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CASE REPORT
Year : 2020  |  Volume : 13  |  Issue : 6  |  Page : 700-702  

Recurrent episodes of acute psychosis like illness: A very delayed presentation of Sheehan syndrome


1 Department of General Medicine, R.G.Kar Medical College and Hospital, Kolkata, West Bengal, India
2 Department of General Medicine, Midnapore Medical College and Hospital, Midnapore, West Bengal, India

Date of Submission25-Mar-2020
Date of Decision10-Jun-2020
Date of Acceptance03-Jul-2020
Date of Web Publication6-Nov-2020

Correspondence Address:
Atanu Chandra
Department of Medicine, R.G. Kar Medical College and Hospital, Kolkata, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mjdrdypu.mjdrdypu_111_20

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  Abstract 


Sheehan syndrome, also known as postpartum pituitary gland necrosis, is a very serious complication of postpartum hemorrhage. Although it is a rare disorder in western countries, it is not much uncommon in developing countries like India. We describe 46-year-old female from a rural area of eastern part of India who presented with recurrent episodes of acute psychosis like illness over the last few years. When she came to our medical emergency unit, she was found to have hypoglycemia, hyponatremia, and hypotension. Further evaluation revealed history of postpartum hemorrhage during her last childbirth 17 years back which was followed by amenorrhea, lactation failure, and associated psychiatric illness. Magnetic resonance imaging pituitary showed empty sella and there were decreased levels of all the anterior pituitary hormones. All these parameters led to the final diagnosis of Sheehan syndrome which was diagnosed after a significant delay.

Keywords: Hypoglycemia, hyponatremia, postpartum hemorrhage, Sheehan syndrome


How to cite this article:
Chandra A, Roy S, Chakraborty U, Ganai S. Recurrent episodes of acute psychosis like illness: A very delayed presentation of Sheehan syndrome. Med J DY Patil Vidyapeeth 2020;13:700-2

How to cite this URL:
Chandra A, Roy S, Chakraborty U, Ganai S. Recurrent episodes of acute psychosis like illness: A very delayed presentation of Sheehan syndrome. Med J DY Patil Vidyapeeth [serial online] 2020 [cited 2020 Dec 1];13:700-2. Available from: https://www.mjdrdypv.org/text.asp?2020/13/6/700/300123




  Introduction Top


Sheehan syndrome is a comparitively rare but serious complication of post partum hemorrhage. The pathogenesis is attributed to postpartum pituitary necrosis which has an insidious course with protean manifestations. We report a case of a 46 year old female who presented with recurrent episodes of psychosis like illness, and a thorough clinical and biochemical evaluation prompted the diagnosis of Sheehan's syndrome 17 years from her last child birth.


  Case Report Top


We describe 46-years-old female from a rural area of eastern part of India who presented to us with an acute psychosis like illness. She had similar history of such episodes in the past few years for which she got admitted to the local hospital and managed by giving intravenous fluids and injections. She was found to have very low blood glucose (22 mg/dl) on initial evaluation which was corrected by infusion of 25% dextrose solution. There was no history of intake of any oral antidiabetic drugs by her or any of the family members. Her sensorium got improved with infusion of dextrose.

There was a history of postpartum hemorrhage following home-delivery 17 years back for which she had to be transfused with four units of blood. It was followed by amenorrhea, lactation failure and associated psychiatric illness. On examination, the patient was listless, drowsy, and had hoarse voice and dry lusterless skin [Figure 1]. Pallor was remarkable with nonpitting pedal edema, and a delayed ankle jerk. On further examination, blood pressure was lower than normal with significant postural drop blood pressure was (98/70 mm of Hg on supine and 90/56 mm of Hg on standing). Axillary and pubic hairs were also absent. However, there was no jaundice, cyanosis, clubbing, hepatosplenomegaly, lymphadenopathy, or mucocutaneous pigmentation. Cardiovascular, respiratory system, and gastrointestinal system examination were unremarkable. She was on antipsychotic drugs for the last 6 years.
Figure 1: Dry lusterless skin of panhypopituitarism

