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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 16  |  Issue : 3  |  Page : 96-98

Precipitation of Sheehan's syndrome following severe dengue infection


1 Department of General Medicine, Christian Medical College, Vellore, Tamil Nadu, India
2 Department of Endocrinology, Christian Medical College, Vellore, Tamil Nadu, India

Date of Web Publication9-Nov-2018

Correspondence Address:
Dr. Ajay Kumar Mishra
Department of Medicine, Christian Medical College, Vellore - 632 004, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/cmi.cmi_24_18

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  Abstract 

Sheehan's syndrome as described by Sheehan refers to necrosis of pituitary gland due to severe postpartum hemorrhage. Although its incidence is decreasing in developed countries, it still continues to be one of the most common causes of hypopituitarism in the underdeveloped and developing countries. The clinical presentation can be variable, and emphasis must be on adequate obstetric history to avoid undue delay in diagnosis. We report a 42-year-old female with the unmasking of Sheehan's syndrome who had lactation failure and amenorrhea for 9 years, developing panhypopituitarism following an episode of severe dengue infection.

Keywords: Dengue fever, hyponatremia, panhypopituitarism, Sheehan


How to cite this article:
Mishra AK, Mahesh D M. Precipitation of Sheehan's syndrome following severe dengue infection. Curr Med Issues 2018;16:96-8

How to cite this URL:
Mishra AK, Mahesh D M. Precipitation of Sheehan's syndrome following severe dengue infection. Curr Med Issues [serial online] 2018 [cited 2018 Dec 15];16:96-8. Available from: http://www.cmijournal.org/text.asp?2018/16/3/96/245037


  Introduction Top


Sheehan's syndrome is named after HL Sheehan, who first described ischemic necrosis of pituitary occurring during severe hypotension or shock secondary to postpartum hemorrhage in 1937.[1] In a study in by Zargar et al., the prevalence was 3% in women above 20 years and with noninstitutional delivery in two-thirds.[2] Lactation failure and secondary amenorrhea are commonest presentations. Clinical diagnosis is made either immediately or in some years after of the obstetric event based on the extent of ischemic injury to pituitary during the initial insult and severity of individual symptoms.


  Case Report Top


A 42-year-old homemaker from Vellore was admitted with high grade, intermittent fever associated with chills, lethargy, and myalgia for 1-week duration. She complained of multiple episodes of nonbilious vomiting, persistent upper abdominal pain after defervescence of fever. On evaluation, she was found to have elevated pancreatic enzymes, serum lipase 369 (n = 13–60 U/L), serum amylase 339 (n = 28–100 U/L), and computerized tomography abdomen showing hyperdense pancreatic parenchyma confirming the diagnosis of acute pancreatitis. Etiological workup was negative for gallstones, drugs, and alcohol. And also common infectious causes such as malaria, scrub typhus, leptospirosis, and enteric fever was negative. She had severe thrombocytopenia (platelet count 4800/cumm), positive dengue IgM, and negative dengue IgG suggesting primary acute dengue infection. She deteriorated further with the development of multiorgan failure with associated acute lung injury and acute myocarditis with elevated cardiac enzymes serum troponin levels of 205 [n ≤14 pg/ml] and creatinine kinase-MB myocardial fraction of 10.5 (n ≤3.8 ng. ml) compromising her left ventricular (LV) function with ejection fraction (EF) of 20% causing hypotension. She was managed conservatively in the intensive care unit and was discharged in a stable condition with a normal platelet count (154,000/cumm), improved cardiac function (LV-EF 58%), and normalized pancreatic enzymes (serum amylase 63 U/L and serum lipase 56 U/L). During the hospital stay, her serum sodium was normal.

Three days after discharge, she was readmitted with a headache along with loose stools and vomiting. At this time, she was found to have persistent hyponatremia (Na-112) in spite of adequate fluid replacement. In view of hyponatremia in a normovolemic status, a possible diagnosis of hypocortisolism was considered. Her 8 am serum cortisol was 2.01 (5–24 ug/dl). The diagnosis of secondary adrenal insufficiency was confirmed with a short synacthen test showing a low value of 6.35 (normal response >20 ug/dl) and inappropriately normal adrenocorticotropin hormone levels 26.3 (9–52 pg/ml). Her other hormonal profile uncovered other pituitary axis involvement with a low free T4 0.33 (0.8–1.8 ng/dl) and total T4 3.0 (4.5–12 ug/dl) and inappropriately normal thyroid-stimulating hormone 1.751 (0.3–4.5 IU/ml) suggestive of central hypothyroidism. A detailed history revealed lactation failure and amenorrhea for 9 years after her 3rd delivery when she had postpartum hemorrhage with neonatal death soon after the delivery. Her general examination divulged sparse axillary and genital hair, Tanner stage 3 breast, madarosis, fine wrinkles around her eyes and lips along with a dry, and coarse skin. She was found to have a low normal prolactin 2.44 (1.9–23 ng/ml). Her follicle-stimulating hormone was 5.1 IU/ml (inappropriately low in spite of being amenorrheic for many years). Hence, a diagnosis of secondary panhypopituitarism was made, and she was started on steroid replacement followed by levothyroxine and cyclical hormone replacement by estrogen-progesterone pills. Although this patient had been amenorrheic for the last 8 years, she had no history of involvement of the other anterior pituitary axis being involved, explained by the normal serum sodium at the onset of severe dengue illness. Following the severe dengue infection, she developed the clinical and biochemical evidence of panhypopituitarism and thus unmasking of Sheehan's syndrome that had led to amenorrhea and lactation failure.


