Visual outcomes of audio-luminous biofeedback training for a child with idiopathic nystagmus / Desfechos visuais de treinamento de biofeedback audioluminoso em criança com nistagmo idiopático

ABSTRACT Microperimetry biofeedback training is a vision rehabilitation method that involves the training of attention and oculomotor control, and the rehabilitation of poorly located and non-functional preferred retinal loci. It can significantly improve distance and near visual acuity in age-related macular degeneration. Previous studies have shown that biofeedback training using electrical nystagmography can reduce nystagmus amplitude and increase foveation time. However, these improvements have not been sustained following training sessions. We hereby report a pediatric case of idiopathic nystagmus in an 11-year old patient treated with microperimetric biofeedback to improve visual acuity and fixation stability. The training had a beneficial impact, positively affecting fixation stability as well as distance and near reading vision. Subjectively, improvement in quality of life was also reported. Conversely to previous studies, the positive effects in this case were maintained for as long as twelve months following therapy. To the best of our knowledge, this is the first case with long-term benefits to be reported in the literature.(AU)

RESUMO O treinamento de biofeedback por microperimetria é um método de reabilitação da visão que envolve treinamento de atenção, controle oculomotor e reabilitação do locus preferencial de fixação da retina. Esse treinamento pode melhorar significativamente a acuidade visual para longe e perto na degeneração macular relacionada à idade. Estudos anteriores mostraram que o treinamento de biofeedback usando a nistagmografia elétrica pode reduzir a amplitude do nistagmo e aumentar o período de foveação. Entretanto, os resultados não se mantiveram após o término das sessões. Aqui é relatado um caso de tratamento com biofeedback por microperimetria para melhorar a acuidade visual e a estabilidade de fixação em uma criança de 11 anos de idade. O treinamento teve impacto benéfico e afetou positivamente a estabilidade da fixação e a visão para longe, para perto e de leitura. Subjetivamente, foi relatada melhoria da qualidade de vida. Em contraste com estudos anteriores, os efeitos positivos foram mantidos até 12 meses após a terapia. Até onde sabemos, este é o primeiro caso na literatura que relata benefícios de longo prazo.(AU)

Management of Benign Paroxysmal Positional Vertigo in an Adult With Severe Osteogenesis Imperfecta.

The purpose of this case report was to describe the evaluation and management of atypical benign paroxysmal positional vertigo (BPPV) in an adult with severe osteogenesis imperfecta. A 29-year-old male was referred to a physical therapist with extensive experience in vestibular rehabilitation who provided horizontal canal BPPV treatment with a canalith repositioning maneuver over two treatment sessions. The individual's symptoms had reduced by 65% and his nystagmus during the roll test was reduced. Extreme care is needed to safely reposition individuals living with severe osteogenesis imperfecta, but the repositioning can reduce symptoms and improve quality of life. Laryngoscope, 130:2241-2244, 2020.

Compensación vestibular / Vestibular compensation

INTRODUCCIÓN Y OBJETIVO: La compensación vestibular es el conjunto de procesos que se ponen en marcha cuando tiene lugar una lesión a nivel vestibular sea cual sea el origen y la magnitud de la misma. a vez establecida la lesión los mecanismos de compensación del daño son variados y se establecen diferentes líneas de actuación. Para conocer cómo mejorar el estado de nuestros pacientes es importante saber cómo funciona la compensación vestibular y a qué niveles podemos actuar para acelerar el proceso de recuperación. CONCLUSIONES: Es importante conocer los mecanismos de compensación vestibular para adecuar la terapia a cada paciente y así mejorar su calidad de vida

INTRODUCTION AND OBJECTIVE: Vestibular compensation is the term used to describe the mechanisms triggered when there is damage in the vestibular system regardless of its origin. When suffering from an injure in vestibular area there are a wide range of compensatory responses that will involve different approaches. In order to improve the quality of life for our patients and to correctly work with them to accelerate the restoration process it is important to become acquainted with how vestibular compensation works. CONCLUSIONS: Vestibular compensation mechanisms are important to adapt the therapy to each patient and thus improve their quality of life

Revisión sobre la aplicación de la realidad virtual en la rehabilitación vestibular? / Review on the application of virtual reality to vestibular rehabilitation

