Journal of Pediatric Rehabilitation Medicine: An Interdisciplinary Approach 11 (2018) 57–70 57

DOI 10.3233/PRM-170507
IOS Press

Review Article

Interventions to improve sensory and motor

outcomes for young children with central
hypotonia: A systematic review
Ginny Palega,∗ , Mark Romnessb and Roslyn Livingstonec
a
Montgomery County Infants and Toddlers Program, Rockville, MD, USA
b
Department of Orthopaedic Surgery, University of Virginia, Charlottesville, VA, USA
c
Sunny Hill Health Centre for Children, Vancouver, BC, Canada

Accepted 30 December 2017

Abstract.
OBJECTIVE: To evaluate evidence supporting physical and occupational therapy interventions used to improve sensory and
motor outcomes for children 0–6 years with central hypotonia.
METHODS: Four electronic databases were searched from 1996 to March 2017. Level of evidence and study conduct was eval-
uated using American Academy of Cerebral Palsy and Developmental Medicine criteria. Traffic lighting classification identified
interventions that were green (proven effective), yellow (possibly effective) or red (proven ineffective or contraindicated).
RESULTS: Thirty-seven articles were included. Nine studies measured orthotic interventions while four distinct studies pub-
lished over nine articles measured treadmill interventions. Remaining studies measured impact of compression garments, mas-
sage, motor and sensori-motor interventions, positioning and mobility interventions.
CONCLUSIONS: Green light evidence supports treadmill training (to promote ambulation and gait characteristics) and massage
(to positively affect muscle tone, motor development and use of vision) for infants with Down syndrome. These interventions are
considered Yellow (possibly effective) for other populations. Green light evidence supports impact of orthoses on foot alignment
for ambulatory children with hypotonia, while impact on gait characteristics is Yellow light and motor development may be neg-
atively impacted (Red light) in pre-ambulatory children. All other interventions rated Yellow (possibly effective) and therapists
should monitor using sensitive outcome measures.

Keywords: Congenital hypotonia, hypotonic, children

1. Introduction central hypotonia represents 60% to 80% of cases [4].

Hypotonia, or reduced resistance to passive move-
ment [1] may be identified in 4.25% of neonates born
at or above 35 weeks gestation [2]. While congenital
hypotonia may be central or peripheral in origin [3],
∗ Correspondingauthor: Ginny Paleg, Montgomery County In-
fants and Toddlers Program, 420 Hillmoor Dr, Silver Spring, MD
20901, USA. Tel.: +1 301 452 4656; E-mail: ginny@paleg.com.
Central hypotonia is concurrent with many genetic dis-
orders such as Down, Rett, Joubert and Prader-Willi
syndromes, but a similar presentation is also seen with
children where no underlying cause can be identified.
There are reports in the literature of children with hy-
potonic cerebral palsy (CP), but some surveillance reg-
isters exclude hypotonia as a CP sub-type [5] while
others describe the prevalence as high as 5.1% [6].
Many children with idiopathic as well as other
causes of hypotonia are referred to physical therapy

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58 G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia

Table 1
Search terms and strategy
Population Diagnosis Therapy type Intervention
Child* Down syndrome Physical Therap* Treadmill training
Toddler* Rett syndrome Physiotherap* Massage
Infant* Joubert syndrome Occupational Therap* Orthot*
‘Low tone’ Prader-Willi Rehabilitation Orthos*
‘muscle tone’ Mowat-Wilson
Hypoton* Developmental Delay
Search Strategy Sample (Medline or CINAHL)
1996–2017 – English Language – (child* OR toddler* OR infant*) AND (hypoton* OR
‘low tone’ OR ‘muscle tone’) AND (Down syndrome OR Rett OR Joubert) AND (physi-
cal therap* OR occupational therap* OR rehabilitation OR physiotherapy*)
∗ = truncation of key words.