  Discussion Top


Ischemic necrosis of the pituitary secondary to postpartum hemorrhage is classically known as Sheehan's syndrome. It has been found to affect 0.05%–3% of women above 20 years of age depending on population (developed vs. developing nation) and institutional or noninstitutional deliveries.[2] Pituitary infarction can occur secondary to vasospasm, thrombosis, or vascular compression of the hypophyseal circulation[3] and manifests as hypopituitarism with varying degrees of anterior pituitary dysfunction and clinical picture from being asymptomatic to acute circulatory collapse with severe hyponatremia. Although panhypopituitarism is most common (86%), only selective pituitary deficiencies can be seen in up to 14% of patients.[3],[4] Clinical symptoms and signs are specific to deficiency of anterior pituitary hormones. The most common finding is prolactin deficiency (agalactia) with postpartum lactational failure followed by gonadotropin deficiency (amenorrhea, breast atrophy, infertility, and loss of secondary sexual characters).[5],[6] Fine wrinkling around the mouth and the eyes are thought to be a classical clinical sign suggesting growth hormone and estrogen deficiency.[5] Features of primary hypothyroidism can be biochemical or clinical. Hyponatremia is the most common electrolyte disturbance and can be seen in 33%–69% of patients.[5] In view of the severity of hyponatremia which can be refractory to fluid replacement, patients may present with altered sensorium, seizures, and coma. Hyponatremia which is due to glucocorticoid deficiency and promptly corrects after administration of steroids.[6] Adrenocortical insufficiency if undetected can present as an adrenal crisis in case of stress like an infection.

Reports of panhypopituitarism secondary to pituitary necrosis resembling Sheehan's syndrome are reported following snake envenomation and severe dengue infection.[7],[8] Injury to pituitary in both the scenarios has been attributed to ischemia. The mechanism by which Russell's viper envenomation leads to pituitary ischemia has been reported to be pituitary hemorrhage and severe hypotension.[9] Vector-borne flavivirus infection dengue has been reported to precipitate the same in a pregnant patient with severe dengue infection and hypotension.[8],[10] Although the presence of intracranial microhemorrhages and macrohemorrhages are reported in patients with dengue, the presence of pituitary hemorrhage has not been reported.[11]

Magnetic resonance imaging scanning is the most sensitive test for investigating hypothalamopituitary region. In early stages, pituitary can be large, which gradually atrophies and ultimately resulting in empty sella. Treatment essentially involves replacing deficient hormones. In an acute setup, glucocorticoid replacement is started first, following which replacement for thyroid hormone and hormone replacement therapy can be initiated. In view of the delayed presentation of Sheehan's, a detailed history of pregnancy, lactation, and menses is crucial in ruling out hypopituitarism. Signs though common can be missed in the absence of high suspicion. As Sheehan's is one of the most common etiologies of panhypopituitarism, it should be excluded in any women developing features of hypocortisolism during stress. Panhypopituitarism resembling Sheehan's syndrome is also a rare manifestation following snake envenomation and severe dengue infection.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

This study was self-funded.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Sheehan HL. Post-partum necrosis of anterior pituitary. J Pathol Bacteriol Chichester 1937;45:189-214s.  Back to cited text no. 1
    
2.
Zargar AH, Singh B, Laway BA, Masoodi SR, Wani AI, Bashir MI, et al. Epidemiologic aspects of postpartum pituitary hypofunction (Sheehan's syndrome). Fertil Steril 2005;84:523-8.  Back to cited text no. 2
    
3.
Kovacs K. Sheehan syndrome. Lancet 2003;361:520-2.  Back to cited text no. 3
    
4.
Zollner N. Hypopituitarism (Seehan's syndrome) with hyperlipemia and xanthomen. Dtsch Med Wochenschr 1955;80:999-1002.  Back to cited text no. 4
    
5.
Shivaprasad C. Sheehan's syndrome: Newer advances. Indian J Endocrinol Metab 2011;15 Suppl 3:S203-7.  Back to cited text no. 5
    
6.
Wani AM, Hussain WM, Al Mejally MA, Banjar AA, Ali KS, Khoujah AM, et al. Practice of symptomatic treatment in the era of evidence-based medicine: Report of 2 cases of diagnosis of Sheehan's syndrome delayed till eighth decade. BMJ Case Rep 2010;2010. pii: bcr0920092276.  Back to cited text no. 6
    
7.
Antonypillai CN, Wass JA, Warrell DA, Rajaratnam HN. Hypopituitarism following envenoming by Russell's vipers (Daboia siamensis and D. russelii) resembling Sheehan's syndrome:First case report from Sri Lanka, a review of the literature and recommendations for endocrine management. QJM 2011;104:97-108.  Back to cited text no. 7
    
8.
Pooja P, Dixit N, Kilapadi K, Sebastian A, Gopal S. Sheehan's syndrome in a dengue parturient. Sri Lankan J Anaesthesiol 2017;26:69-71.  Back to cited text no. 8
    
9.
Tun-Pe, Phillips RE, Warrell DA, Moore RA, Tin-Nu-Swe, Myint-Lwin, et al. Acute and chronic pituitary failure resembling Sheehan's syndrome following bites by Russell's viper in burma. Lancet 1987;2:763-7.  Back to cited text no. 9
    
10.
Koshy M, Mishra AK, Agrawal B, Kurup AR, Hansdak SG. Dengue fever complicated by hemophagocytosis. Oxf Med Case Reports 2016;2016:121-4.  Back to cited text no. 10
    
11.
Vanjare HA, Mannam P, Mishra AK, Karuppusami R, Carey RA, Abraham AM, et al. Brain imaging in cases with positive serology for dengue with neurologic symptoms: A clinicoradiologic correlation. AJNR Am J Neuroradiol 2018;39:699-703.  Back to cited text no. 11
    




 

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