Recurrence of Benign Paroxysmal Positional Vertigo

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Otology & Neurotology 33:437Y443 Ó 2012, Otology & Neurotology, Inc. Recurrence of Benign Paroxysmal Positional Vertigo Paz Pérez, Virginia Franco, Paz Cuesta, Patricia Aldama, María Jesús Alvarez, and Juan Carlos Méndez Department of ENT, Otoneurology Unit, Hospital de Cabueñes, Gijón, Spain Objective: To determine the recurrence rate of benign positional paroxysmal vertigo (BPPV) and the factors associated to such recurrences. Study Design: Prospective study. Method: Sixty-nine consecutive patients treated for first episode of BPPV. Study period: 63 months. Mean follow-up: 47 months. Results: The recurrence rate was 27%. Fifty percent of recurrences occurred in the first 6 months. Nineteen patients had 1 or more recurrence of BPPV; 10 had 1 recurrence, 7 patients had 2, and 2 patients had 3 recurrences. There was no significant difference in the recurrence rate according to sex, age, side, cause of BPPV, or instability after successful treatment. Multicanal BPPV (log-rank, p = 0.024) and anterior canal BPPV (logrank, p = 0.029) showed a significantly greater tendency to recur and to do so earlier. There was a significant difference in time to recurrence related to the number of maneuvers used to resolve the initial BPPV episode (log-rank, p = 0.023). Except for cases of BPPV secondary to labyrinthitis or neurolabyrinthitis, at least 70% of the recurrences affected a different side and/or different canal than the primary BPPV. Conclusion: The recurrence rate of BPPV is 27%, and relapse largely occurs in the first 6 months. When BPPV recurrence is suspected, every canal on both sides must be investigated because it is the BPPV syndrome that recurs, rather than BPPV affecting a particular side or canal. Complex cases of BPPV have a greater risk of recurrence. Key Words: Benign paroxysmal positional vertigovrecurrencevrisk factor. Otol Neurotol 33:437Y443, 2012. Benign paroxysmal positional vertigo (BPPV) is probably the most common diagnosis at vertigo clinics, accounting for 20% to 40% of all cases (1,2). The incidence of BPPV has been calculated at 0.6% per year, with a lifetime prevalence of 2.4% (1). The age of onset is most commonly between the age of 60 and 70 years, with elderly people being at increased risk (2). It is characterized by rotational vertigo induced by head position changes. Patients typically complain about attacks of vertigo when extending or turning the neck, getting up or lying down, or rolling over in bed. The attacks often are accompanied by a feeling of unsteadiness and loss of confidence while walking (1,2). The diagnosis is confirmed by the Dix-Hallpike positioning test or the roll test in cases of horizontal canal variant BPPV (1,3). The diagnosis of BPPV of the posterior semicircular canal is based on the clinical finding of a transient, upbeating, torsional nystagmus with the upper Address correspondence and reprint requests to Paz Pérez, Ph.D., Arquitecto Reguera, 3, 33004 Oviedo, Spain; E-mail: paz.perez@ sespa.princast.es Presented at the 61th SEORL-PCF (Spanish Society of Otolaryngology and Head Neck Surgery) Congress, Valencia (Spain) November 8, 2010. The authors disclose no conflicts of interest. poles of the eyes beating toward the undermost ear when the patient is rapidly positioned into the lateral headhanging position; this is known as the Dix-Hallpike test (1Y3). For BPPV of the horizontal semicircular canal (HSC VPPB), the diagnosis is based on history and features of the positioning nystagmus provoked by a quick turn of the head to either side with the patient lying supine (Pagnini-McClure s maneuver). It is a paroxysmal, purely horizontal direction-changing nystagmus, directed toward the uppermost ear (apogeotropic) with cupulolithiasis or toward the undermost ear (geotropic) with canalithiasis (1). Posterior semicircular canal involvement is the most common form in BPPV; lateral and anterior canal involvement is less common (2). Involvement may be bilateral or simultaneously affect different canals (4). There are 2 accepted theories that support BPPV, known as cupulolithiasis and canalithiasis (2,3). Cupulolithiasis involves a deposit of dense particulate material becoming abnormally attached to the cupula, changing its specific gravity and making the crista sensitive to linear accelerations, such as the acceleration of gravity. Canalithiasis similarly involves the abnormal presence of dense particles within the semicircular canal, but in this case, the particles are free to move about in the endolymph fluid, creating a pressure gradient in the canal that abnormally stimulates the crista, leading to clinical symptoms. 437

