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      Mal de Debarquement Syndrome: A Rare Entity—A Case Report and Review of the Literature

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          Abstract

          Mal de Debarquement Syndrome (MDS) is a rare, understudied, underdiagnosed, and self-limiting condition. Etiology and incidence are unknown. It is characterized by abnormal sensation of motion/balance reported after travel by air, land, and sea; being reexposed to motion/activity relieves it. Symptoms may last from minutes to years. Workup though required reveals no findings; it is a diagnosis of exclusion. While no efficacious treatment exists, amitriptyline and benzodiazepines as well as supportive therapy have proved to be useful. We have described a 40-year-old Caucasian female who presented for the evaluation of persistent rocking and swaying sensation after a ship cruise which lasted for one week. Patient was treated with benzodiazepines after extensive workup and is now stable. A high index of suspicion is required to make a diagnosis.

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          Most cited references21

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          Readaptation of the Vestibulo-Ocular Reflex Relieves the Mal De Debarquement Syndrome

          The mal de debarquement syndrome (MdDS), a continuous feeling of swaying, rocking, and/or bobbing, generally follows travel on the sea. The associated symptoms cause considerable distress. The underlying neural mechanisms are unknown, and to date there have been no effective treatments for this condition. Results in monkeys and humans suggested that MdDS was caused by maladaptation of the vestibulo-ocular reflex (VOR) to roll of the head during rotation. We studied 24 subjects with persistent MdDS (3 males, 21 females; 19.1 ± 33 months). Physical findings included body oscillation at 0.2 Hz, oscillating vertical nystagmus when the head was rolled from side-to-side in darkness, and unilateral rotation during the Fukuda stepping test. We posited that the maladapted rocking and the physical symptoms could be diminished or extinguished by readapting the VOR. Subjects were treated by rolling the head from side-to-side while watching a rotating full-field visual stimulus. Seventeen of the 24 subjects had a complete or substantial recovery on average for approximately 1 year. Six were initially better, but the symptoms recurred. One subject did not respond to treatment. Thus, readaptation of the VOR has led to a cure or substantial improvement in 70% of the subjects with MdDS. We conclude that the adaptive processes associated with roll-while-rotating are responsible for producing MdDS, and that the symptoms can be reduced or resolved by readapting the VOR.
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            Clinical features and associated syndromes of mal de debarquement.

            To investigate the clinical features and natural history of mal de debarquement (MdD). Retrospective case review with follow-up questionnaire and telephone interviews. University Neurotology Clinic. Patients seen between 1980 and 2006 who developed a persistent sensation of rocking or swaying for at least 3 days after exposure to passive motion. Clinical features,diagnostic testing, and questionnaire responses. Of 64 patients(75% women) identified with MdD, 34 completed follow-up questionnaires and interviews in 2006. Most patients had normal neurological exams, ENGs and brain MRIs. The average age of the first MdD episode was 39+/-13 years. A total of 206 episodes were experienced by 64 patients. Of these, 104 episodes (51%) lasted>1 month; 18%, >1 year; 15%, >2 years; 12%, >4 years, and 11%, >5 years. Eighteen patients (28%) subsequently developed spontaneous episodes of MdD-like symptoms after the initial MdD episode.There was a much higher rate of migraine in patients who went onto develop spontaneous episodes(73%) than in those who did not(22%). Subsequent episodes were longer than earlier ones in most patients who had multiple episodes.Re-exposure to passive motion temporarily decreased symptoms in most patients (66%).Subjective intolerance to visual motion increased (10% to 66%)but self-motion sensitivity did not(37% to 50%) with onset of MdD. The majority of MdD episodes lasting longer than 3 days resolve in less than one year but the probability of resolution declines each year. Many patients experience multiple MdD episodes. Some patients develop spontaneous episodes after the initial motion-triggered episode with migraine being a risk factor.
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              Metabolic and Functional Connectivity Changes in Mal de Debarquement Syndrome

              Background Individuals with mal de debarquement syndrome (MdDS) experience a chronic illusion of self-motion triggered by prolonged exposure to passive motion, such as from sea or air travel. The experience is one of rocking dizziness similar to when the individual was originally on the motion trigger such as a boat or airplane. MdDS represents a prolonged version of a normal phenomenon familiar to most individuals but which persists for months or years in others. It represents a natural example of the neuroplasticity of motion adaptation. However, the localization of where that motion adaptation occurs is unknown. Our goal was to localize metabolic and functional connectivity changes associated with persistent MdDS. Methods Twenty subjects with MdDS lasting a median duration of 17.5 months were compared to 20 normal controls with 18F FDG PET and resting state fMRI. Resting state metabolism and functional connectivity were calculated using age, grey matter volume, and mood and anxiety scores as nuisance covariates. Results MdDS subjects showed increased metabolism in the left entorhinal cortex and amygdala (z>3.3). Areas of relative hypometabolism included the left superior medial gyrus, left middle frontal gyrus, right amygdala, right insula, and clusters in the left superior, middle, and inferior temporal gyri. MdDS subjects showed increased connectivity between the entorhinal cortex/amygdala cluster and posterior visual and vestibular processing areas including middle temporal gyrus, motion sensitive area MT/V5, superior parietal lobule, and primary visual cortex, while showing decreased connectivity to multiple prefrontal areas. Conclusion These data show an association between resting state metabolic activity and functional connectivity between the entorhinal cortex and amygdala in a human disorder of abnormal motion perception. We propose a model for how these biological substrates can allow a limited period of motion exposure to lead to chronic perceptions of self-motion.
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                Author and article information

                Journal
                Case Rep Otolaryngol
                Case Rep Otolaryngol
                CRIOT
                Case Reports in Otolaryngology
                Hindawi Publishing Corporation
                2090-6765
                2090-6773
                2015
                5 August 2015
                : 2015
                : 918475
                Affiliations
                1Geriatric and Palliative Medicine Division, Internal Medicine Department, University of Michigan Health System, 4260 Plymouth Road, Ann Arbor, MI 48109, USA
                2Family Medicine Department, Saint Mary Mercy Hospital, 36475 Five Mile, Livonia, MI 48154, USA
                3School of Public Health, University of Texas, 1200 Pressler Street, Houston, TX 77030, USA
                Author notes

                Academic Editor: Robert Stokroos

                Article
                10.1155/2015/918475
                4540991
                26346344
                7a6f130f-8923-44e6-84c2-769791d6d107
                Copyright © 2015 Veronica Nwagwu et al.

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 21 June 2015
                : 22 July 2015
                Categories
                Case Report

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