenia is an important feature of dengue fever, which may elicit PA. A review of the literature showed nine reported cases of dengue hemorrhagic fever-induced PA[1],[2],[3],[4],[5],[6],[7],[8]. We report a case of PA due to dengue fever-induced-thrombocytopenia which was managed conservatively.

  2. Case report 

This study was approved by the Institutional review board, and informed consent was obtained from the patient.

A 58-year-old male patient presented to the emergency department with a history of fever of 102 °F, myalgia and retro-orbital pain for 5 d, a sudden onset of severe headache, left-sided hemiparesis, and altered sensorium for 1 d. There was no history of any petechial rash or spontaneous mucosal bleeding. He was diagnosed with pituitary macroadenoma [Figure 1]A & [Figure 1]B 7 months ago after complaints of diminished vision and psychosis, yet had not undergone a complete hormonal workup. He was advised transsphenoidal surgery for treating the pituitary macroadenoma, but he refused and hence was not on any treatment. There was no history of smoking, alcohol use, or substance abuse. On examination, he had a pulse rate of 108 bpm (normal: 60-100 bpm), blood pressure of 100/64 mmHg (normal: 100-140/60-90 mmHg), respiratory rate of 22/min (normal: 13-19/min), SpO2 92% (normal: ≥95%) at room air, altered sensorium with a Glasgow coma scale score of 10/15, disoriented, left-sided hemiparesis and left extensor plantar reflex, and the rest of physical examination was unremarkable. The pupils were reactive to light and normal in size. Visual acuity and field of vision could not be tested due to the altered sensorium.

Figure 1: A & B: T1W magnetic resonance imaging of brain sagittal and coronal section showing lobulated sellar-suprasellar mass (35 mm× 60 mm × 55 mm) suggestive of pituitary macroadenoma (arrows). C & D: Non-contrast computed tomography head showing iso to hypodense mass (28 mm× 41 mm× 42 mm) mass suggestive of pituitary macroadenoma with intratumoral hemorrhage and right temporal and capsulo-ganglionic (36 mm× 38 mm) hemorrhage (arrows).

Click here to view

Investigations showed thrombocytopenia (platelet counts 21 000/mm3, normal: 50 000-150 000/mm3) with positive dengue serology (both NS1 antigen and immunoglobulin M). Non-contrast computed tomography of the head was suggestive of pituitary macroadenoma with intratumoral hemorrhage and right temporal and capsulo-ganglionic (36 mm×38 mm) hemorrhage [Figure 1]C & [Figure 1]D. On hormonal evaluation, serum thyroid stimulating hormone was 0.23 mIU/L (normal: 0.34-5.60 mIU/L), serum FT4 was 0.55 ng/mL (normal: 0.61-1.12 ng/mL), serum prolactin was >200 ng/mL (normal: 2.64-13.13 ng/mL), 8:00 AM serum cortisol was 4.25 mcg/dL (normal: 5-25 mcg/dL), serum testosterone was 68 ng/dL (normal: 270-1 070 ng/dL) and luteinizing hormone was 2.2 mIU/mL (normal: 1.8-8.6 mIU/mL).

According to the UK PA guideline, when having a PA score of 3/10, a patient should be managed conservatively[9]. Our patient was managed with oxygen therapy, intravenous fluids, 5 units of platelet transfusion, antibiotic, mannitol, and steroid (dexamethasone 4 mg intravenous 8 hourly with gradual tapering). The patient was also started with a tablet of thyroxin 50 mcg daily and a tab of cabergoline 0.5 mg half tablet weekly. He responded well to the conservative treatment with improvement in oxygen saturation and sensorium and started to take orally on the 5th day of admission. The patient was discharged with a prescription of thyroxine 50 mcg peroral daily, prednisolone 5 mg peroral daily, and tab cabergoline 0.5 mg half tablet peroral weekly. A follow-up visit after two weeks showed significant improvement in clinical condition with residual left-sided hemiparesis.

  3. Discussion 

We report a case of PA in a 58-year-old male due to dengue fever-induced-thrombocytopenia. PA is a catastrophic disorder manifested as a sudden onset of intrapituitary hemorrhage and/or infarction, generally within a pre-existing pituitary tumor[9]. The incidence of PA in pituitary tumors is about 2%-12% around the world[9]. The important risk factors for PA are cerebral angiographic procedures, systemic hypertension, surgeries, head injury, coagulopathies, and drugs (gonadotropin-releasing hormoneanalogs, dopamine receptor analogs, etc.)[10]. The proposed pathophysiology for PA include reduced blood flow resulting in infarction, acute increase in blood flow, stimulation of the pituitary gland from dynamic tests, and coagulopathies from thrombocytopeniaor anticoagulation[9]. Our patient had dengue fever-induced-thrombocytopenia, which precipitated apoplexy. In dengue hemorrhagic fever and dengue shock syndrome, hypotension and hypovolemia may trigger apoplexy in patients with pre-existing pituitary adenoma[2].

