Six-minute walk test to assess muscular fatigability and mobility in children with congenital myasthenic syndrome: a pilot study

Bulut, Numan; Yağcıoğlu, Güllü Aydın; Demir, Aykut; Alemdaroğlu-Gürbüz, İpek; Haliloğlu, Göknur; Tunca, Öznur

doi:10.1590/1806-9282.20250307

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ORIGINAL ARTICLE • Rev. Assoc. Med. Bras. 72 (1) • 2026 • https://doi.org/10.1590/1806-9282.20250307 linkcopy

Six-minute walk test to assess muscular fatigability and mobility in children with congenital myasthenic syndrome: a pilot study

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OBJECTIVE: The aims of this study were to determine whether the six-minute walk test predicts muscular fatigability in ambulant children with congenital myasthenic syndrome and to examine the relationship between the six-minute walk test and mobility tests.

METHODS: Eight children (four girls and four boys) with congenital myasthenic syndrome (mean age: 11.75±3.54 years) were included. Five children had COLQ variants. The distance of each minute for the six-minute walk test was determined to evaluate fatigability. The mobility tests included the 10-m walk/run test, rising from the floor, and climbing up and down four stairs.

RESULTS: The mean six-minute walk test distance was 463.38±132.59 m. There was no difference between the first and last minute, between the first and last two minutes, and between any of the minutes during the six-minute walk test (p>0.05). The mean times for performing the 10-m walk/run test, rising from the floor, and climbing up and down four stairs were 8.59±3.36, 6.38±4.49, 4.38±5.07, and 3.35±3.69 s, respectively. The strong correlations were determined between six-minute walk test and all the mobility tests, except for climbing down four stairs (p< 0.05, r_s=-0.72 to -0.83).

CONCLUSION: The six-minute walk test did not provide sufficient evidence to predict muscular fatigability. However, this preliminary study suggested that the six-minute walk test may be used by clinicians and researchers to assess mobility in congenital myasthenic syndrome.

KEYWORDS:
Congenital myasthenic syndrome; Six-minute walk test; Functional performance; Muscle fatigue; Outcome assessment

	Gender	Mutation type	Age (years)	BMI (kg/m²)	Symptom onset age (years)	Functional level*	Family history	Consanguinity
C1	F	COLQ	10	17.16	3	1	No	Yes
C2	M	COLQ	15	32.05	5	3	No	Yes
C3	F	CHRNE	13	16.63	4	1	No	No
C4	F	CHAT	10	17.33	2	1	No	Yes
C5	M	COLQ	15	15.19	0	1	Yes	Yes
C6	M	COLQ	9	14.26	0	1	No	Yes
C7	M	COLQ	16	16.85	0	1	Yes	Yes
C8	F	CHRNE	6	12.80	1	1	No	No

Variables	Children with congenital myasthenic syndrome (n=8)
Variables	Mean±SD	Median (IQR)
Walking distance at 1st min (m)	76.38±19.60	81.00 (56.25–89.25)
Walking distance at 2nd min (m)	76.25±19.70	78.50 (60.25–90.25)
Walking distance at 3rd min (m)	79.56±23.17	83.00 (61.25–98.00)
Walking distance at 4th min (m)	77.56±23.55	79.00 (60.25–97.75)
Walking distance at 5th min (m)	77.63±24.85	86.00 (56.50–94.00)
Walking distance at 6th min (m)	75.75±24.99	83.00 (52.50–92.50)
Six-minute walk test (m)	463.38±132.59	490.50 (354.50–558.00)
10-m walk/run test (s)	8.59±3.36	7.67 (5.93–11.31)
Rising from the floor (s)	6.38±4.49	5.14 (3.31–7.94)
Climbing up four stairs (s)	4.38±5.07	2.55 (2.30–3.60)
Climbing down four stairs (s)	3.35±3.69	2.22 (1.74–2.54)

Measures		Six-minute walk test	10-m walk/run test	Rising from the floor	Climbing up four stairs	Climbing down four stairs
Six-minute walk test	r_s		-0.81	-0.83	-0.72	-0.21
Six-minute walk test	p		0.02*	0.01*	0.04*	0.61
10-m walk/run test	r_s	-0.81		0.74	0.44	-0.10
10-m walk/run test	p	0.02*		0.04*	0.28	0.82
Rising from the floor	r_s	-0.83	0.74		0.68	0.50
Rising from the floor	p	0.01*	0.04*		0.06	0.21
Climbing up four stairs	r_s	-0.72	0.44	0.68		0.64
Climbing up four stairs	p	0.04*	0.28	0.06		0.09
Climbing down four stairs	r_s	-0.21	-0.10	0.50	0.64
Climbing down four stairs	p	0.61	0.82	0.21	0.09

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[T1FN1] *According to Vignos Scale; C: Case; CHAT: choline acetyltransferase; CHRNE: acetylcholine receptor epsilon (∊) subunit; COLQ: collagen like tail subunit of asymmetric acetylcholinesterase; F: female; M: male; BMI: body mass index.