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Standardized Finger-Nose Test Validity for Coordination Assessment in an

Ataxic Disorder

Article  in  The Canadian journal of neurological sciences. Le journal canadien des sciences neurologiques · November 2004
DOI: 10.1017/S031716710000367X · Source: PubMed

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 Journal of the Neurological Sciences 347 (2014) 341–344

Contents lists available at ScienceDirect

Journal of the Neurological Sciences

journal homepage: www.elsevier.com/locate/jns

Short communication

The Virtual Peg Insertion Test as an assessment of upper limb

coordination in ARSACS patients: A pilot study
Cynthia Gagnon a,c,d,⁎, Caroline Lavoie a,b,c,d, Isabelle Lessard a,b,c,d, Jean Mathieu a,b,c,d, Bernard Brais e,
Jean-Pierre Bouchard f, Marie-Christine Fluet g, Roger Gassert g, Olivier Lambercy g
a
Groupe de recherche interdisciplinaire sur les maladies neuromusculaires (GRIMN), Québec, Canada
b
Clinique des maladies neuromusculaires, Centre de réadaptation en déficience physique Le Parcours du Centre de santé et de services sociaux de Jonquière, Québec, Canada
c
Centre hospitalier affilié universitaire régional (CAUR) de Chicoutimi, Centre de santé et de services sociaux de Chicoutimi, Chicoutimi, Québec, Canada
d
School of Rehabilitation, Faculty of Medicine and Health Sciences, Université de Sherbrooke, Québec, Canada
e
Montreal Neurological Institute, McGill University, Québec, Canada
f
Department of Medicine, Faculty of Medicine, Université Laval, Québec, Canada
g
Rehabilitation Engineering Laboratory, ETH Zurich, Switzerland

a r t i c l e i n f o a b s t r a c t

Article history: Objective: This paper introduces a novel assessment tool to provide clinicians with quantitative and more objec-
Received 28 February 2014 tive measures of upper limb coordination in patients suffering from Autosomal Recessive Spastic Ataxia of
Received in revised form 18 September 2014 Charlevoix–Saguenay (ARSACS). The Virtual Peg Insertion Test (VPIT) involves manipulating an instrumented
Accepted 19 September 2014 handle in order to move nine pegs into nine holes displayed in a virtual environment. The main outcome mea-
Available online 28 September 2014
sures were the number of zero-crossings of the hand acceleration vector, as a measure of movement coordination
and the total time required to complete the insertion of the nine pegs, as a measure of overall upper limb perfor-
Keywords:
Instrument
mance.
Coordination Results: 8\9 patients with ARSACS were able to complete five repetitions with the VPIT. Patients were found to be
Spastic Ataxia of Charlevoix–Saguenay type significantly less coordinated and slower than age-matched healthy subjects (p b 0.01). Performance of ARSACS
User–computer interface patients was positively correlated with the Nine-Hole Peg Test (r = 0.85, p b 0.01) and with age (r = 0.93,
Outcome assessment (health care) p b 0.01), indicative of the degenerative nature of the disease.
Instrumented assessment tool Conclusion(s): This study presents preliminary results on the use of a robotics and virtual reality assessment tool
with ARSACS patients. Results highlight its potential to assess impaired coordination and monitor its progression
over time.
© 2014 Elsevier B.V. All rights reserved.

1. Introduction
The Autosomal Recessive Spastic Ataxia of Charlevoix–Saguenay
(ARSACS) is a genetic disorder that was first described in Québec,
Canada. ARSACS manifests as a developmental and later neurodegener-
ative disorder affecting the central and peripheral nervous systems [1].
Clinically, it presents mixed pyramidal, cerebellar and neuropathic fea-
tures of variable intensities. Disease onset is in early childhood, and
symptoms become obvious in the late teens or early twenties as the
disease progresses [1]. Early signs are slight ataxia of the four extremi-
ties and spasticity in the legs for most patients. Clinical features may
also include dysarthria, nystagmus, hypermyelination of retinal fibers
and urinary urge emergency [2]. Upper extremity coordination is
⁎ Corresponding author at: Groupe de recherche interdisciplinaire sur les maladies
neuromusculaires, 2230 de l'Hôpital, cp 1200, Jonquière, QC G7X 7X2, Canada. Tel.: +1
418 695 7700(#2756) (office).
E-mail address: cynthia.gagnon4@usherbrooke.ca (C. Gagnon).
greatly affected, with young adult patients often already performing
below normative values [3]. Patients have a progressive decline of
functional independence and social participation as they get older [4],
which in turn is strongly correlated with impaired upper limb coordina-
tion [4].
Assessment of upper limb coordination could be a key variable to
follow in upcoming therapeutic trials. Several instruments have been
used to quantify coordination, including the finger–nose test [3] and
the Nine Hole Peg Test (NHPT) [5]. However, these measures suffer
from several limitations when applied to the ARSACS population. First,
they only provide a crude estimate of coordination by reporting the
time required to perform a task. Second, the rate of disease progression
in ARSACS is currently unknown, but clinically a rather slow course is
observed, which underlines the need for more sensitive instruments
to document disease progression over short time periods. Finally, the
presence of distal neuropathy in several patients, resulting in muscle
wasting, often leads to poor fine prehension. This in turn strongly influ-
ences the results of coordination tests involving this function (e.g. the
NHPT).

