Affiliation:
1. Department of Endocrinology and Metabolism, Hamad General Hospital Hamad Medical Corporation Doha Qatar
2. Internal Medicine University Hospital of Coventry and Warwickshire (UHCW) Coventry UK
3. Department of Internal Medicine Punjab Medical College/Faisalabad Medical University Faisalabad Pakistan
4. Department of Internal Medicine University of Missouri‐Kansas City Kansas City Missouri USA
5. Department of Internal Medicine, Hamad General Hospital Hamad Medical Corporation Doha Qatar
6. Department of infectious diseases University of Missouri‐Kansas City Missouri USA
7. Department of Family Medicine, Hamad General Hospital Hamad Medical Corporation Doha Qatar
Abstract
AbstractIntroductionInsulin‐like growth factor‐2 (IGF‐2)‐mediated hypoglycemia is a rare yet clinically significant entity with considerable morbidity and mortality. Existing literature is limited and fails to offer a comprehensive understanding of its clinical trajectory, management and prognostication.MethodsSystematic review of English‐language articles reporting primary patient data on IMH was searched using electronic databases (PubMed, Scopus and Embase) from any date up to 21 December 2022. Data were analysed in STATA‐16.ResultsThe systematic review contains 172 studies, including 1 Randomised controlled trial, 1 prospective observational study, 5 retrospective observational studies, 150 case reports, 11 case series and 4 conference abstracts. A total of 233 patients were analysed, averaging 60.6 ± 17.1 years in age, with comparable proportions of males and females. The commonest tumours associated with Insulin‐like Growth Factor‐2‐mediated hypoglycaemia were fibrous tumours (N = 124, 53.2%), followed by non‐fibrous tumours originating from the liver (N = 21, 9%), hemangiopericytomas (N = 20, 8.5%) and mesotheliomas (N = 11, 4.7%). Hypoglycaemia was the presenting feature of NICT in 42% of cases. Predominant clinical features included loss of consciousness (26.7%) and confusion (21%). The mean IGF‐2 and IGF‐1 levels were 882.3 ± 630.6 ng/dL and 41.8 ± 47.8, respectively, with no significant correlation between these levels and patient outcomes. Surgical removal was the most employed treatment modality (47.2%), followed by medication therapy. The recovery rate was 77%, with chronic liver disease (CLD) significantly associated with a poor outcome (OR: 7.23, P: 0.03). Tumours originating from fibrous tissues were significantly associated with recovery (p < .001). In the logistic regression model, CLD remained a significant predictor of poor outcomes.ConclusionThis systematic review highlights that most non‐islet‐cell tumour‐hypoglycaemia (NICTH) is due to fibrous tumours. NICTs demonstrate a variable prognosis, which is fair if originating from fibrous tissue. Management such as octreotide, corticosteroids, diazoxide, embolization, radiotherapy and surgical resection have disparate success rates.