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Neurotologic manifestations of the fibromyalgia syndrome
Journal of the Neurological Sciences 196 (2002) 77 – 80 www.elsevier.com/locate/jns
Neurotologic manifestations of the fibromyalgia syndrome ¨ zer b, Gu¨nhan Karakurum c, Ercan Madenci a Yıldırım A. Bayazıt b,*, Savasß Gu¨rsoy a, Enver O a
Department of Physical Medicine and Rehabilitation, Faculty of Medicine, Gaziantep University, Kolejtepe, Gaziantep, Turkey b Department of Otolaryngology, Faculty of Medicine, Gaziantep University, Kolejtepe, Gaziantep, Turkey c Department of Orthopedics, Faculty of Medicine, Gaziantep University, Kolejtepe, Gaziantep, Turkey Received 14 November 2001; accepted 28 January 2002
Abstract The purpose was to assess otologic symptoms, and audiologic and vestibular findings in fibromyalgia (FM) syndrome. Twenty-four female patients with FM syndrome (FMS) were included in the study. The assessments were based on history, physical examination, audiometry, bithermal caloric testing and auditory brainstem response (ABR) testing. Dizziness was the most common complaint of the patients, and was followed by tinnitus, hearing loss and vertigo. Almost 50% of the patients had some sort of otologic symptoms. Dix – Halpike maneuver proved positional rotary vertigo in 5 (20.8%) patients. The audiometry results of 23 patients were normal. None of the patients had abnormal bithermal caloric testing. Although there were a variety of ABR abnormalities, the ABR results of the patients with and without cochleovestibular symptoms were not significantly different ( p < 0.05). In conclusion, FM patients can complain otologic symptoms even though they do not have any clinically or audiologically detectable ear disease. A neural disintegration or some other events related to neural mediators may be the mechanisms involved in the pathogenesis of otoneurologic as well as systemic manifestations of the disease that possibly leads to abnormal perception of the stimuli coming from internal or external environment. D 2002 Elsevier Science B.V. All rights reserved. Keywords: Fibromyalgia syndrome; Vertigo; Dizziness; Tinnitus; Hearing loss
1. Introduction Fibromyalgia (FM) is a syndrome of unknown etiology, and is characterized by chronic widespread pain, increased tenderness on palpation, and some additional symptoms like disrupted sleep, stiffness, fatigue, psychological disease and cold intolerance. This syndrome is mostly seen in females. Some other disorders like irritable bowel syndrome, migraine and premenstrual syndrome are also related to this specific syndrome [1]. Recent research has shown some sort of neuroendocrine dysfunctions in FM syndrome (FMS), including serotonin deficiency, increased cerebrospinal fluid substance P, disturbance of non-REM sleep, and involvement of the caudate nuclei as well as thalami as detected by brain SPECT [2]. It would not be surprising to encounter otologic symptoms in FMS in which the patients could suffer from multisystem symptoms. However, little is known about the otologic
*
Corresponding author. Tel.: +90-342-336-5400; fax: +90-342-3365505. E-mail address: [email protected] (Y.A. Bayazıt).
manifestations of the disease. For the purpose of this study, otologic symptoms were investigated in patients with FMS.
2. Materials and methods Twenty-four female patients who were diagnosed as having FMS were included in the study. The ages ranged between 21 and 46 years (mean 30.6). The diagnosis of FMS was made on the basis of the criteria of the American College of Rheumatology, 1990 [3]. Briefly, the criteria were diffuse aches and stiffness in the muscle and tendon insertions on digital palpation with an approximate force of 4 kg (the amount of pressure required to blanch a thumbnail) lasting for at least 3 months. To meet the diagnostic criteria, pain must be in 11 or more out of the 18 specified tender point sites. The patients with the objective sign of articular or periarticular disease, erythrocyte sedimentation rate less than 10 mm/h (Westergren), negative latex fixation test, normal ceratine phosphokinase values, and obvious underlying disease such as diabetes mellitus, chronic renal insufficiency, epilepsy, multiple sclerosis, or hypothyroidism were not admitted to the study.
