This review highlights several gaps within the existing literature for both medical and non-medical costs. Medical costs should include a lifetime horizon and explore insurance type in greater detail. Non-medical cost literature is completely lacking outside of a caregiver burden proxy (lifetime income).

The available cost literature for sickle cell disease (SCD) is heterogenous in study design, data sources, population, and analysis methods. These differences all contribute to difficulty in conducting meta-analyses for further synthesis beyond what we present.

Future US cost research should give more detailed attention to medical and non-medical costs to fairly evaluate new and existing health technologies for SCD patients, referring to the Second Panel on Cost Effectiveness in Health and Medicine to gain insight on important cost measurements necessary for value considerations.