438 P. PÉREZ ET AL. BPPV is found in isolation and termed primary or idiopathic BPPV in approximately 50% to 70% of all cases. The most common cause of secondary BPPV is head trauma, representing 7% to 17% of all cases. Other causes of secondary BPPV are viral neurolabyrinthitis or vestibular neuronitis (which is involved in up to 15% of BPPV cases), Ménière s disease (5%), migraines, otologic and nonotologic surgery, and prolonged bed rest (2). Particle repositioning maneuvers are effective treatment for BPPV, leading to the resolution of the positioning nystagmus in 70% to 100% of cases (2,3,5). Repositioning maneuvers are believed to treat BPPV by moving the canaliths from the semicircular canal to the vestibule where they are absorbed (1). These repositioning maneuvers are Epley s canalith repositioning maneuver (modified or as originally described), Semont s liberatory maneuver, and Brandt-Daroff redistribution exercises, for posterior canal BPPV. Horizontal contralateral rolls, log rolls, and forced prolonged positioning are used in the treatment for horizontal semicircular canal-bppv (1,5). The number of maneuvers required to resolve the symptoms varies from 1 to 3 (range, 1.5 maneuvers) (5). Nevertheless, there are complex cases that need 10 or more sessions. The reported recurrence rate of BPPV is 20% to 30%, but there are series with as much as a 50% recurrence rate (6,7). Aspects such as age, sex, the presence of residual instability after the repositioning maneuvers, complexity of primary BPPV treatment, and cause have been related to the development of recurrences, sometimes with controversial results (6,8,9). The purpose of this study was to determine the rate of BPPV recurrence, analyze time to recurrence, and identify the factors affecting the development of recurrent BPPV. PATIENTS AND METHODS A prospective study design was used. This prospective series comprised 69 consecutive patients treated for the first episode of BPPV at our clinic s otoneurology unit from January 2005 to December 2006. Patients who referred having previously presented symptoms compatible with BPPV, irrespective of whether it was diagnosed or treated, were not taken into consideration. Patients with a clinical examination, laboratory findings, and imaging studies suggesting conditions affecting the central nervous system also were excluded from the study. BPPV was diagnosed based on the results of the Dix-Hallpike and supine head-turning tests. The Dix-Hallpike test for vertical canal BPPV was positive if nystagmus was recorded with appropriate positioning, latency, duration, and fatigability, and if it reversed when the patient resumed a sitting position. Horizontal semicircular canal-bppv diagnoses were confirmed by horizontal direction-changing positional nystagmus concurrent with vertigo triggered by the supine head-turning test. Patients were classified as canalolithiasis and cupulolithiasis horizontal semicircular canal-bppv types according to the direction of the nystagmus (horizontal geotropic and apogeotropic nystagmus, respectively). From 2005 to 2008, all patients were examined with Frenzel lenses during their evaluation. Since January 2009, diagnostic and treatment maneuvers are conducted using binocularvideonystagmography(vo425b,interacoustics,denmark). The patients were treated using the appropriate repositioning maneuver for their type of BPPV (largely theepley maneuver for vertical canal BPPV and the Lempert maneuver for horizontal canal BPPV). Patients were reevaluated 7 days after treatment. Success was defined as the absence of nystagmus and positional vertigo. The maneuvers were repeated weekly until successful repositioning had been achieved. The date of the last successful maneuver was established as the start of follow-up. Patients were asked to return to our clinic if they reexperienced symptoms of BPPV. We established a preferential route to examine patients suspected of having a recurrence within 1 week. Date of recurrence, canal and type of BPPV, and the number of maneuvers required for relief were recorded for each recurrence. Follow-up ended in March 2010. Every patient not revisited at the clinic was then interviewed by phone to confirm that they had not experienced BPPV symptoms. Description of the Series at the Onset of BPPV The mean age of the 69 individuals was 62 T 13 years (range, 28Y82 yr); 25 (36%) were men, and 44 (64%) were women. Forty-five patients (65%) presented idiopathic BPPV. Twelve cases (17%) presented after acute unilateral peripheral vestibulopathy, 4 cases (6%) after labyrinthitis (2 of them after otologic surgery), and 6 patients (9%) developed BPPV after head trauma. One patient presented ipsilateral vestibular schwannoma, and in 1 patient, BPPV was triggered after bed rest because of nonotologic surgery. The right side was affected in 40 patients (58%), the left ear in 27, and there were 2 cases with bilateral involvement. Forty-four (64%) of the BPPV patients were identified with the PC variant, 4 (6%) were identified with the HC variant, and 4 (6%) were identified with an AC variant. Eleven patients (15%) had concomitant AC and PC BPPV of the same ear, 4 (6%) patients presented horizontal and posterior semicircular canal involvement of the same labyrinth, and 2 patients (3%) had bilateral posterior canal BPPV. All the cases were consistent with the canalithiasis variant of BPPV. The number of canalith-repositioning maneuvers required to treat the condition ranged from 1 to 9 maneuvers, with a mean of 2. Statistical Analysis Statistical analysis of the data was performed using SPSS software (Statistical Package for the Social Sciences, version 17.0; SPSS Inc., Chicago, IL, USA); W 2 testing and Fisher s exact test (if any of the values obtained in the 2 2 table was G5) were used for statistical comparison between groups. Quantitative variable scores were assessed using the 2-sample t test for independent samples, and the Wilcoxon nonparametric signed-rank method was used for 2 related samples. Normal adjustment was assessed using Kolmogorov-Smirnov test. Kaplan-Meier estimates were used to estimate the percentage of recurrence-free patients, and log-rank tests were used to compare subgroups. The level of significance was defined as a p value of less than 0.05. RESULTS The study period was 5 years and 3 months. Median follow-up time was 47 months (range, 36Y63 mo). Two patients moved to other cities at 8 and 44 months,

RECURRENCE OF BPPV 439 respectively. This time was considered the end of their follow-up period, given that BPPV relapse could not be confirmed. Two other patients died from cancer at 2 and 8 months. These patients are appropriately considered in the statistical analysis (Kaplan-Meier method). Of the 69 patients, 19 had 1 or more BPPV recurrence. This yields a recurrence rate of 27%. Of these 19 patients, 10 had 1 recurrence, 7 had 2 recurrences, and 2 patients had 3 recurrences. Fifty percent of the recurrences occurred within 6 months of the initial repositioning maneuver. The rest occurred during the remaining 18 months, except for 1 patient who experienced recurrence after 3 years (Fig. 1). There was no significant difference in the recurrence rate according to sex, age, or affected side (log-rank, p = 0.93, p = 0.79, and p = 0.4, respectively). Eleven (21%) of the 52 patients with BPPV onset affecting a single canal and 8 (48%) of the 17 patients with multiple canal or bilateral BPPV at onset experienced a recurrence. The rate of recurrence among these 2 groups showed statistical significant differences (W 2, p =0.042), and the log-rank test revealed that there also was a significant difference between them in time to recurrence (log-rank, p = 0.024) (Fig. 2). When single canal cases were analyzed, there was no significant difference in the rate of recurrence in HC-type BPPV and vertical canal BPPV: of the 4 horizontal canal cases, 1 (25%) developed a recurrence; of the 48 cases with vertical canal BPPV, 8 cases (16%) had a recurrence. When vertical canal BPPV was considered, the rate of recurrence among posterior and superior canal BPPV showed statistical differences: of the 44 cases with posterior canal BPPV, 6 cases recurred (13%), whereas 2 (50%) of the FIG. 2. Kaplan-Meier estimation for effect of the number of canals affected on BPPV recurrence. Recurrence was significantly more likely when multiple canals were affected (p =0.024). 4 cases of anterior canal BPPV experienced a BPPV relapse (log-rank, p = 0.029) (Fig. 3). There was a significant difference in the number of maneuvers required to resolve the BPPV between single-canal and multiple- or bilateral-type BPPV. Single- FIG. 1. Kaplan-Meier estimation of time to recurrence (first relapse). FIG. 3. Kaplan-Meier estimation for the effect of each vertical canal on BPPV recurrence. Recurrence was significantly more likely when superior canal was involved (p = 0.029). (PC indicates posterior canal BPPV; SC, superior canal BPPV).