INTRODUCCIÓN Y OBJETIVO: Las nuevas tecnologías nos conceden nuevas fórmulas para analizar y modificar la relación del sujeto con el medio. La realidad virtual sería su máxima expresión. Explicaremos los fundamentos básicos que se aplican en estas tecnologías, las cuales poseen muchos elementos en común con la organización de nuestro sentido del equilibrio. MÉTODO: Revisión narrativa. RESULTADOS: Expondremos las características técnicas y los mecanismos de compensación aplicados en los dispositivos de rehabilitación vestibular. DISCUSIÓN: Revisaremos algunos estudios que analizan las ventajas e inconvenientes de la realidad virtual frente a la terapia física convencional. CONCLUSIONES: Los sistemas de seguimiento ocular, postural o de giro cefálico aplicados a la realidad virtual, pueden emplearse en el desarrollo de dispositivos diagnósticos o de rehabilitación vestibular. Se necesitas más estudios que comparen su eficacia respecto a la terapia física tradicional

INTRODUCTION AND OBJECTIVE: New technologies grant us new formulas to analyze and modify the subject's relationship with the environment. Virtual reality would be its maximum expression. We will explain the basic foundations that are applied in these technologies, which have many elements in common with the organization of our sense of balance. METHOD: Narrative review. RESULTS: We will present the technical characteristics and compensation mechanisms applied in vestibular rehabilitation devices. DISCUSSION: We will review some studies that analyze the advantages and disadvantages of virtual reality compared to conventional physical therapy. CONCLUSIONS: The ocular, postural or head turn monitoring systems applied to virtual reality can be used in the development of diagnostic or vestibular rehabilitation devices. More studies are needed that compare their effectiveness with respect to traditional physical therapy

Acute vestibular syndrome in cerebellar stroke : A case report and review of the literature.

The current paper reports on a patient with recurrent rotational vertigo and persistent dizziness and imbalance lasting several weeks, who underwent extensive neuro-otological and radiological examinations. Pathological findings initially included right-sided benign paroxysmal positional vertigo (BPPV), persistent horizontal spontaneous nystagmus (SPN) to the left, and a pathological bedside and video head impulse test (HIT) on the left. The pathological HIT on the left and the SPN to the left indicated a central origin. Therefore, cranial magnetic resonance imaging was performed which revealed a left-sided ischemic stroke in the territory of the medial branch of the posterior inferior cerebellar artery (mPICA).

[A randomized controlled trial on short-term efficacy of the modified Semont maneuver for the treatment of posterior canal benign paroxysmal positional vertigo].

Objective:To observe the short-term efficacy of modified Semont maneuver for posterior canal benign paroxysmal positional vertigo (PC-BPPV)Method:This was a prospective randomized controlled trial on 130 PC-BPPV patients. Subjects were randomized divided into two groups: modified Semont (65 patients),and Epley(65 patients).Each maneuver was repeated twice,and the presence of sequelae,nystagmus and vertigo on positional testing were evaluated 3rd day and 1 week after treatment.Result:Five patients were lost to follow up (all five in the modified Semont group),and three patients failed to complete treatment (all three in the Epley group). The sequelae at the 3rd day and one week after modified Semont maneuver were 27 and 9,while 41 and 15 in Epley group. The efficacy rates at the 3rd day and one week after modified Semont maneuver were 91.7% and 98.3%,and 91.9% and 96.8% in Epley group retrospectively. The sequelae and short-term effective rate of patients in modified Semont group was no difference when compared with that in Epley group (P>0.05).Conclusion:Modified Semont maneuver represents a simple rapid and effective approach to the treatment of posterior canal benign paroxysmal positional vertigo.

Síndrome de nistagmo vertical hacia abajo: reporte de 2 casos y revisión de la literatura / Downbeat nystagmus syndrome: a report of 2 cases and literature review

El síndrome de nistagmo vertical hacia abajo (NVA) es una forma común de nistagmo de fijación adquirido que se presenta con nistagmo persistente con fase rápida en dirección descendente, mareo, oscilopsia y alteraciones de la marcha. Se considera un trastorno vestíbulo-cerebelar debido a un defecto en las células de Purkinje en el flóculo del cerebelo. Las causas reportadas con mayor frecuencia son los trastornos degenerativos cerebelares e isquemia cerebelar, sin embargo, en un gran porcentaje de los pacientes la etiología permanece incierta (forma idiopática). El NVA se puede dar en un contexto más amplio de neuropatía somatosensorial y ataxia cerebelar en el síndrome CANVAS. Las medidas terapéuticas incluyen evitar la posición supina y prona al descansar, rehabilitación vestibular y tratamiento farmacológico con aminopiridinas, entre otros. En este artículo presentamos dos casos de NVA así como la revisión de la literatura.

