Benign paroxysmal positional vertigo (BPPV) is the most common vestibular disorder in adults; the age of onset is most commonly between the 5th and 7th decades.1 Symptoms are characterized by imbalance and brief attacks of vertigo occurring after specific position changes because of the presence of debris (thought to be fragmented endolymph particles) in the semicircular canal.2 Considered rare in children,3 the literature describing the incidence of BPPV in pediatrics is limited. D'Agostino et al4 reported the case of a 10-year-old child with horizontal canal BPPV with complaints of vertigo in the supine position. Positional testing revealed bilateral geotropic nystagmus with greater intensity of symptoms on the left, indicating right horizontal canal canalithiasis. The patient was treated successfully with a prolonged positional maneuver lying on his right side. Saka et al5 reported findings consistent with horizontal canal cupulolithiasis BPPV in an 11-year-old boy and horizontal canal canalithiasis in a 3-year-old girl. The authors concluded that BPPV has been studied less extensively in children than in the adult population. Among all patients complaining of vertigo or dizziness, the percentage of cases of BPPV in children was only 3%.
Although it is possible for each of the 3 semicircular canals to be involved, the majority of all BPPV cases are in the posterior canal.2 According to the canalithiasis theory, otoconial debris from the utricle are caught in the posterior semicircular canal and start to move when the head position is changed quickly in relation to gravity. Posterior canal BPPV is diagnosed by the Dix-Hallpike (DH) test, which involves bringing the patient from an upright to supine position, with the head turned 45° to 1 side and neck extended 20°. This test will elicit symptoms of vertigo along with transient upbeating, torsional nystagmus with the upper poles of the eyes beating toward the affected ear.6 The DH test is considered the reference standard test for the diagnosis of posterior canal BPPV,1 despite a sensitivity of 82% and a specificity of 71.7 The lack of an alternative diagnostic test limits the availability of rigorous sensitivity and specificity data.
Treatment for posterior canal BPPV includes the canalith repositioning maneuver (CRM) also referred to as the Epley maneuver.8 The CRM moves the otoconia from the posterior semicircular canal to the vestibule through a series of head movements. This maneuver therefore repositions the offending otoconia out of the posterior canal, which had been producing the vertigo in BPPV. A Cochrane review reported a statistically significant effect in favor of the CRM compared with control treatment.9
The prevalence of vertigo in 10-year-old children is estimated to be 5.7%, with the most common cause being vestibular migraine that accounts for almost 40% of vertigo diagnoses.10 The most common cause of episodic vertigo in children between the ages of 2 and 5 years is benign paroxysmal vertigo of childhood (BPVC) thought to be a variant of migraine, characterized by short-lived attacks of vertigo and postural imbalance that resolves spontaneously.11,12 The International Classification of Headache Disorder, second edition, defines benign paroxysmal vertigo (1.3.3) as recurrent (at least 5) attacks of severe vertigo resolving spontaneously after minutes to hours, and has been labeled as periodic syndromes of childhood, migraine equivalent or migraine precursors. Neurological function tests including electroencephalogram are normal between attacks in cases of BPVC.13 Diagnosis of BPVC and vestibular migraines in childhood is based on history taking. Children with BPVC complain of dizziness that is not position dependent and is not constant. Chang and Young14 studied the use of vestibular-evoked myogenic potential and caloric testing to diagnose children with BPVC and found that 70% of children with BPVC demonstrated abnormalities on both modes of testing. Lindskog et al11 found that approximately 21% of patients with BPVC developed migraine during a follow-up of 13 to 20 years. Marcelli et al15 reported that children with BPPV had a normal vestibular examination with the exception of a positive DH test, whereas children with BPVC had a positive vestibular examination and a negative DH test. Children with BPVC also have a positive family history of migraine and motion sickness.
