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 ::  Abstract
 :: Introduction
 :: Case Report
 :: Discussion
 ::  References
 ::  Article Figures
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CASE REPORT
Year : 2013  |  Volume : 59  |  Issue : 4  |  Page : 318-320

Recurrent hypoglycemia…: A less well-known presentation of Sheehan's syndrome


Department of Medicine, Government Medical College and S.S.G. Hospital, Vadodara, Gujarat, India

Date of Submission20-Oct-2012
Date of Decision23-Dec-2012
Date of Acceptance27-Jan-2013
Date of Web Publication17-Dec-2013

Correspondence Address:
R V Dosi
Department of Medicine, Government Medical College and S.S.G. Hospital, Vadodara, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0022-3859.123167

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 :: Abstract 

Sheehan's syndrome is a rare cause of recurrent hypoglycemia. We present a case of a middle aged woman who presented with recurrent life threatening hypoglycemia. Based on a past history of severe post partum hemmorhage and a clinical picture of myxedema and secondary amennorhea, laboratory and neuroimaging was undertaken to confirm a diagnosis of Sheehan's syndrome. The patient responded to treatment with thyroxine and steroids in replacement doses. The case report emphasises the importance of early suspicion and management of this easily missed and treatable condition.


Keywords: Hypoglycemia, Sheehan′s syndrome, panhypopitutarism


How to cite this article:
Dosi R V, Bhatt N R, Patell R D, Raj R R. Recurrent hypoglycemia…: A less well-known presentation of Sheehan's syndrome. J Postgrad Med 2013;59:318-20

How to cite this URL:
Dosi R V, Bhatt N R, Patell R D, Raj R R. Recurrent hypoglycemia…: A less well-known presentation of Sheehan's syndrome. J Postgrad Med [serial online] 2013 [cited 2019 Nov 19];59:318-20. Available from: http://www.jpgmonline.com/text.asp?2013/59/4/318/123167



 :: Introduction Top


Sheehan's syndrome, first described by Sheehan in 1937, [1] is a well-known cause of panhypopitutarism secondary to pituitary apoplexy. This syndrome generally occurs after an intra-or post-partum bleeding episode characterized by severe hypotension or hemorrhagic shock. The degree of hypopituitarism is variable ranging from a single hormone deficiency to a general disorder. Moreover, the onset in most cases is several months or even years after the inciting delivery, so it is often misrecognized and not adequately treated. The presenting symptoms are also extremely variable. Recurrent hypoglycemia due to growth hormone, adrenal and other counter regulatory hormone deficiency is a less well-known complication of Sheehan's syndrome. [2] We report a case of a patient who had recurrent episodes of altered sensorium due to hypoglycemia which recovered after replacement with glucocorticoids and thyroid hormones.


 :: Case Report Top


An Indian 31-year-old female patient presented to the emergency department of our hospital in July 2012 with a sudden loss of consciousness. Her relatives informed us that she was anxious, perspiring and confused before she lost consciousness. On examination she was pale, cold, and clammy with a random blood glucose level of 2.49 mmol/L. Her pulse was 92/min and blood pressure was 90/60 mm Hg with a pale and rough skin. She regained consciousness on administration of intravenous glucose. On eliciting a detailed history she was found to have episodes of recurrent loss of consciousness since 6 months accompanied by symptoms of generalized fatigue, weakness, lethargy, and anorexia. She showed signs of hoarseness of voice, dry skin, thinning of hair, facial puffiness, non-pitting pedal edema, and a delayed ankle jerk all of which point to a clinical diagnosis of myxedema. Moreover, the investigations also showed a low free triiodothyronine (T3) and low free thyroxine (T4) with a normal thyroid stimulating hormone further confirming the diagnosis of secondary (central) hypothyroidism. The initial investigations showed a normocytic normochromic anemia with normal indices and normal reticulocyte count.

Based on her previous medical history, it was revealed that the woman had a full term vaginal delivery with obstructed labor after which she developed post-partum hemorrhage and had to be transfused with multiple units of blood 8 years ago. She had a failure of lactation and amenorrhea since then for which she did not seek medical attention. This history suggests a case of hypopituitarism. The elaborate endocrinological investigations further done were also compatible with our diagnosis of central hypopituitarism [Table 1]. A magnetic resonance imaging of the brain showed an "empty sella" appearance with a pituitary flattened against the sellar floor with decreased volume, thus, confirming a diagnosis of Sheehan's syndrome [Figure 1].
Table 1: Laboratory investigations of a patient presenting with recurrent hypoglycemia and diagnosed with Sheehan's syndrome


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Figure 1: Magnetic resonance imaging suggestive of empty sella suggestive of pituitary atrophy

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Treatment was based on administration of saline infusion 25% dextrose with hydrocortisone and levothyroxine in replacement doses. Her blood sugar was monitored regularly and she was finally discharged on oral levothyroxine, prednisolone, Vitamin D and calcium. Although sex hormone replacement therapy was considered it was decided against taking the patient's personal preference into consideration.


