Expanded Dengue Syndrome: A Case of Subarachnoid Haemorrhage, Cranial Diabetes Insipidus, and Haemophagocytic Lymphohistiosis

Dengue fever is a mosquito-borne viral infection common in tropical countries with increasing incidence. The clinical manifestations can range from asymptomatic or mild infection to multiorgan failure. The latter is also called “Expanded dengue syndrome,” and it carries a high rate of mortality and morbidity. Intensive care management of such complicated cases is a challenging task for the treating physician, which requires intense monitoring and a multidisciplinary approach for decision making. We report an atypical case of an expanded dengue syndrome presented with subarachnoid haemorrhage associated with moderate thrombocytopenia, cranial diabetes insipidus, and haemophagocytic lymphohistiosis in a young healthy female patient.

Optic Chiasmatic Hypothalamic Glioma Presented as with Panhypopituitarism with Bilateral Primary Optic Atrophy

Optic chiasmatic hypothalamic gliomas are among common primary neoplasm of the optic nerve. It presents with decreased vision as well as features of hypopituitarism due to infiltration of the hypothalamus. In this case report, a young man presented with loss of libido, increased thirst, gradual loss of vision, bilateral optic atrophy and visual field defect. The MRI of sellar and paraseller region showed optic chiasmaticglioma with hypothalamic extension and biochemical parameter showed panhypopituitarism. We diagnosed this patient as a case of Optic chiasmatic hypothalamic gliomas (OCHGS) with panhypopituitarism with partial cranial diabetes insipidus with bilateral primary optic atrophy.The patient underwent surgery after adequate hormonal treatment but died due to post-operative complications Bangladesh Med J. 2020 Sept; 49(3) : 49-52

Chronic Lymphocytic Leukaemia Complicated by Cranial Diabetes Insipidus - A Case Report

Here we describe a case of 55-year-old lady who was admitted in hospital for evaluation of recurrent anaemia, polyuria and polydipsia with history of splenectomy 9 months back. Physical examination revealed anaemia, dehydration and scar mark of splenectomy. Initial laboratory tests were suggestive of chronic lymphocytic leukaemia (CLL) and further bone marrow examination & immunophenotyping confirmed the diagnosis. At the same time Polyuria & polydipsia was evaluated by water deprivation test and diabetes insipidus was diagnosed.

Postpartum Cranial Diabetes Insipidus

Cranial Diabetes Insipidus is a rare diagnosis and rarer still postpartum. We present the case of 24-year-old woman who developed CDI following pregnancy. The patient had developed persistent polydipsia and polyuria 5 months following her first pregnancy. The pregnancy had been complicated by Gestational Diabetes Mellitus, Obstetric Cholestasis and a Postpartum Haemorrhage which had required a 3 unit transfusion of blood. The patient reported feeling fatigued and lightheaded and stated that she had needed to drink water frequently: around 8 litres throughout the day and 4 litres overnight. She reported that she had been unable to breastfeed but the rest of her systemic enquiry was unremarkable. The patient had attributed her symptoms of lethargy to sleepless nights with her new born baby and the polyuria as a consequence of labour and as such had presented for review at her primary care Physician 18 months following delivery. Initial investigations revealed a fasting blood glucose of 4.9 mmol/l, an Adj. Calcium of 2.23 mmol/l and a fasting urine osmolality of 85 mmol/kg. A diagnosis of DI was suspected and was confirmed by water deprivation test: the patient had an inappropriately dilute urine osmolality of 111 mmol/kg when compared to the serum osmolality of 301 mOsm/Kg at the start of the test and her urine failed to concentrate as water was withheld. Administration of DDAVP resulted in appropriate concentration of urine and therefore confirmed the diagnosis specifically as Cranial Diabetes Insipidus. Blood tests revealed normal anterior pituitary function: TSH was 2.78 mU/L, Prolactin was 361 mU/l, LH and FSH were 23.6 U/L and 5.3 U/L, IGF and GH were 197 ug/L and 0.1 ug/l and ACTH was 10 mU/L. Her basal cortisol was 392 nmol/l and was stimulated to 593 nmol/l by SST. MRI Pituitary revealed an unusually flat and broad pituitary gland with a possible tiny lesion in the posterior pituitary suggestive of an adenoma. The patient was established on DDAVP replacement therapy and her quality of life improved: she enjoyed restful sleep and reported less exhaustion. There was no anterior pituitary hormone deficiency 28 months following delivery. DI is a rare diagnosis with an estimated prevalence of 1 in 25000 people. CDI has been commonly reported as being caused by infiltrative or inflammatory pituitary disease, as an iatrogenic sequelae of pituitary surgery or as a result of a congenital defect in the production of vasopressin. We suspect that in this case the patient’s PPH may have resulted in isolated cranial diabetes insipidus though the significance of the MRI scan findings remains unclear. The learning points highlighted by this case are that CDI can occur following pregnancy in an isolated form without anterior pituitary hormone deficiency. We also highlight that patients may misattribute significant symptoms and signs of DI as being a normal part of the postpartum period resulting in a delayed diagnosis.

