INTRODUCTION
Benign paroxysmal positional vertigo (BPPV) is a common vestibular disorder of the inner ear in adults, with a lifetime prevalence of 2.4% [1]. This condition most often arises between ages 50 and 70 and disproportionately affects women, with a female-to-male ratio of 2.4:1 [2]. Pathophysiologically, BPPV arises when otoconia detach from the utricular macula of the inner ear. These particles then migrate into the semicircular canals and either float freely (canalolithiasis) [3] or adhere to the cupula (cupulolithiasis) [4]. In rare cases, otoconia may become lodged within the canal, a condition known as canalith jam [5]. Such displacement causes cupular deflection, eliciting brief episodes of spinning vertigo in response to changes in head position [6,7]. BPPV is diagnosed based on the Bárány Society criteria, which combine a clinical history of positional vertigo and/or dizziness with characteristic findings on positional maneuvers, such as the Dix-Hallpike test for anterior/posterior canal involvement or the supine roll test for horizontal canal BPPV [8]. Approximately 50% to 70% of BPPV cases are idiopathic, with no identifiable etiology [9]. In a minority of patients, BPPV co-occurs with otologic disorders or arises in individuals with a history of such conditions [10]. This presents challenges due to overlapping symptoms, such as vertigo and dizziness [11]. Often, these symptoms are attributed to the primary disorder, causing BPPV to be overlooked. Alternatively, failure to thoroughly investigate underlying otologic pathology can result in misclassifying BPPV as idiopathic [11,12]. When co-occurring with an otologic disorder, BPPV most often involves the ipsilateral ear and may manifest concomitantly with or after the primary disorder. However, it remains uncertain whether this association is causal or coincidental. Indeed, most instances lack a definitive causal otologic disease [13]; thus, idiopathic BPPV remains the most common diagnosis [13]. Therefore, identifying potential underlying causes through structured history-taking is essential [14].
Although various otologic disorders—such as acute unilateral vestibulopathy/vestibular neuritis, Menière’s disease, labyrinthitis, and other conditions—have been reported in association with BPPV, a detailed overview of the co-occurrence of otologic disorders and BPPV is currently lacking. Since these otologic disorders each have distinct pathophysiological mechanisms, the processes driving their co-occurrence with BPPV may differ from those underlying idiopathic cases [13]. Therefore, the aim of this study was to systematically investigate the co-occurrence of otologic disorders and BPPV.
MATERIALS AND METHODS
Registration and protocol
The methods and results for this systematic review are reported following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines [15]. The protocol was registered with the International Prospective Register of Systematic Reviews (PROSPERO) (www.crd.york.ac.uk/prospero; registration no. CRD42024501512).
Data sources and screening
The final systematic search was conducted on November 7, 2024, in the PubMed and Web of Science (Core collection) databases. During a later review of the search strategy, additional keywords were identified as missing. Therefore, a secondary search was performed on December 25, 2024. The search queries were as follows: “Adult” and “Benign Paroxysmal Positional Vertigo” (Population), “Otologic disorders” (Outcome). No Comparison and Intervention were included. The term “Otologic disorders” encompassed a range of conditions such as “vestibular neuritis,” “labyrinthitis,” “sudden sensorineural hearing loss,” “Menière’s disease,” “post-surgical complications,” “benign recurrent vertigo,” “autoimmune inner ear disease,” and “congenital.” Specific search queries were used (Supplementary Tables 1 and 2), and these queries were developed by two reviewers (MK and SK). No filters were applied.
Study selection
The relevant articles were selected using predetermined inclusion and exclusion criteria (as outlined below). All publications were first imported into EndNote X9 software. Subsequently, the “Find Duplicates” option was applied to remove publications appearing in multiple databases. Two reviewers (MK and SK) independently screened the articles by title and abstract (stage 1) and then conducted a full-text review of the articles (stage 2). After each stage, discrepancies between the reviewers regarding the inclusion and exclusion criteria were discussed in consensus meetings. Finally, the references of the articles included after stage 2 were also screened, and relevant articles were included to ensure that no pertinent articles were overlooked.