(PT) and/or occupational therapy (OT) services with
concerns regarding developmental and motor delays.
Although believed to be beneficial [4], the effective-
ness of PT and OT interventions for children with cen-
tral hypotonia is unclear and clinical practice guide-
lines for young children combine low-level evidence
with expert opinion [7]. Therapeutic interventions by
OTs and PTs for children with hypotonia often fo-
cus on achieving developmental milestones, improving
posture and postural stability, strength, endurance and
enhancing function and activity [8].
While hypotonia from metabolic, genetic and neu-
rologic pathologies may differ from idiopathic hypo-
tonia, interventions for children with central hypoto-
nia (regardless of specific etiology) should be based
on the best available evidence and, in the absence of
strong research with the specific population, interven-
tions should be based on best-practice for children with
similar impairments [9]. Accordingly, the purpose of
this review was to identify and evaluate the evidence
supporting interventions used by PTs and/or OTs to en-
hance motor and sensory outcomes for children aged
birth to 6 years-of-age with central hypotonia.
2. Method
The Preferred Reporting Items for Systematic Re-
views and Meta-Analyses (PRISMA) statement was
used to structure this review [10]. An electronic data-
base search was undertaken in July 2016 and up-
dated March 2017 for articles published from 1996
to present. We included studies conducted in the
last 20 years, as these would meet similar report-
ing standards. Databases included CINAHL; Medline;
EMBASE; and EBM Reviews. See Table 1 for search
terms and a sample search strategy. No limits were
placed on study design or publication status in the ini-
tial search. Primary research studies published in En-
glish in peer-reviewed journals were included if 50%
or more participants were explicitly described as hy-
potonic or diagnosed with syndromes associated with
hypotonia (e.g. Down syndrome) and measured impact
of PT and/or OT interventions on motor or sensory
outcomes. Exclusion criteria included; studies focused
on other types of outcomes (e.g. behavioral, commu-
nication, activities of daily living); interventions not
commonly used by OTs or PTs in typical early in-
tervention settings (e.g. robotic trainers); interventions
requiring an advanced professional certification (e.g.
hippotherapy); studies involving children with hypoto-
nia not central in origin (e.g. Spinal Muscular Atrophy,
Spina Bifida, Spinal Cord Injury, Brachial Plexus In-
jury, etc.); and studies published as abstracts only.
Data were extracted from articles meeting inclusion
criteria (see Table 2). American Academy of Cerebral
Palsy and Developmental Medicine (AACPDM) [11]
criteria was used to identify evidence level and
AACPDM quality rating was used for individual stud-
ies evidence level I–III. AACPDM levels are as fol-
lows: Level I – systematic review (SR) of randomized
controlled trials (RCTs), and large RCTs; Level II –
smaller RCT’s and SR of cohort studies; Level III – co-
hort studies with concurrent controls, and SR of case
control studies; Level IV – case series, cohort studies
with historical controls, and case-control studies; Level
V evidence – expert opinion or case studies. The traffic
lighting classification system [12] was used to identify
interventions that were green (proven effective), yel-
low (possibly effective) or red (proven ineffective or
contra-indicated). All three authors agreed on studies
to be read full text, articles meeting inclusion criteria,
content of evidence table, level of evidence and quality
ratings. Disagreements were resolved through discus-
sion at all stages.
 Table 2
Included articles
Reference Design AACPDM Participants N N Age Intervention Results
Level treatment Controls
An 2015 [13] Case V Hypotonic 1 ∼ 19 mos Swing: vestibular stimulation Improved Bayley III mental and mo-
study CP tor function scores.
Angulo- RCT II Down 16 High 14 Low 10 mos As Ulrich 2008. HI group sig higher velocity, cadence,
Barrosa syndrome Intensity Intensity and lower double support percentage.
2008A [14] (HI) (LG)
Angulo- RCT II Down 16 (HI) 14 (LG) 10 mos As Ulrich 2008 HI: higher levels of leg activity.
Barrosa syndrome
2008B [15]
Buccieri [16] Case V Flatfoot and 1 ∼ 25 mos Foot orthosis plus weekly PT PDMS scores improved 12 mos over
study GM delay 5 mos.
Gawand [17] 2 group III Down 14 14 14.9 and Adaptive chair Stat sig difference motor and manipu-
study syndrome 15.1 mos lation scores and right hand grasp sta-
bility and mobility.
George [18] Case V Hypotonic 1 ∼ 19 mos Foot orthoses, 22 days: standing Shoes plus orthoses improved rise to
study CP and cruising only stand, standing, lowering, cruising,
and stepping forward.
Hernandez- RCT II Down 12 11 Mean 30 mins massage vs reading 2 x Increased fine and gross motor func-
Reif [19] syndrome 24.5 mos week: 2 mos tioning and less severe limb hypo-
tonicity than controls.
Karimi [20] RCT II Down 12 12 6–18 mos OT vs OT plus herbal preparation Both groups improved in motor
syndrome and binding 3x/wk 45 mins x 6 skills. Intervention group higher
mos GMFM scores (not stat sig).
LaForme- 2 group III Down 5 5 13–29 mos 10 weekly sensory motor group Sig increase lying rolling, crawling
Fiss [21] study syndrome kneeling, and GMFM total score.
Logan [22] Case V Down 1 ∼ 13 mos Switch adapted ride-on toy car Increased movement, mobility and
study syndrome socialization.
Looper RCT II Down 10 7 578 days TT at home; 0.2m/s, 8 mins All sig increased GMFM scores. At
2010 [25] syndrome (SD 188) day, 5 days/week. Intervention: 1 month of walking, controls higher
SMO’s 8 hrs/day 5 days/week. GMFM scores than SMO group.
Controls: SMOs once walking.
Looper RCT II Down 10 7 578 days No group differences hand support in
2011 [24] syndrome (SD 188) upright. All decreased 2-hand support
over time.
Looper Repeated IV Down 6 ∼ 4–7 yrs SMO vs foot orthosis or barefoot Longer cycle time with SMO’s than
2012 [23] Measures syndrome foot orthoses or barefoot walking;
and lower cadence than barefoot.
G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia

Lotan 2005 [26] Case V Rett 1 ∼ 5 yrs Adaptive seating, standing and Decreased scoliosis from 29 degrees
study syndrome lying to 20 degrees over 18 mos.
Lotan 2012 [27] SSRD AB V Rett 3 ∼ 3–5 yrs 18 mos conductive education GM function improvements post in-
syndrome tervention.
59
 60
Table 2, continued
Reference Design AACPDM Participants N N Age Intervention Results
Level Treatment Controls
Mahoney [28] 2 groups III CP (23); Down 28 22 Mean Developmental vs NDT inter- GM skills and quality of movement
non- Syndrome (27) 14.3/13.9 vention over 6 mos. improved irrespective of interven-
randomized mos tion or diagnosis.
Martin [29] Repeated IV Down 17 ∼ 3.6–8 yrs SMO’s plus shoes vs shoes SMOs plus shoes sig improvement
measures Syndrome (mean 5.10 alone standing, walking running, balance.
yrs)
Mattern- Pre-test IV Developmental 12 (6 with ∼ Mean 30.4 Treadmill training Group Stat sig increased standing and
Baxter [30] Post-test delay hypotonia) mos 2x/wk x14wks walking skills and mobility scores.
Mintz- RCT II Benign 14 15 8–12 mos PT 1x week vs 1 x month Weekly PT group walked earlier, no
Itkin [31] hypotonia sig differences by 18 mos.
Paleg [32] Case V Hypotonic CP 1 ∼ 18 months Benik Vest 2–6 hrs/day No change in GMFM score, but im-
study proved vision, alertness, head con-
trol, laughing and rolling.
Parent- Case V Noonan 1 ∼ 3 years 10 SMO × 8 wks 2x weekly PT Improved scores on PDMS-2, gait
Nichols [33] study syndrome mos parameters and decreased pain.
Purpura [34] RCT II Down 10 10 1–3 mos to Massage by parents 20 mins Faster development of visual acuity
syndrome 12 mos daily and stereopsis. Stat sig difference at
age 5–6 mos.
Ross [35] RCT II Hypotonia and 13 12 18 mos– Soft orthosis plus GM therapy No difference in gait parameters.
flexible flat 5 yrs vs GM therapy alone Sig modified arch index.
feet
Santos [36] Case IV Down 5 5 TD 36 mos Ankle weights 1/3 weight Increased touching of foot to sur-
control syndrome lower limb face when weights removed.
Sayers [37] Case series IV Down 5 ∼ 18–38 mos Exercises with ankle weights Improved motor development.
syndrome
Schaefer- Case V Rett syndrome 1 ∼ 5 yrs Ambulation devices Improved gait with metal shopping
Campion [38] study cart and anterior 4-wheeled walker.
Selby- Repeated IV Down 16 10 TD 3–6 yrs Shoes only vs shoe plus or- Orthoses plus shoes improved gait
Silverstein [39] Measures/ syndrome thoses more than orthoses alone.
Case control
Silva [40] Randomized II Cerebral palsy 14 14 2 yrs 5 mos Qigong massage daily Sig improved PDMS scores. Sen-
crossover and Down by parent sory responses no treatment effect.
syndrome
Tamminga [41] SSRD IV Down 2 ∼ 19 and 24 Two styles of SMO’s vs shoes One style better for both subjects,
ABAB syndrome mos other better for one subject vs
shoes.
Ulrich RCT II Down 15 15 10 mos 8 min/day 5 days/wk home TT Walked with help 74 days and inde-
2001* [43] syndrome pendently 101 days earlier.
Ulrich RCT II Down 16 High In- 14 Low 10 mos LG = 8 min/day, 5x/wk at 0.15 HI group increased stepping and at-
G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia

2008 [42] syndrome tensity (HI) Intensity m/s. HI = added ankle weights tained Bailey III motor milestones
(LG) after 10 steps/min; belt speed earlier.
to 0.30 m/s; 8–12 min/day
Wang [44] Case IV Down 20 30 TD 3–6 yrs Jumping training Improved scores for floor walk,
control syndrome beam walk and jumping.
 Table 2, continued
Reference Design AACPDM Participants N N Age Intervention Results
Level Treatment Controls
Washington SSRD I CP, Down 4–2 w/ (own 9–18 mos Contoured foam seating Improved postural alignment,
[49] alternating Syndrome and hypotonia controls) play, and social interactions.
treatments hypotonia
Wentz [45] 3 group IV Down 10 < 11 9 historical (Early) Tummy time 90 mins/day initi- Stat sig improved motor scores
study syndrome mos controls 40.9 days- ated before and after 11 wks of for early group at 1, 2, and 3 mos
9 > 11 mos (late) age following intervention initiation.
95 days
Wu 2007 [46] 2 groups vs IV Down 16 HI 15 Mean 10 As Ulrich 2008 HI: sig earlier walking onset and
historical syndrome 14 LG historical mos more advanced gait patterns.
controls controls [43]
Wu 2008 [48] RCT II Down 16 HI 14 LG Mean 10 As Ulrich 2008 HI group chose to step over an ob-
syndrome mos ject earlier than LG group.
Wu 2010 [47] RCT II Down 16 HI 14 LG 10 mos As Ulrich 2008 Sig advanced joint kinematics.
syndrome HI: peak ankle plantar flexion at
or before toe-off; increased for-
ward thigh swing duration after
toe-off i.
AACPDM = American Academy of Cerebral Palsy and Developmental Medicine; CP = cerebral palsy; GM = gross motor; GMFM = Gross Motor Function Measure; hrs = hours; HI = high
intensity training; LG = low intensity generalized training; min = minutes; mos = months; m/s = meters per second; OT = Occupational Therapy; PEDI: Pediatric Evaluation of Disability
Inventory; PDMS = Peabody Developmental Motor Scales; PT = Physical Therapy; RCT = randomized controlled trial; sig = significant; SMO = supramalleolar orthosis; SSRD = single
subject research design; stat = statistically; TD = typically developing; TT = treadmill training; vs = versus; wk = weeks; yrs = years.
G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia
61
62 G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia

Fig. 1. PRISMA flow diagram of the search results. From: Moher D, Liberati A, Tetzlaff J, Altman DG, The PRISMA Group (2009). Pre-
ferred Reporting Items for Systematic Reviews and Meta-Analyses: The PRISMA Statement. PLoS Med 6(6): e1000097. doi:10.1371/journal.
pmed1000097.

