Sickle cell disease (SCD), also known as sickle cell anemia or hemoglobin S disease, is a genetic disorder caused by a mutation in one of the genes encoding hemoglobin [1] . It is characterized by increased susceptibility to infections, organ damage, and episodes of vaso-occlusive crises (VOC) [2] [3] . VOCs represent the most common acute complication and the primary reason for hospitalization in sickle cell disease [4] [5] . All organs are potential targets of VOC [4] . It often requires multidisciplinary care which can lead to significant healthcare expenditures [3] [6] [7] . Hospitalization for VOC management involves costs related to medications, diagnostic tests, and inpatient care. In Togo, no study has determined the economic impact of sickle cell disease on families or on the country budget. A median monthly economic burden of around N76,711 (USD385) per person was found in Nigeria. That’s a lot of money for people in a low-income country [8] .

National Center for Research and Care for Sickle Cell Patients (CNRSD) has been operational since November 2018 in Togo [9] . Patients suffering from acute complications of sickle cell disease are hospitalized in this center. It has been observed that some patients are hospitalized several times within the same month or quarter, often struggling to afford their care due to financial constraints. The objective of this study was to evaluate the cost of managing a painful VOC at CNRSD in 2024.

This was a cross-sectional descriptive survey conducted at the CNRSD from 1 June to 31 August 2024. A structured pre-tested interviewer-administered quiz was used to collect primary data from patients hospitalized at the CNRSD for painful VOC. Painful VOC was defined as bone or joint pain, pain in the ribs or spine, pelvis, skull, or abdominal pain. Patients were included if they consented to participate by signing the questionnaire form, which served as proof of consent. For patients under 18 years of age, consent was provided by a parent who brought the child to the hospital.

Exclusion criteria included indigent patients who received assistance from the social service, patients referred to the CNRSD after receiving care in another facility, and patients who died during hospitalization.

In addition to the quiz used to collect medical and social data (Appendix 1), each patient included in the study was asked to carefully record all expenses incurred during hospitalization.

When the patient was discharged, the various invoices were collected to complete the patient’s quiz. Parameters analyzed included age, presence of comorbidities, patient phenotype, duration of hospitalization, hospital-related expenditures, and the patient’s average income. Costs were calculated in CFA francs (XOF). Total care costs were derived by summing consultation fees, medication expenses, diagnostic test charges, and hospitalization fees. The tariffs in effect at the CNRSD during the study period are provided in Appendix 2.

During the study period, 765 patients were admitted for painful VOC. Of these, 300 patients (39.22% of the target) were included in the study. Most of the patients were of the SS genotype (n = 192). The monthly incidence of VOC was 255, equating to an average of 8.5 VOC managed daily at the CNRSD.

The median age of the patients was 16.5 years, with an interquartile range of [7; 23] and extremes of 2 and 60 years. The cohort included 178 females and 122 males, with a sex ratio of 0.68. Most patients (61.33%) were under 20 years of age, as shown in Figure 1 .

The duration of hospitalization was longer in patients with the SS phenotype, as shown in Table 1 .

¹Kruskal-Wallis rank sum test; Min = minimum; Max = maximum; SD = standard deviation.

The study population was predominantly young, and the majority had no income ( Figure 2 ).

The median costs of the various requirements for managing a painful VOC are in Table 2 .

Q₁ = First quartile; Q₃ = Third quartile.

The consultation fee, which was 1500 XOF (€2.29), must be included. Thus, in 2024, the median cost of managing a painful VOC at the CNRSD was 64,130 XOF (€97.77).

The hospitalization duration for SS patients was generally longer, leading to correspondingly higher expenses ( Table 3 ).

The cost of managing a VOC varied with the patient’s age; it was lower among younger patients (below the median age), as shown in Table 4 .

The presence of comorbidity (e.g., asthma, hypertension, diabetes, history of stroke) did not affect the cost of managing a painful VOC ( Table 5 ).

¹Kruskal-Wallis rank sum test; IQR = Interquartile range.

¹Wilcoxon rank sum test.

The aim of this study was to determine the average cost of managing a VOC. The cost is relatively high, with a median expenditure of 64,130 XOF (€97.77) for a single painful VOC episode requiring an average hospital stay of 3.1 days. This cost exceeds the financial means of most of the Togolese population, where the minimum wage has been set at 52,500 XOF (€80) since January 2023.

¹Wilcoxon rank sum test; IQR = Interquartile range.

During this study, laboratory tests included a complete blood count, malaria testing (thick smear or rapid diagnostic test), renal function tests, liver function tests, and lactate dehydrogenase levels. No imaging tests, such as X-rays or abdominal ultrasounds, were performed.

Sickle cell disease (SCD) is a public health priority in several African countries, and the direct costs of managing its acute complications have attracted the attention of many researchers. Studies by Ngolet L. O. et al. in the Democratic Republic of Congo (2013) [7] , Kafando E. et al. in Burkina Faso (2017) [6] , and Fiawo et al. in Togo (2020) [3] evaluated the direct costs of managing acute SCD complications (VOC, infections, acute anemic crises, etc.).