The clinical presentation of PA is highly variable and is largely depending on the extent of hemorrhage, necrosis, and edema. Clinical manifestations include headache, nausea, vomiting, diminished visual acuity, visual field defects, ocular paresis, and altered mental status[9]. Our patient presented with headache, altered sensorium, and left-side hemiparesis. PA is an endocrine emergency and the most common hormonal deficiency is corticotrophin deficiency, occurring in up to 80% of cases, resulting in severe hemodynamic instability and hyponatremia[10]. PA can also result in other hormonal deficits like growth hormone, thyrotropic, and gonadotropic deficiency[9]. A comparison of the present case with previously reported cases of dengue fever-induced PA is shown in [Table 1].

Table 1: Summary of cases of pituitary apoplexy induced by dengue hemorrhagic fever.

Click here to view

Computed tomography brain is a suitable choice of imaging. PA can be managed both conservatively and by surgical debridement as per the UK guideline suggests[9]. Medical management is aimed at hemodynamic stabilization, correction of electrolyte imbalance, and parenteral steroids to correct secondary adrenal insufficiency that occur from acute corticotrophin deficiency[9],[10]. PA is an endocrine emergency and glucocorticoid should be started immediately to prevent hemodynamic instability, neurological complications, and mortality[10]. Transsphenoidal surgical decompression should be considered if the patient has a progressive neuro-ophthalmic deficit and deterioration of consciousness[9],[10]. Our patient was managed medically with supportive treatment including steroids and thyroxine and showed significant improvement with residual left-side hemiparesis.

In conclusion, dengue hemorrhagic fever-induced PA is rare and requires a high index of suspicion. Clinicians should strongly suspect PA in patients with or without pituitary adenoma and dengue hemorrhagic fever who develop a rapid onset of severe headache and neuro-ophthalmic deficit and should be managed urgently to prevent adverse clinical outcomes.

Conflict of interest statement

The authors report no conflict of interest.

Funding

This study received no extramural funding.

Authors’ contributions

HRN: concept, design, intellectual content, literature search, data acquisition, manuscript preparation; RA: literature search, editing, and review; BM: intellectual content, data acquisition; RPA: clinical studies, data acquisition, manuscript editing, and review.

 

  References 
1.Tan SK, Seow CJ, Tan E, Chau YP, Dalan R. Pituitary apoplexy secondary to thrombocytopenia due to dengue hemorrhagic fever: A case report and review of the literature. Endocr Pract 2014; 20(4): 58-64.  
    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(5): e97-e101.  
    3.KumarV, Kataria R, Mehta VS. Dengue hemorrhagic fever: A rare cause of pituitary tumor hemorrhage and reversible vision loss. Indian J Ophthalmol 2011; 59(4): 311-312.  
    4.Mishra SS, Panigrahi S, Das S. Dengue hemorrhagic fever: A rare cause of pituitary apoplexy. Neurol India 2014; 62(1): 92-93.  
    5.Balaparameswara Rao SJ, Savardekar AR, Nandeesh BN, Arivazhagan A. Management dilemmasina rare case of pituitary apoplexy in the setting of dengue hemorrhagic fever. Surg Neurol Int 2017; 8: 4.  
    6.Sree Harsha Varma CH, Amaresh Reddy P, Pradeep TVS, Sindhu Sree R. A rare case of pituitary apoplexy secondary to dengue haemorrhagic fever. Indian J Endocrino Metabol 2017; 21(Suppl 1): S46.  
    7.Singh A, Gupta R, Gunjan G, Bhatoe HS, Sharma D. Pituitary apoplexy in setting of dengue hemorrhagic fever with thrombocytopenia: Case report and review of literature. Romanian Neurosurgery 2018; 32(2): 297-302.  
    8.Thomas M, Robert A, Rajole P, Robert P. A rare case of pituitary apoplexy secondary to dengue fever-induced thrombocytopenia. Cureus 2019; 11(8): e5323.  
    9.Briet C, Salenave S, Bonneville JF, Laws ER, Chanson P. Pituitary apoplexy. Endocr Rev 2015; 36(6): 622-645.  
    10.Glezer A, Bronstein MD. Pituitary apoplexy: Pathophysiology, diagnosis and management. Arch Endocrinol Metab 2015; 59(3): 259-264.  
    

Publisher’s note The Publisher of the Journal remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

  [Figure 1]
 
 
  [Table 1]