Inclusion and exclusion criteria
The inclusion and exclusion criteria can be found in Supplementary Table 3. The screening process was conducted sequentially, focusing first on study design, followed by population, and finally on outcomes. Regarding study design, all original studies written in English, ranging from full-text clinical trials to case reports, were included. Conference abstracts/reports, letters, animal studies, editorials, (systematic) reviews, and meta-analyses were excluded. Concerning the study population, only studies involving adult BPPV patients (≥18 years) were selected. Studies involving healthy subjects, model studies, animal studies, and temporal bone studies were excluded. Regarding outcomes, only studies that reported otologic disorders in conjunction with BPPV were considered. An otologic disorder was defined as a condition primarily originating from the inner ear, middle ear, ear canal or vestibulocochlear nerve disorder. Therefore, conditions with mixed origin (e.g. vestibular migraine), were excluded. Articles reporting light/heavy cupula or hypotheses of BPPV were excluded. Patients with functional vestibular disorders (e.g., persistent postural perceptual dizziness, mal de debarquement syndrome), cerebellar disorders, and central nervous system disorders (e.g., stroke, posterior circulation, multiple sclerosis) were excluded. Head trauma may cause detachment of the otoliths. However, head trauma in the absence of objective evidence of inner ear damage is not an otologic disease. Traumatic BPPV was therefore considered to be neither idiopathic nor secondary BPPV and excluded from the study.
Quality assessment
The Quality in Prognosis Studies (QUIPS) tool was applied to evaluate the risk of bias. The level of evidence was determined following the EBRO-platform (Evidence-Based Guideline Development) [16]. Level A1, A2, B, C, or D could be given, based on different items such as the number of participants and statistical power (Supplementary Table 4). The risk of bias and level of evidence were independently evaluated by both reviewers, who then discussed their findings during a consensus meeting. Each study’s overall risk of bias score was assigned according to the checklist guidelines, categorized as “low risk of bias,” “uncertain risk of bias,” or “high risk of bias.” Four types of biases were considered.
(1) Selection bias: “low risk of bias” in case diagnostic criteria of otologic disorders/BPPV were clearly reported, “uncertain risk of bias” in case diagnostic criteria were not clearly reported. (2) Attrition bias: “low risk of bias” in case the drop-out rate was below 20%, “uncertain risk of bias” in case the drop-out rate was not reported, and “high risk of bias” in case the drop-out rate was above 20%. (3) Detection bias: “low risk of bias” if co-occurring otologic disorders/BPPV were clearly reported, with all conditions quantitatively presented and no missing data. “Uncertain risk of bias” if co-occurring otologic disorders/BPPV were reported but not all were quantitatively presented, and no missing data was present. “High risk of bias” if co-occurring otologic disorders/BPPV were reported but not all were quantitatively presented, and missing data was present. (4) Publication bias: “low risk of bias” in case the statistical analysis was clearly reported, “uncertain risk of bias” in case the statistical analysis was not clearly reported, “high risk of bias” in case the statistical analysis was not appropriate for the design of the study [17].
Interrater reliability was assessed using Cohen’s kappa. Kappa values were classified as follows: no agreement (values <0), slight (0.01–0.20), fair (0.21–0.40), moderate (0.41–0.60), substantial (0.61–0.80), and almost perfect agreement (0.81–1.00) [18]. Cohen’s kappa was applied to evaluate the four types of bias: selection, attrition, detection, and publication bias.
Data extraction
Relevant data from the included articles were extracted by both reviewers (for more details, see Supplementary Tables 5 and 6). Otologic disorders included outer, middle and inner ear, as well as eight cranial nerve pathologies. In case the otologic disorder was not clearly reported, those were labelled as missing. The characteristics of the patients were extracted, including disease percentage (%), total number of patients in the study, the number of patients in the study reporting otologic disorders/BPPV, the number of idiopathic cases of BPPV, and non-otologic causes of BPPV. Two groups of articles were identified. Group 1: studies about BPPV patients in which co-occurring otologic disorders were reported; Group 2: studies about otologic disorders in which co-occurring BPPV was reported. The frequency of co-occurring otologic disorders and BPPV was analyzed in both groups.
