Treatment of sleep-disordered breathing with positional therapy: long-term results.

PURPOSE: The aim of the present study was to assess the efficacy of a sleep position trainer (SPT) in patients with an established diagnosis of positional obstructive sleep apnea and to evaluate the adherence after 1-year follow-up. METHODS: Polysomnography (PSG) was performed at baseline and after 1 year of SPT use. Patients received questionnaires to assess treatment satisfaction and subjective adherence. Data on objective adherence and number of vibrations initiated by the SPT were collected from the SPT device. RESULTS: Nine out of 58 patients stopped using the SPT during the first year of treatment (16%). Thirty-four middle-aged and overweight patients underwent a PSG after 1 year of SPT use (male/female ratio, 28/6; overall apnea/hypopnea index (AHI), 16/h). A significant reduction in overall AHI to 6/h was observed using treatment (p < 0.001). The median percentage of supine sleep decreased significantly to 1% with SPT (p < 0.001). The mean objective SPT use in 28 patients was 7.3 ± 0.9 h/night and 69 ± 26% of the nights. Furthermore, 75% of the patients reported a better sleep quality since the start of SPT treatment. CONCLUSIONS: Long-term treatment with the SPT was found to be effective in reducing overall AHI. Time spent sleeping in supine position was reduced to almost zero in the continuing users. Patient satisfaction was high when using the SPT.

Mal de Debarquement Syndrome: a survey on subtypes, misdiagnoses, onset and associated psychological features.

INTRODUCTION: Mal de Debarquement Syndrome (MdDS) is a neurological condition typically characterized by a sensation of motion, that persists longer than a month following exposure to passive motion (e.g., cruise, flight, etc.). The most common form of MdDS is motion triggered (MT). However, recently it has been acknowledged that some patients develop typical MdDS symptoms without an apparent motion trigger. These cases are identified here as spontaneous or other onset (SO) MdDS. This study aimed to address similarities and differences between the MdDS subtypes. Diagnostic procedures were compared and extensive diagnostic guidelines were proposed. Second, potential triggers and associated psychological components of MdDS were revealed. METHODS: This was a retrospective online survey study for MT and SO MdDS patients. Participants were required to respond to a set of comprehensive questions regarding epidemiological details, as well as the diagnostic procedures and onset triggers. RESULTS: There were 370 patients who participated in the surveys. It is indicated that MdDS is often misdiagnosed; more so for the SO group. In addition to the apparent self-motion, both groups reported associated levels of stress, anxiety and depression. DISCUSSION: It appears at present that both MdDS subtypes are still poorly recognised. This was the first attempt to evaluate the diagnostic differences between MdDS subtypes and to propose a set of comprehensive diagnostic guidelines for both MdDS subtypes. In addition, the current research addressed that associated symptoms such as stress, anxiety and depression should also be considered when treating patients. We hope this study will help the medical community to broaden their awareness and diagnostic knowledge of this condition.

Prevalence of residual excessive sleepiness during effective oral appliance therapy for sleep-disordered breathing.

BACKGROUND: Oral appliance therapy with a mandibular advancement device (OAm) can yield to complete therapeutic response (apnea-hypopnea index [AHI]<5 events/h), though some patients show little or no improvement in daytime sleepiness. The prevalence of residual excessive sleepiness (RES) despite effective treatment with OAm therapy is unknown. We aimed to determine the prevalence of RES in patients treated with a titratable custom-made duobloc OAm. METHODS: A prevalence study was performed, collecting data from 185 patients with an established diagnosis of sleep-disordered breathing (SDB) under OAm therapy with a titratable custom-made duobloc device (baseline data were male:female ratio, 129:56; age, 48±9 years; body mass index [BMI], 27±4 kg/m2; Epworth Sleepiness Scale [ESS] score, 10±5; and AHI, 19±12 events/h). A full-night polysomnography was performed at baseline and after 3 months of OAm therapy. Daytime sleepiness was assessed using the ESS with RES defined as an ESS score of 11 or higher out of 24, despite complete therapeutic response. RESULTS: Out of 185 patients, 84 patients (45%) showed a complete therapeutic response with an AHI of <5 events per hour after 3 months of OAm therapy. Despite this normalization of AHI, 27 out of these 84 patients (32%) showed RES and had a significantly higher baseline ESS (15±4 vs. 9±4; P<.001) and were younger (43±9 vs. 47±9; P=.028) compared to patients without RES. CONCLUSION: RES under OAm therapy showed a prevalence of up to 32% in SDB patients effectively treated with respect to AHI. Patients with RES were younger and had higher baseline daytime sleepiness.

