Systematic review of the patient burden of generalised myasthenia gravis in Europe, the Middle East, and Africa

McCallion, J; Borsi, A; Noel, W; Lee, J; Karmous, W; Sattler, S; Boggia, GM; Hardy, EJ; Mitchell, CR; Mitchell, SA; Gilhus, Nils Erik

doi:10.1186/s12883-024-03553-y

BMC Neurology

Table 3 Summary of trends identified in extracted data

From: Systematic review of the patient burden of generalised myasthenia gravis in Europe, the Middle East, and Africa

Comparison	Generic QoL	Symptom-specific QoL	Disease-specific QoL
gMG patients vs healthy controls	Significant difference (gMG worse): Bartel 1995 (POMS) [9] De Freitas Fregonezi 2006 (SF-36) [16] Jordan 2017 (VAS) [28] Numerical difference (significance not reported): Padua 2001 (SF-36) [38]	Significant difference (gMG worse): Bartel 1995 (IPAT B-score) [9] Happe 2004 (SDS, SAS, PSQI, SSA) [23] Jordan 2017 (CES-D, PSQI, FSMC) [28, 29] Sitek 2009 (BDI) [48] Tascilar 2018 (PSQI, FSS, HAM-A, HAM-D) [52]	Significant difference (gMG worse): Jordan 2017 (MGFS, MG-ADL, MG-QoL 15) [28, 29]
gMG patients vs healthy controls	No significant difference: Happe 2004 (QLI) [23] Jordan 2017 (VAS cognitive) [29]	No significant difference: Bartel 1995 (IPAT A-score) [9] Happe 2004 (ESS) [23] Tascilar 2018 (ESS, adjusted PSQI, adjusted FSS) [52]
gMG vs oMG	Significant difference (gMG worse): gMG vs oMG vs bMG: Stojanov 2019 (SF-36) [50] Numerical difference (significance not reported): gmG vs oMG: Cioncoloni 2016 (subjective measure) [15]	Significant difference (gMG worse): gMG vs oMG: Akkan Suzan 2022 (FIS total, FIS cognitive) [1] gMG vs oMG vs remission: Hoffmann 2016 (CFQ, ISI) [24] gMG vs oMG vs bMG: Stojanov 2019 (HAM-A) [50]	Significant difference (gMG worse): gMG vs oMG vs bMG: Stojanov 2019 (revised MG-QoL 15) [50] With vs without current gMG symptoms: Westerberg 2018 (MG-QoL 15) [55] Numerical difference (significance not reported): gMG vs oMG: De Lapiscina 2012 (MG-QoL 15) [17]
gMG vs oMG		No significant difference: Akkan Suzan 2022 (ESS, FIS physical, FIS social, FAS, BDI) [1] Alanazy 2019 (PHQ-9) [3] De Lapiscina 2012 (ESS, PSQI) [17] Hoffmann 2016 (HADS-D, HAS-A) [24] Significant difference (oMG worse): gMG vs oMG vs bMG: Stojanov 2019 (HAM-D) [50]
MG severity (stage I +)	Significant difference (higher stage worse): MGFA: Basta 2012 (SF-36) [10] Osserman: Busch 1996 (modified EORTC QLQ) [13] MGFA: Dewilde 2022 (EQ-5D-5L) [18] Osserman: Padua 2001 (SF-36) [38] MGFA: Raggi 2010 (SF-36) [41] MGFA: Stojanov 2019 (SF-36) [50] MGFA: Szczudlik 2020 (SF-36) [51]	Significant difference (higher stage worse): Osserman: Aysal 2013 (BAI, HAM-D, HAM-A) [6] MGFA: Stojanov 2019 (HAM-A, HAM-D) [50]	Significant difference (higher stage worse): MGFA: Stojanov 2019 (revised MG-QoL 15) [50]
MG severity (stage I +)		No significant difference: Osserman: Aysal 2013 (BDI) [6]