The purpose of this case report is to present the cases of 2 children with complaints of vertigo as a result of left posterior canal BPPV as well as possible BPVC or vestibular migraine because of a family history of migraines and a history of motion sickness as well as persistent symptoms after resolution of BPPV.
DESCRIPTION OF THE CASES
Patient A was an 11-year-old boy with a history of migraines, and complaints of vertigo with positional changes and head movement lasting 4 years. He had not been treated for this problem by any other medical professionals, and was referred to our vestibular clinic by a neurologist for a full vestibular evaluation. Patient A had an audiogram performed as well as a CT scan, both of which were normal. He had a 4-year history of headaches lasting hours in duration that were relieved with sleep. Headaches were triggered by bright lights, loud noises, and occasionally accompanied by dizziness, nausea, and motion sickness. Patient A had difficulty riding the school bus, concentrating in class, and participating in wrestling during gym class because of complaints of vertigo while bending over. Subjective complaints were difficult to obtain because of a lack of awareness of when the symptoms occurred and triggers of the symptoms. Lack of sleep and difficulty concentrating on homework were identified as problems because of dizziness and fatigue. No vertigo was reported with rolling over in bed; however, the patient did report some symptoms of falling with lying flat and sitting up from the supine position. He reported room-spinning sensations with quick movements of his head, standing up and sitting down, and rocking movement after getting out of a car. Given the age and awareness of his symptoms, it was difficult to obtain a thorough history; however, given complaints of positional symptoms with bending over, and bed mobility, a diagnosis of BPPV was suspected. Because of complaints of motion sickness and vertigo, Patient A was limited functionally in walking outdoors, walking in the dark, and taking any form of transportation. Patient A's goal for therapy was “To not feel dizzy and the ground doesn't move.”
Patient B was a 9-year-old boy with reports of episodic heavy-headed sensation, intermittent tinnitus in the left ear, and vertigo while moving from sit to stand, and bending over. Symptoms lasted about 5 to 10 minutes, occurring 2 times per week over the course of about 1 year. Patient B did not have a personal or family history of migraines before onset of episodes; he did, however, have a positive history of motion sickness, which had worsened over the past year since onset of his vertigo. The migraine history was significant for a recent onset of headaches around the time of his vertiginous episodes occurring on the right side of his head and lasting less than 1 hour. His pain was described as sharp, and intense, relieved by Tylenol, occurring at varying frequencies from once a week to several months without headaches. The patient had been referred by a pediatric resident for vestibular evaluation and treatment. He had not undergone any diagnostic tests or received any treatment from another medical professional.
DESCRIPTION OF INTERVENTION
Patient A presented to the vestibular clinic with normal oculomotor findings, and an unremarkable vestibular examination with the exception of results of the left DH test. Dix- Hallpike testing on the left side revealed left torsional upbeat nystagmus lasting approximately 40 seconds with severe complaints of vertigo in the test position. Given the positive result in the left DH test and complaints of vertigo, a diagnosis of left posterior canal BPPV was confirmed. During the initial evaluation, a CRM was performed to treat left posterior canalithiasis. Patient A was seen for 4 more visits during which repeated CRMs were performed because of continued presence of upbeat left torsion nystagmus in the DH test. Patient A was allowed to rest in between repeated maneuvers and did not report any lasting side effects with multiple treatments. Gordon and Gadoth6 reported that a higher percentage of patients were symptom free after repeated maneuvers in 1 single session compared with 1 maneuver during each session although the difference was not statistically significant. After the fourth visit, because of difficulties coordinating with the patient's busy school schedule, the patient and his mother were trained to carry out the self-CRM to be performed at home to continue treatment of BPPV. Radtke et al16 have reported greater success with use of self-CRM for self-treatment of BPPV performed 3 times a day until they were symptom free for 24 hours compared with Brandt-Daroff exercises. The patient's mother reported they were not compliant with performance of the self-maneuver; however, she did report the symptoms were greatly improved. The therapist did not see the patient again; however, after 2 months during a follow-up phone call the patient's mother reported he was free of vertiginous symptoms and had not had any migraines for several months.