Evaluation of the impact of a clinical pathway on the organization of a multidisciplinary dental sleep clinic.

PURPOSE: Clinical pathways are used to organize complex care processes by providing structure and standardization. The multidisciplinary approach of oral appliance (OA) therapy for sleep-disordered breathing (SDB) is a complex and dynamic process suitable for such a structured pathway approach. METHODS: A clinical pathway for patients referred for OA therapy was developed and implemented. The aim of this study was to evaluate the impact of this clinical pathway on the time to delivery of the OA and the organization of the multidisciplinary dental sleep clinic (MDSC). The latter was achieved using the care process self-evaluation tool (CPSET). RESULTS: First, development and implementation of the clinical pathway gave structure and shortened the mean time to delivery by 102 days (240 ± 70 vs. 138 ± 33 days) (Mann-Whitney U: P < 0.001). Second, the CPSET scores were obtained in a cohort of 49 healthcare professionals involved in the pathway. Overall, patient-focused organization received the highest scores (80.5 ± 9.0%), whereas cooperation with primary care received the lowest score (66.7 ± 12.4%). CONCLUSIONS: This is the first project on clinical pathways in OA therapy for SDB. The implementation of the pathway in our MDSC has created a significant shortening of the time to delivery. A first evaluation of the clinical pathway using the CPSET scores indicates that all disciplines involved should be thoroughly informed in an ongoing approach.

Evaluation of the effects of anti-motion sickness drugs on subjective sleepiness and cognitive performance of healthy males.

This study aimed to investigate the clinical and cognitive side effects of baclofen (10 mg), meclizine (25 mg), dimenhydrinate (40 mg) plus cinnarizine (25 mg) and promethazine (25 mg) plus d-amphetamine (10 mg). The study had a double-blind, placebo controlled, repeated measures design and was conducted on healthy male volunteers. The psychomotor vigilance test, the Sternberg working memory task, the implicit memory test and the automated Operation Span (Ospan) task were performed. The Stanford, the Karolinska and the Epworth Sleepiness scale determined the degree of sleepiness. The Profile of Mood States (POMS) evaluated mood states and adverse effects were reported on a 22-item questionnaire. Letter recalls and time for solving mathematical problems, recorded during the Ospan task, were impaired by baclofen and dimenhydrinate-cinnarizine respectively, suggesting an influence of these drugs on the working memory. Significant side effects for baclofen were: sleepiness, tiredness, blurred vision, concentration problems and dizziness whereas for dimenhydrinate-cinnarizine only sleepiness and blurred vision were reported. Meclizine decreased the accuracy on the Sternberg working memory task and thus seemed to affect short-term memory. A reported side effect was increased sleepiness. Promethazine plus d-amphetamine did not affect any of the tested cognitive functions. However, many side effects such as sleepiness, dry mouth, dizziness, vertigo, confusion, insomnia and tremors were reported. The results show that meclizine and dimenhydrinate combined with cinnarizine were the two drugs with the most acceptable combination of side effects.

Treatment of obstructive sleep apnea using a custom-made titratable duobloc oral appliance: a prospective clinical study.