Statistical analysis
Descriptive statistics (number and percentage) were used to show the frequency of each disorder. The percentage of each disorder was calculated by dividing the total number of patients reporting a specific disorder by the total number of patients included in the same study and multiplying the resulting proportion by 100. It was decided not to divide by the total number of patients included across all studies because patients did not have the opportunity to report a specific disorder in each study. Next, the study-specific proportions for each disease (e.g., Menière’s disease, acute unilateral vestibulopathy/vestibular neuritis, labyrinthitis, etc.) were synthesized through meta-analyses, which were conducted to more accurately assess the heterogeneity across studies and estimate the overall prevalence. The meta-analyses were conducted for the two groups of studies separately (BPPV with co-occurring otologic disorders, and vice versa). Logit-transformed proportions and variances were calculated, and a random-effects model was used to estimate the pooled prevalences [19,20] along with corresponding 95% CI. A 95% prediction interval (PI) was also reported, which indicated a range of plausible true prevalence values for a new study from the same population [21]. Heterogeneity was assessed using Cochran’s Q statistic, its degrees of freedom (df), and its corresponding P-value. Higgins’ I2 (%) was used to quantify the proportion of heterogeneity attributable to true between-study variability [22]. The outliers were identified using Baujat plots and excluded as deemed necessary. Funnel plots were generated to assess potential publication biases. The final results were visualized using forest and funnel plots. Finally, a one-sample proportion test was conducted to compare the observed proportions with the known prevalence reported in the general population. All statistical analyses were conducted using R version 4.2.2 [23] with the metafor package version 3.8.1 [24].
RESULTS
The initial search retrieved 3,345 articles. After removing 690 duplicates via EndNote (Clarivate Analytics), 2,655 records underwent title and abstract screening, and full-text review resulted in 61 included articles [10,25-84]. The secondary search yielded 62 articles; after removing 22 duplicates with EndNote, 40 remained for title and abstract screening. After full-text review, seven articles were found to meet the inclusion criteria [85-91]. In total, 68 articles were included in this study (Fig. 1).
Group 1: co-occurring otologic disorders in patients with BPPV—descriptive statistics
Nineteen studies [10,29,30,38,39,45,53,54,56,59,61,62,65,67,72,76,78,80,83] reported on otologic comorbidities among patients with BPPV. Across these studies, 11.4% of individuals with BPPV exhibited at least one co-occurring otologic disorder. Sudden sensorineural hearing loss was the most frequently observed condition (found in 11.9% of patients with BPPV in studies of this condition), followed by acute unilateral vestibulopathy/vestibular neuritis (5.0%), whereas labyrinthitis occurred in 2.8% of cases (Table 1). Furthermore, a variety of other otologic and non-otologic disorders were reported (Supplementary Tables 7 and 8, respectively).
Meta-analyses for group 1
Acute unilateral vestibulopathy/vestibular neuritis
A meta-analysis of co-occurring acute unilateral vestibulopathy/vestibular neuritis in patients with BPPV initially included 12 studies; after excluding two outliers identified via Baujat plot (Karlberg 2000 [10] and Waissbluth 2023 [80]), 10 studies remained for the final analysis. The pooled prevalence was 6.5% (95% CI, 3.7%–11.2%). Heterogeneity remained high (Q=126.31, df=9, P<0.001; I2=92.6%), with individual study proportions ranging from 1.7% to 23.6%. The 95% PI indicated that the true prevalence rate might lie between 1.1% and 30.7%. Funnel-plot asymmetry indicated possible publication bias, likely due to underreporting of smaller studies reporting higher prevalences (Fig. 2).
