Abstract
OBJECTIVE:
We studied the clinical features of benign paroxysmal positional vertigo (BPPV) associated with Meniere’s disease.
METHODS:
The medical records of 120 patients with BPPV was retrospectively analyzed. Complete otolaryngological, audiological, and neurotological evaluation results were available for all patients, including nystagmography. All patients were diagnosed using the Dix-Hallpike test or roll test and treated with the canalith repositioning procedure. The outcomes were compared among the three groups.
RESULTS:
A series of 120 BPPV cases. Results showed that Group A and Group B based on the following features: unilateral semicircular canal BPPV occurred more often than bilateral BPPV and the posterior semicircular canal was the most common canal involved. Additionally, Meniere’s disease patients with multiple semicircular canal BPPV required repeated canalith repositioning procedures and had a higher recurrence rate.
CONCLUSION:
A lower treatment success rate and a higher recurrence rate were found in the BPPV patients with Meniere’s disease compared with the patients without Meniere’s disease. The recurrence rate was highest in the patients with multiple semicircular canal BPPV with Meniere’s disease.
Keywords
Introduction
Benign paroxysmal positional vertigo (BPPV) is the most common form of inner ear vertigo. BPPV occurs after head trauma and viral neurolabyrinthitis and may occur following surgery and prolonged bed rest [17]. BPPV can also be idiopathic or secondary to chronic otitis media, vestibular neuritis, Meniere’s disease, vertebro-basilar ischemia, or age. The patient experiences a severe rotatory vertigo lasting a few seconds that is brought on by a change in head position, such as lying back quickly, turning in bed, reaching for the top shelf, or bending. The Dix-Hallpike positional maneuver is diagnostic. In this maneuver, the patient experiences the usual vertigo along with geotropic torsional nystagmus when brought rapidly from a sitting position to a lying position with the head hanging and turned with the affected ear down. The Dix-Hallpike maneuver was considered positive for posterior (or anterior) semicircular canal (SCC) BPPV when vertigo was provoked, accompanied by a burst of torsional-vertical two-component nystagmus with the typical characteristics of latency, crescendo, and transience. The supine roll test was considered positive for horizontal SCC BPPV when intense vertigo was provoked, accompanied by horizontal geotropic (canalolithiasis) or apogeotropic (cupulolithiasis or canalolithiasis of the short arm of the horizontal SCC) paroxysmal nystagmus. The pathophysiology of BPPV is the dislodgement of otoconia from the utricle; otoconia are microscopic crystals of calcium carbonate that drift into the ampulla of the posterior SCC, where they stimulate the end-organ and trigger an attack of vertigo. BPPV can also less commonly affect the lateral SCC or anterior SCC; in this case, the vertigo and nystagmus begin similarly, although the direction of the nystagmus is different and is oriented in the plane of its respective SCC.
Presumably, any inner ear disease that detaches otoconia but does not totally destroy SCC functions can induce secondary BPPV. Reports that idiopathic and secondary cases of BPPV differ in several respects suggest that the pathology or pathophysiology of secondary BPPV may differ quantitatively or qualitatively from idiopathic BPPV. However, few studies have focused on secondary BPPV, which may be an underdiagnosed entity.
Meniere’s disease is another common vestibular entity characterized by recurring postural vertigo that persists for a time ranging from minutes to hours and is accompanied by hearing impairment, tinnitus, and a feeling of pressure in the affected ear. Occasionally, the vertigo is preceded by amplified ear noises, increased ear pressure, or reduced hearing acuity [15, 16]. Benign paroxysmal positional vertigo may be associated with Meniere’s disease and may occur at any stage of this disease. However, Meniere’s disease may be considered one cause of persistent vertigo in patients with BPPV, which poses difficulties in obtaining the correct diagnosis and aggravates the ability to predict the prognosis [17].
The aim of this study was to investigate a group of patients who presented with BPPV in conjunction with Meniere’s disease and who were diagnosed and treated in the otolaryngology department during the past 5 years. The clinical and nystagmographic features and the treatment outcomes of this group were studied and compared with a group with idiopathic BPPV.