PURPOSE: This prospective clinical study investigates the efficacy of a specific custom-made titratable mandibular advancement device (MAD) for the treatment of obstructive sleep apnea (OSA). This MAD has attachments in the frontal teeth area that allow for progressive titration of the mandible. METHODS: Sixty-one adult OSA patients were included (age, 46.7 ± 9.0 years; male/female ratio, 45/16; apnea-hypopnea index (AHI), 23.2 ± 15.4 events/h sleep; body mass index, 27.9 ± 4.1 kg/m²). After an adaptation period, titration started based on a protocol of symptomatic benefit or upon reaching the physiological limits of protrusion. As a primary outcome, treatment response was defined as an objective reduction in AHI following MAD treatment of ≥50 % compared to baseline, and treatment success as a reduction in AHI with MAD to less than 5 and 10 events/h sleep. Compliance failure was defined as an inability to continue treatment. RESULTS: A statistically significant decrease was observed in AHI, from 23.4 ± 15.7 at baseline to 8.9 ± 8.6 events/h with MAD (p < 0.01). Treatment response was achieved in 42 out of 61 patients (68.8 %), whereas 42.6 % met criteria of AHI < 5 and 63.9 % achieved an AHI < 10 events/h sleep, respectively. Four patients (6.6 %) were considered as "compliance failures." CONCLUSIONS: The present study has evaluated the efficacy of a specific custom-made titratable MAD in terms of sleep apnea reduction.

Impact of type D personality on adherence to oral appliance therapy for sleep-disordered breathing.

PURPOSE: Type D personality, defined as a combination of social inhibition and negative affectivity, has been associated with poor medication adherence and lower adherence to continuous positive airway pressure in patients with sleep-disordered breathing. Up to this date, the association of patient's personality with adherence with a mandibular advancement device (MAD) has not been studied. The purposes of this study were to examine the association between type D personality and poor adherence to MAD treatment and to examine the impact of type D personality on perceived side effects during this treatment. METHODS: Eighty-two patients out of 113 patients with a known baseline type D scale who have started MAD treatment between June 2006 and December 2009 were included. Information about side effects and adherence were collected via a postal questionnaire. Thirty-three patients were using a monobloc MAD and 49 patients were using a duobloc MAD. RESULTS: Forty-five percent of type D patients discontinued MAD treatment, whereas only 15 % of non-type D patients reported treatment discontinuation. The odds ratio for treatment discontinuation was 6.03 (95 % confidence interval 1.22-29.81; p = 0.027) for type D personality, adjusted for age, gender, MAD type (monobloc or duobloc), and decrease in apnea severity. In continuing MAD users, no significant difference in perceived side effects was reported between the personality types. CONCLUSION: This is the first study to examine the relationship between type D personality and adherence to MAD treatment. Type D patients reported a significantly higher discontinuation rate when compared to patients without type D personality.

Atypical adenolymphoma and glomus caroticum tumour: a rare coincidence.

We describe the rare simultaneous appearance of an atypical adenolymphoma with a glomus caroticum tumour on the same side of the neck in a middle-aged man. This case report is the first to describe this coexistence. Due to the atypical, cyst-like presentation of the Warthin's tumour, a final diagnosis was made only after surgical resection and histopathological examination. Both the adenolymphoma and glomus caroticum tumour were successfully removed surgically.

Cochlear implantation in posttraumatic bilateral temporal bone fracture.

Temporal bone (TB) fractures can cause loss of audiovestibular function. Four cases of profound hearing impairment following bilateral TB fracture are presented in this report. All patients received a cochlear implant. All 4 patients became regular users of their implants. None of the patients experienced facial nerve stimulation. Implant-aided audiometry demonstrated a hearing threshold of 28 dB HL. The performance in speech understanding was comparable to standard postlingual adult patients implanted. We believe that cochlear implantation in patients suffering from profound sensorineural hearing losses secondary to TB fractures can be an effective tool for rehabilitation.

Reliability of utricular function testing sinusoidal translation profile during unilateral centrifugation.

