Eur J Gen Med 2016;13(2):155-157 Case Report DOI : 10.15197/ejgm.1457 Recurrent Hypoglycaemia: an Uncommon Presentation in Sheehan Syndrome HK Aggarwal 1 , Deepak Jain 1 , Sunil Pawar 1 , Anshul Mittal 1 ,Promil Jain 2 Malign Servikal Lenf Nodlarinin Ayiriminda ve Yönetiminde Ultrasonografjnin Rolü ÖZET ABSTRACT Sheehan sendromu nadir fakat ciddi bir postpartum komplikasyondur. Sheehan’s syndrome is a rare but potentially serious postpartum com- En sık ve erken belirtileri arasında laktasyon yetmezliği ve amenore - plication. Though most common and early symptoms are lactation dir. Bazı durumlarda Sheehan sendromu hipotiroidizm ve adrenal failure and amenorrhea, some cases might be relatively asymptomatic yetmezliği özellikleri baskın olduğunda daha sonraki yıllarda tanı konu - which are diagnosed in later years when features of hypothyroidism lan asemptomatik olabilir. Adrenal yetersizlik nedeni ile tekrarlayan and adrenal insuffjciency predominate. Recurrent hypoglycaemia, due hipoglisemi Sheehan sendromunun nadir görülen bir komplikasyonudur. to adrenal insuffjciency, though described is a rare complication of Burada, tekrarlayan hipoglisemi atakları ile sunulan bir Sheehan send - Sheehan syndrome. Here we report a case of Sheehan syndrome which romu olgusu bildirildi. presented with recurrent episodes of hypoglycaemia, Anahtar kelimeler: Sheehan sendromu, hipoglisemi, hipotroidizm, ad - Key words: Sheehan’s syndrome, hypoglycaemia, hypothyroidism, ad- renal yetersizlik renal insuffjciency INTRODUCTION on detailed history and investigations was diagnosed as sheehan syndrome. Sheehan’s syndrome is a rare but potentially serious postpartum complication. First described by Sheehan in 1937; it is the ischemic necrosis of the pituitary gland CASE secondary to shock due to obstetric hemorrhage (1). It A 48-year-old female patient presented to the outpatient is a well-known cause of hypopituitarism secondary to department of our hospital in January 2014 with history pituitary apoplexy. Most common presenting symptoms of multiple episodes of sudden loss of consciousness for of Sheehan's syndrome are lactational failure, prolonged past 15 years. Patient also complained of generalized amenorrhea or oligomenorrhea after delivery (2). Rarely, swelling of body and weakness with inability to do house- a woman with Sheehan syndrome might be relatively as- hold work since past 5 years. On detailed history it was ymptomatic, and the diagnosis is not made until years revealed that the woman had multiple admissions in past later , with features of hypothyroidism and adrenal insuf- for episodes of unconsciousness during which her blood fjciency (2). Recurrent hypoglycaemia and hyponatremia glucose was recorded below 40 mg with frequency of epi- though described in literature are less known complica- sodes around 2 per year. During all these episodes patient tion of Sheehan's syndrome with only few case reports regained consciousness on administration of intravenous (3). We report a case in which patient who had recur- glucose. There was no history of any other drug intake. rent episodes of altered sensorium due to hypoglycaemia Sharma University of Health Sciences Department of Medicine 1 , Pathology 2 , Rohtak, Correspondence: Deepak Jain Sharma University of Health Sciences Department of Medicine , Rohtak, India. India. Received: 18.12.2014, Accepted: 20.10.2015 E-mail: jaindeepakdr@gmail.com European Journal of General Medicine
Recurrent hypoglycaemia in Sheehan syndrome Patient 17 years back had intrauterine death of seven DISCUSSION month foetus following which she developed post-partum Adenohypophyseal ischemic necrosis following hypo per- haemorrhage and had to be transfused with multiple fusion is the most common cause of adenohypophysal units of blood. She has amenorrhea since then for which insuffjciency.Vasospasm, thrombosis, and vascular com - she did not seek medical attention. pression of the hypophyseal arteries could be the possible On examination she was conscious, oriented to time, causes of Sheehan's syndrome. The classical condition as- place and person. Her pulse rate was 70 per min, regular , sociated is postpartum hemorrhage with cardiovascular low volume and blood pressure was 90/60 mm Hg with collapse (4). The most common antecedent obstetric a pale and rough skin. She had hoarseness of voice, dry events described with this syndrome includes, obstetric skin, thinning of hair , facial puffjness, non-pitting pedal haemorrhage (82%), shock (47%), blood transfusion (43%), oedema. Her systemic examination was unremarkable. and peripartum hysterectomy (7%) (5). Patient had weakness of proximal muscles in both upper The diagnosis can be made reliably in the presence of lac- and lower limbs with power 4/5. Distal muscle power was tational failure, prolonged amenorrhea. However , other essentially normal. Her ankle refmex was present but de - signs of adenohypophysal insuffjciency like hypothyroid - layed. Sensory system examination was essentially nor- ism and adrenal failure are often delayed and subtle lead- mal. Initial investigations showed a anemia with normal ing to the diagnosis being missed. In some cases the pitu- indices and reticulocyte count. Her ESR, renal function, itary necrosis is only partial and the syndrome can present liver function tests and serum electrolytes were normal. in atypical and incomplete forms further complicating the Her thyroid profjle showed low free triiodothyronine (FT3) diagnosis (6). Recurrent hypoglycaemia and hyponatremia and free thyroxine (FT4) with normal thyroid stimulating are rare presentation of sheehan syndrome. Although de- hormone which pointed towards the diagnosis of central scribed earlier , there are only few case reports of sheehan hypothyroidism. Her fasting 0800 hrs serum cortisol levels syndrome presenting as recurrent hypoglycaemia. The were found to be 1.23 µg/ml which was very low. female in our case presented with symptoms of recur- Elaborate endocrinological investigations were done rent loss of consciousness which were documented due which showed LH, FSH defjciency and GH, ACTH insuf - to recurrent hypoglycaemia. Such recurrent episodes of fjciency further confjrming our diagnosis of central hypo - altered sensorium 15 years after puerperal haemorrhage, pituitarism (Table 1). Her vitamin D levels and DEXA score is a rare presentation of Sheehan's syndrome. were normal. Patient’s plasma insulin and C-peptide lev - els were normal ruling out the possibility of insulinoma as reason behind hypoglycaemia. Her ECG, chest X-ray PA and abdominal ultrasonography showed no abnormal- ity with normal adrenals. A magnetic resonance imaging of the pituitary gland showed an "empty sella" appear- ance with hypoplastic pituitary fmattened against the sel - lar fmoor with normal bright spot of posterior pituitary (Figure 1). Patient was treated with hydrocortisone 15 mg in morning and 5 mg in evening. L-thyroxine in a dose of 0.075 mg was started, to be titrated on basis of subse- quent T4 levels. Somatotrophin was started in a dose of 1 mg qd. On regular follow up patient improved symptom- atically with no episodes of hypoglycaemia. She has been further advised to increase the dose of hydrocortisone during any stressful event. Figure 1. MRI showing empty sella" appearance with hypoplastic pituitary fmattened against the sellar fmoor with normal bright spot of posterior pituatory Eur J Gen Med 2016 13(2): x-x 156
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