The burden of congenital hyperinsulinism in the United Kingdom: a cost of illness study

Orphanet Journal of Rare Diseases
Sana EljamelRichard Thompson

Abstract

Congenital hyperinsulinism (CHI) is a rare, genetic disease which causes persistent hypoglycaemia, typically in new-borns. Patients with the diffuse disease variant often require near-total surgical removal of the pancreas, causing insulin-dependent diabetes mellitus (IDDM). The CHI economic burden is currently unknown. This study aimed to estimate the annual cost of illness (COI) of CHI patients in the UK from a service provider perspective (National Health Service, NHS and Personal Social Services), and to explore cost distribution within the patient population. The model was based on standard practice of two CHI centres of excellence. Model inputs were informed by a pragmatic literature review, NHS Reference Costs (2015-2016) and the British National Formulary (2017). Only direct costs to the NHS and Personal Social Services were considered. A prevalence-based approach was used and annual costs incurred at all ages were calculated. A deterministic sensitivity analysis (DSA; run at 10%) identified major cost drivers. The COI of CHI patients to the NHS was £3,408,398.59 annually and average cost per patient was £2124.95. Cost distribution was skewed among CHI patients, with 5.9% of patients (95 patients in their first year of ...Continue Reading

References

Jun 8, 1999·Journal of Pediatric Surgery·H N LovvornN S Adzick
Jun 26, 2003·European Journal of Endocrinology·Thomas MeissnerErtan Mayatepek
Oct 12, 2004·Quality of Life Research : an International Journal of Quality of Life Aspects of Treatment, Care and Rehabilitation·Michael G SawyerPeter A Baghurst
Apr 9, 2005·Journal of Pediatric Nursing·Linda SteinkraussCharles A Stanley
Sep 26, 2007·European Journal of Endocrinology·K Mazor-AronovitchH Landau
Sep 24, 2008·Expert Opinion on Investigational Drugs·Laurence Guy Howes, Divina Gracila Brillante
Oct 5, 2011·Orphanet Journal of Rare Diseases·Jean-Baptiste ArnouxPascale de Lonlay
Jul 25, 2013·Orphanet Journal of Rare Diseases·Pierrick RolletMarc Dunoyer
Mar 22, 2014·The New England Journal of Medicine·Senthil SenniappanKhalid Hussain
Jun 18, 2014·The Journal of Clinical Endocrinology and Metabolism·Huseyin DemirbilekKhalid Hussain
Aug 1, 2014·Annals of Pediatric Endocrinology & Metabolism·Tohru Yorifuji
Jul 18, 2015·World Journal of Diabetes·Chiara MameliGian Vincenzo Zuccotti
Sep 1, 2015·Journal of Clinical Research in Pediatric Endocrinology·Klára RoženkováKhalid Hussain
Oct 4, 2016·The Journal of Clinical Endocrinology and Metabolism·Marie SzymanowskiJean-Baptiste Arnoux
Apr 16, 2017·Orphanet Journal of Rare Diseases·Indraneel BanerjeeMark J Dunne

❮ Previous
Next ❯

Citations

Aug 11, 2020·International Journal of Endocrinology·Somayyeh HashemianAmirhossein Sahebkar
Apr 15, 2021·Orphanet Journal of Rare Diseases·Lidia García-PérezPedro Serrano-Aguilar
Aug 19, 2021·The Journal of Clinical Endocrinology and Metabolism·Myat WinKathryn Beardsall

❮ Previous
Next ❯

Methods Mentioned

BETA
pancreatectomy
pancreatectomies

Software Mentioned

GOSH
NORCHI

Related Concepts

Trending Feeds

COVID-19

Coronaviruses encompass a large family of viruses that cause the common cold as well as more serious diseases, such as the ongoing outbreak of coronavirus disease 2019 (COVID-19; formally known as 2019-nCoV). Coronaviruses can spread from animals to humans; symptoms include fever, cough, shortness of breath, and breathing difficulties; in more severe cases, infection can lead to death. This feed covers recent research on COVID-19.

Blastomycosis

Blastomycosis fungal infections spread through inhaling Blastomyces dermatitidis spores. Discover the latest research on blastomycosis fungal infections here.

Nuclear Pore Complex in ALS/FTD

Alterations in nucleocytoplasmic transport, controlled by the nuclear pore complex, may be involved in the pathomechanism underlying multiple neurodegenerative diseases including Amyotrophic Lateral Sclerosis and Frontotemporal Dementia. Here is the latest research on the nuclear pore complex in ALS and FTD.

Applications of Molecular Barcoding

The concept of molecular barcoding is that each original DNA or RNA molecule is attached to a unique sequence barcode. Sequence reads having different barcodes represent different original molecules, while sequence reads having the same barcode are results of PCR duplication from one original molecule. Discover the latest research on molecular barcoding here.

Chronic Fatigue Syndrome

Chronic fatigue syndrome is a disease characterized by unexplained disabling fatigue; the pathology of which is incompletely understood. Discover the latest research on chronic fatigue syndrome here.

Evolution of Pluripotency

Pluripotency refers to the ability of a cell to develop into three primary germ cell layers of the embryo. This feed focuses on the mechanisms that underlie the evolution of pluripotency. Here is the latest research.

Position Effect Variegation

Position Effect Variagation occurs when a gene is inactivated due to its positioning near heterochromatic regions within a chromosome. Discover the latest research on Position Effect Variagation here.

STING Receptor Agonists

Stimulator of IFN genes (STING) are a group of transmembrane proteins that are involved in the induction of type I interferon that is important in the innate immune response. The stimulation of STING has been an active area of research in the treatment of cancer and infectious diseases. Here is the latest research on STING receptor agonists.

Microbicide

Microbicides are products that can be applied to vaginal or rectal mucosal surfaces with the goal of preventing, or at least significantly reducing, the transmission of sexually transmitted infections. Here is the latest research on microbicides.