Downbeat nystagmus syndrome (DBN) is a frequent form of acquired fixation nystagmus, it presents with persisting nistagmus with fast phases directed downward, dizziness, oscillopsia and gait disturbances. It is considered a vestibulocerebellar disorder due to a bilateral defect of the Purkinje cells in the cerebellar flocculus. Most reported causes are degenerative disorders of the cerebellum and cerebellar ischemia, nevertheless the etiology remains unknown in a large percentage of patients (idiopathic form). DBN may present in a broader context of somatosensory neuropathy and cerebellar ataxia as in CANVAS syndrome. Therapeutic measures includes avoiding the supine and prone position when resting, vestibular rehabilitation, and pharmacologic treatment with aminopyridines, among others. In this article we present two cases of DBN and review of literature.

A multicenter randomized double-blind study: comparison of the Epley, Semont, and sham maneuvers for the treatment of posterior canal benign paroxysmal positional vertigo.

We evaluated the short-term efficacy of Epley, Semont, and sham maneuvers for resolving posterior canal benign paroxysmal positional vertigo (BPPV) in a prospective multicenter randomized double-blind controlled study. Subjects were randomly divided into three groups: Epley (36 patients), Semont (32 patients), and sham (Epley maneuver for the unaffected side, 31 patients). Out of 14 institutes which participated in this study, 5 institutes had previous experience of the Epley but not the Semont maneuver and the other 9 had previous experience of both maneuvers. Each maneuver was repeated twice if there was still positional vertigo or nystagmus on day 0, and the presence of nystagmus and vertigo on positional testing were evaluated immediately, 1 day, and 1 week after treatment. After the first maneuver, the Epley group showed a significantly higher resolution rate of positional nystagmus than the Semont or sham groups (63.9, 37.5, and 38.7%, respectively). After the second maneuver, the resolution rate (83.3%) of the Epley group was significantly higher than that (51.6%) of the sham group. At 1 day and 1 week after treatment, the resolution rate of the Epley group was significantly higher than those of the other groups. Similar results were seen for the resolution of positional vertigo. The Epley maneuver showed persistent resolution rates of positional vertigo and nystagmus without a fatigue phenomenon. The Epley maneuver was significantly more effective per maneuver than Semont or sham maneuvers for the short-term treatment of posterior canal BPPV. The Semont maneuver showed a higher success rate than the sham maneuver, but it was not significantly different.

Acute VOR gain differences for outward vs. inward head impulses.

BACKGROUND: Vestibular rehabilitation is a sub-specialization within the practice of physical therapy that includes treatments designed to reduce gaze instability. Gaze stability exercises are commonly given for head rotations to the left and right, even in subjects with one healthy vestibular system (as in unilateral loss). Few studies have investigated the difference in the angular vestibular ocular reflex gain (aVOR) measured in the acute phase after deafferentation for ipsilesional head rotations that move the head away from center or towards center. OBJECTIVE: The purpose of this study was to compare differences in acute aVOR gain when the head was passively rotated outward from an initially centered position (neck neutral) versus the head being rotated inward. METHODS: We recorded head and eye velocity using video head impulse test equipment in patients with unilateral vestibular pathology scheduled for tumor resection via retrosigmoid approach (n=5) or labyrinthectomy due to Meniere's disease (n=2). RESULTS: We found 1) no difference in the ipsilesional aVOR gain for inward or outward directed head impulse rotations and 2) head velocity is inversely correlated with aVOR gain for ipsilesional but not contralesional rotations. CONCLUSIONS: Bedside testing of the ipsilesional aVOR following acute vestibular ablation can be done with head impulse rotations to either side. In the acute stages, physical therapists should prescribe ipsilesional and contralesional gaze stability exercises.

Natural course of positional down-beating nystagmus of peripheral origin.