The unilateral centrifugation test is one of the few vestibular tests that evaluate the utricles side by side. During this test, a subject is rotated about an earth vertical axis at high rotation speeds (e.g. 400 degrees/s) and translated sideways along the interaural axis to align the axis of rotation consecutively with the right and the left utricle. The combined rotation and translation induces ocular counter rolling (OCR), which is measured using three-dimensional video-oculography. Recently, a new model has been proposed to analyse the OCR. The model is based on contributions from both the semicircular canals and the utricles. Concomitant with the new model a new stimulation profile using a sinusoidal translation profile during the unilateral centrifugation has been introduced [1]. The current study presents the test-retest reliability as well as the robustness of the new stimulation method, based on data of 67 healthy subjects. Test-retest reliability was based on repeated measurements of a group of subjects. To test the robustness of the new sinusoidal translation paradigm, we investigated the effect of a different amplitude of the sinusoidal translation (6 cm instead of 4 cm) and of an offset in translation (from -3 to +5 cm, instead of from -4 to +4 cm) on the parameters. Several statistical measures were used to reflect the reliability: intraclass correlation coefficient (ICC), the "coefficient of variation of the method error" and the "minimal difference" (MD). All relevant variables from the physiological model for the OCR induced by unilateral centrifugation show a good to excellent reliability during the test-retest study and the relevant parameters remain unaffected by the changes applied to the translation profile (p > 0.05) as predicted by the model. Additionally, all observed differences are smaller than the MD values calculated in the test-retest part of the study.

A new model for utricular function testing using a sinusoidal translation profile during unilateral centrifugation.

The utricle plays an important role in orientation with respect to gravity. The unilateral centrifugation test allows a side-by-side investigation of both utricles. During this test, the subject is rotated about an earth-vertical axis at high rotation speeds (e.g. 400°/s) and translated along an interaural axis to consecutively align the axis of rotation with the left and the right utricle. A simple sinusoidal translation profile (0.013 Hz; amplitude = 4 cm) was chosen. The combined rotation and translation induces ocular counter rolling (OCR), which is measured using 3-D video-oculography. This OCR is the sum of the reflexes generated by both the semicircular canals and the utricles. In this paper, we present a new physiological model that decomposes this total OCR into a canal and a utricular contribution, modelled by a second-order transfer function and a combination of 2 sine functions, respectively. This model yields parameters such as canal gain, cupular and adaptation time constants and a velocity storage component for the canals. Utricular gain, bias, phase and the asymmetry between the left and the right utricle are characteristic parameters generated by the model for the utricles. The model is presented along with the results of 10 healthy subjects and 2 patients with a unilateral vestibular loss due to acoustic neuroma surgery to illustrate the effectiveness of the model.

Transcranial magnetic stimulation (TMS) in tinnitus patients.

The objective of this paper is to present Transcranial Magnetic Stimulation (TMS), a new and highly promising technique in tinnitus modulation. We conducted a Pubmed and Web of Science search using the words 'Tinnitus', 'TMS' and 'Transcranial Magnetic Stimulation'. We report on the most relevant studies relating to the effects and stimulation parameters of TMS in tinnitus patients. It has already proved possible to reduce tinnitus using TMS and rTMS in selected patient populations with specific stimulation parameters. Intrinsic and extrinsic factors were shown to determine the amount of tinnitus reduction. Though many studies point out that tinnitus reduction can be obtained using TMS, a lot of questions still remain concerning stimulation parameters and optimal patient selection.

Tenotomy of the tensor tympani and stapedius tendons in Ménière's disease.

UNLABELLED: Tenotomy of the tensor tympani and stapedius tendons in Ménière's disease. OBJECTIVE: In Ménière's disease (MD), when patients have incapacitating vertigo that is resistant to drug treatment, an intratympanic gentamicin application (ITG) is often proposed. Recently, some authors suggested that tenotomy, sectioning of the tensor tympani and stapedius tendons, could be a promising treatment. We examined whether tenotomy (ST) has additional benefit, compared to ITG alone, with respect to tinnitus, vertigo, and quality of life. METHODOLOGY: We conducted a retrospective survey of the charts of 24 patients with MD who underwent ITG, or ITG plus ST. Baseline data and follow-up assessments were obtained, using the Ménière's Disease Outcomes Questionnaire (MDOQ), the Dizziness Handicap Inventory (DHI), vertigo frequency per month, tinnitus visual analogue scale, and functional level. Failure was determined by the need for an additional procedure. RESULTS: ITG was performed on 15 patients, and 9 patients underwent ITG plus ST. The procedure was sufficient in 53% of the ITG group and in 22% of the ITG plus ST group. No significant difference was found between the two groups concerning MDOQ scores, DHI, functional level, vertigo frequency, and tinnitus. In the ITG group, we found a significant improvement in number of vertigo attacks and the tinnitus visual analogue scale. In the ITG plus ST group, there was a significant reduction in vertigo attacks, but not in tinnitus. CONCLUSION: This preliminary study suggests no additional benefit of stapedius and tensor tympani tenotomy in the treatment of Ménière's disease patients.

