SFEBES2021 Poster Presentations Late Breaking (60 abstracts)
Unexplained hypoglycaemia in a patient with craniopharyngioma and GAD positive encephalitis
Umme Rubab , Ei Thuzar Aung , Charlotte Woodward , Temi Olabampe , Clifford Ediale , Thomas Flamini , Lorna Pain , Usman Hassan , Adam Townsend , Tala Balafshan & Ram Prakash Narayanan
Whiston Hospital, St Helen’s and Knowsley Teaching Hospitals NHS Trust, Prescot, United Kingdom
Case report: A 55 years old Caucasian gentleman presented with recurrent episodes of unexplained hypoglycaemia with slurred speech, lethargy, myoclonic jerks and seizures. He had background of craniopharyngioma at the age of 17 and underwent surgery but no radiotherapy. Subsequently he was started on hormonal replacement with desmopressin, levothyroxine, hydrocortisone, testosterone and genotrophin and remained stable on treatment for 38 years. Hypoglycaemia work up revealed blood glucose of 2.2 mmol/l with insulin inappropriately detectable at 16 pmol/l and C peptide 510 pmol/l (NR-190-990). On second occasion, blood glucose was 2.2 mmol/l with high insulin and C Peptide at 77 pmol/l and C-peptide 1184 pmol/l respectively. CT pancreas and gut hormones profile were normal. Extensive neurological work up showed positive GAD antibodies, encephalopathic pattern in EEG and reduced activity in the right basal ganglia on DAT scan. A diagnosis of GAD positive autoimmune encephalitis and Parkinson’s plus syndrome was reached. He was given Prednisolone 60 mg for encephalitis in addition to physiological hydrocortisone dose resulting in termination of seizures and hypoglycaemia.
Discussion: Literature search showed impaired response of counter-regulatory hormones to hypoglycaemia in patients with craniopharyngioma who had undergone surgery extending to the hypothalamus. Anti-GAD antibodies are linked with destruction of Beta cells leading to type-1 diabetes. We wonder if in early phase of B cell destruction, excess insulin is released from preformed insulin containing granules causing hypoglycaemia. One case report demonstrates reactive hypoglycaemia in a non-diabetic patient with positive GAD antibodies. GAD antibodies are also linked with several neurological syndromes. In our patient, the clinical conundrum arises from the fact that he remained stable for 38 years post-operatively then developed GAD positive encephalitis and hypoglycaemia. Whether there is a unifying diagnosis linking his hypoglycaemia, GAD positivity, neurological sequelae and underlying craniopharyngioma remains to be answered.
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Endocrine Abstracts
ISSN 1470-3947 (print) | ISSN 1479-6848 (online)
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