The aim of this study was to assess the natural course of positional down-beating nystagmus (pDBN) and vertigo in patients with no evidence of central nervous system involvement and of presumed peripheral origin. Fifty-three patients with pDBN had a complete otoneurological examination. All subjects, apart from three (excluded from the study), showed no additional neurological signs and normal brain imaging. Patients were randomly assigned to two groups: with or without treatment with exercise. Patients were seen again after 24 h, and then weekly for up to 6 months. Forty-seven patients (94%) showed pDBN in the straight head-hanging position and in a Dix-Hallpike position. A torsional component was detected in 17 patients (34%). The mean latency and duration of pDBN was 4.7 ± 5 s and 40.1 ± 22 s, respectively. After 2 weeks, only 12 patients (24%) still had pDBN and all but one patient had recovered by 1 month. Twenty patients (40%) were diagnosed with a typical posterior canal benign paroxysmal positional vertigo (PC BPPV) before or after pDBN. This study assessed for the first time the natural course of presumed peripheral pDBN, which was characterized by a spontaneous remission in 24 patients in the first week and in 49 patients within 4 weeks. pDBN is much more common than previously suggested, with about the same frequency as lateral canal BPPV. Furthermore, the clinical characteristics of pDBN have been highlighted, as well as its possible relationship to PC BPPV.

[Visual neurorehabilitation of patients with cerebral damage using botulinum toxin]. / ción visual con toxina botulínica en pacientes con daño cerebral.

BACKGROUND: The neurorehabilitation of the patient with cerebral damage implies the reestablishment of the visual functions. Botulinum toxin can be considerate as a less invasive alternative for treatment. OBJECTIVE: to demonstrate the answer to the treatment using botulinum toxin of the visual motor alterations in patients with cerebral damage. METHODS: Descriptive study of patients with visual alterations associated to cerebral damage. The visual treatment included three areas: sensorial, refracting and motor under quimiodenervation with botulinum toxin, of May 2009 to May 2010. RESULTS: 48 patients were studied, age 22,4 years ± 23. The strabismus were: esotropia 52%, exotropia 39,5%, vertical 8%, nystagmus 4%. 50% of the patients had psychomotor delay. Some of the most important causes of cerebral damage were: Down syndrome, epilepsy, tumor, hydrocephalus, neuroinfection, infantile cerebral paralysis, multiple sclerosis, metabolic syndrome, cranial trauma, congenital cardiopathy, ventricular hemorrhage, cerebrovascular stroke. The dose of botulinum toxin was 8,1 UI ± 3. We registered good results in 56.5%, regular 23,9% and bad 19,5%. The global percentage of rehabilitation was 69% of correction with a r of Pearson of 0,5. DISCUSSION: Patients with cerebral damage have diverse types of visuomotor alterations, strabismus and nystagmus.Use of botulinum toxin as a paralytic muscle agent is a good alternative in these cases. CONCLUSION: The botulinum toxin is an effective option for the visual rehabilitation in patients with cerebral damage and prevents the progression of more cerebral changes secondary to strabismus.

Double-blind randomized trial on short-term efficacy of the Semont maneuver for the treatment of posterior canal benign paroxysmal positional vertigo.

The need for Class I and II studies on the efficacy of Semont's liberatory maneuver (SLM) in the treatment of posterior canal benign paroxysmal positional vertigo (PC-BPPV) motivated the present double-blind randomized trial on the short-term efficacy of SLM. A total of 342 patients with unilateral PC-BPPV were recruited for a multicenter study. Patients were randomly assigned to treatment by SLM (n = 174) or sham treatment (n = 168). Subjects were followed up twice (1 and 24 h) with the Dix-Hallpike maneuver by blinded examiners. At the 1 and 24 h follow-up, 79.3 and 86.8%, respectively, of patients undergoing SLM had recovered from vertigo, compared to none of the patients undergoing the sham maneuver (p < 0.0001). Patients who manifested liberatory nystagmus at the end of SLM showed a significantly higher percentage of recovery (87.1 vs. 55.7%; p < 0.0001). To the best of our knowledge, this is the first Class I study on the efficacy of SLM. SLM proved highly effective with respect to the sham maneuver (p < 0.0001). Liberatory nystagmus was demonstrated to be a useful prognostic factor for the efficacy of treatment. The present Class I study of efficacy of SLM changes the level of recommendation of the maneuver for treating PC-BPPV from level C to level B.

A cupulolith repositioning maneuver in the treatment of horizontal canal cupulolithiasis.

OBJECTIVE: We evaluated outcomes and their significance of a new treatment method for horizontal canal cupulolithiasis that could be applied regardless of the side of the cupula where otoliths are attached. METHODS: Consecutive 78 patients who showed persistent apogeotropic horizontal canal positional vertigo (horizontal canal cupulolithiasis) were enrolled, and they were treated with the new cupulolith repositioning maneuver. RESULTS: Horizontal semicircular canal cupulolithiasis was alleviated in 97.4% of patients, after an average of 2.1 repetitions of the maneuver. Otoliths were suspected to be attached to the canal side of the cupula in 30 cases and the utricular side in 44 cases. CONCLUSION: The cupulolith repositioning maneuver is an effective method for treating horizontal canal cupulolithiasis. It may also provide an insight into the side of the cupula where otoliths are attached.