Menière’s disease
Thirteen studies were initially included regarding the prevalence of co-occurring Menière’s disease in patients with BPPV. However, two outliers (Karlberg 2000 [10] and Hughes 1997 [45]) were excluded based on Baujat plot results. The pooled prevalence of the 11 remaining studies was 5.3% (95% CI, 3.4%–8.2%). Heterogeneity remained high (Q=64.80, df=10, P<0.001; I2=89.4%), with individual study estimates ranging from 1.3% to 17.5%. The 95% PI spanned 1.3%–19%, indicating that the true prevalence of co-occurrence could vary within this range. Funnel-plot asymmetry suggested potential publication bias (Fig. 3).
Sudden sensorineural hearing loss and labyrinthitis
The pooled prevalence of sudden sensorineural hearing loss was 11.8% (95% CI, 3.1%–35.7%; 95% PI, 0.4%–80.5%), and for labyrinthitis it was 2.9% (95% CI, 0.9%–8.4%; 95% PI, 0.3%–21.4%). Both analyses exhibited high heterogeneity (I2=98.3% and I2=75.9%, respectively) (Fig. 4). Because only five studies contributed data on sudden sensorineural hearing loss and four on labyrinthitis, outlier detection and publication bias assessments were not performed.
Group 2: co-occurring BPPV in patients with otologic disorders—descriptive statistics
A total of 49 studies [25-28,31-37,40-44,46-52,55,57,58,60,63,64,66,68-71,73-75,77,79,81,82,84-91] evaluated the prevalence of BPPV among patients with primary otologic diagnoses. Across these studies, 9.7% of patients with otologic disorders had co-occurring BPPV. Menière’s disease demonstrated the highest co-occurrence rate at 10.2%, while post-surgical complications were least frequently associated (3.7%) (Table 2).
Meta-analyses for group 2
Acute unilateral vestibulopathy/vestibular neuritis
A meta-analysis was conducted regarding co-occurring BPPV in patients with acute unilateral vestibulopathy/vestibular neuritis. Initially, 11 studies were included. After excluding two case reports (Rambold 2004 [71] and Zapala 2006 [82]) and one outlier (Harada 1993 [43]), eight studies remained for the final meta-analysis. The pooled prevalence of BPPV in this population was 10.4% (95% CI, 6.2%–16.8%). Significant heterogeneity persisted (Q=64.57, df=7, P<0.001; I2=87.2%), with prevalence estimates ranging from 3.2% to 21.3%. The 95% PI suggested that the true prevalence could range from 2.5% to 34.6% (Fig. 5).
Menière’s disease
Ten studies were initially included regarding the assessment of co-occurring BPPV in patients with Menière’s disease. However, two case reports (Baker 2020 [28] and Psillas 2011 [70]) and one outlier [Li 2010 [60]), identified via Baujat plot, were excluded, leaving seven studies for meta-analysis. The pooled prevalence was 9.1% (95% CI, 4%–19.1%). Despite these exclusions, heterogeneity remained high (Q=223.34, df=6, P<0.001; I2=97.1%), with study-specific estimates ranging from 2.4% to 32.4%. The 95% PI suggested that the true prevalence could lie between 0.9% and 52.2% (Fig. 6).
Sudden sensorineural hearing loss and post-surgical complications
Co-occurring BPPV was reported in seven studies of patients with sudden sensorineural hearing loss, with a pooled prevalence of 9% (95% CI, 5.6%–14.4%; 95% PI, 2.5%–28.4%). Among 11 studies examining post-surgical complications, the pooled prevalence of BPPV was 5.2% (95% CI, 3.3%–8.3%; 95% PI, 1.2%–19.3%). Both meta-analyses exhibited high heterogeneity (I2=91.8% and I2=80.2%, respectively) (Fig. 7). Baujat and funnel plots for these analyses are provided in Supplementary Figs. 1 and 2. Based on a one-sample proportion test, the observed prevalence of co-occurrence (9.7%–11.4%) was found to be significantly higher than the 2.4% prevalence in the general population (P<0.001).