Patient B was seen for initial evaluation and presented with normal oculomotor findings and normal vestibular examination, with the exception of a positive right head thrust. Upon testing for BPPV, right DH testing revealed right beating torsional nystagmus with unclear vertical component, with patient complaints of “tripping” sensation lasting less than 30 seconds. In the left DH position, the patient exhibited left torsion upbeating nystagmus and complaints of vertigo with reproduction of symptoms. Left CRM was performed twice in a follow-up session, and by the third visit Patient B reported no symptoms of vertigo and bilateral DH testing positions were negative. Patient B reported his motion sickness had improved and he was even able to ride on bumper cars at the amusement park without symptoms. He was discharged because of complete resolution of symptoms of positional vertigo.
DESCRIPTION OF OUTCOMES
Outcome measures used included the Visual Analog Scale (VAS) and the Dizziness Handicap Inventory (DHI). The DHI is a questionnaire developed to evaluate the self-perceived disability as a result of vestibular system disease and is reliable and valid for use in patients with vestibular disorders.17 The VAS was used to identify intensity of dizziness and level of imbalance. The VAS has been tested for validity and reliability when used in measuring pain, but has also been used for estimating vertigo and unsteadiness18 (Figure 1). After initial evaluation with each subsequent visit, Patient A described decreased intensity and duration of vertiginous symptoms during positional testing. Objective measures of symptoms pre- and posttreatment showed decreased levels of dizziness and imbalance on the VAS for both boys (Table 1); however, the DHI score increased posttreatment for Patient A and decreased posttreatment for Patient B (Table 1).
Vertigo is considered rare in children, and there are few conditions in pediatrics that can cause vertigo as a symptom. Subjective history is frequently used to establish a diagnosis for migraines and vestibular disorders. Evaluating children with vertigo as their primary complaint is difficult because of the limitations in obtaining an accurate subjective history as a result of the child's age and/or cognitive level. The children in this case report had complaints of vertigo that were position dependent; however, it was difficult to determine the exact trigger of their symptoms because they did not limit their activities as a result of their symptoms. Patient A was able to describe a sensation of “the floor moving up and down,” which is different from a spinning sensation commonly described by adults. Patient B had complaints of vertigo but also reported a sensation of “tripping” during testing. Duration of vertigo was also much longer in Patient B than would be typical of BPPV in adults. Details such as duration of vertigo, and associated symptoms during the episode, which are typically used to determine cause of vertigo in adults, were difficult to obtain from these patients and different from typical presentation for BPPV. The subjective interview did not point clearly to 1 specific diagnosis as the origin of their vertigo; however, the diagnosis in this case was made by performing the DH test as a screen to rule BPPV in or out. The test was used to reproduce these patients' symptoms, and elicited nystagmus indicating both of these children had left posterior canalithiasis BPPV and supported further treatment of their vertigo.
The use of subjective questionnaires like the DHI can also point to specific diagnoses. Questions 1, 5, 13, and 25 on the DHI specifically ask questions related to symptoms of BPPV. An affirmative answer to these questions would suggest that BPPV testing is warranted. The DHI is not a validated outcome measure for children, and given the difficulty with the self-report of symptoms in children it is difficult to determine the cause of their persistent symptoms. It is also difficult to determine whether their responses on the DHI are accurate and whether the instrument is able to detect change. The VAS seemed to be more sensitive to change in these 2 cases as it is a simpler tool to measure the outcome of the treatments. Because both children were unclear in identifying their symptoms and the effect on their daily activities, they had difficulty answering some of the questions on the DHI, especially since some of the questions applied to adults. These 2 children did not answer affirmatively on the BPPV questions; however, they tested positive on the DH test, which may indicate that the DHI is not a useful tool for the pediatric population.