Improvement of fine motor skills in children with visual impairment: an explorative study.

In this study we analysed the potential spin-off of magnifier training on the fine-motor skills of visually impaired children. The fine-motor skills of 4- and 5-year-old visually impaired children were assessed using the manual skills test for children (6-12 years) with a visual impairment (ManuVis) and movement assessment for children (Movement ABC), before and after receiving a 12-sessions training within a 6-weeks period. The training was designed to practice the use of a stand magnifier, as part of a larger research project on low-vision aids. In this study, fifteen children trained with a magnifier; seven without. Sixteen children had nystagmus. In this group head orientation (ocular torticollis) was monitored. Results showed an age-related progress in children's fine-motor skills after the training, irrespective of magnifier condition: performance speed of the ManuVis items went from 333.4s to 273.6s on average. Accuracy in the writing tasks also increased. Finally, for the children with nystagmus, an increase of ocular torticollis was found. These results suggest a careful reconsideration of which intervention is most effective for enhancing perceptuomotor performance in visually impaired children: specific 'fine-motor' training or 'non-specific' visual-attention training with a magnifier.

A case report of HTLV-I associated myelopathy presenting with cerebellar ataxia and nystagmus.

HTLV-I associated myelopathy/tropical spastic paraparesis (HAM/TSP) is characterized by spastic paraparesis in the lower extremities, and urinary disturbance. HAM/TSP has also been less frequently associated with cerebellar syndromes and nystagmus. We report a case of HAM/TSP presenting with cerebellar ataxia and nystagmus. The patient was a 73-year-old woman who was born in southern Japan. At age 41, she developed pain and spasticity in the bilateral lower limbs and gradually progressive gait disturbance. At age 57, she was diagnosed with HAM/TSP based on spastic paraparesis in the lower limbs, urinary disturbance and positive anti HTLV-I antibody in serum and cerebrospinal fluid. In June 2008, she was referred to our university and hospitalized for rehabilitation. Twenty days later, she experienced rotatory vertigo sensation. Magnetic resonance imaging revealed pontocerebellar atrophy. The patient presented with cerebellar signs in the upper limbs, gaze-evoked nystagmus in the sitting position and right-beating horizontal nystagmus in the supine and head-hanging positions. Electronystagmography (ENG) showed horizontal saccadic overshoot dysmetria and horizontal saccadic pursuit. Nystagmus is rare among the literature on HAM/TSP. ENG is helpful to evaluate and confirm the cerebellar syndromes of HAM/TSP.

Efficacy of a new home treatment device for benign paroxysmal positional vertigo.

OBJECTIVE: To prospectively test the efficacy of a device for the home treatment of benign paroxysmal positional vertigo (BPPV). DESIGN: Multicenter prospective cohort study. SETTING: Community and tertiary care clinic offices and patient homes. PATIENTS: Forty patients with active BPPV. INTERVENTIONS: Training on and treatment with a home treatment device (The DizzyFIX) as their primary therapy technique. MAIN OUTCOME MEASURE: The Dix-Hallpike maneuver at 1 week after treatment. RESULTS: Patients using the home treatment device had no evidence of nystagmus in posttreatment Dix-Hallpike maneuvers at 1 week in 88% of cases (n = 40). This rate was comparable to standard treatment. There were no significant complications. CONCLUSION: The use of this device enables patients with an established diagnosis of posterior canal BPPV to safely conduct an effective particle repositioning maneuver and achieve success rates similar to those found with the standard Epley maneuver.

[Computerized methods for therapy and rehabilitation of patients suffering dizziness and balance disorder].

A new computerized method is proposed for correction and inhibition of unfavorable illusory (vertigo), vestibular-oculomotor (nystagmus), and vestibular-postural (balance) responses. This method allows to teach the subject to block generalization of an afferent signal to effector mechanisms of the central nervous system by developing a fixational reflex, utilizing a delayed biofeedback. Three variant ofthis method were employed, depending on the kind of stimulation programs aimed at inducing abnormal responses of a certain sensory modality (visual, vestibular or combined). The study involved 36 subjects divided into 3 equal groups one of which had been taught by a visual or vestibular technique, and the last one by a combined. Each group included approximately equal numbers of subjects with vestibulopathies of an organic (peripheral or central) or psychogenic origin. It was demonstrated that the method proposed allows, with the help of computerized stimulation programs, to provoce abnormal illusory and vestibular-optooculomotor responses in the subject, and inhibit them with the help of development of a fixational reflex. Patients were taught using three various variants of the method. A subsequent comparative analysis of results obtained has shown dependence of their effectiveness on the level of disturbance in the vestibular system: the visual variant of correction was the most effective for subjects with peripheral vestibulopathia; for subjects with central vestibulopathia, the vestibular variant was more effective; the combined variant of correction was the most effective for subjects with psychogenic vestibulopathia.