Risk of bias and level of evidence
In the assessment of inter-rater agreement, Cohen kappa analysis yielded scores of 0.83 for selection bias, 0.78 for attrition bias, 0.82 for detection bias, and 0.76 for publication bias—indicating substantial to almost perfect agreement between the two raters across all domains. Regarding level of evidence, all interventional cross-sectional studies with control groups were classified as level B evidence due to the inclusion of these controls (n=14). Most studies (n=53) were graded as level C, primarily due to small sample sizes and non-comparative designs. Only one study—a randomized, double-blind trial with an adequate number of participants—achieved a level A2 rating (Supplementary Table 9).
DISCUSSION
This study investigated the co-occurrence of otologic disorders and BPPV. The findings revealed that 11.4% of patients with BPPV had co-occurring otologic disorders (group 1), whereas 9.7% of patients with otologic disorders had co-occurring BPPV (group 2). In group 1, sudden sensorineural hearing loss was the most prevalent condition, followed by acute unilateral vestibulopathy/vestibular neuritis. In group 2, Menière’s disease and acute unilateral vestibulopathy/vestibular neuritis were the most common. However, both groups exhibited substantial heterogeneity, reflecting considerable variability in prevalence rates across the included studies.
Compared with the general population, BPPV appeared to co-occur more frequently in patients with certain otologic disorders. This finding underscores the importance of incorporating vestibular evaluation into the diagnostic workup. Early identification of coexisting BPPV may enable targeted vestibular management, improving treatment strategies and patient outcomes. The pooled prevalence rates for groups 1 and 2 differed. For instance, acute unilateral vestibulopathy/vestibular neuritis occurred in 6.5% of patients with BPPV, whereas BPPV was present in 10.4% of patients with acute unilateral vestibulopathy/vestibular neuritis. Several factors might account for these discrepancies.
First, diagnostic priorities may influence outcomes. Many clinicians focus on positional tests—such as the Dix–Hallpike or supine roll maneuvers—when positional vertigo is the primary concern. This emphasis often expedites the diagnosis of BPPV, since these tests are both quick and specific, potentially reducing the extent of history-taking and assessment [11]. This review demonstrated that the prevalence of secondary otologic disorders associated with BPPV, such as acute unilateral vestibulopathy/vestibular neuritis and Menière’s disease, ranges from 3.5% to 12%. In clinical practice, however, most patients with vestibular symptoms receive a single diagnosis: multiple vestibular disorders are identified in only 3.7% [92] to 30.1% [93,94] of cases. These findings underscore the importance of explicitly screening for underlying otologic disorders when diagnosing BPPV [95], and vice versa. BPPV and other otologic conditions (e.g., vestibular hypofunction] may share overlapping symptoms, such as dizziness triggered by rapid head movements [94]. At a minimum, explicit screening should include structured history-taking, such as the four-step approach [14].
Second, follow-up protocols varied across studies. BPPV often resolves rapidly following canalith repositioning maneuvers and may not warrant prolonged monitoring, whereas otologic disorders typically require extended follow-up [96]. This phenomenon may have increased the likelihood of detecting BPPV in these patients over time.
In this review, acute unilateral vestibulopathy/vestibular neuritis, Menière’s disease, and sudden sensorineural hearing loss were reported more frequently, while other disorders were less commonly documented. This pattern suggests that these otologic conditions and BPPV may share underlying etiologies.
Acute unilateral vestibulopathy/vestibular neuritis and BPPV
The relationship between BPPV and acute unilateral vestibulopathy/vestibular neuritis remains unclear but may involve inflammation of the (primarily superior) vestibular nerve or ischemia of the anterior vestibular artery. Both the superior vestibular nerve and the anterior vestibular artery supply the utricle and the anterior and lateral semicircular canals. Consequently, inflammation or ischemia can damage these structures, potentially causing otoconia to detach from the utricle. Once detached, otoconia may migrate into the semicircular canals [97,98]. This phenomenon—BPPV following acute unilateral vestibulopathy/vestibular neuritis—is known as Lindsay-Hemenway syndrome [99].
