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. 2022 May 31;22(1):1078.
doi: 10.1186/s12889-022-13342-2.

Range of invasive meningococcal disease sequelae and health economic application - a systematic and clinical review

Jing Shen  1   2 , Najida Begum  3 , Yara Ruiz-Garcia  4 , Federico Martinon-Torres  5   6   7 , Rafik Bekkat-Berkani  4 , Kinga Meszaros  8
Affiliations

Range of invasive meningococcal disease sequelae and health economic application - a systematic and clinical review

Jing Shen et al. BMC Public Health. .

Abstract

Background: Invasive meningococcal disease (IMD) is uncommon, life-threatening, with many diverse sequelae. The aims were to: 1) comprehensively characterise the sequelae; 2) have a systematic application for sequelae impact in economic evaluation (EE).

Methods: Sequelae categorised as physical/neurological or psychological/behavioural were identified from a systematic review of IMD observational studies (OS) and EEs in high-income countries (published 2001-2020). A comprehensive map and EE-relevant list, respectively, included physical/neurological sequelae reported in ≥2OS and ≥ 2OS + 2EE (≥1OS and ≥ 1OS + 1EE for psychological/behavioural). Sequelae proportions were selected from the highest quality studies reporting most sequelae. Three medical experts independently evaluated the clinical impact of findings.

Results: Sixty-Six OS and 34 EE reported IMD sequelae. The comprehensive map included 44 sequelae (30 physical/neurological, 14 psychological/behavioural), of which 18 (14 physical/neurological and 4 psychological/behavioural) were EE-relevant. Experts validated the study and identified gaps due to limited evidence, underreporting of psychological/behavioural sequelae in survivors/their families, and occurrence of multiple sequelae in the acute phase and long-term.

Conclusions: The considerable burden of IMD sequelae on survivors and their families is potentially underestimated in EE, due to underreporting and poorly-defined subtle sequelae. When assessing IMD burden and potential interventions e.g., vaccination, sequelae range and duration, underreporting, and indirect burden on dependents should be considered.

Keywords: Economic evaluation; Meningococcal infection; Sequelae; Systematic review.

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Conflict of interest statement

KM, YRG and RBB are employees in the GSK group of companies. KM, RBB hold shares in the GSK group of companies. JS was employee and hold share in the GSK group of companies during the conduct of the study. NB is a freelance consultant for the GSK group of companies and therefore received consulting fees during the conduct of the study. FMT reports personal fees, non-financial support and/or grants from Ablynx, Jansen, GSK, Regeneron, Medimmune, Pfizer, MSD Sanofi Pasteur, Novavax, Astra Zeneca, Novartis, Seqirus, Roche, Abott, Instituto de Salud Carlos III and Biofabri, outside the submitted work. JS, NB, YRG, FMT, RBB and KM declare no other financial and non-financial relationships and activities.

Figures

Fig. 1
Fig. 1
Sequelae selection criteria for comprehensive map (from OS) and EE-relevant list (from OS+EE), with sequela proportions from studies with highest SIGN rating and number of outcomes. EE: economic evaluation; OS: observational study; SIGN: Scottish Intercollegiate Guidelines Network. Sequelae selected for the comprehensive map must be reported in at least 2 observational studies (OS) for physical/neurological sequelae and at least 1 OS for psychological/behavioural sequelae. Sequelae proportions were taken from highest SIGN-rated studies reporting the greatest number of sequelae. Sequelae from this map relevant to economic evaluation (EE) must also be reported in at least 2 health economic studies (HES) for physical/neurological sequelae and at least 1 HES for psychological/behavioural sequelae
Fig. 2
Fig. 2
Distribution by geographic location of OS (with age at IMD diagnosis) and EE, and by IMD serogroup. ACWY/B IMD: single or multiple serogroup(s) A,C,W,Y or B invasive meningococcal disease; EE: economic evaluation; EU European Union (i.e., Austria, Germany, Lithuania, Netherlands, Spain, Switzerland, UK); OS: observational study; UK United Kingdom; US United States; SIGN: Scottish Intercollegiate Guidelines Network. Sequelae selection criteria for comprehensive map (from OS) and EE-relevant list (from OS+EE), with sequela proportions from studies with highest SIGN rating and number of outcomes. The distribution of the 66 observational studies (OS) and 34 health economic studies (HES) by country (if > 2 studies conducted per country), and by serogroup
Fig. 3
Fig. 3
Number of IMD sequelae by category reported in A) each OS and B) each EE. AU: Australia; BE: Belgium; CA: Canada; CH: Switzerland; CZ: Czech Republic; DK: Denmark; EE: economic evaluation; EU: European Union; FR: France; GE: Germany; GR: Greece; IC: Iceland; IMD: invasive meningococcal disease; IR: Ireland; IS: Israel; IT: Italy; N.: number; NL: Netherlands; OS: observational study; PO: Poland; SE: Sweden; SL: Slovakia; SP: Spain; UK: United Kingdom; US: United States (of America) *Note: studies reporting number but not type of sequelae or grouping sequelae into a composite outcome. The number of sequelae reported per observational study (OS) and per health economic study (HES) categorised as physical, neurological or psychological/behavioural
Fig. 4
Fig. 4
Time from IMD onset to follow-up study reporting each physical/neurological and psychological/behavioural sequela, by age group at IMD onset. The time from IMD onset (for age groups 0–4 years, 5–17 years and 18+ years) to follow-up study reporting each physical/neurological and psychological/behavioural sequela is presented
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References

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