Healthcare Resource Utilization and the Cost of Care for Mucopolysaccharidosis I Patients in Iran

Davari, Majid; Nabizadeh, Azita; Kadivar, Maliheh; Asl, Akbar Abdollahi

doi:10.1016/j.vhri.2019.01.009

Cited by 3 publications

(1 citation statement)

References 24 publications

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“…We also found a significantly increased length of hospitalization and cost of care in patients with MPS, which is congruent with findings from previous studies for other procedures. More specifically, a total cost-analysis study by Davari et al [ 29 ] recorded that the average annual cost of treatment for MPS-I patients is $87,971.99, a quantity 16.2 times the GDP per capita in Iran. A separate cost-analysis study by Conner et al [ 30 ] recorded an average cost per patient per month of $10,715 for an average annual cost of $128,580 for medical care of patients with severe MPS-I.…”

Section: Discussionmentioning

confidence: 99%

Post-Tonsillectomy Outcomes in Children with Mucopolysaccharidosis and Obstructive Sleep Apnea

Elwell,

Mancuso,

Wolter

et al. 2023

Journal of Otolaryngology - Head & Neck Surgery

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Objective To describe the incidence of respiratory complications, postoperative hemorrhage, length of stay, and cost of care in children with mucopolysaccharidosis (MPS) undergoing adenotonsillectomy (AT). Methods Analysis of the 2009, 2012, and 2016 editions of the Healthcare Cost and Utilization Project Kids’ Inpatient Database (HCUP KID) identified 24,700 children who underwent AT (40 children with MPS). Demographics, respiratory complications, postoperative hemorrhage, length of stay, and total cost were compared across children with and without MPS. Results Children with MPS had a higher likelihood of being male (P < 0.017). There was a higher rate of respiratory complications in children with MPS compared with children without MPS [6/40 (15%) vs. 586/24,660 (2.4%), P < 0.001], which remained significant after adjusting for sex [adjusted odds ratio 6.88 (95% CI 2.87–16.46)]. There was also a higher risk of postoperative hemorrhage [4/40 (10%) vs. 444/24,660 (1.8%), P < 0.001), with sex-adjusted odds ratio of 5.97 (95% CI 2.12–16.86). Median (IQR) length of stay was increased in children with MPS (3 days, 1–4) compared with children without MPS (1 day, 1–2, P < 0.001). There was an increase in median (IQR) charges for hospital stay in children with MPS compared with their peers [$33,016 ($23,208.50–$72,280.50 vs. $15,383 ($9937–$24,462), P < 0.001]. Conclusions Children with MPS undergoing AT had an increased risk of respiratory complications, postoperative hemorrhage, longer length of stay, and a higher cost of treatment when compared with children without MPS. This information may help inform interventional, perioperative, and postoperative decision making.

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Section: Discussionmentioning

confidence: 99%

Post-Tonsillectomy Outcomes in Children with Mucopolysaccharidosis and Obstructive Sleep Apnea

Elwell,

Mancuso,

Wolter

et al. 2023

Journal of Otolaryngology - Head & Neck Surgery

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Cost-effectiveness of miglustat versus symptomatic therapy of Niemann–Pick disease type C

2022

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Health service utilization, economic burden and quality of life of patients with mucopolysaccharidosis in China

Kang,

Fang,

Yang

et al. 2024

Orphanet J Rare Dis

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BackgroundPatients with mucopolysaccharidosis (MPS) often face delayed diagnoses, limited treatment options and high healthcare costs, that may significantly affect patients' quality of life. The objective of this study was to understand medical service utilization related to diagnosis and treatment, economic burden during diagnosis period, and health-related quality of life among MPS patients in China.MethodsA series of patients diagnosed with MPS registered in the national patient organization were recruited for a cross-sectional survey from May to July 2019. Information were collected from patients or their parents via phone interview, including demographic data, utilization of services related to diagnosis and treatment, total cost during the period of MPS diagnosis and health-related quality of life (HRQoL). HRQoL was assessed by PedsQL 4.0 Generic Core Scale (PedsQL) and 36-item short-form health survey (SF-36) depending on the age of patients with MPS and compared with the general Chinese population.ResultsA total of 180 MPS patients (50, 67, 15, 46, 1 and 1 for type I, II, III, IV, VI and VII), with a mean age of 9.54 years and 137 (76.11%) males, were included in analysis. The mean age at first visit to a medical doctor for MPS related symptoms was 3.65 ± 2.58 years old, while only 12 patients (6.67%) were diagnosed on their first visit. The mean diagnostic delay, which is defined as the time between the first visit to a medical doctor for MPS related symptoms and the final diagnosis, was 9.42 months, with no significant difference between types. The average number of misdiagnosis was 4.56. Before the confirmed diagnosis, the patients made an average of 6.31 visits and visited 4.3 hospitals. During diagnosis period, the mean of ¥81,086.72 direct medical costs accounted for 63.75% of the total cost. Only 32.78% of the patients had ever received specific treatments. The mean scores of PedsQL and SF-36 of patients were significantly lower than the Chinese norms. Household annual income per person, specific treatment use and MPS subtype were significantly associated HRQoL of patients.ConclusionThe results highlight challenges faced by MPS patients in terms of diagnosis, access to specific treatments, economic burden and low HRQoL. There is an urgent need to improve early detection and diagnosis, create fair and consistent mechanisms to increase access to specialized treatment and reduce the economic burden of MPS patients in China.

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Healthcare Resource Utilization and the Cost of Care for Mucopolysaccharidosis I Patients in Iran

Cited by 3 publications

References 24 publications

Post-Tonsillectomy Outcomes in Children with Mucopolysaccharidosis and Obstructive Sleep Apnea

Post-Tonsillectomy Outcomes in Children with Mucopolysaccharidosis and Obstructive Sleep Apnea

Cost-effectiveness of miglustat versus symptomatic therapy of Niemann–Pick disease type C

Health service utilization, economic burden and quality of life of patients with mucopolysaccharidosis in China

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