Acute Sheehan’s syndrome manifesting initially with diabetes insipidus postpartum: a case report and systematic literature review

Purpose Acute Sheehan’s syndrome is a rare, but potentially life-threatening, obstetric event that can be complicated by diabetes insipidus. Little information on the diagnosis and treatment of Sheehan’s syndrome with diabetes insipidus is available. We report on a 28-year-old patient who developed acute Sheehan’s syndrome with diabetes insipidus after giving birth, and on a systematic review of similar cases. Methods We performed a systematic review of the literature cataloged in PubMed and Google Scholar using the keywords “Sheehan syndrome” OR “Sheehan's syndrome” AND “diabetes insipidus” to identify relevant case reports published between 1990 and 2021. Eight Reports met the inclusion criteria (English-language abstracts available, onset in the puerperium, information about the day of the onset). Results In the present case, postpartum curettage was necessary to remove the residual placenta. The total amount of blood loss was severe (2500 ml). On the second day postpartal, the patient developed polyuria. Laboratory analysis revealed hypernatremia with increased serum osmolality and decreased urinary osmolality. Hormone analysis showed partial hypopituitarism involving the thyroid, corticotropic, and gonadotropic axes. The prolactin level was elevated. Brain magnetic resonance imaging showed pituitary gland infarction. Desmopressin therapy was initiated and resolved the polyuria. Hormone replacement therapy was administered. Four months later, the patient was well, with partial diabetes insipidus. The literature review indicated that this case was typical in terms of symptoms and disease onset. Most reported cases involve hypotension and peripartum hemorrhage, but some patients without hemorrhage also develop Sheehan’s syndrome. Elevated prolactin levels are uncommon and associated with poor prognosis in patients with Sheehan’s syndrome. Conclusion Acute Sheehan’s syndrome with diabetes insipidus involves nearly all pituitary hormone axes, indicating severe disease. Prolactin elevation could suggest that a case of Sheehan’s syndrome is severe.

Sheehan’s syndrome with hypothyroidism: a rare case report

Sheehan's syndrome is a form of hypopituitarism caused by pituitary gland necrosis caused by hemorrhagic shock during pregnancy. It's a rare issue with a wide range of symptoms and a long time to diagnose. A 40-year-old female presented with a giddiness which was rotatory type followed by headache, vomiting associated with nausea, decreased appetite for one week. She has experienced excessive vaginal bleeding and secondary amenorrhea exists for 20 years. Patients diagnosed to have acute liver injury, hyponatremia, and acute gastritis. The thyroid profile showed hypothyroidism and the patient started on appropriate medication. Hypopituitarism due to Sheehan's disease was discovered after a thorough clinical examination, endocrine investigations, and a pituitary magnetic resonance scan. Following the start of hormone replacement therapy, she showed significant improvement. The current case demonstrates that undiagnosed Sheehan's syndrome is linked to long-term morbidity, and we want to emphasize the importance of a high index of suspicion for early diagnosis of the syndrome during routine clinical visits to avoid complications that can arise from delayed diagnosis.

Management of a patient with Sheehan’s syndrome and diabetes insipidus complicated by recurrent hyponatremia

Summary A 38-year-old female was initially seen in the intensive care unit after severe postpartum hemorrhage. She was initially diagnosed to have Sheehan’s syndrome and after discharge, she was diagnosed to have a vesicovaginal fistula which initially caused a delay in diagnosing diabetes insipidus in the patient because she was having urinary incontinence. The patient had the vesicovaginal fistula repaired and was on replacement with levothyroxine, prednisone, and desmopressin. Years after her diagnosis, the patient experienced recurrent episodes of hyponatremia in the setting of desmopressin therapy. This case highlights the challenges of diagnosing diabetes insipidus in a patient with Sheehan’s syndrome and a vesicovaginal fistula, as well as the long-term management of central diabetes insipidus in a resource-limited setting. Learning points Sheehan’s syndrome is rarely associated with diabetes insipidus, and in our patient, it was initially missed due to a vesicovaginal fistula which caused urinary incontinence. Water intoxication is more common in young children and older adults but can occur years after initiating treatment with desmopressin in adults and should be kept in mind when treating patients with hyponatremia who have hypopituitarism associated with diabetes insipidus. Water intoxication is much more common in patients with diabetes insipidus being treated with intranasal desmopressin than in those using the oral preparations.

Misleading normal TSH and persistently elevated creatine kinase: clues to the diagnosis of chronic Sheehan’s syndrome

A 53-year-old woman was referred for medical evaluation of therapy-resistant dyslipidaemia accompanied by elevated creatine kinase levels. Because cessation or alteration of her medication did not improve laboratory abnormalities, hypothyroidism was considered, despite the fact that thyroid stimulating hormone levels were within the reference interval. On further evaluation, she was found to have panhypopituitarism and empty sella turcica as shown by MRI. These findings were unexpected since there was no clinical suspicion during detailed evaluation. When supplementary questions were asked, she brought up a history of severe postpartum haemorrhage 30 years ago, for which she underwent a hysterectomy. Based on these findings, the patient was diagnosed with Sheehan’s syndrome. This syndrome is a rare but potentially life-threatening complication of postpartum haemorrhage, characterised by varying degrees of hypopituitarism that are most commonly presented many years after delivery. The patient recovered after adequate hormone replacement therapy.

Co-presentation of Acute Sheehan Syndrome with Raised Intracranial Pressure in a Post-partum Lady

Sheehan’s syndrome (SS) or necrosis of pituitary gland is a rare complication of severe postpartum hemorrhage. It may cause hypopituitarism immediately or several years later, depending on the degree of tissue destruction. Sheehan’s syndrome though rare is still one of the commonest causes of hypopituitarism in developing countries like ours. The presence of an intercurrent infection and administration of thyroxine exacerbated her corticosteroid insufficiency. Intracranial hypertension (IH) manifested as bilateral optic disc swelling with reduced visual acuity, bilateral sixth nerve palsies, and impaired consciousness. Intracranial hypertension (IH) has been associated with hypocortisolism caused by either primary adrenocortical insufficiency or corticosteroid withdrawal. The author describes a case of a young lady with IH with acute SS who presented on 3rd day postpartum after lower uterine cesarean section with acute severe symptomatic hyponatremia which was complicated by postpartum hemorrhage. The clinical manifestations of IH resolved with corticosteroid replacement

Recurrent hypoglycaemia and dilated cardiomyopathy: delayed presentation of Sheehan’s syndrome

Sheehan’s syndrome (SS) is ischaemic necrosis of the pituitary gland due to massive postpartum haemorrhage. The clinical manifestations may vary from subtle to life-threatening and may present immediately after delivery or many years later. We present a case history of a 58-year-old non-diabetic woman who had undetected SS and presented with two unusual manifestations, including recurrent hypoglycaemia and dilated cardiomyopathy 34 years after delivery. The dilated cardiomyopathy reversed partially after treatment.