MG severity within gMG (stage II +)	Significant difference (higher stage worse): Osserman: Bachmann 2008 (EORTC QLQ) [7] MGFA: Szczudlik 2020 (SF-36) [51] Numerical difference (significance not reported between gMG stages): MGFA: Basta 2012 (SF-36) [10] Osserman: Busch 1996 (modified EORTC QLQ) [13] Osserman: De Freitas Fregonezi 2006 (SF-36) [16] MGFA: Dewilde 2022 (EQ-5D-5L) [18] Osserman: Padua 2001 (SF-36) [38] MGFA: Raggi 2010 (SF-36) [41] MGFA: Stojanov 2019 (SF-36) [50]	Significant difference (higher stage worse): Osserman: Aysal 2013 (BAI, HAM-D, HAM-A) [6] Numerical difference (significance not reported between gMG stages): MGFA: Stojanov 2019 (HAM-A, HAM-D) [50]	Numerical difference (significance not reported between gMG stages): MGFA: Stojanov 2019 (revised MG-QoL 15) [50]
MG severity within gMG (stage II +)		No significant difference: Osserman: Aysal 2013 (BDI) [6]
Age	Significant difference (older worse): > 45 vs ≤ 45 years: Bachmann 2008 (EORTC QLQ) [7]	NR	NR
Sex	No significant difference: De Freitas Fregonezi 2006 (SF-36) [16]	Significant difference (female worse): Ruiter 2021 (CIS-f) [46]	NR
Country	NR	Significant difference (Estonia worse): Estonia vs Sweden, Sabre 2017 (FSS) [47]	NR
Impact of treatment	Significant difference (improvement): Thymectomy: Ambrogi 2012 (SF-36) [4] Thymectomy, failure vs response: Busch 1996 (EORTC QLQ) [13] Numerical difference (significance not reported): Rituximab: Peres 2017 (EQ-5D) [40] Thymectomy: Roth 2002 (subjective evaluation) [44]	Significant difference (combination better): Prednisolone alone vs prednisolone with azathioprine/pyridostigmine: Aysal 2013 (BAI, HAM-D, HAM-A total, HAM-A somatic) [6]	Significant difference (improvement): Thymectomy: Baram 2021 (MG-ADL) [8] Methotrexate: Rodolico 2021 (MG-ADL) [43] Standard care: Thomsen 2021 (MG-ADL, MG-QoL 15) [54] Numerical difference (significance not reported): Rituximab: Peres 2017 (MG-QoL 15) [40]
Impact of treatment	No significant difference: Thymectomy, open vs minimally invasive: Bachmann 2008 (EORTC QLQ) [7] Thymectomised vs not thymectomised: Padua 2001 (SF-36) [38]	No significant difference: Prednisolone alone vs prednisolone and azathioprine/pyridostigmine: Aysal 2013 (BDI, HAM-A psychic) [6]	No significant difference: Thymectomy, tThx vs aThx vs sThx: Ruckert (MG-ADL) 2003 [45]
MuSK + MG vs AChR + MG	Significant difference (AChR worse): Stankovic 2018 (MSPSS, SF-36) [49]	No significant difference: Stankovic 2018 (HAM-A, HAM-D) [49]	NR
MuSK + MG vs AChR + MG	No significant difference: Stankovic 2018 (AIS) [49]		NR
PASS-postive vs PASS-negative	Significant difference (PASS-negative worse): Andersen 2021 (EQ-5D-3L, EQ-5D-VAS) [5]	Significant difference (PASS-negative worse): Andersen 2021 (MFI-20, MDI) [5]	Significant difference (PASS-negative worse): Andersen 2021 (MG-ADL) [5]
With vs without psychiatric disorders	Significant difference (psychiatric disorders worse): Kotan 2016 (SF-36, MSPSS, PAIS-SR) [31]	Significant difference (psychiatric disorders worse): Kotan 2016 (HADS-A, HADS-D) [31]	NR
With vs without psychiatric disorders	No significant difference: Kotan 2016 (PTGI) [31]		NR

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