Methods
Data were collected from January 2013 to January 2018 by participating investigators at baseline and the 6-month follow-up. All patients included in this study had a history of rotatory vertigo that lasted seconds. All patients had rotatory vertigo with direction-changing reversible torsional or geotropic horizontal nystagmus triggered by the Dix-Hallpike or supine roll test, respectively. Repositioning maneuvers were performed if the results of the tests were positive. One hundred and twenty patients examined at the ENT department received a BPPV diagnosis. Among them, 21 patients had a previous diagnosis of Meniere’s disease. The clinical records of these patients were retrospectively reviewed, including 8 with Meniere’s disease with multiple SCC BPPV (group A) and 13 with single semicircular canal BPPV (group B).
The patient’s age at the initial BPPV diagnosis, gender, and duration of symptoms were recorded. We used the following scale to evaluate the severity of the vertiginous symptoms [5]: 1, slight vertigo in the provoking position without autonomic symptoms; 2, severe vertigo with nausea; and 3, severe vertigo with severe nausea, vomiting, or hypotension. Patients with any clinical, laboratory, or imaging findings suggesting a disorder of the central nervous system were excluded. Patients with idiopathic BPPV who were examined and treated during the same period were used as a control group. The protocol of the study was reviewed and approved by the local Institutional Review Board.
For all patients, complete otolaryngological, audiological, and neurotological evaluations were performed, including pure tone audiometry, measurements of acoustic immittance, and occasionally auditory brain stem response testing. Eye movements were recorded by electronystagmography or videonystagmography using a standard test protocol of visual and vestibular stimulation, which was described elsewhere [12]. Patients with idiopathic BPPV who were examined and treated during the same period were used as a control group (n = 99, BPPV without Meniere’s disease).The nystagmographic data from patients in groups A and B were compared with data from 99 patients with idiopathic BPPV who underwent testing under similar conditions.
The diagnosis of Meniere’s disease was based on the guidelines of the American Academy of Otolaryngology–Head and Neck Surgery (AAO-HNS) [1]. The majority of these patients had undergone disease-specific tests in the past, such as the glycerol test and electrocochleography. All 120 patients underwent the Dix-Hallpike maneuver and the supine roll test. The Dix-Hallpike maneuver was considered positive for posterior (or anterior) SCC BPPV when vertigo was provoked and accompanied by a burst of torsional-vertical two-component nystagmus with the typical characteristics of latency, crescendo, and transience. The supine roll test was considered positive for horizontal SCC BPPV when intense vertigo was provoked and accompanied by horizontal geotropic (canalolithiasis) or apogeotropic (cupulolithiasis or canalolithiasis of the short arm of the horizontal SCC) paroxysmal nystagmus [2].
Posterior SCC BPPV was treated using the modified Epley canalith repositioning procedure [11], and horizontal SCC BPPV was treated using the barbecue maneuver or the Gufoni maneuver [10]. Repeat treatment sessions were performed every 2 or 3 days (in cases of failure or incomplete remission of the symptoms) for a maximum of 7 sessions. Assessment of the success of the treatment included both the patient’s report of relief from vertigo for at least 2 months and a negative Dix-Hallpike or supine roll test result. In case of recurrence of the symptoms, the canalith repositioning procedure was repeated following the same plan. Follow-up data were available for most patients at 6 months.
Continuous variables were expressed as the mean±SD, and categorical variables were expressed as frequencies and percentages. The significance of any difference between groups was evaluated using a t-test for independent samples. The Chi-square test and Fisher’s exact test (if the expected frequencies were less than 5) were used for the statistical analyses, and P < 0.05 was considered significant.
Results
A total of 120 patients, including 68 men and 52 women, were diagnosed with unilateral BPPV and treated with the repositioning maneuver. The male-female incidence ratio was 1.3:1. The patients’ ages ranged from 20 to 76 years, with an average age of 48.8±2.8 years.