http://dx.doi.org/10.1016/j.jns.2014.09.032
0022-510X/© 2014 Elsevier B.V. All rights reserved.
342 C. Gagnon et al. / Journal of the Neurological Sciences 347 (2014) 341–344

With the objective of refining the assessment of coordination, we
investigated the use of the Virtual Peg Insertion Test (VPIT) [6,7] a
robotic assessment consisting in a goal-directed functional task combin-
ing haptic feedback and virtual reality.The pilot study will explore the
use of the VPIT and assess coordination among ARSACS patients.
2. Methods
The VPIT combines a haptic device (Phantom Omni, SensAble Tech-
nologies, USA) equipped with a custom-made handle instrumented
with three force sensors, and a virtual reality environment displaying
a pegboard task. Pegs can be grasped in the virtual environment by pre-
cisely aligning a cursor representing the position of the handle of the
haptic device with one of the nine pegs (distal coordination), and grasp-
ing the handle over a force threshold of 2 N (prehension strength).
While holding the grasp and displacing the handle of the haptic device,
which requires lifting of the arm against gravity (proximal
coordination), the peg can be transported, and inserted by releasing
the grasp when properly aligned with one of the nine holes (Fig. 1(c)).
During the test, the haptic device renders interaction forces with the vir-
tual pegboard and records trajectories and grasping forces. In addition
to the time to complete the task (Tex) providing a simple measure of
upper limb function, quantitative parameters can be extracted to
evaluate specific impairments. In particular, movement smoothness is
a well-accepted indicator of coordination [8]. Ideal fast and smooth
point-to-point movements have been shown to follow a minimal jerk
(derivative of acceleration) profile [9], which corresponds to a bell-
shaped velocity profile composed of a single velocity peak. Methods to
quantify movement smoothness have been evaluated in the literature
using different metrics based on jerk or velocity traces [10]. A simple
way to evaluate movement smoothness is to compute the time-
normalized number of zero-crossings, i.e. change in sign, of the acceler-
ation profile (Nzc). This measure approximates the number of sub-
movements a movement is composed of [11], and has the advantage