Perioperative management of cranial diabetes insipidus in a patient requiring a tracheostomy

Cranial diabetes insipidus (DI), which can cause life-threatening dehydration, is treated with desmopressin, often intranasally. This is challenging in patients whose nasal airflow is altered, such as those requiring tracheostomy. We report the case of a patient, taking intranasal desmopressin for cranial DI, who underwent partial glossectomy, free-flap reconstruction and tracheostomy. Postoperatively, she could not administer nasal desmopressin due to reduced nasal airflow. She developed uncontrollable thirst, polyuria and hypernatraemia. Symptoms were relieved by switching to an enteric formulation. A literature review showed no cases of patients with DI encountering difficulties following tracheostomy. The Royal Society of Endocrinology recommends perioperative planning for such patients, but gives no specific guidance on medication delivery in the context of altered airway anatomy. Careful perioperative planning is required for head and neck patients with DI, particularly for those undergoing airway alteration that may necessitate a change in the mode of delivery of critical medications.

Arginine-vasopressin infusion in a child with cranial diabetes insipidus during hyperhydration therapy with chemotherapy: a therapeutic challenge

Summary An 11-year-old girl presented with acute lower limb weakness, dehydration, hypernatraemia and secondary rhabdomyolysis on a background of an 8-month history of polyuria. Radiological investigations revealed a suprasellar tumour which was diagnosed on biopsy as a non-metastatic germinoma. Further endocrinological investigations confirmed panhypopituitarism and she commenced desmopressin, hydrocortisone and thyroxine. Her chemotherapeutic regime consisted of etoposide, carboplatin and ifosfamide, the latter of which required 4 litres of hyperhydration therapy daily. During the first course of ifosfamide, titration of oral desmopressin was trialled but this resulted in erratic sodium control leading to disorientation. Based on limited literature, we then trialled an arginine-vasopressin (AVP) infusion. A sliding scale was developed to adjust the AVP dose, with an aim to achieve a urine output of 3–4 mL/kg/h. During the second course of ifosamide, AVP infusion was commenced at the outset and tighter control of urine output and sodium levels was achieved. In conclusion, we found that an AVP infusion during hyperhydration therapy was required to achieve eunatraemia in a patient with cranial diabetes insipidus. Developing an AVP sliding scale requires individual variation; further reports/case series are required to underpin practice. Learning points Certain chemotherapeutic regimens require large fluid volumes of hyperhydration therapy which can result in significant complications secondary to rapid serum sodium shifts in patients with diabetes insipidus. The use of a continuous AVP infusion and titrating with a sliding scale is more effective than oral desmopressin in regulating plasma sodium and fluid balance during hyperhydration therapy. No adverse effects were found in our patient using a continuous AVP infusion. Adjustment of the AVP infusion rate depends on urine output, fluid balance, plasma sodium levels and sensitivity/response of the child to titrated AVP doses.