Menière’s disease and BPPV
Menière’s disease is characterized by endolymphatic hydrops, with potential disruption of the inner ear fluid homeostasis [100]. This imbalance may damage the utricle and saccule, causing otoconia to detach from the utricle. These displaced otoconia can then migrate into the semicircular canals, resulting in secondary BPPV. Conversely, it has been hypothesized that BPPV might contribute to the development of Menière’s disease. Detached otoconia could obstruct the endolymphatic duct, impairing endolymph drainage and leading to hydrops [101]. However, this hypothesis remains speculative and is not yet supported by robust clinical data.
Sudden sensorineural hearing loss and BPPV
Idiopathic sudden sensorineural hearing loss appears to be associated with BPPV. Although the precise mechanism remains unclear, nerve inflammation or vascular injury to the vestibular system—mechanisms similar to those implicated in acute unilateral vestibulopathy/vestibular neuritis—may cause otoconia to detach, resulting in BPPV [4,37,50].
In addition to a potential shared etiology, factors such as selection and publication biases may have influenced our findings. The studies included in this review may have selectively focused on certain otologic disorders, reflecting clinical practices that prioritize diagnosing and reporting those conditions [102]. Likewise, publication bias is possible, since studies demonstrating significant associations with specific disorders are more likely to appear in the literature. Indeed, funnel-plot asymmetries for several disorders suggest that smaller studies with higher prevalence rates may be underreported. Consequently, the prevalence estimates presented here may not fully represent the true distribution of otologic disorders in the BPPV population.
Even with detailed history-taking, identifying the etiology of BPPV or otologic disorders can be challenging, especially in cases of co-occurrence. For instance, this review included two BPPV cases reported following canal wall down surgery [26,34], making it difficult to establish whether BPPV resulted from cholesteatoma extension into the inner ear or from surgical manipulation (e.g., temporal bone drilling). Likewise, three cases of BPPV occurred after surgery to address superior canal dehiscence syndrome (SCDS) [31,73,81], and distinguishing whether BPPV reflects the underlying SCDS or is a complication of the procedure continues to pose a challenge. While BPPV may not necessarily develop from these disorders, overlapping symptoms (e.g., vertigo) can mimic BPPV and complicate its diagnosis. Therefore, in such cases, careful evaluation is essential to accurately identify any potential BPPV. Lastly, several additional otologic disorders—such as bilateral vestibulopathy and vestibular schwannoma—have been reported to co-occur with BPPV [10,45]. Although these co-occurrences appear uncommon, clinicians should remain mindful of them.
This systematic review has several limitations. First, some of the included studies lacked detailed clinical data, which may limit the generalizability of the findings and hinder accurate assessment of the relationship between otologic disorders and BPPV. Second, because most studies were observational, establishing causal relationships was challenging. Finally, high heterogeneity (I2>90%) was observed across studies, likely reflecting clinical and methodological differences. Although PIs were provided to aid interpretation, the relatively small number of studies precluded further analyses to explore sources of heterogeneity.
To conclude, the co-occurrence of otologic disorders and BPPV is prevalent (9.7%–11.4%). The present findings underscore the need for explicit mutual screening of BPPV and otologic disorders. Moreover, they may help clarify the underlying etiologies of BPPV.
HIGHLIGHTS
▪ Benign paroxysmal positional vertigo (BPPV) co-occurs with otologic disorders in 9.7%–11.4% of cases, exceeding the prevalence of BPPV in the general population (2.4%).
▪ Differences in diagnostic priorities and follow-up protocols may account for the variability in co-occurrence prevalence across studies.
▪ Overlapping symptoms frequently result in misdiagnosis or delayed identification of BPPV in patients with otologic disorders and vice versa. This underscores the necessity of structured history-taking and detailed vestibular assessments.
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