Stridor due to a bridge-like subglottic stenosis in a 10-week-old male infant.

We present an infant with post-intubation stridor caused by a bridge-like subglottic stenosis. At the age of 6 weeks he suffered from a RSV infection with the need for endotracheal intubation. At week 10 acute respiratory distress required a re-intubation. Flexible endoscopy was suggestive for laryngomalacia. Rigid endoscopy revealed a subglottic laterolateral mucosal bridge resulting in a doubling of the airway lumen. Histopathological examination showed a fibrinoid pseudomembrane. Follow up endoscopy showed a grade 1 posterior subglottic stenosis without respiratory compromise. This is the first case in the literature of an infant with a post-intubation bridge-like fibrinoid pseudomembranous subglottic lesion.

Vestibular evoked myogenic potentials: test-retest reliability and normative values obtained with a feedback method for the sternocleidomastoid muscle contraction.

OBJECTIVE: To determine normal limits and to analyse the test-retest reliability of the vestibular evoked myogenic potentials (VEMPs) parameters. METHODS: The VEMP procedure was repeated on different test days to analyze test-retest differences. We calculated several reliability parameters: intraclass reliability coefficient (ICC), method error (ME), coefficient of variation of the method error (CV(ME)), standard error of measurement (SEM) and minimal difference (MD) for test-retest measurements. Normal values for left-right differences, based on the interaural ratio (IAR), were determined. RESULTS: For each VEMP parameter, the ICC values indicated excellent reliability, except for p13 and corrected amplitude (fair to good reliability). The CV(ME)) values were less than 7% for p13, n23, threshold, MRV(females) and MRV(males). For the parameters corrected amplitude and raw amplitude, the CV(ME)) values exceeded 15%. The 95% IAR prediction intervals (PIs) were also largest for the parameters raw amplitude and corrected amplitude. CONCLUSION: In order to evaluate a VEMP outcome in a patient, the VEMP parameters and IAR values can be compared with the 95% PI of the normal values. When successive measurements are performed within the same subjects, the minimal difference (MD) serves as a tool to decide whether these differences are clinically relevant or not.

Case report: "auditory neuropathy" in a newborn caused by a cerebellopontine angle arachnoid cyst.

We present a 6-week-old girl, referred because of failed newborn hearing screening in the right ear. Click-evoked oto-acoustic emissions were present in both ears, auditory brainstem responses (ABR) were present in the left but totally absent in the right ear. A magnetic resonance imaging (MRI) study revealed a large arachnoid cyst in the right cerebellopontine angle (CPA) and a diagnosis of "auditory neuropathy/auditory dyssynchrony" was established. A microsurgical resection of the cyst wall and fenestration was performed by a retro sigmoid approach. This is the first case in the literature of auditory neuropathy (AN) in an infant caused by a cerebellopontine angle arachnoid cyst.

Functional imaging using computational fluid dynamics to predict treatment success of mandibular advancement devices in sleep-disordered breathing.

Mandibular advancement devices (MADs) have emerged as a popular alternative for the treatment of sleep-disordered breathing. These devices bring the mandibula forward in order to increase upper airway (UA) volume and prevent total UA collapse during sleep. However, the precise mechanism of action appears to be quite complex and is not yet completely understood; this might explain interindividual variation in treatment success. We examined whether an UA model, that combines imaging techniques and computational fluid dynamics (CFD), allows for a prediction of the treatment outcome with MADs. Ten patients that were treated with a custom-made mandibular advancement device (MAD), underwent split-night polysomnography. The morning after the sleep study, a low radiation dose CT scan was scheduled with and without the MAD. The CT examinations allowed for a comparison between the change in UA volume and the anatomical characteristics through the conversion to three-dimensional computer models. Furthermore, the change in UA resistance could be calculated through flow simulations with CFD. Boundary conditions for the model such as mass flow rate and pressure distributions were obtained during the split-night polysomnography. Therefore, the flow modeling was based on a patient specific geometry and patient specific boundary conditions. The results indicated that a decrease in UA resistance and an increase in UA volume correlate with both a clinical and an objective improvement. The results of this pilot study suggest that the outcome of MAD treatment can be predicted using the described UA model.

