Epidemiological, humanistic, and economic burden of illness of lower limb spasticity in adults: a systematic review

Figures & data

Table 1 Reported incidence and prevalence of conditions associated with lower limb spasticity

Condition	Annual incidence	Prevalence	References
Stroke	30–485 per 100,000	40–600 per 100,000	Benamer et al^Citation39 Saposnik and Del Brutto^Citation40 Hirtz et al^Citation41 Feigin et al^Citation34 Burke and Venketasubramanian^Citation42
Multiple sclerosis	–	2–350 per 100,000	Cristiano et al^Citation43 Cooper et al^Citation44 Poppe et al^Citation45 Hirtz et al^Citation41
Cerebral palsy	–	240–360 per 100,000	Yeargin-Allsopp et al^Citation46
Traumatic brain injury	100–235 per 100,000	–	Hirtz et al^Citation41 Tagliaferri et al^Citation47
Spinal cord injury	0.2–8 per 100,000	22–90 per 100,000	Hagen et al^Citation48 Wyndaele et al^Citation49 Cripps et al^Citation36 Hirtz et al^Citation41 Devivo^Citation37

Figure 1 Flow of literature.

Abbreviation: LLS, lower limb spasticity.

Table 2 Characteristics of studies included in this review

Reference	Underlying pathology	Country	Participants (n)	Focus of study
Marciniak et al^Citation22	Cerebral palsy	Not reported	91	Complications
Noonan et al^Citation21	Cerebral palsy	USA	77	Prevalence, complications
Burbaud et al^Citation29	Lower limb spasticity of any cause	France	870	Treatment patterns, cost of treatment
Marchese et al^Citation31	Lower limb spasticity of any cause	France, Germany, Greece, Sweden, UK	127	Treatment patterns
Zamli and Xiong^Citation32	Lower limb spasticity of any cause	New Zealand	74	Treatment patterns
Hoang et al^Citation14	Multiple sclerosis	Australia	156	Prevalence
Nogueira et al^Citation33	Multiple sclerosis	Brazil	61	Quality of life
Salci et al^Citation16	Multiple sclerosis	Turkey	51	Prevalence
Marcotte et al^Citation15	Multiple sclerosis	USA	17	Prevalence, complications
Sosnoff et al^Citation17	Multiple sclerosis	USA	34	Prevalence, complications
Wagner and Kremer^Citation18	Multiple sclerosis	USA	42	Prevalence
Singer et al^Citation20	Primary lateral sclerosis	USA	25	Prevalence
Diong et al^Citation25	Spinal cord injury	Australia	80	Complications
Voerman et al^Citation24	Spinal cord injury	The Netherlands	19	Complications, quality of life
Kwah et al^Citation26	Stroke	Australia	165	Complications
Pang et al^Citation23	Stroke	Canada	39	Complications
Laurent et al^Citation27	Stroke	France	39	Complications
Urban et al^Citation6	Stroke	Germany	211	Prevalence
Hara^Citation28	Stroke	Japan	296	Complications
Dajpratham et al^Citation12	Stroke	Thailand	327	Prevalence, treatment patterns
Soyuer and Ozturk^Citation13	Stroke	Turkey	100	Prevalence, complications
Leathley et al^Citation3	Stroke	UK	106	Prevalence, predictive factors
Esquenazi et al^Citation30	Stroke and traumatic brain injury	USA	487	Treatment patterns
Singer et al^Citation19	Traumatic brain injury	Australia	105	Prevalence, complications