Groups A and B included 7 men and 14 women with a duration of BPPV symptoms of 64±7.2 days. Five cases (23.8%) had horizontal semicircular canal BPPV, 8 cases (38.1%) had posterior SCC BPPV, and 8 cases (38.1%) had multi-semicircular canal BPPV. The 99 cases of idiopathic BPPV without Meniere’s disease in the control group included 38 females and 61 males with a duration of BPPV symptoms of 28.1±7.2 days; twenty of these cases (20.2%) had horizontal SCC BPPV, 62 cases (62.6%) had posterior semicircular canal BPPV, 10 cases (10.1%) had anterior semicircular canal BPPV and 7 cases (7.1%) had multi-semicircular canal BPPV. No significant differences were found between the experimental groups and the control group in the gender ratio and duration of BPPV symptoms (P > 0.05, Chi-square test).
Thirteen cases had unilateral Meniere’s disease ipsilateral to the SCC responsible for the BPPV. Eight cases had Meniere’s disease with unilateral BPPV. Of these, 4 cases had BPPV on the side with more severe Meniere’s disease.
Analysis of Nystagmography in Three Groups of Patients with BPPV
Analysis of Nystagmography in Three Groups of Patients with BPPV
Comparison of the Efficacy of Canalith Repositioning in Three Groups of Patients with BPPV
P for Posterior semicircular canal, H for Horizontal semicircular canal, A for Anterior semicircular canal.
The procedure performed in group A proved successful in 1 case (12.5%), and 4 cases (50.0%) needed more therapy sessions for treatment. Conversely, in group B the procedure was successful in 2 case (15.4%), and 9 cases (69.2%) needed more therapy sessions for treatment. In the control group, 30 cases (30.3%) had one therapy session, and 53 cases (53.5%) needed more therapy sessions for treatment. The overall treatment success rate of the experimental group was 76.2% (16/21), whereas the overall treatment success rate for the control group was 83.8% 83/99); this difference was not significant (P > 0.05). However, the patients with Meniere’s disease required more therapy sessions for treatment of the disease. At the 6-month follow-up in groups A and B, 10 patients presented recurrence of BPPV (50.0% and 46.2). In contrast, the recurrence rate in the control group was only 20.2% (P < 0.05).
Comparison of Efficacy of Canalith Repositioning for different semicircular canals
P for Posterior semicircular canal, H for Horizontal semicircular canal, A for Anterior semicircular canal.
Statistically significant for P < 0.05.
The incidence of Meniere’s disease among patients with BPPV has been reported with a wide range of 0.5% to 45% [3, 7–9]. Low incidence rates were reported in earlier studies. For example, Katsarkas and Kirkham [9] found that 3 of 255 patients with BPPV (0.8%) had Meniere’s disease. More recently, Karlberg et al. [8] found only 16 patients with Meniere’s disease in a group of 2,847 patients (0.5%). However, Hughes and Proctor [7] reported that 45 of 151 (29.8%) patients with BPPV had associated Meniere’s disease, which was the highest incidence reported to date. One possible explanation for the difference was the inclusion of patients who had vestibular function tests on more than one occasion, which resulted in the selection of patients with recurrent disorders, such as Meniere’s disease. However, the prevalence rate of BPPV associated with Meniere’s disease has gradually increased as research into BPPV has increased in recent years for the following reasons: 1) the use of the canalith repositioning procedure in an increasing number of patients with Meniere’s disease to eliminate the rate of missed diagnoses in patients with BPPV associated with Meniere’s disease and 2) disease diagnostic methods for Meniere’s disease have increased, such as the electrocochleogram and glycerol test in pure tone audiometry.