3. Results mill training was reported over six articles [14,15,42,

Thirty seven articles met inclusion criteria [13–49]
(see Fig. 1 for PRISMA flow chart) and are summa-
rized in Table 2. See online appendix A for details of
excluded studies.
Two relevant systematic reviews [50,51] were iden-
tified, but since they included some studies not meet-
ing inclusion criteria for this review they are refer-
enced only in the discussion. The 37 included arti-
cles represent 31 individual studies. Nine studies [16,
18,23–25,29,33,35,39,41]. measured orthotic interven-
tions while four studies [14,15,25,30,42,43,46–48]
measured treadmill interventions. One study on tread-
46–48] and another study investigating use of supra-
malleolar orthoses (SMOs) with treadmill training was
reported over two articles [24,25]. Remaining stud-
ies measured impact of compression garments [32],
massage [19,34,40], motor [27,28,31,36,37,44,45] and
sensori-motor [13,20,21] interventions, positioning
and mobility interventions [17,22,26,38,49]. Of 31
individual studies, 20 included children with Down
syndrome, while children with Rett syndrome, CP
with hypotonia and developmental delays were repre-
sented in fewer studies. A wide range of study designs
were identified including nine individual randomized
controlled trials (RCTs) or randomized crossover de-
 G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia
signs [19,20,25,31,34,35,40,42,43] as well as a num-
ber of non-randomized group designs such as pre-test
post-test, repeated measure and case-control designs.
Single-subject designs and case studies were also rep-
resented.
Results were divided into seven different types
of clinical interventions; compression garments, mas-
sage, motor interventions, positioning and mobility
interventions, orthotic interventions, treadmill train-
ing, and sensory interventions. Motor interventions
were all different and included Conductive Education,
Neurodevelopmental Therapy (NDT), ankle weights,
jumping training and tummy time as well as different
intensities of PT. Positioning and mobility devices in-
cluded adaptive seating, standing and lying systems,
walking aids and early power mobility. See Table 3
for a summary of positive versus inconclusive results
across AACPDM evidence levels with details of out-
come measures used. Statistically significant results
are shown by bold type and mainly achieved in obser-
vational rather than experimental designs.
Evidence supporting the seven clinical intervention
areas is summarized in Table 4 with AACPDM evi-
dence level [11] and quality rating for individual stud-
ies achieving AACPDM [11] evidence levels I-III. See
online appendix B for AACPDM [11] quality rating
detail. Interventions supported by statistically signif-
icant positive results from moderate or high quality,
level I or II group studies were accorded a green light
or proven effective recommendation. Other than mas-
sage and treadmill training for infants with Down syn-
drome, other interventions achieved yellow light indi-
cating that although results may be positive, outcomes
should be measured to ensure benefit for individual
children. Evidence supporting orthotic interventions is
mixed, with possible negative effects for use of or-
thoses with children who are not yet walking indepen-
dently [25], yellow evidence for type and timing of in-
troduction of orthoses and green light for promoting
better stability and foot structure and alignment in chil-
dren who are walking independently.
4. Discussion
In this review, the strongest evidence was found to
support massage (to improve muscle tone, use of vision
and gross motor skills) and treadmill training (to ac-
celerate onset of walking and improve gait parameters)
for children with Down syndrome. Only one treadmill
training study including children with a diagnosis other
63
than Down syndrome met inclusion criteria for this
review [30]. Treadmill training should be standard of
care for infants with Down syndrome [52] and may
be reasonably supported as an intervention for young
children with other causes of hypotonia [53]. This was
confirmed by a systematic review [50] despite having
different inclusion criteria than this review. A major
limitation however, is that of the nine primary studies
that met our inclusion criteria, six used the same data
set and this warrants caution in application of results.
Based on the best available evidence for effective
treadmill training protocols for children with Down
syndrome, treadmill training for children with hypoto-
nia from other causes may benefit from home based
treadmill training using speed of 0.33–0.67 miles per
hour (or 0.15–0.35 meters per second), starting at a
lower speed and increasing as the child gains skill.
Starting around 10 months of age for 8–12 minutes per
day, 5 days a week, increasing time as the child pro-
gresses and possibly adding light ankle weights to in-
crease resistance may be effective. As with adapting
interventions shown to be effective for a different pop-
ulation, therapists should be careful to measure out-
comes with children to ensure desired outcomes. In the
case of treadmill training, progress should be recorded
with a valid and reliable outcome measure. Therapists
should also monitor vital signs especially in children
with cardiac concerns.
Most early intervention programs encourage natural
environments and routines and treadmill interventions
may be challenging to incorporate into this model. The
use of typical infant walkers, reverse postural control
walkers, push toys and/or gait trainers have not been
studied in this same population, but these interven-
tions may be more effectively incorporated into nat-
ural environments and routines. Positive results have
been demonstrated from gait trainer interventions with
children with intellectual disability, many presenting
with hypotonia [54]. More rigorous research is re-
quired with group designs to support the effectiveness
of gait trainer or ‘over-ground’ training for children
with hypotonia.
There is also good evidence supporting use of mas-
sage with infants with Down syndrome. Infant mas-
sage is also helpful for parent bonding with children
who may have self-regulation, sensory and motor con-
cerns [55]. Although the difference in visual acuity and
stereopsis [34] was not maintained as the children got
older, it could be argued that stimulating earlier use of
vision might impact other outcomes like cognition or
visual motor control and should be considered for chil-
 64
Table 3
Outcomes
Positive outcomes No change or inconclusive
OUTCOME by Level I Level II Level III Level IV Level V Level I Level II Level III Level IV Level V
AACPDM
evidence levels
COMPRESSION GARMENTS
GMFM Paleg [32]
Ability to roll Paleg [32]
MASSAGE
Muscle Tone: ALT Hernandez-
Reif [19]
Visual Acuity and Purpura [34]
Stereopsis
Sensory process- Silva [40]
ing; SSC
Gross and fine Hernandez-
motor: DIPYC Reif [19]
Gross motor: Silva [40]
PDMS
MOTOR INTERVENTIONS
Gross motor: Mintz-
AIMS Itkin [31]
Movement Quality: Mahoney [28]
TIME
Gross motor: Mahoney [28]
PDMS
Ability to kick Santos [36]
Gross motor: RFES Lotan [27]
Balance and jump- Wang [44]
ing: BOT
Gross motor: Wentz [45]
Bailey III
Gross motor: Lotan [27]
RSGMS
Hand function Lotan
Developmental Sayers [37]
quotient
ORTHOTIC INTERVENTIONS
Arch Index Ross [35]
Foot alignment Selby-
G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia

Silverstein [39]
Standing hands- Looper [24]
free
Gait parameters Parent-Nichols [33] Ross [35]
 Table 3, continued
Positive outcomes No change or inconclusive
OUTCOME by Level I Level II Level III Level IV Level V Level I Level II Level III Level IV Level V
AACPDM
evidence levels
GMFM Martin [29]
Tamminga [41]
Gross motor: Buccieri [16]
PDMS George [18]
Parent-
Nichols [33]
Balance: BOT Martin [29]
Pain
Parent-
Nichols [33]
POSITIONING AND MOBILITY DEVICES
Mobility Logan [22]
Distance walked Schaefer
-Campion [38]
Postural alignment Washington [49] Lotan [26]
Fine motor skills Gawand [17]
TREADMILL TRAINING
Leg movement Angulo-
Barrosa [15]
Age of walking Ulrich Wu 2007 [46]
2001 [43]
Bayley III Ulrich
2008 [42]
Gait parameters Angulo- Wu 07 [46]
Barrosa [14]
Wu
2008 [48]
Wu
2010 [47]
PEDI mobility Mattern-
Baxter [30]
FMS Mattern-
Baxter [30]
GMFM Mattern-
Dimensions D & E Baxter [30]
SENSORI-MOTOR INTERVENTIONS
Bayley III An [13]
G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia

GMFM La Forme-Fiss [21] Karimi [20]

GAS La Forme-Fiss [21]
AIMS = Alberta Infant Motor Scale; ALT = muscle tone rating; BOT = Bruininks Osoretsky Test of Motor Proficiency; DPIYC = Developmental Profile for Infants and Young Children;
GAS = Goal Attainment Scaling; GMFM = Gross Motor Function Measure; PDMS = Peabody Developmental Motor Scales; PEDI = Pediatric Evaluation of Disability Inventory; RFES =
Rett Functional Evaluation Scale; RSGMS = Rett Syndrome Gross Motor Scale; SSC = Sense and Self regulation Checklist; TIME = Toddler Infant Motor Evaluation.
65
66 G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia

Table 4
dren with other causes of hypotonia. Expert opinion Traffic lighting synthesis
suggests that early intervention providers, certified in
infant massage, train parents and caregivers to pro-
vide traditional massage strokes given at a medium
level [56]. This training should include attention to
state (arousal versus relaxation), stress signals (e.g. fin-
ger splaying, yawning or eye aversion) and enhance
caregiver child bonding as well as the child’s aware-
ness of body segments.
The evidence surrounding use of orthotic interven-
tions is more controversial, with a mix of positive, in-
conclusive and negative results. From a biomechanical
perspective, orthoses should provide children with hy-
potonia a more neutral and stable foot position how-
ever, children should also be given barefoot time to
explore the sensation of active weight bearing and
weight shifting. Kathy Martin and Julia Looper (per-
sonal communication March 2017) suggest that chil-
dren who are pulling to stand and have good ex-
ploratory behavior should be allowed to move with-
out the restriction of full-time orthotic use. Those that
do not have the stability at the foot and ankle to move
and explore in the standing position, should be pro-
vided with orthoses that give them enough stability to
explore their environment in order to maximize cog-
nitive, motor and social development. When children
pull to stand and cruise, a wearing schedule of 50%
waking time is suggested.
The systematic review of orthotics was inconclu-
sive [51], but did not divide the studies on children
before and after the onset of independent ambula-
tion. The authors questioned the reliability and valid-
ity of the outcome measure used to support the con-
tention that SMOs and foot orthotics improved foot
alignment [35]. However, numerous references suggest
that the Staheli arch index [57] is reliable [58], has
clinical utility and is sensitive in identifying flatfoot
in preschool children [59] and may be considered a
gold standard measure in individuals with Down syn-
drome [60].
The only study we identified that included only chil-
dren with benign hypotonia (and excluded children
with Down syndrome or CP) did not meet inclusion
criteria as it was published in conference proceedings
only. However, the researchers identified significantly
improved gait characteristics when the children wore
SMOs as opposed to a shoes only condition, and care-
givers reported improved balance and functional skills
such as ability to jump [61]. One case study [32] sup-
porting use of compression garments with children consider inexpensive neoprene models before more ex-
with hypotonia met inclusion criteria. Therapists may pensive alternatives such as full body lycra garments.
 G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia
No strong recommendations can be made regarding
motor interventions since higher evidence level studies
were inconclusive. However early implementation of
tummy time, 90 min/day before 11 weeks of age [45]
appears to promote motor development and should be
combined with 2–4 sessions per month early interven-
tion. When a child is not sitting by 8 months (ad-
justed age), early intervention therapists should con-
sider the introduction of adapted toys, supported sitting
and standing devices as well as increasing activity for
participation in natural routines with an enriched envi-
ronment [62].
Augmentative mobility (use of gait trainers and
early manual and power mobility) is recommended for
children who are not moving and exploring their envi-
ronment between 9 and 12 months corrected age [54,
63]. Three included studies described use of a sup-
ported seating device for young children with hypoto-
nia [17,26,49], while one also described other postural
management interventions, such as stander, floor seat
and a lying positioning system [26]. Although, the evi-
dence supporting seating, standing and augmented mo-
bility with children of any diagnostic group is lower
level [54,63,64] evidence is mainly positive and these
interventions should be considered for children with
hypotonia who are not sitting, standing or walking at a
similar age to their peers.
Surveys of Physical (PT) and Occupational Ther-
apists (OT) suggest that the definition of hypotonia
should include decreased strength, decreased activity
tolerance, delayed motor skills development, rounded
shoulder posture with leaning onto supports, hyper-
mobile joints and increased flexibility [8,65]. How-
ever, for hypotonia to be classed as a diagnosis, these
characteristics need to be further defined and quanti-
fied [66]. While there are reports of PTs and OTs us-
ing range of motion (ROM), manual muscle testing and
tests of ligamentous laxity, [67] there currently exists
no valid and reliable way to measure hypotonia [68].
The ALT Scale [19] attempts to quantify hypotonia us-
ing an inverse of the Modified Ashworth Scale [69].
Govender [70] has published a clinical algorithm based
on a previous literature review [68], survey of rele-
vant clinicians (OTs, PTs, neurologists and pediatri-
cians) [71] and a Delphi consensus of experts [72].
This work requires further development and validation.
Recently the Hammersmith Infant Neurological Ex-
amination (HINE), has been suggested as a valid and