Table 3 Prevalence of LLS in different conditions

Study	Country	n	Muscle groups	Instrument	Prevalence of LLS
Stroke
Dajpratham et al^Citation12	Thailand	327	Knee flexors	MAS	37%
Leathley et al^Citation3	UK	106	Hip and knee	TAS, TASMAX	28%–31%
Leathley et al^Citation3	UK	106	Ankle	TAS, TASMAX	32%
Urban et al^Citation6	Germany	211	Any LL	MAS	35%
Soyuer and Ozturk^Citation13	Turkey	100	Any LL	AS	29%
Multiple sclerosis
Hoang et al^Citation14	Australia	156	Any LLS	Tardieu Scale	66%
Sosnoff et al^Citation17	USA	34	Gastrocnemius-soleus (ankle flexor)	MAS	44%
Wagner and Kremer^Citation18	USA	42	Ankle flexors	MAS, ISM, dynamometer	69%
Hoang et al^Citation14	Australia	156	Ankle flexors	Tardieu Scale	48%
Marcotte et al^Citation15	USA	17	Knee	MAS	47%
Hoang et al^Citation14	Australia	156	Knee extensors	Tardieu Scale	41%
Salci et al^Citation16	Turkey	51	Knee	MAS	Mean score 4.33
Cerebral palsy
Noonan et al^Citation21	USA	77	Knee	AS	25% none or mild 56% moderate 19% severe
Traumatic brain injury
Singer et al^Citation19	Australia	105	Any LL	AS	13%
Primary lateral sclerosis
Singer et al^Citation20	USA	25	Any LL	NR	92%

Abbreviations: MAS, Modified Ashworth Scale; TAS, Tone Assessment Scale; TASMAX, Tone Assessment Scale, Maximum score at any one joint; AS, Ashworth Scale; NR, not reported; LL, lower limb; LLS, lower limb spasticity; ISM, instrumented spasticity measure.

Table 4 Summary of risk of complications associated with lower limb spasticity in different underlying pathologies

Underlying pathology	Contractures	Limited mobility	Falls	Deep vein thrombosis	Pain
Cerebral palsy		Increased risk^Citation22			Knee spasticity increases risk of hip pain^Citation21
Multiple sclerosis		Increased risk^Citation15,Citation17
Spinal cord injury	No increased risk^Citation25				No association^Citation24
Stroke	Contradictory evidence on risk^Citation26,Citation27	Increased risk^Citation23	Contradictory evidence on risk^Citation13,Citation23	Increased risk in spastic calf^Citation28
Traumatic brain injury	No increased risk^Citation19

Table S1 Search string for MEDLINE-indexed publications

	Search algorithm	Hits
1. Quality of life string	(“quality of life”[tiab] OR “quality-of-life”[tiab] OR “patient reported outcome”[tiab] OR “patient-reported outcome”[tiab] OR “patient reported outcomes”[tiab] OR “patient-reported outcomes”[tiab] OR satisfaction[tiab] OR utility[tiab] OR disability[tiab] OR “functional status”[tiab] OR “physical function”[tiab] OR burden[tiab] OR carer[tiab] OR pain[tiab] OR mobility[tiab] OR ‘self care’[tiab] OR hygiene[tiab] OR falls[tiab])	326,689
2. Economic string	(Cost[MeSH] OR economic[MeSH] OR “burden of illness”[tiab] OR “illness burden”[tiab] OR “cost benefit”[tiab] OR “cost-benefit”[tiab] OR “cost-effective”[tiab] OR “cost effective”[tiab] OR expenditure[MeSH] OR “quality-adjusted life year”[tiab] OR “quality adjusted life year”[tiab] OR qaly[tiab] OR “life years saved”[tiab] OR deductible[tiab] OR insurance[tiab] OR “value of life”[tiab] OR budget[MeSH] OR fiscal[tiab] OR funding[tiab] OR financial[tiab] OR finance[tiab] OR price[tiab] OR pricing[tiab])	110,282
3. Epidemiologic string	(Incidence[tiab] OR prevalence[tiab] OR incidence[MeSH] OR prevalence[MeSH] OR epidemiology[MeSH] OR “epidemiological data”[tiab] OR epidemiology[tiab] OR epidemiologic[tiab] OR epidemiological[tiab] OR “unmet need”[tiab] OR access[tiab])	414,403
4.	1 OR 2 OR 3	753,166
5. Disease string	((spasticity[tiab] OR palsy[tiab] OR rigidity[tiab] OR hemiparesis[tiab] OR hemiplegia[tiab] OR “brain injury”[tiab] OR stroke[tiab] OR “multiple sclerosis”[tiab] OR hypertonic*[tiab] OR “cerebral palsy”[tiab] OR botulinum[tiab]) AND (leg[tiab] OR “lower limb”[tiab] OR calf[tiab] OR thigh[tiab] OR knee[tiab] OR foot[tiab] OR ankle[tiab] OR hip[tiab]))	3,951
6.	4 AND 5	1,457
7.	NOT (child NOT (adult AND child))	1,326
8.	NOT (Review[pt] NOT (systematic[tiab] OR “meta analysis”[tiab] OR (indirect[tiab] OR mixed[tiab] AND “treatment comparison”[tiab])))	1,188