440 P. PÉREZ ET AL. FIG. 4. Kaplan-Meier estimation for effect of the number of maneuvers required to manage initial episode on BPPV recurrence. Recurrence was significantly more likely when more than 3 maneuvers were used to control the first BPPV episode (p =0.023). canal BPPV needed a mean of 1.2 maneuvers (SD, 0.5), whereas multiple canal BPPV required 4.5 maneuvers (SD, 2.3) (Student s t test, p G 0.001). Predictably, the log-rank test revealed ( p = 0.023) that there was a significant difference in time to recurrence related to the number of maneuvers used to resolve the initial BPPV episode (Fig. 4). Concerning the cause of BPPV, we found no significant differences related to idiopathic and secondary BPPV, or among secondary BPPV types (W 2 test, p =0.71), despite the fact that posttraumatic BPPV had a larger rate of recurrence, represented most multicanal cases, and needed more repositioning maneuvers for resolution (Table 1). Likewise, time to recurrence of primary orsecondarybppvshowednostatisticaldifferences (log-rank, p =0.24). The average number of maneuvers required to resolve the initial BPPV was 2.5 (SD, 2.22). Nevertheless, the first recurrence of BPPV needed an average of 1.5 maneuvers (SD,1.2) This difference was significant (Wilcoxon test, p = 0.023). The cases with a second recurrence needed an average of 2 maneuvers (SD, 1.6), but this difference was not statistically significant (Wilcoxon test, p = 0.25). Seven (37%) of the 19 cases with a first recurrence of BPPV affected the contralateral ear. Six (50%) of the 12 cases occurring on the same side affected a different canal. Moreover, 5 (71%) of the 7 cases with contralateral side recurrence also changed the affected canal. As shown on Table 1, all these series (original, first, second, and third recurrence) finally showed right-side and PCtype BPPV prevalence. Note that every patient with more than 1 recurrence changed the affected canal, the affected side or both. It is interesting to find that all cases of BPPV secondary to labyrinthitis or neurolabyrinthitis recurred in the same ear and canal. Of the 45 cases of idiopathic BPPV, 10 (71%) recurred on the same side, and 4 (29%) TABLE 1. Characteristics of benign positional paroxysmal vertigo recurrences Primary benign positional paroxysmal vertigo First recurrence Second recurrence Third recurrence Cause Side Canal n Side Canal n Side Canal n Side Canal n Side and canal maintained Id D P 1 D P 1 Id D p 1 D P 1 AUPV D P 4 D P 1 Lab D P 2 D P 1 Id I P 1 I P 2 Same side, canal changed Id D H 1 D P 1 D H 1 D H X Id D P and S 2 D P 1 Id D P and S 4 D P 1 Id D P and H 6 D P 2 D H a 6 Id I S 1 I P 1 Same canal, side changed Id D P 1 I P 1 Id I P 1 D P 1 Side and canal changed Id I S 1 D H 2 D H 2 Id D P and S 8 I P 6 T D P and H 2 I P 1 D P 1 T I P and S 5 D H 1 I P 2 Id I P and S 6 I P and S 3 D P 2 I P and H 4 Id D P 1 D S 2 I P a 3 Bilateral T B P and P 1 I P 1 AUPV indicates acute unilateral peripheral vestibulopathy; D, right; H, horizontal canal; I, left; Id, idiopathic; Lab, labyrinthitis; N, number of maneuvers required to resolve the benign positional paroxysmal vertigo episode; P, posterior canal; S, superior canal; T, posttraumatic; 1, not treated. a Cupulolithiasis-type benign positional paroxysmal vertigo.