We found 21 patients with Meniere’s disease in a group of 120 patients with BPPV (17.5%). The larger proportion of patients with Meniere’s disease and associated BPPV observed recently might be explained by the increased awareness of BPPV over the past decade due to the development of the canalith repositioning procedures; as a result, the Dix-Hallpike test and the supine roll test might be the most commonly performed of the more than ten tests for patients with known Meniere’s disease. Additionally, BPPV is now widely diagnosed in a timely and accurate manner, which has increased the population of patients with diagnosed BPPV. Finally, significant progress in the diagnosis of horizontal SCC BPPV, which is quite common in Meniere’s disease, has been recently achieved. Discrepancies in the reported incidence among studies might be due to differences in the study design, different patient populations, and/or the use of various inclusion and exclusion criteria for diagnosis. Meniere’s disease is quite difficult to diagnose despite the published criteria. Older studies [3, 9] obviously did not use the AAO-HNS 1995 criteria, whereas more recent studies [8] used additional measures, such as positive electrocochleographic findings.
The above pathogenetic mechanisms have been further supported by temporal bone studies. Morita et al. [13] found significant differences in the incidence of cupular and free-floating deposits in the posterior and lateral SCCs between temporal bones in patients with and without Meniere’s disease. The authors observed that the incidence of these deposits was associated with the duration of disease rather than aging. Although most authors support the idea that BPPV is secondary to Meniere’s disease [7, 8], Paparella [14] has proposed that Meniere’s disease secondary to BPPV is also possible, because loose otoconia can cause a decrease in endolymphatic absorption, resulting in endolymphatic hydrops.
Several authors have stated that secondary BPPV has specific clinical characteristics that differ from those of idiopathic BPPV. We found this hypothesis to be true in our patients with BPPV in association with Meniere’s disease. The features that distinguish this clinical entity from idiopathic BPPV are briefly discussed below.
The first difference is the longer duration of symptoms. One probable explanation could be either different pathogenetic mechanisms or treatment difficulties. Posterior SCC involvement appears to be a cardinal feature of secondary BPPV. We found 8 patients with posterior SCC BPPV for a rate of 38.1% compared to a rate of 62.6% in patients with idiopathic BPPV. There is no clear explanation for the posterior SCC predilection or the discrepancies between studies. This finding might be attributed to the recently achieved progress in the diagnosis of posterior SCC BPPV and to a different pathogenetic mechanism. Therefore, posterior SCC could be more susceptible to lithiasis than the other canals. Thus, we may assume that anatomic factors predominate in Meniere’s-associated BPPV over the gravity factor, which is responsible for the predilection for the posterior SCC of idiopathic BPPV.
The finding of more frequent canal paresis in our patients is not surprising, because this finding is common in patients with Meniere’s disease, especially in the advanced stage [12].
Finally, the treatment of patients with BPPV associated with Meniere’s disease appears to be less effective and more time-consuming than the treatment of patients with idiopathic BPPV. We had success with the first canalith repositioning procedure in only 12.5% of the patients in group A, whereas we had a success rate of 15.4% in group B.
High recurrence rates were reported in most previous studies [4] and were also found in the present study. Even authors who reported successful treatment results [4] found recurrence rates as high as 50%. Several factors may explain the possible worse treatment results and higher rate of recurrence in patients with both BPPV and Meniere’s disease [6]. First, repeated hydropic distention may reduce the elasticity of the membranous labyrinth and result in partial collapse or adhesion of the SCC, which may exhibit partial obstruction. Accordingly, multiple canalith repositioning procedures may be needed for effective treatment and thus a higher rate of failure is possible. Second, partial obstruction may also be due to a dilated saccule or adhesion of otoliths to the membranous labyrinth. Finally, periodic hydropic distention, which is observed during the natural course of Meniere’s disease, may result in repeated release of otoconia and manifestation of BPPV attacks.
In conclusion, patients with BPPV associated with Meniere’s disease differ from the patients with idiopathic BPPV in the following ways: 1) a longer duration of symptoms; 2) more frequent involvement of the posterior SCC; 3) a greater incidence of canal paresis; and 4) poorer treatment results and 5) a higher rate of recurrence. The above findings imply that BPPV associated with Meniere’s disease differs from idiopathic BPPV in terms of several clinical features and thus may follow a different clinical course and respond less effectively to treatment.
Declaration of interest
The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
Footnotes
Acknowledgments
This study was supported by the Foundation of Guangzhou Twelfth People’s Hospital scientific research projects (2017-3-29) and (2017-3-22).