 :: Discussion Top


Hypoglycemia in a case of hypopituitarism is more often seen in pituitary apoplexy than Sheehan's syndrome. Hypoglycemia with hyponatremia has been found more often in cases of acute Sheehan's syndrome. [3],[4] Vasospasm, thrombosis, and vascular compression of the hypophyseal arteries could be the possible causes of Sheehan's syndrome. [5] The most common antecedent obstetric events in this syndrome includes, obstetric hemorrhage (82%), shock (47%), blood transfusion (43%), and peripartum hysterectomy (7%), a higher incidence in home deliveries has also been found. [6] The prevelance of this syndrome has been found to be as high as 3.21% in women between the ages 20 years and 39 years and 4.18% in those above 40 years in the Kashmir valley of the subcontinent. [7] A delay in diagnosis after the inciting event is common, as late as 13 years. [5] Our patient was diagnosed 8 years after vaginal delivery in the hospital during which she suffered obstetric hemorrhage.

Common signs include the absence of axillary hair (93%) and pubic hair (93%), dry skin (82%), and pallor (70%). [6] In this case, the patient's history showed a failure of lactation following her last delivery and secondary amenorrhea since then. This along with the central hypothyroidism in the patient led to the diagnosis. On pelvic exam and sonogram she was found to have normal uterus and ovaries and the low follicle stimulating hormone and luteinizing hormone values, which further proved hypogonadotrophic hypogonadism as a cause of her secondary amenorrhea.

The Sheehan's syndrome in our case report presented with symptoms of recurrent loss of consciousness due to hypoglycemia (fasting intolerance), hypothyroidism and anemia. Such recurrent episodes of altered sensorium due to fasting intolerances 8 years after puerperial hemorrhage, is a rare presenting feature of Sheehan's syndrome. However, a retrospective study conducted in Turkey [8] for the incidence and causes of hypoglycemia showed that endocrine deficiency (20%) was the second most common cause of hypoglycemia after diabetic treatments (42%). The leading endocrine deficiency being panhypopituitarism and Sheehan's was the most common cause of panhypopiuitarism (44%). Another case review conducted by Ozkan and Colak [9] showed 3 out of 20 cases (15%) of Sheehan's syndrome presented with hypoglycemia.

Cortisol deficiency results in glycogen depletion by causing anorexia and weight loss. This increases reliance on gluconeogenesis pathway. The low-level of gluconeogeneiss precursors (due to cortisol deficiency) and the glycogen depletion results in impaired ability to tolerate fasting. Moreover, the growth hormone deficiency also contributes to the hypoglycemia. [10] This patient was found to have low levels of cortisol and growth hormone, which due to impaired fasting tolerance and led to recurrent episodes of hypoglycemia and loss of consciousness due to it. This improved after treatment with hydrocortisone followed by levothyroxine, in that order to prevent adrenal crisis. [11]

To conclude, in a woman having a previous pregnancy with post-partum hemorrhage and secondary amenorrhea, Sheehan's syndrome can be a cause of recurrent episodes of altered sensorium due to hypoglycemia (fasting intolerance). Thus, our case report aims to draw attention to an easily treated condition, which if not suspected and diagnosed early can have disastrous complications.

 
 :: References Top

1.Anfuso S, Patrelli TS, Soncini E, Chiodera P, Fadda GM, Nardelli GB. A case report of Sheehan's syndrome with acute onset, hyponatremia and severe anemia. Acta Biomed 2009;80:73-6.  Back to cited text no. 1
[PUBMED]    
2.Dosi RV, Saraiya S, Patell RD, Sonune NN, Sheth SD. An unusual association of sheehan's syndrome with pancytopenia. J Case Rep 2012;2:21-3.  Back to cited text no. 2
    
3.Krishna K, Singh P, Pradhan AB. Atypical presentations of Sheehan's syndrome. J Assoc Physicians India 2006;54:747-8.  Back to cited text no. 3
[PUBMED]    
4.Tessnow AH, Wilson JD. The changing face of Sheehan's syndrome. Am J Med Sci 2010;340:402-6.  Back to cited text no. 4
[PUBMED]    
5.Shivaprasad C. Sheehan's syndrome: Newer advances. Indian J Endocrinol Metab 2011;15:S203-7.  Back to cited text no. 5
[PUBMED]    
6.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. 6
[PUBMED]    
7.Zargar AH, Singh B, Laway BA, Masoodi 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. 7
[PUBMED]    
8.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. 8
    
9.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. 9
[PUBMED]    
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. 18 th ed., Vol. 2. New York: McGraw Hill; 2012. p. 3003-9.  Back to cited text no. 10
    
11.Singhania P, Singh S, Banerjee R, Singhania B, Banerjee I, Maitra S. Hyponatremia - A rare and emergency presentation of Sheehans Syndrome. Pak J Med Sci 2010;26:713-5.  Back to cited text no. 11
    


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