Dedication to Mingjia Dai, Ph.D. for Discovery of the First Successful Treatment of the Mal de Debarquement Syndrome

Cohen, Bernard

doi:10.3389/fneur.2019.01196

Cited by 6 publications

(5 citation statements)

References 19 publications

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“…Of note is a possible parallel between roll-induced yaw drift and the utility of the Fukuda stepping test in the clinical MdDS assessment, in which the presence of left or right turning/drifting is examined while the patient takes alternating steps in place with the eyes closed and the arms extended forward (10, 12, 26, 66). The stepping motion may cause subtle roll head movement or possibly stand in for such since velocity storage can be activated by multiple sensory modalities (24,(67)(68)(69).…”

Section: Discussionmentioning

confidence: 99%

“…Based on the experimental data from macaque monkeys, it was hypothesized that the velocity storage mechanism of the central vestibular circuitry would acquire an inappropriate sense of vertical (pitch) motion in association with roll motion when conditioned with RWR, and furthermore that similar maladaptation might underlie MdDS (13). Velocity storage is a working memory-like mechanism of self-motion and spatial orientation that supports brainstem ocular and postural reflexes but is also thought to contribute to the perception of self-motion in the cerebral cortex (20)(21)(22)(23)(24). Thus, it follows that dysfunction Abbreviations: MdDS, mal de débarquement syndrome; OVAR, o -vertical axis rotation; PWR, pitch while rotating; RWR, roll while rotating; VOR, vestibulo-ocular reflex.…”

Section: Introductionmentioning

confidence: 99%

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Lasting alteration of spatial orientation induced by passive motion in rabbits and its possible relevance to mal de débarquement syndrome

Maruta

2023

Front. Neurol.

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BackgroundMal de débarquement syndrome (MdDS) is a chronic disorder of spatial orientation with a persistent false sensation of self-motion, whose onset typically follows prolonged exposure to passive motion of a transport vehicle. Development of similar but transient after-sensations mimicking the exposed motion and associated postural instability, indicative of central vestibular adaptation, are common. The cause of MdDS is thought to be a subsequent failure to readapt to a stationary environment. However, vestibular plasticity pertinent to this illness has not been studied sufficiently. Because the rabbit's eye movement is sensitive to three-dimensional spatial orientation, characterizing maladaptation of the vestibulo-ocular reflex (VOR) induced in the animal may open an approach to understanding MdDS.MethodsThree rabbits underwent a series of 2-h conditioning with an unnatural repetitive motion that involved a complex combination of roll, pitch, and yaw movements in a head-based reference frame, consisting of periodic rolling in darkness in a frame of reference that rotated about an earth-vertical axis. Eye movement in three dimensions was sampled during the conditioning stimulus as well as during test stimuli before and up to several days after conditioning.ResultsDuring roll-while-rotating conditioning, the roll component of the VOR was compensatory to the oscillation about the corresponding axis, but the pitch component was not, initially prominently phase-leading the head pitch motion but subsequently becoming patently phase-delayed. Unidirectional yaw nystagmus, weak but directionally compensatory to the earth-vertical axis rotation, was seen throughout the period of conditioning. After conditioning, simple side-to-side rolling induced an abnormal yaw ocular drift in the direction that opposed the nystagmus seen during conditioning, indicating a maladaptive change in spatial orientation. The impact of conditioning appeared to be partially retained even after 1 week and could be partially reversed or cumulated depending on the rotation direction in the subsequent conditioning.ConclusionThe observed reversible long-term maladaptation of spatial orientation as well as the depth of knowledge available in relation to the vestibular cerebellar circuits in this species support the potential utility of a rabbit model in MdDS research.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Lasting alteration of spatial orientation induced by passive motion in rabbits and its possible relevance to mal de débarquement syndrome

Maruta

2023

Front. Neurol.

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“…[26]. Der zugrundeliegende Pathomechanismus ist unklar, die pathophysiologischen Erklärungen des MdDS reichen von einer Funktionsstörung des Geschwindigkeitsspeichers und des vestibulo-cerebellären Nodulus [27] über hypothetische ‚Pseudohalluzinationen' des vestibulären Gedächtnisses [28] bis zu allgemeinen Vorstellungen abnormer Synchronisationen sensomotorischer Netzwerke [24,29] oder somatoformer funktioneller Beschwerden. Während eines Schifftransports treten vor allem niederfrequente Bewegungsreize von 0,1-0,4 Hz auf [30], was auch der vorwiegenden Frequenz der empfundenen und gemessenen Körperschwankungen bei Patienten mit MdDS entspricht [31,32].…”

Section: Bewegungskrankheitunclassified

“…Entsprechend dieser verschiedenen Entstehungstheorien sind die Therapieversuche und -empfehlungen vielfältig. Sie umfassen Medikamente wie Tranquilizer oder Antidepressiva, visuelle oder vestibuläre Bewegungsreizprogramme, nicht-invasive transkranielle Magnetstimulationen sowie ‚Neueineichungen' des vestibulo-okulären Reflexes [24,25,27,37,38]. Bislang fehlen jedoch überzeugende, kontrollierte, prospektive Therapiestudien.…”