reliable tool to identify hypotonia between 3 and 24
months as well as to identify underlying biomarkers of
cerebral palsy [62].
67
4.1. Study limitations
Most notably, the original intention was to search
for evidence regarding interventions for children with
idiopathic hypotonia. However, the majority of the re-
search identified, and the highest levels of evidence
apply to children with Down syndrome, and it is ac-
knowledged that this evidence cannot necessarily be
directly applied to children with hypotonia from other
causes. Children with hypotonia from a variety of
causes make up a significant portion of referrals to
early intervention services and the lack of evidence
supporting intervention for these children is challeng-
ing for therapists. Consequently we chose to search for
evidence supporting interventions for children with all
forms of central hypotonia to assist therapists in iden-
tifying interventions that could reasonably be applied
to their clients with careful monitoring and appropriate
measurement of outcomes.
It should also be noted, that the search conducted in
this review was wide ranging as we included a broad
range of populations and interventions. We sought to
address the most common interventions conducted by
OTs and PTs with the aim of influencing motor and
sensory outcomes, but excluded a number of daily liv-
ing, play and assistive technology interventions that
may influence these outcomes. The search was system-
atic and covered several different databases, but inclu-
sion criteria were limited to studies published in En-
glish, in peer-reviewed literature. We may have missed
studies published in other languages or grey litera-
ture. We did not have access to translation services for
the two studies we found where only the abstract was
available in English.
The quality rating used in this review was only appli-
cable to evidence levels I-III, and therefore, no quality
rating is provided for 19 out of the 31 included primary
studies. The low numbers of participants involved and
low evidence level in the majority of studies included
warrants caution in interpretation of results. Although
a number of higher evidence level studies were iden-
tified in this review, a number of the publications in-
clude the same data set. We chose to rate the study as a
whole, rather than providing separate ratings for each
publication, and recommendations cannot be given the
same weight as if these were separate studies. Addi-
tionally, the traffic lighting classification system [12]
used provides a green light (proven effective) rating for
interventions supported by positive results from evi-
dence level II group studies. We chose to use this clas-
sification as it is appropriate for rehabilitation interven-
68 G. Paleg et al. / Interventions to improve sensory and motor outcomes for young children with central hypotonia
tions and allows discrimination between smaller num-
ber and lower evidence level studies. If we had cho-
sen a more stringent rating designed for discriminat-
ing between high number medical interventions such
as GRADE [73], all evidence would have been rated as
low or very low.
5. Conclusion
Green light evidence supports treadmill training (to
promote ambulation and gait characteristics) and mas-
sage (to positively affect muscle tone, motor develop-
ment and use of vision) for infants with Down syn-
drome only. Foot orthoses should be used with cau-
tion for pre-ambulatory children, but may be help-
ful in maintaining foot alignment for ambulatory chil-
dren with hypotonia from various causes. For children
with central hypotonia (other than Down syndrome),
all therapeutic interventions discussed in this review
can be considered yellow (possibly effective) and valid
and reliable outcome measures should be used, in order
to ensure appropriate use.
Lack of a clear diagnosis is often a barrier to im-
plementation of early intervention services but when
hypotonia is interfering with function and/or develop-
ment, we recommend they be found eligible for early
intervention services as ‘atypical’. Evidence-informed
clinical practice requires that the clinician be able to
identify the condition (based on agreed upon defini-
tion), measure the severity, apply an intervention and
then re-measure the severity with a valid reliable tool.
None of these conditions can be met for children with
central hypotonia. Further research is warranted into
all aspects of hypotonia including definition, measure-
ment, assessment and intervention.
Acknowledgments
Sunny Hill Health Centre for Children Clinical Li-
brary services for assistance in conducting the search
and retrieval of documents. Dr Meir Lotan for sharing
his extensive research and Drs Julia Looper and Kathy
Martin for their willingness to discuss their views on
use of orthotic interventions in regards to this review.
Conflict of interest
The first author has worked as an educational con-
sultant for various manufacturers and suppliers of gait
trainers and standers. Funding from these sources did
not influence or bias the content of this work. The sec-
ond and third authors report no conflict of interest. The
authors alone are responsible for the content and writ-
ing of the paper.
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