In the DRC, the average cost of managing a simple VOC (mono- or bifocal pain without fever, lasting less than 5 days) was 67,095 XOF (€102.43), while a severe VOC (multifocal pain with fever, anemia aggravation, lasting more than 5 days) cost 199,365 XOF (€304.37) [7] . Kafando et al. reported an average hospital stay of 4.5 days for VOC and 7.5 days for infections, with an average patient expenditure of 125,616 XOF (€191.50) [6] . At the Sylvanus Olympio and Campus University Teaching Hospitals in Lomé, Fiawo M. et al. estimated the average direct cost of hospitalization for acute SCD complications at 231884.6 ± 175547.03 XOF (€353.5 ± €267.62).

These studies consistently demonstrate high expenses (exceeding the minimum wage in all three countries) for an episode of acute SCD complications, confirming the financial burden on affected individuals.

Only 11.33% (n = 34) of our patients had a stable monthly income above 100,000 XOF (€152). This highlights the economic challenges faced by patients and their families in managing painful VOC. Gbadoe A. D. et al. have described the difficulties encountered by low income countries in managing chronic diseases, due to low health budgets and the precarious socio-economic conditions of their populations [10] . The same observations of the financial burden of sickle cell disease on families have been observed in Nigeria [8] and Gabon [11] .

The median cost at the CNRSD (64,130 XOF) is lower than the costs reported by other studies, which could be explained by two factors. First, CNRSD fees are generally reduced by 30% - 50% compared to public hospital rates in Togo, a measure intended to improve financial accessibility for patients at the country’s only SCD care and research center. Second, our study focused on the direct costs of managing painful VOC, while other authors evaluated the costs of managing all acute complications, such as infections, thrombotic events, and acute anemic crises, which are typically more expensive [12] .

As in other studies, most of our patients (61.33%; n = 184) were under 20 years old. This reflects the increased frequency of VOC during childhood and teenage [13] . The cost of managing a painful VOC varied with age and hemoglobin phenotype but was not influenced by the presence of comorbidities. This is likely due to the longer hospitalization durations observed in patients with the SS phenotype and the higher medication requirements in adults.

VOC, the leading cause of hospitalization in SCD patients, imposes significant financial burdens, increasing the vulnerability of affected individuals. During hospitalization for a painful VOC, pharmacy costs constitute the largest expense, followed by diagnostic tests. The relatively high cost of VOC management can lead to delayed consultations. We call on public authorities to provide support that facilitates access to quality care for acute SCD complications in general and painful VOCs in particular.

Quiz for survey

Datasheet N˚ Investigator name:

Date: ---- /---- /----

A- Social and demographic information

I- Surname: First name:

Age: Sex: F M

II- Are your biological parents alive?:

2.1- Father Yes No

2.2- Mother Yes No

III- Who pays for this hospitalization?

3.1- Father Yes No

3.2- Mother Yes No

3.3- Both parents Yes No

3.4- Yourself Yes No

IV- What is your occupation?

4.1 = civil servant 4.3 = worker

4.2 = craftsman 4.4 = unemployed

4.5 = Other (please specify)

V- What is your father’s occupation?

5.1 = civil servant 5.3 = worker

5.2 = craftsman 5.4 = unemployed

5.5 = Other (please specify)

VI- What is your mom’s occupation?

6.1 = civil servant 6.3 = worker

6.2 = craftswoman 6.4 = unemployed

6.5 = Other (please specify)

VII- What is your average monthly income?

7.1: No income 7.2: less than 52,500

7.3: from 52,500 to 100,000 7.4: From 100,000 to 150,000

7.5: from 150,000 to 200,000 7.6: more than 200,000

7.7: variable

VIII- How much does Dad earn on average per month?

8.1: No income 8.2: less than 52,500

8.3: from 52,500 to 100,000 8.4: from 100,000 to 150,000

8.5: from 150,000 to 200,000 8.6: more than 200,000

8.7: variable

IX- How much does Mom earn on average per month?

9.1: No income 9.2: less than 52,500

9.3: from 52,500 to 100,000 9.4: from 100,000 to 150,000

9.5: from 150,000 to 200,000 9.6: more than 200,000

9.7: variable

B- Medical information

10- Do you have an abnormality other than sickle cell?

10.1- Yes No

If yes to the previous question, please specify.

10.2 Asthma

Diabetes

High blood pressure

Other (please specify)

11- What kind of sickle cell disease do you have?

11.1: SS 11.2: SC 11.3: SBêta-thalassemia

12- How many hospitalizations do you have on average per year? ......................

13- Where do you have pain?

13.1- limbs 13.2- spinal column 13.3- pelvis bones

13.4- ribs 13.5- stomach ache 13.6- skull

13.7- Other (please specify) ………………………..

C- Hospitalization-related expenses

Total amount =

Prescriptions (drugs and medical consumables) =

Medical tests =

Hospitalization fees =

Number of days in hospital: ………………