RECURRENCE OF BPPV 441 FIG. 5. Kaplan-Meier estimation of time to second recurrence. changed to the contralateral ear. All cases of posttraumatic BPPV changed side and canal or turned from bilateral into unilateral. We found no statistically significant differences in rate of recurrence or time to recurrence between patients with residual instability after successful treatment of BPPV ( p = 0.79 log-rank test). Nine (47%) of the 19 patients who had a recurrence presented a second relapse (Fig. 5). Seven belonged to the idiopathic BPPV group (i.e., 50% of recurrent idiopathic BPPV cases can experience a second recurrence), and 2 originated in the posttraumatic group. There were no second recurrences in the group with BPPV secondary to labyrinthitis or neurolabyrinthitis. The 2 patients with a third recurrence belonged to idiopathic group. DISCUSSION Various authors have reported different recurrence rates of BPPV, ranging from 7% to 50% (6,7,10,11). Our results suggest that a quarter of the patients (27%) with BPPV can develop a recurrence and be expected to return to the clinic. Our attention focuses on several aspects. The series can be described as small after 2 years of patient enrollment. However, we did not include patients who previously had positional symptoms, even if they were not diagnosed with or treated as BPPV. This decision was made to have a patient cohort in which we were certain that the subjects were presenting their first attack of BPPV, even if this meant reducing the sample size. Discrepancies among recurrence rates can be related to the follow-up methodology used. Studies often are based on questionnaires or telephone interviews (6,8,10,12). Our study was conducted in a purely prospective manner, as patients were told to return to the clinic if they suspected BPPV, regardless of whether they had scheduled visits for other reasons. We established a preferential route to asses these patients within 1 week. All suspected recurrences were thus either confirmed or ruled out, as all the patients, including those whose examinations were negative, referred symptoms when they were evaluated. This procedure also prevented the possible spontaneous resolution of BPPV recurrence (it is estimated that spontaneous resolution of BPPV occurs after 4Y6 wk (2,4,8)). High rates of spontaneous resolution of BPPV have been reported but usually rely on subjective symptoms. We also must consider that a significant number of the patients reporting spontaneous improvement had their symptoms reduced by avoiding provocative positions rather than an actual cure (1). Therefore, we can confirm the accuracy of the follow-up with regard to recurrences. The procedure did not increase our work load unnecessarily, as only 3 of the returning patients did not actually present a recurrence of BPPV. Another interesting aspect is the fact that sex had no impact on recurrence. BPPV is more prevalent in women. This has been related to the greater prevalence of osteoporosis among female subjects (13,14), and Brandt (7) found a female-to-male recurrence ratio of 3:2. However, our series shows no difference between sexes when BPPV develops. Other studies have shown that sex does not have a significant effect on the outcome of BPPV treatment (9,10). In the series studied by Brandt (7), the recurrence rate of patients in their 70s was half that of those in their 60s, and Rashad (12) found that time to recurrence was significantly longer in patients younger than 40 years. In our study, however, we found no association between age and BPPV recurrence. The same is true of the affected side. BPPV most commonly affects the right side, but there is no difference in the rate of recurrence between right- and left-sided BPPV. Also, in general, every recurrence has a preference for the right side despite the fact that 37% of all recurrences affect the contralateral ear. Together with the fact that multiple recurrences occurred in idiopathic and traumatic BPPV, this suggests that these patients do not have a particular problem with one of their ears but that they have a condition involving their 2 labyrinths leading to the development of BPPV, sometimes affecting 1 ear, sometimes the opposite side or even both. Tanimoto et al. (9) have found that endolymphatic hydrops can be considered the most important risk factor for recurrence of BPPV. In their series, the first BPPV episode, recurrent BPPV, and endolymphatic hydrops affected the same ear, and these patients often had more than 1 BPPV relapse. This suggests that endolymphatic hydrops can lead to repeated inner ear damage, giving rise to frequent ipsilateral BPPV episodes. None of our patients had Ménière s disease, so we were unable to confirm these observations. Patients diagnosed with BPPV after acute unilateral peripheral vestibulopathy or labyrinthitis were the only

