2015 Section on Pediatrics Knowledge Translation Lecture

Rine, Rose Marie; Dannenbaum, Elizabeth; Szabo, Joanne S

doi:10.1097/pep.0000000000000226

Cited by 11 publications

(8 citation statements)

References 20 publications

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“…Other congenital disorders with abnormal or missing semicircular canals and their incidence include Waardenburg (1/40,000; Elmaleh-Bergès et al 2013), Noonan (1/1000-1/2500; Naficy et al 1997), Wildervanck (1/100 in females with congenital hearing loss; Abu-Amero et al 2014), Goldenhar (1/3500-1/25,000; Bisdas et al 2005), and branchiooto-renal (BOR) syndrome (1/40,000;Laclef et al 2003). Despite the low incidence of any one congen-ital vestibular disorder, vestibular impairment occurs in 5.3-10 % of US children (Li et al 2016;Rine et al 2016).…”

Section: Symptoms and Prevalence Of Congenital Vestibular Disordersmentioning

confidence: 99%

A New Model for Congenital Vestibular Disorders

Lilian

Seal

Popratiloff

et al. 2018

JARO

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Many developmental disorders of the inner ear are manifested clinically as delayed motor development and challenges in maintaining posture and balance, indicating involvement of central vestibular circuits. How the vestibular circuitry is rewired in pediatric cases is poorly understood due to lack of a suitable animal model. Based on this, our lab designed and validated a chick embryo model to study vestibular development in congenital vestibular disorders. The developing inner ear or Botocyst^on the right side of 2-day-old chick embryos (E2) was surgically rotated 180°in the anterior-posterior axis, forming the Banterior-posterior axis rotated otocyst chick^or ARO chick. The ARO chick has a reproducible pathology of a sac with truncated or missing semicircular canals. A sac is the most common inner ear defect found in children with congenital vestibular disorders. In E13 ARO chicks, the sac contained all three cristae and maculae utriculi and sacculi, but the superior crista and macula utriculi were shortened in anteriorposterior extent. Also, the number of principal cells of the tangential vestibular nucleus, a major avian vestibular nucleus, was decreased 66 % on the rotated side. After hatching, no difference was detected between ARO and normal chicks in their righting reflex times. However, unlike normal chicks, ARO hatchlings had a constant, right head tilt, and after performing the righting reflex, ARO chicks stumbled and walked with a widened base. Identifying the structure and function of abnormally developed brain regions in ARO chicks may assist in improving treatments for patients with congenital vestibular disorder.

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Section: Symptoms and Prevalence Of Congenital Vestibular Disordersmentioning

confidence: 99%

A New Model for Congenital Vestibular Disorders

Lilian

Seal

Popratiloff

et al. 2018

JARO

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“…The history of her research is beautifully summarized in an article published in Pediatric Physical Therapy. 10 Dr. Rine and colleagues first determined that vestibular hypofunction, in the absence of other neurological conditions, caused abnormal development of the postural control system and a progressive delay of gross motor abilities. 11,12 In these studies, functional impairments were measured using the Peabody Developmental Motor Scales (PDMS), Sensory Organization Test (SOT), and a modification of the Motor Control Test (MCT) with electromyography.…”

Section: A History Lessonmentioning

confidence: 99%

“…The children improved their ability to use the somatosensory and visual systems for postural control, and no longer stepped or fell on the MCT (i.e., rapid toesup tip). 10,11 Braswell-Christy and Rine then developed an intervention focused on improving DVA and reading and tested the intervention in two children with hypofunction. 15 The intervention included gaze stabilization exercises (GSE) modified for children by using a laptop and PowerPoint presentations set to automatically change slides every few seconds.…”

Section: A History Lessonmentioning

confidence: 99%

Considerations for Testing and Treating Children with Central Vestibular Impairments

Christy

2018

Semin Hear

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This perspective explores common pediatric diagnoses that could present with central vestibular pathway dysfunction, leading to delays in motor development and postural control, and gaze instability. Specifically, the following diagnoses are considered: cerebral palsy, myelomeningocele, vestibular migraine, attention-deficit hyperactivity disorder, developmental coordination disorder, concussion, childhood cancer, congenital muscular torticollis, adolescent idiopathic scoliosis, and autism. Suggestions for clinical screening, vestibular function testing, and vestibular rehabilitation for children with these diagnoses are based on evidence for the efficacy of testing and interventions for children with peripheral vestibular hypofunction. More research is needed to explore peripheral and central vestibular function in children with these diagnoses. Testing and intervention methods may need to be modified to accommodate for the specific behavior and motor challenges that some children might present. Researchers should develop technology so that gaze stabilization exercises can be delivered in a fun, functional, and effective way.

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“…In addition, Waardenburg (15), Noonan (16), Wildervanck (17), Goldenhar (18), and branchio-otorenal syndromes (BOR) (20) form a sac-like inner ear during development. Epidemiological studies reveal that vestibular disorders affect about 3.3 million children in the US alone (5.3-10%) (24,25). Due to the major role that the vestibular system plays in daily living, vestibular symptoms can impose life-altering disabilities that become a major healthcare burden.…”

Section: Introductionmentioning

confidence: 99%

Understanding the Pathophysiology of Congenital Vestibular Disorders: Current Challenges and Future Directions

et al. 2021

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In congenital vestibular disorders (CVDs), children develop an abnormal inner ear before birth and face postnatal challenges to maintain posture, balance, walking, eye-hand coordination, eye tracking, or reading. Only limited information on inner ear pathology is acquired from clinical imaging of the temporal bone or studying histological slides of the temporal bone. A more comprehensive and precise assessment and determination of the underlying mechanisms necessitate analyses of the disorders at the cellular level, which can be achieved using animal models. Two main criteria for a suitable animal model are first, a pathology that mirrors the human disorder, and second, a reproducible experimental outcome leading to statistical power. With over 40 genes that affect inner ear development, the phenotypic abnormalities resulting from congenital vestibular disorders (CVDs) are highly variable. Nonetheless, there is a large subset of CVDs that form a common phenotype of a sac-like inner ear with the semicircular canals missing or dysplastic, and discrete abnormalities in the vestibular sensory organs. We have focused the review on this subset, but to advance research on CVDs we have added other CVDs not forming a sac-like inner ear. We have included examples of animal models used to study these CVDs. Presently, little is known about the central pathology resulting from CVDs at the cellular level in the central vestibular neural network, except for preliminary studies on a chick model that show significant loss of second-order, vestibular reflex projection neurons.

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2015 Section on Pediatrics Knowledge Translation Lecture

Abstract: Our participation in knowledge translation is presented, to include discussion of possible barriers, challenges, and opportunities for facilitating collaboration and joint efforts of clinical and research practice.

Cited by 11 publications

References 20 publications

A New Model for Congenital Vestibular Disorders

A New Model for Congenital Vestibular Disorders

Considerations for Testing and Treating Children with Central Vestibular Impairments

Understanding the Pathophysiology of Congenital Vestibular Disorders: Current Challenges and Future Directions

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