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The patient's basic investigations revealed hemoglobin - 8.8 g/dl, total leukocyte count - 5400/cmm, differential leukocyte counts - 64% polymorphs, 33% lymphocytes, 3% eosinophils, platelet counts - 2.52 lac/cmm, erythrocyte sedimentation rate - 50 mm/h, serum sodium - 126 mmol/L, serum potassium - 4.6 mmol/L, total calcium - 2.58 mmol/L, and phosphorous - 1.16 mmol/L. Liver and renal function tests were normal. Her chest X-ray, electrocardiographs did not reveal any significant abnormality. Patients pituitary hormone profile was thyroid-stimulating hormone (TSH) - 2.5 mIU/L (0.5–4.7 mIU/L), FT3-1.2 pmol/L (3.1–6.8 pmol/L), FT4-8.5 pmol/L (12–22 pmol/L) (suggestive of central hypothyroidism), serum cortisol at 8 am was 122 nmol/L (138–690 nmol/L), follicle stimulating hormone (FSH) - 3.6 IU/L (3.5-12.5 IU/L), luteinizing hormone (LH) - 4.5 IU/L (2.4–12.6 IU/L), and prolactin - 102.6 mIU/ml (40–530 mIU/ml). During the hospital stay, the patient had multiple episodes of hypoglycemia, despite dextrose infusions. Patient's magnetic resonance imaging brain was done which revealed partial empty sella, thereby confirming the diagnosis of Sheehan's syndrome [Figure 2].
Figure 2: Magnetic resonance imaging pituitary showing partial empty sella

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The treatment was based on administration of saline infusion 25% dextrose with hydrocortisone followed by levothyroxine in replacement doses. The patient had dramatic improvement of symptoms, and after 5–6 days, she maintained normal blood sugar level without dextrose infusion. She was finally discharged on oral levothyroxine (100 μg/day), oral hydrocortisone tablet (in two divided doses), Vitamin D, and calcium.


  Discussion Top


Sheehan's syndrome (postpartum pituitary necrosis due to bleeding and hypovolemia) was first described by Sheehan in 1937.[1] Although the exact pathogenesis of the disease is not known, a few contributory factors such as vasospasm, thrombosis, and vascular compression of the hypophyseal arteries had been postulated. Enlargement of the pituitary gland during pregnancy, disseminated intravascular coagulation and some forms of autoimmunity have also been suggested to play a role in the pathogenesis of this condition.[2] The common antecedent obstetric events in decreasing order of frequency include obstetric hemorrhage, shock, blood transfusion, and peripartum hysterectomy. A higher frequency in home deliveries has also been found.[3] The symptoms may occur immediately or present in a delayed fashion depending on the degree of damage to the pituitary. Many of the patients remain undetected until years later, when signs of pituitary insufficiency become evident.[4]

A study in the Kashmir valley of the subcontinent showed the prevalence of this syndrome to be as high as 3.21% in women between the ages of 20 years and 39 years and 4.18% in those above 40 years.[5] This condition often presents months even after many years of delivery and the presenting symptoms are also nonspecific and extremely variable. Hence, it is often misrecognized and not treated appropriately thereby causing poor quality of life for the patient.[6] Our patient was diagnosed about 17 years after delivery in the hospital and there was excessive blood loss during delivery for which she had to be transfused with four units of blood.