Outpatient assessment of neurovisual functions in children with Cerebral Palsy.

This study examined the feasibility of the Atkinson Battery for Child Development for Examining Functional Vision (Atkinson, Anker, Rae, et al., 2002) to evaluate neurovisual functions of children with neurodevelopmental disorders in outpatient setting. A total of 90 patients underwent a comprehensive evaluation. Among these, a group of 33 children with Cerebral Palsy (CP), mean age 6 years, with different types of CP (26% diplegic, 37% hemiplegic and 37% tetraplegic) were selected to constitute the cohort of the study. Visual sensory measures as well as higher level visual functions were considered. Overall, 73% patients had impairments at the assessment protocol, the majority of which presenting difficulties on both visuoperceptual and visuospatial tasks (79%). Subgroups of participants presented similar profiles of impairments with spared basic visuocognitive abilities and limitations in visuoperceptual and visuospatial domains. The Atkinson's battery proved to be valuable for evaluation in outpatient setting and follow-up testing. Some limitations emerged. For the definition of personalized and detailed rehabilitation programs a breakdown of the different components of vision and subsequent in-depth evaluation are needed.

Efficacy of postural restriction in treating benign paroxysmal positional vertigo.

OBJECTIVE: To investigate the efficacy of postural restriction after canalith repositioning in treating benign paroxysmal positional vertigo (BPPV). DESIGN: Prospective trial of patients with postural restriction vs those without postural restriction after treatment. PATIENTS: Patients with classic BPPV and with BPPV without nystagmus were treated using the modified Epley canalith repositioning procedure. Patients were randomly separated into 2 groups. The first group was instructed to wear a cervical collar and to maintain an upright head position for 2 days. The second group had no motion restriction. After 5 days, the patients were followed up and evaluated using the Dix-Hallpike test. RESULTS: In the first group, 56 of 62 ears healed after the first maneuver, and the remaining ears healed after the second. In the second group, 45 of 57 ears healed after the first maneuver, 6 after the second, and 5 (with subsequent postural restriction) after the third (1 ear did not improve). Five patients in the first group and 3 patients in the second group had BPPV without nystagmus; all of these patients healed after a single maneuver. The difference between the 2 groups in the number of maneuvers required for treatment was statistically significant (P<.05). The number of patients who required a third maneuver was significantly higher in the second group (P<.05). CONCLUSIONS: Postural restriction enhances the therapeutic effect of canalith repositioning in the treatment of posterior semicircular canal BPPV. The long-term efficacy of postural restriction in preventing BPPV recurrence has not been demonstrated.

Social and visual function in nystagmus.

AIM: To investigate the influence of nystagmus on visual and social function and determine if parents are able to assess visual and social function in children with nystagmus. METHOD: A postal questionnaire comprising 14 questions related to visual function (VF-14) and questions pertaining to social function were sent to all 1013 members of the Nystagmus Network-a UK based organisation for nystagmus sufferers and their families. Visual and social function scores were compared by regression analysis. RESULTS: 180 adult, 233 parent, and 124 child questionnaires were returned. Idiopathic nystagmus was the most common cause. In adults the mean VF-14 score indicated very low visual function, in the same range as patients assessed in low vision services. Children's visual function scored better than adults, between scores of patients with age related macular disease and corneal grafts. There was a strong correlation between perceived visual and social function for adults (p<0.001) and parental assessment of their children (p<0.001), but not between child self assessment of visual and social function. There was strong correlation between parental and child assessment of visual and social function (p<0.001, p<0.001) CONCLUSION: Questionnaires indicated that nystagmus is associated with very low visual function. There is a strong correlation between visual and social impairment. The authors have shown for the first time in an ophthalmic disease that parents are able to estimate the impact of nystagmus on their child both in terms of visual and social functioning, although they underestimate the impact of nystagmus on emotional aspects of wellbeing.