Table 1.
Overview of the studies included
| Author (year) [reference] | Title of paper | Genetic type of HSP | No. of subjects | Treatment | Outcome measures | Type of study |
|---|---|---|---|---|---|---|
| Physical therapy | ||||||
| Marsden et al. (2011) [23] | The effects of functional electrical stimulation on walking in hereditary and spontaneous spastic paraparesis | Not specified | 11 patients (6 with a family history) | Bilateral stimulation of the common peroneal nerve (BICP) (n = 8) or preferred pattern of stimulation (PREF) (n = 3) | Dorsiflexor torque, range of movement of dorsiflexion and degree of toe clearance while walking, 10-Meter Walking Test (10 MWT) for walking speed, Physiological Cost Index (PCI) | Uncontrolled prospective study |
| Bertolucci et al. (2015) [24] | Robotic gait training improves motor skills and quality of life in hereditary spastic paraplegia | SPG4 – SPG5 – SPG7 – SPG11 | 13 patients (SPG4 n = 5; SPG5 n = 1; SPG7 n = 6; SPG11 n = 1) | Lokomat, for three weekly sessions, for a 6-week long period | Berg Balance Scale (BBS), the Timed Up and Go Test (TUG), 6 Minutes Walking Test (6 MWT), 10 MWT, the Modified Ashworth Scale (MAS), PCI, Hospital Anxiety and Depression Scale, and the SF-36 scale | Uncontrolled prospective study |
| Zhang et al. (2014) [25] | The effect of hydrotherapy treatment on gait characteristics of hereditary spastic paraparesis patients | Not specified | 9 patients | 10-week hydrotherapy program | MAS and gait analysis in 3 domains (spatiotemporal, kinematic, and kinetic) | Uncontrolled prospective study |
| Denton et al. (2016) [26] | Superficial warming and cooling of the leg affects walking speed and neuromuscular impairments in people with spastic paraparesis | SPG3a – SPG4 – SPG10 – not specified | 22 patients (8 received a genetic diagnosis, SPG3a n = 1; SPG4 n = 6; SPG10 n = 1); 19 matched healthy controls | Single session of cooling or warming of the leg for 30 min | 10 MWT, foot tap time, slow and fast stretches, maximal isometric muscle strength at the ankle, tibial nerve conduction studies, and motor-evoked potentials | Controlled prospective study |
| Denton et al. (2018) [27] | Effects of superficial heating and insulation on walking speed in people with hereditary and spontaneous spastic paraparesis: a randomised crossover study | Not specified | 21 patients | Superficial heating of the leg combined or not with insulation | 10 MWT and foot tap time | Randomized crossover study |
| Samuel et al. (2013) [28] | Physical therapy interventions for the patients with hereditary spastic paraparesis—an exploratory case reports | Not specified | 2 patients | Structured 8-week intensive rehabilitation program or SEIRP, including stretching, strength, and functional exercises for 60–90 min per day, 6 days a week, 8 weeks | TUG, Functional Reach Test (FRT), 10 MWT, and 2-min walking test (2 MWT) | Case series |
| Interventional and surgical therapy | ||||||
| Kai et al. (2014) [29] | Long-term results of selective dorsal rhizotomy for hereditary spastic paraparesis | Not specified | 4 patients | Selective dorsal rhizotomy (SDR) under neurophysiological monitoring | Ashworth score and spasms frequency score | Case series |
| Sharma et al. (2016) [30] | Selective dorsal rhizotomy for hereditary spastic paraparesis in children | Not specified | 2 patients | SDR | MAS sum score, Gross Motor Function Measure Score Sheet (GMFM-88), and muscle strength | Retrospective study |
| Non-invasive stimulation techniques | ||||||
| Antczak et al. (2019) [39] | The effect of repetitive transcranial magnetic stimulation on motor symptoms in hereditary spastic paraplegia | SPG3a – SPG7 – not specified | 9 patients (SPG3a n = 1; SPG7 n = 1) | 5 daily sessions of 10-Hz repetitive transcranial magnetic stimulation (rTMS) over the bilateral primary motor cortex of lower limb muscles vs. sham rTMS over the same area (40 trains lasting 7.5 s per hemisphere per session) | 10 MWT, TUG, muscle strength measured by a dynamometer, MAS | Randomized cross-over controlled trial |
| Bastani et al. (2021) [40] | A randomized controlled trial of the effect of repetitive transcranial magnetic stimulation of the motor cortex on lower extremity spasticity in hereditary spastic paraplegia | Not specified | 8 patients | Active or sham rTMS over the vertex (5 daily sessions of 5-Hz rTMS, 5 trains per session, each lasting 1 min) | MAS, Fugl-Meyer Assessment (FMA-LE), 10 MWT, SF-36 | Randomized controlled trial |
| Carra et al. (2022) [41] | Controversies and clinical applications of non-invasive transspinal magnetic stimulation: a critical review and exploratory trial in hereditary spastic paraplegia | SPG4 – SPG33 | 3 patients (SPG4 n = 1; SPG33 n = 2) | Transpinal magnetic stimulation (TsMS) at the level of the second thoracic vertebra by continuous theta burst session of a hundred three-pulse bursts at 50 Hz repeated after a 90-min interval | H-reflex recovery curve to paired stimuli | Open-label pilot uncontrolled trial |
| Krause et al. (2004) [42] | Lumbar repetitive magnetic stimulation reduces spastic tone increase of the lower limbs | Not specified | 2 patients with familial spasticity among 15 spinal lesion patients; 10 matched controls | Repetitive magnetic stimulation over unilateral L3-L4 roots (20 Hz, 2000 pulses, 1 session) | MAS, pendulum test | Open-label trial |
| Ardolino et al. (2021) [43] | Spinal direct current stimulation (tsDCS) in hereditary spastic paraplegias (HSP): a sham controlled crossover study | SPG3a – SPG4 – SPG7 – SPG15 | 11 patients (SPG3a n = 1; SPG4 n = 8; SPG7 n = 1; SPG15 n = 1) | Five daily sessions of anodal transpinal direct current stimulation (tsDCS) over the thoracic spinal cord (T10-T12) vs. sham tsDCS (intensity current: 2 mA, duration of stimulation: 20 min) | Motor-evoked potentials (MEPs), the H-reflex, F-waves, Ashworth scale, the Five Minute Walking Test, and the Spastic Paraplegia Rating Scale (SPRS) | Randomized cross-over controlled trial |
10 MWT 10 Meter Walking Test, 2 MWT 2 Minutes Walking Test, 6 MWT 6 Minutes Walking Test, BBS Berg Balance Scale, BICP bilateral stimulation of the common peroneal nerve, FMA-LE Fugl-Meyer Assessment, FRT Functional Reach Test, GMFM-88 Gross Motor Function Measure Score Sheet, MAS Modified Ashworth Scale, MEPs motor-evoked potentials, PCI Physiological Cost Index, rTMS repetitive transcranial magnetic stimulation, SDR selective dorsal rhizotomy, SF-36 Short Form Health Survey 36, SPRS Spastic Paraplegia Rating Scale, tsDCS transpinal direct current stimulation, TsMS transpinal magnetic stimulation, TUG Timed Up and Go Test