442 P. PÉREZ ET AL. group in which all recurrences affected the same side. Also, these patients only had 1 recurrence. Unlike endolymphatic hydrops, which causes a recurrent chronic course affecting the inner ear, vestibular neuritis and labyrinthitis involve a limited process which, once stabilized, prevents new BPPV development. Posttraumatic BPPV deserves special consideration. Traumatic brain injury has been reported to be a common cause of BPPV (9,12,15). Traumatic BPPV has been described as more complex, often affecting the superior canal or more than 1 canal and sometimes being bilateral. In the series studied by Kansu et al. (10), a history of head trauma was one of the factors contributing to recurrence; conversely, no tendency to recur was observed by Ahn et al. (11) in patients who developed BPPV after traumatic brain injury compared with idiopathic BPPV. In our series, 3 cases of traumatic BPPV presented recurrence, and 2 of them had 2 recurrences. They all had 2 canals affected in the primary episode and changed side and canal when they recurred. Therefore, our series confirms the suspicion that cases of traumatic BPPV are complex and are particularly predisposed to relapse. It is not surprising to find that patients with complex BPPV affecting more than 1 canal or requiring a large number of maneuvers tended to relapse. This can be related to the condition of their labyrinths, with inflammatory changes leading to repeated BPPV episodes. Recently, Horii et al. (16) defined intractable BPPV in cases of either persistent nystagmus or frequent recurrence, finding that intractable BPPV patients often showed stenosis and semicircular canal filling defects on magnetic resonance imaging of the inner ear. They hypothesized that abnormal lesions in the canal may act as a source of debris, and in many instances, small otoconial debris can be scattered from a large plug to various canals at various times, resulting in a high rate of recurrence and heterogeneous clinical symptoms. This also can be related to the common canal changes observed in our patients. Many patients report prolonged mild imbalance, even after successful treatment of BPPV. The origin of these residual symptoms is unknown, but it has been speculated that they reflect otolith dysfunction (17,18). We tested the impact of residual instability on the development of BPPV recurrence, but it was not found to be a risk factor. In our series, residual symptoms were more commonly encountered in patients with BPPV secondary to vestibular deficits (37%) than in those with idiopathic BPPV (18%). We can speculate that residual instability is associated with vestibular asymmetry instead of otolith dysfunction. Recently, Farally (19) showed that persistent dizziness after resolution of BPPV may be due to mental stress and is affected by the duration and recurrence of vertigo. Note the apparent reduction in the number of maneuvers required to resolve first BPPV recurrences (Table 1). In the series studied by Tanimoto et al. (9), patients with recurrent BPPV tended to require more canalith repositioning procedures than primary cases, and he hypothesized that conditions that predispose patients to repeated otoconial release may cause not only recurrence but also labyrinth resistance to repositioning treatment. All the first episodes of BPPV and the first recurrences were consistent with canalithiasis, so the impact of this possible factor on the development of recurrences cannot be evaluated. Only 2 patients, in their second recurrences, developed cupulolithiasis in BPPV, one in the horizontal and the other in the posterior semicircular canal (Table 1). These were the patients who required the largest number of maneuvers to resolve the recurrence. They may be responsible for the slight increase in the average number of maneuvers required to resolve the second recurrence. In our study, we considered the effect of the time since the beginning of the study, which could act as a bias. During this time, we have gained experience in the assessment and treatment of BPPV. In the last years, we have routinely included videonystagmography viewing and recording. Eye movement videotaping clarifies features of complex nystagmus patterns, particularly mixed combinations, and is available both during treatment and after treatment for review. It has been very helpful for improving decision making regarding diagnostic testing and repositioning maneuvers and could have improved repositioning maneuver efficiency. CONCLUSION BPPV relapses are frequent, affecting a quarter of all patients. Fifty percent of recurrences present within 6 months. 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