Note: The limits for the MEDLINE search were: only items with abstracts, humans, published in the last 10 years (October 2002 to October 2012) and field: Title/Abstract.

Table S2 Search string for EMBASE-indexed publications

	Search algorithm	Hits
1. Quality of life string	(‘quality of life’:ab,ti OR ‘quality-of-life’:ab,ti OR ‘patient reported outcome’:ab,ti OR ‘patient-reported outcome’:ab,ti OR ‘patient reported outcomes’:ab,ti OR ‘patient-reported outcomes’:ab,ti OR ‘satisfaction’:ab,ti OR ‘utility’:ab,ti OR ‘disability’:ab,ti OR ‘functional status’:ab,ti OR ‘physical function’:ab,ti OR burden:ab,ti OR carer:ab,ti OR pain:ab,ti OR mobility:ab,ti OR ‘self care’:ab,ti OR hygiene:ab,ti OR falls:ab,ti)	388,882
2. Economic string	(‘cost’/exp OR economic OR ‘burden of illness’:ab,ti OR ‘illness burden’:ab,ti OR ‘cost benefit’:ab,ti OR ‘cost-benefit’:ab,ti OR ‘cost-effective’:ab,ti OR ‘cost effective’:ab,ti OR expenditure OR ‘quality-adjusted life years’:ab,ti OR ‘quality adjusted life years’:ab,ti OR qaly:ab,ti OR ‘life years saved’:ab,ti OR deductible:ab,ti OR insurance:ab,ti OR ‘value of life’:ab,ti OR ‘budget’/exp OR fiscal:ab,ti OR funding:ab,ti OR financial:ab,ti OR finance:ab,ti OR price OR pricing:ab,ti)	221,870
3. Epidemiologic string	(‘incidence’/exp OR ‘prevalence’/exp OR ‘epidemiology’/exp OR morbidity:ab,ti OR mortality:ab,ti OR survival:ab,ti OR ‘unmet need’:ab,ti OR access:ab,ti)	835,121
4.	1 OR 2 OR 3	12,235,931
5. Disease string	((spasticity:ab,ti OR palsy:ab,ti OR rigidity:ab,ti OR hemiparesis:ab,ti OR hemiplegia:ab,ti OR “brain injury”:ab,ti OR stroke:ab,ti OR “multiple sclerosis”:ab,ti OR hypertonic*:ab,ti OR “cerebral palsy”:ab,ti OR botulinum:ab,ti) AND (leg:ab,ti OR “lower limb”:ab,ti OR calf:ab,ti OR thigh:ab,ti OR knee:ab,ti OR foot:ab,ti OR ankle:ab,ti OR hip:ab,ti))	6,026
6.	4 AND 5	2,674
7.	NOT (child NOT (adult AND child))	2,352
8.	NOT (Review[pt] NOT systematic:ab,ti OR “meta analysis”:ab,ti OR indirect:ab,ti OR mixed:ab,ti AND “treatment comparison”:ab,ti)	1,918

Note: The limits for the EMBASE search were: only items with abstracts, humans, published in the last 10 years (October 2002 to October 2012) and field: Title/Abstract.

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