Fig. 1. Position traces during a typical repetition of the Virtual Peg Insertion Test (VPIT) for (a) one healthy subject (H2, repetition 2, dominant hand) and (b) one ARSACS patient (A3,
repetition 2, dominant hand). Based on position and velocity thresholds, the task can be decomposed into gross movements (peg-to-hole and hole-to-peg) and fine position adjustments
(approach of a hole or a peg). Velocity and acceleration profiles are reported for one representative peg insertion for each subject. The upper limb coordination metric Nzc is computed from
the number of zero-crossings (Z) of the acceleration vector during the gross movement (shaded area), normalized over the movement duration (T). (c): the VPIT with one subject
performing the test. (d): mean (SE) total execution time (Tex) in function of age for ARSACS patients and healthy subjects (average over both tested hands), with linear regressions (dashed
lines). (e): mean (SE) Nzc for healthy subjects and ARSACS patients and both tested hands (p-value from two-way ANOVA). (f): Comparison of mean (SE) total execution time of the VPIT
(Tex) and mean time to complete the Nine Hole Peg Test (NHPT), average over both tested hands, with linear regression (dashed line). HD/HND: healthy subjects dominant/nondominant
hand, AD/AND: ARSACS patients dominant/nondominant hand.
 C. Gagnon et al. / Journal of the Neurological Sciences 347 (2014) 341–344
of being independent of movement amplitude, speed or movement
duration.
A sample of nine patients based on maximum variance sampling
(age and clinical disease severity, as subjectively assessed by the
treating neurologist) were recruited among the registry of the Neuro-
muscular Clinic of the Centre de Santé et de Services Sociaux (CSSS)
de Jonquière, Canada (A1–A9, 5 females, age 26 to 59 (mean ± SE:
41.6 ± 4.3)). Patients with molecular diagnosis of ARSACS were includ-
ed. Exclusion criteria was the presence of other diseases affecting upper
limb coordination. Nine age-matched healthy subjects (H1–H9, 5 fe-
males, age 23 to 59 (mean ± SE: 39.2 ± 4.0)) were enrolled in this
study to collect baseline data for performance comparison. All subjects
gave informed consent prior to enrolment. The study received ethical
approval from the CSSS Chicoutimi.
Subjects performed five repetitions of the VPIT with their dominant
hand (i.e. insertion of the nine pegs per repetition), followed by five
repetitions with the nondominant hand. Rest periods were given in be-
tween each repetition, and one initial test trial was allowed for each
hand. For data analysis, only the five repetitions were considered. Tex
was averaged over all completed repetitions, while Nzc was calculated
for each gross peg-to-hole movement, and then averaged over all com-
pleted repetitions. Other indicators of upper limb function could be
extracted from the VPIT data (see [6]), however given the small sample
size, only the robust and previously described coordination metric Nzc
was analyzed in this pilot study, as this feature is the most relevant for
ARSACS patients.
A 2-way ANOVA was performed to test for the effects of subject
groups and tested hands. For comparison with clinical scales, subjects
were also tested on the NHPT on the same day. The NHPT is commonly
used in the ARSACS population to evaluate hand function, and its valid-
ity has been documented in Frieidrich Ataxia [12]. Correlation analyses
were performed using Pearson's linear correlation coefficient. Signifi-
cance levels of all statistical tests were set at 0.05.
3. Results
All patients were able to use the VPIT and 8/9 patients completed all
five repetitions with both hands. One patient stopped after 2 repetitions
with the dominant hand, reporting shoulder fatigue and difficulties in
understanding the task. Fig. 1(a)–(b) shows position traces during
representative repetitions for one healthy subject and one ARSACS pa-
tient, with the nine peg insertions divided into gross movements
(peg-to-hole and hole-to-peg) and fine position adjustments (approach
of a hole or a peg). In patients, gross movements were typically jerky
and not straight, especially when reaching for the holes located further
away from the body, where the arm is in a more extended position
(upper row in Fig. 1(b)). This is further illustrated in velocity traces by
a large number of small amplitude velocity peaks throughout the
movement.
At the group level, no significant effect of the tested hand on task
performance was found (F1,3 = 0.88, p = 0.43), which motivated the
averaging of results from both hands for the correlation analyses. How-
ever, there was a significant effect of the subject group (F1,3 = 12.57,
p b 0.01). ARSACS patients were significantly slower in the execution
of the VPIT (F1,3 = 21.6, p b 0.01) than age-matched healthy subjects,
and a strong correlation between age and Tex was observed (r = 0.93,
Fig. 1(d)). A similar trend in performance decrease as a function of age
was also observed in healthy subjects (r = 0.57), but at a significantly
slower rate (significant interaction effect of group-age on Tex
(p b 0.01) using a post-hoc ANCOVA analysis). Younger patients
(b 30 years old) already exhibited abnormally high execution times, in
the range of 60 year old healthy adults. The proposed measure of
upper limb coordination (Nzc) was significantly different between sub-
ject groups (F1,3 = 19.99, p b 0.01, Fig. 1(e)), with patients performing
peg-to-hole movements with a larger number of sub-movements, i.e. in
343
a less coordinated way. A positive correlation was observed between Tex
and the clinically validated NHPT (r = 0.85, p b 0.01, Fig. 1(f)).
4. Discussions
This paper presents results of a pilot study using the VPIT as a way to
quantitatively evaluate coordination in ARSACS patients. The functional
task to achieve during the VPIT requires upper limb strength, gross
prehension, and coordination. This makes it a promising task to assess
upper limb impairment in ARSACS patients, as these usually have suffi-
cient strength and gross prehension capabilities. All patients were able
to perform at least one complete repetition of the test even the ones
with impaired fine prehension.
Performance in the VPIT was strongly correlated with age, which can
be a proxy for disease progression as symptoms are present already
from childhood [13]. Older patients exhibited longer execution times,
which was expected considering the progressive nature of the patholo-
gy. These observations are well in accordance with the results of previ-
ous work using a variant of the finger–nose test [3]. Execution times
correlated well with the conventional NHPT, underlining the ability of
the VPIT to provide an overall evaluation of hand function, while
offering additional insights on upper limb coordination impairment,
and permitting testing of more severe patients who lack the dexterity
to grasp and hold the pegs of the conventional NHPT. Deficits in coordi-
nation were observed in ARSACS patients in the form of significantly
less smooth movements compared to healthy subjects, as also observed
in multiple sclerosis and stroke patients [6,7].
The VPIT further offers the possibility to extract other metrics from
trajectories and grasping force profiles. In complement to the proposed
coordination metric, additional parameters could contribute to more
finely assess and better understand upper limb function impairment
in ARSACS patients and should be investigated in future studies with a
larger patient sample.
5. Conclusion
These preliminary results demonstrate the potential of using the
VPIT with ARSACS patients, highlight its potential to evaluate upper
limb impairment, in particular impaired coordination, and monitor its
progression over time. Future work will build on these results with a
larger patient sample, including younger patients, in order to validate
the test with clinical measures of upper limb function and determine
the ability of the test to detect early pathologic performance.
Declaration of conflicts of interest
MCF owns shares of ReHaptix Gmbh, Zurich, Switzerland, a startup
company commercializing the Virtual Peg Insertion Test. All other
authors declare to have no competing interest.
Acknowledgments
This work is supported by the National Center of Competence in
Research on Neural Plasticity and Repair of the Swiss National Science
Foundation, the Gebert-Rüf Foundation (GRS-044/12), a ZNZ-McGill
collaborative grant, and the CIHR-Charlevoix–Saguenay Ataxia Founda-
tion (TR2-119 189) Emerging team grant. Cynthia Gagnon holds a
FRQ-S career grant (Chercheur-Junior 1). Marie-Christine Fluet,
Roger Gassert and Olivier Lambercy are members of the Neurosci-
ence Center Zurich (ZNZ) and the Rehabilitation Initiative and Tech-
nology Platform Zurich (RITZ).
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