Section: Bewegungskrankheitunclassified

Seltenere, aber klinisch relevante episodische Schwindelsyndrome

Becker‐Bense

Huppert

2021

Fortschr Neurol Psychiatr

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ZusammenfassungDie Differentialdiagnostik der selteneren, episodischen Schwindelsyndrome kann im klinischen Alltag eine Herausforderung darstellen, insbesondere wenn sie keine im Intervall messbaren Befunde in der neuro-ophthalmologischen oder -otologischen Routinediagnostik hinterlassen. Ursächlich für diese episodischen Schwindelsyndrome können physiologische Reaktionen aufgrund intersensorischer Inkongruenzen oder angeborene bzw. erworbene neuroanatomische/neurophysiologische Varianten sein, die zu vestibulären Reizsyndromen führen. In dieser Übersicht fokussieren wir auf die folgenden, aus unserer Sicht wichtigen vestibulären Syndrome: Bewegungskrankheit, Mal de Debarquement Syndrom, Visuelle Höhenintoleranz, Vestibularisparoxysmie, Zervikaler Schwindel, Episodische Ataxie Typ II und Syndrome eines dritten mobilen Fensters wie das Syndrom der Dehiszenz des superioren Kanals. Die Ausprägung reicht von milden Symptomen mit geringer Belastung bis hin zu schweren Krankheitsbildern mit relevanter Alltagseinschränkung. Sie können vom Kindes- oder Jugendalter bis ins Senium auftreten, teilweise mit abweichender Symptomatik. Durch gezielte Anamnese und ggf. erweiterte vestibuläre Diagnostik in einem spezialisierten Zentrum lassen sich diese Syndrome oft klar herausarbeiten und einer erfolgreichen Therapie zuführen.

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“…A recent sequel animal-based study also supports that the effect of VOR maladaptation can be systematically cumulated or reversed by the choice of the vestibular stimulus ( 46 ). Together, VOR readaptation has come to be recognized as the first effective treatment for MdDS ( 47 ).…”

Section: Introductionmentioning

confidence: 99%

Symptom reduction in mal de débarquement syndrome with attenuation of the velocity storage contribution in the central vestibular pathways

Maruta,

Cho,

Raphan

et al. 2024

Front. Rehabil. Sci.

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BackgroundThe velocity storage mechanism of the central vestibular system is closely associated with the vestibulo-ocular reflex (VOR), but also contributes to the sense of orientation in space and the perception of self-motion. We postulate that mal de débarquement syndrome (MdDS) is a consequence of inappropriate sensory adaptation of velocity storage. The premise that a maladapted velocity storage may be corrected by spatial readaptation of the VOR has recently been translated into the development of the first effective treatment for MdDS. However, this treatment's initial impact may be reversed by subsequent re-triggering events. Presently, we hypothesized that MdDS symptoms could alternatively be reduced by attenuating the velocity storage contribution in the central vestibular pathways.MethodsForty-three patients with MdDS (aged 47 ± 14 yo; 36 women) were randomly assigned to two treatment groups and followed for 6 months. The horizontal VOR was tested with chair rotation during laboratory visits, and the strength of velocity storage was quantified with model-based parameters—the time constant (Tc) and the gain of coupling from the vestibular primary afferent signals (g0). To attenuate velocity storage, Group 1 underwent a progressively intensifying series of low-frequency earth-vertical oscillatory rotation coupled to conflicting visual stimuli. Group 2 underwent an established protocol combining head tilts and visual stimulation, designed to correct maladapted spatial orientation but not change the velocity storage strength. The symptom severity was self-rated on an 11-point scale and reported before and up to 6 months after the treatment.ResultsIn Group 1, velocity storage was modified through reduction of g0 (p < 0.001) but not Tc. The symptom rating was at least halved initially in 43% of Group 1 (p = 0.04), the majority of whom retained a similar level of improvement during the 6-month follow-up period. In Group 2, no systematic change was induced in the parameters of velocity storage strength, as expected. The symptom rating was at least halved initially in 80% of Group 2 (p < 0.001), but paralleling previous findings, symptoms often returned subsequently.ConclusionAttenuation of velocity storage shows promise as a lasting remedy for MdDS that can complement the VOR readaptation approach.

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Dedication to Mingjia Dai, Ph.D. for Discovery of the First Successful Treatment of the Mal de Debarquement Syndrome

Cited by 6 publications

References 19 publications

Lasting alteration of spatial orientation induced by passive motion in rabbits and its possible relevance to mal de débarquement syndrome

Lasting alteration of spatial orientation induced by passive motion in rabbits and its possible relevance to mal de débarquement syndrome

Seltenere, aber klinisch relevante episodische Schwindelsyndrome

Symptom reduction in mal de débarquement syndrome with attenuation of the velocity storage contribution in the central vestibular pathways

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