According to McCaslin et al,19 the most commonly reported disorders in the literature associated with dizziness/vertigo in children are migraine headache, BPVC, and otitis media. Trauma and vestibular neuritis are also cited as common causes of dizziness in children. Wiener-Vacher20 performed a retrospective chart review of more than 2000 children over a 14-year period and found that migrainous vertigo was the most commonly diagnosed vestibular disease accounting for nearly 25% of cases; BPVC represented 20% of diagnoses.
Given the history of migraines and motion sickness, a diagnosis of BPVC/vestibular migraine was suspected; however, both patients presented with a positive DH test indicating BPPV. Symptoms of vertigo in BPVC are not induced by movements of the head or specific positioning, and typically have a sudden onset and last from a few seconds to several minutes.21 Given the patients' familial histories of migraines and their own histories of migraines, BPVC cannot be ruled out as a contributing cause for these patients' symptoms. Precipitating factors for BPVC attack are similar to those of migraine including lack of sleep, hormonal changes, intake of certain foods, and stress.21 Most agree that BPVC is a variant of migraine21; however, it is difficult to distinguish between vestibular migraine and BPVC (Table 2). To date no internationally approved diagnostic criterion for vestibular migraine exists (Table 2).22 Erbek et al23 in a sample of 4- to 17-year-olds found 34% to have migraine and 12% to have BPPV. A third potential diagnosis for pediatric vertigo is vestibular paroxysmia, which presents with brief and frequent vertiginous attacks, thought to be caused by neurovascular cross-compression at the root entry zone of cranial nerve VIII.24 Diagnostic criteria for vestibular paroxysmia include the occurrence of at least 5 episodes lasting seconds to minutes, occurring at rest and with certain head positions or position changes (not BPPV-specific positions) and which respond to treatment with low-dose sodium channel-blocking antiepileptics such as carbamazepine.24 Patients with vestibular paroxysmia have normal results for neuro-otological examinations (including the DH test) (Table 2). Lehnen et al24 report this diagnosis accounts for about 4% of children. Figure 2 illustrates a clinical decision-making algorithm that can assist the reader in determining what could be the cause of a patient's vertigo symptoms. Figure 2 and Table 2 are not comprehensive of all possible diagnoses that cause vertigo; however, it does illustrate differential diagnoses for the patients in this case series.
These 2 cases present a dilemma in determining what could be the cause of their symptoms. Both patients had positive findings on DH testing; however, after resolution of BPPV they continued to have other symptoms. There is a high prevalence of BPPV in patients with migraine.25 Although both patients were treated for BPPV in the same canal, Patient B had much greater improvement in symptoms upon follow-up compared with Patient A. Both patients reported a decrease in intensity of dizziness and imbalance posttreatment; however, both patients continued to report dizziness posttreatment, and this could possibly be due to their history of motion sickness and migraines. Patients with vestibular migraines or BPVC may benefit from continued vestibular therapy to address residual symptoms of dizziness after treatment of BPPV once their migraine symptoms have been medically managed.
Limitations in this case series were lack of validated measures for children as well as difficulty scheduling Patient A for follow-up appointments because of school commitments. Perhaps the length of time with symptoms is a predictor of how well the patient will recover once treated. Further research into the incidence and etiology of BPPV, BPVC, and vestibular migraine in children would be helpful in guiding clinicians to better diagnose patients who present with vertigo. Greater awareness of differential diagnoses for pediatric vertigo, as well as the use of the DH test to screen patients for BPPV, which is a condition physical therapists can treat, is important to keep in mind when treating children with vertigo. In addition, if the DH test is negative, a referral to the physician would be warranted to explore other diagnoses that can be managed medically. Further research could serve to validate outcome measures such as the DHI for pediatric patients with vestibular disorders.
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Keywords:Copyright © 2016 Academy of Pediatric Physical Therapy of the American Physical Therapy Association
benign paroxysmal positional vertigo; case reports; child; differential diagnosis; male; physical therapy