Internal consistency and factor analysis of the Dutch version of the Dizziness Handicap Inventory.

CONCLUSIONS: The Dutch (Belgium) translation of the Dizziness Handicap Inventory (DHI) has proven to be as consistent as the original version. In addition to the three original subscales, factor analysis revealed a fourth component scoring self-perceived effects of insufficient functioning of the vestibulo-ocular reflex (VOR). Focus should be on the DHI total score in order to compare future results with the existing literature. OBJECTIVE: To conduct a factor analysis and to determine its internal consistency. MATERIALS AND METHODS: Charts of 214 outpatients, referred with dizziness or imbalance of vestibular and non-vestibular origin, were reviewed. RESULTS: The Cronbach's alpha coefficients for internal consistency were high for the total scale and good for the subscales. Corrected item-total correlations ranged from 0.71 for 'restricted travelling' to 0.29 for 'difficulties reading', when items were correlated with their respective subtotals, and ranged from 0.69 (restricted participation in social activities) to 0.33 (stressed relationships), when correlated with the total score. A principal component analysis with orthogonal rotation was conducted, suggesting a four-factor solution. Two factors were related to vestibular handicap, referring to the original functional and emotional subcategories. The remaining two factors related to vestibular disability, documenting motion sensitivity (original physical subscale) and insufficient VOR functioning.

Organisation of a universal newborn hearing screening programme in Flanders.

OBJECTIVE: Since 1998 an integrated universal newborn hearing screening programme (UNHSP) based on automated auditory brainstem response (AABR) has been implemented in Flanders. The protocol of the UNHSP is based on guidelines defined by the American Academy of Paediatrics (AAP). The aim of this paper is to report on the screening protocol and to assess its feasibility. METHODOLOGY: Descriptive study based upon an analysis of the screening results in the neonatal non-NICU population of Flanders between 1999 and 2004. The UNHSP, organized by Kind en Gezin (K&G), uses a 2-stage protocol: children with a refer at the first screening test are retested, and those with a refer at the retest are referred to a certified centre. Screening and referral centres communicate their data to a central database at K&G. RESULTS: From the beginning of 1999 until the end of 2004 a screening was offered to 97.91% of all eligible babies in Flanders; 91.5% of these babies were screened by K&G using the Algo Portable Newborn Screener. Three-quarters of the referred babies had a confirmed hearing loss. In 57.6% of these babies, hearing loss was bilateral. Some babies had a temporary hearing problem. The false positive rate after two tests was 0.53 per thousand. All ascertained babies started early intervention, most of them before the age of 4 months. CONCLUSIONS: K&G has succeeded in organizing a new, well-structured community-based UNHSP according to the guidelines of the AAP on Neonatal Hearing Screening.

The use of flupirtine in treatment of tinnitus.

CONCLUSION: Flupirtine, a functional NMDA antagonist, does not seem to be efficacious in the treatment of tinnitus. OBJECTIVES: The purpose of this study was to investigate whether flupirtine has any beneficial effect on tinnitus perception. PATIENTS AND METHODS: Twenty-four patients were selected (6 female and 18 male patients) with continuous subjective tinnitus. Eight patients suffered left-sided tinnitus, 4 right-sided tinnitus and 12 bilateral tinnitus. We assessed the burden of the tinnitus by loudness visual analogue scale (VAS) and tinnitus questionnaire (TQ) according to Hallam et al., and Hiller and Goebel. All patients were treated with a 2 x 100 mg daily dosage of oral flupirtine for 3 weeks in an open prospective design. RESULTS: There was no statistical effect on VAS and TQ of the treatment with flupirtine. Only one patient (4.2%) experienced a positive effect on the tinnitus but discontinued the treatment because of amnesia and concentration disorders.