The most common presentation of Sheehan's syndrome is the postpartum acute pituitary failure with stoppage of menstruation and failure of lactation. Common clinical features include loss of axillary and pubic hair (93%), dry skin (82%), and pallor (70%).[3] Different symptoms will occur depending on specific deficiencies of anterior pituitary hormones. Deficiency of prolactin can cause lactation failure. Gonadotropic hormone deficiency may cause genital hair loss and amenorrhea. Adrenocorticotropic hormone deficiency can result in generalized fatigue, weakness, and hypoglycemia. Growth hormone causes fatigability, decreased quality of life, and weight loss. Central hypothyroidism is clinically similar to primary hypothyroidism, with the exception of inappropriately low TSH levels in the background of low T3 and T4 levels. The diagnosis of partial hormone deficiencies are often difficult to elicit.[7] In our patient, there was a history of failure of lactation following her last delivery along with secondary amenorrhea. There were also features of central hypothyroidism. Lower abdominal sonogram revealed normal uterus and ovaries and there was low FSH and LH values than the reference range which further proved pituitary failure as the cause of her secondary amenorrhea.

Hypoglycemia with hyponatremia has been found more often in cases of acute Sheehan's syndrome. Severe hypoglycemia is a less common manifestations of Sheehan's syndrome and is even rarer as delayed presentation.[7]

However, a case series showed 15% of Sheehan's syndrome (3 out of 20 cases) presented with hypoglycemia.[8] Another retrospective study conducted in Turkey showed that endocrinopathy (20%) was the second-most common cause of hypoglycemia after diabetic treatment (42%) related side effects.[9]

Recurrent hypoglycemia in Sheehan's syndrome is due to growth hormone, adrenal, and other counter regulatory hormone deficiency. Deficiency of cortisol results in glycogen depletion by causing anorexia and weight loss thereby enhancing the gluconeogenesis pathway. The low level of precursors of gluconeogenesis (due to cortisol deficiency) and depletion of glycogen results in impaired ability to tolerate fasting. In addition, the growth hormone deficiency also contributes to the hypoglycemia.[10]

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

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Sheehan HL. Postpartum necrosis of the anterior pituitary. J Path Bacteriol 1937;45:189-214.  Back to cited text no. 1
    
2.
Haddock L. Sheehan's syndrome in post-partum hemorrhage. In: A Textbook of Postpartum Hemorrhage: A Comprehensive Guide to Evaluation, Management and Surgical Intervention. CB Lynch, LG Keith, AB Lalonde, M Karoshi, editors. 1st ed.. Dumfriesshire, UK: Sapiens Publishing; 2006.  Back to cited text no. 2
    
3.
Gei-Guardia O, Soto-Herrera E, Gei-Brealey A, Chen-Ku CH. Sheehan syndrome in Costa Rica: Clinical experience with 60 cases. Endocr Pract 2011;17:337-44.  Back to cited text no. 3
    
4.
Sert M, Tetiker T, Kirim S, Kocak M. Clinical report of 28 patients with Sheehan's syndrome. Endocr J 2003;50:297-301.  Back to cited text no. 4
    
5.
Zargar AH, Singh B, Laway BA, Masodoi SR, Wani AI, Bashir MI. Epidemiologic aspects of postpartum pituitary hypofunction (Sheehan's syndrome). Fertil Steril 2005;84:523-8.  Back to cited text no. 5
    
6.
Shivaprasad C. Sheehan's syndrome: Newer advances. Indian J Endocrinol Metab 2011;15 Suppl 3:S203-7.  Back to cited text no. 6
    
7.
Tessnow AH, Wilson JD. The changing face of Sheehan's syndrome. Am J Med Sci 2010;340:402-6.  Back to cited text no. 7
    
8.
Ozkan Y, Colak R. Sheehan syndrome: Clinical and laboratory evaluation of 20 cases. Neuro Endocrinol Lett 2005;26:257-60.  Back to cited text no. 8
    
9.
Güven M, Bayram F, Güven K, Kelestimur F. Evaluation of patients admitted with hypoglycaemia to a teaching hospital in Central Anatolia. Postgrad Med J 2000;76:150-2.  Back to cited text no. 9
    
10.
Davis SN, Cryer PE. Hypoglycemia. In: Longo DL, Kasper MD, Jameson JL, Fauci AS, Hauser SL, Localzo J, editors. Harrison's Principles of Internal Medicine. 18th ed.. Vol. 2. New York: McGraw Hill; 2012. p. 3003-9.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2]



 

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