0022-510X/02/$ - see front matter D 2002 Elsevier Science B.V. All rights reserved. PII: S 0 0 2 2 - 5 1 0 X ( 0 2 ) 0 0 0 3 2 - 1
78
Y.A. Bayazıt et al. / Journal of the Neurological Sciences 196 (2002) 77–80
FM impact questionnaire (FIQ) [4] was applied to all patients. There were two forms of questions. Some of the questions were as follows: ‘‘how bad has your pain been?’’, or ‘‘how bad has your stiffness been?’’. The tenderness sites of the patients are shown in Table 1. The otolaryngologist assessed all the FM patients as well. The assessments were based on the patient’s history, otolaryngological examination, and some audiologic and vestibular tests. In the patient’s history, questions were asked for the presence or absence of hearing loss, tinnitus, vertigo and dizziness. The diagnosis of tinnitus was made by the patient’s history (subjective tinnitus). None of the patients had objective tinnitus. The patients were asked whether they had a hallucination of motion or light headedness for diagnosing vertigo or dizziness, respectively. Each patient underwent standard otolaryngological examination including otoscopy, rhinoscopy and laryngoscopy, and positional testing (Dix – Halpike maneuver) as well as endoscopy when indicated. In the audiological assessment, pure tone and speech audiometry, tympanometry and auditory brainstem response testing (ABR) were performed. Bithermal caloric testing was performed for vestibular assessment, and 20 diopter Frenzel glasses were used. An interear difference greater than 20% was considered unilateral weakness. On audiometry, pure tone averages (PTA) and speech discrimination scores (SDSs) were evaluated. The PTA was calculated at 0.5-, 1-, 2- and 3-kHz frequencies. Tympanometry was made to assess the acoustic reflex arc. ABR results were interpreted for absolute wave latency of the waves, I, III and V, and for interpeak latency of the waves I –III, III –V and I– V. The active ABR electrode was placed on the mastoid being tested, and the electrode placed on the other mastoid was used as a ground, and the vertex or reference electrode was placed in the midline on the forehead at the level of hairline. Each ear was tested separately. ABR (Intelligent Hearing Sytem, multichannel evoked potential system, Miami, FL) parameters were filters 100– 3000 Hz, stimulus intensity of 80 dB HL and stimulation rate of 21.7 rarefaction clicks per second. ABR results were considered normal if interaural difference was 0.2 ms or less for the waves I –III, III – V and I –V IPLs. Results were considered
Table 1 Body sites and rates of the tenderness points Site
Tenderness point
Trapezius Supraspinatus Low cervical Occiput Gluteal Lateral epicondyl Greater trochanter Second rib Knee
21 20 20 18 10 12 0 0 1
(84%) (80%) (80%) (72%) (40%) (48%) (0%) (0%) (4%)
Table 2 Overview of the audiovestibular symptoms of the patients Symptom
Hearing loss, number of patients (%)
Tinnitus, number of patients (%)
Dizziness, number of patients (%)
Vertigo, number of patients (%)
Present Absent
3 (12.5) 21 (87.5)
4 (16.7) 20 (83.3)
9 (37.5) 15 (62.5)
2 (8.3) 22 (91.7)
borderline if the interaural difference for the waves I –III, III –V and I –V IPLs was between 0.2 and 0.3 ms. If the interaural difference was 0.3 ms or greater for the waves I– III, III –V or I– V IPLs, results were considered abnormal. Results were also considered abnormal if the IPLs or absolute latencies were more than 2 standard deviations (2 S.D.) from the equipment norms [5]. The ABR results of the patients with and without cochlear and/or vestibular symptoms were compared with each other using Mann – Whitney U-test.
3. Results Dizziness was the most common symptom of the patients, and was followed by tinnitus, hearing loss and vertigo in the decreasing order (Table 2). Two (8.3%) patients were suffering from both tinnitus and dizziness. One patient had all of the symptoms, and one had both hearing loss and vertigo. The remaining 20 patients described only one of the symptoms. The otolaryngological examination was normal in all patients except one who had type III adhesive otitis media on the left ear. This patient was not suffering from any cochleovestibular symptom, and had her audiovestibular tests within the normal limits, and underwent a tympanoplasty operation for the ear disease. Dix– Halpike maneuver proved positional vertigo, which was rotary, in 5 (20.8%) patients. The test result was normal in other patients. Audiometry revealed that 23 patients had PTAs and SDSs within the normal limits. The PTA was 31 and 28 dB for the right and left ears only in one patient who had a mild bilateral sensorineural hearing loss. SDS of this patient was also diminished (right, 68%; left, 72%). None of the patients had abnormal bithermal caloric testing result or acoustic reflex arc disorder, (Table 3). On ABR, absolute latencies of the waves I, III and V were found abnormal in 1 (4.2%), 2 (8.3%) and 18 (75%) patients, respectively. IPL of the waves I– III, III – V and I– V was prolonged in 15 (62.5%), 11 (45.8%) and 20 (83.3%) Table 3 Audiovestibular test results of the patients Assessment Dix – Halpike, Audiometry, Caloric testing, Acoustic reflex number of number of number of arc, number of patients (%) patients (%) patients (%) patients (%) Abnormal Normal
5 (20.8) 19 (79.2)
1 (4.2) 23 (95.8)
– 24 (100)
– 24 (100)
Y.A. Bayazıt et al. / Journal of the Neurological Sciences 196 (2002) 77–80
79
Table 4 ABR results of FMS patients FMS patient
Absolute latency, mean F S.D. (ms)
Interpeak latency, mean F S.D. (ms)
Amplitude, mean (Av)
I
III
V
I – III
III – V
I–V
I
III
V
Symptom (+) Symptom ( )
1.5 F 0.2 1.6 F 1.7
3.61 F 0.18 3.7 F 0.21
5.82 F 0.11 5.69 F 0.12
2.28 F 0.23 2.5 F 12
2.34 F 0.14 2.55 F 0.9
4.8 F 0.13 4.92 F 0.22
0.41 0.5
0.36 0.45
0.52 0.61
ABR, auditory brainstem response; FMS, fibromyalgia symptom; S.D., standard deviation.
patients, respectively. The ABR latencies were not significantly different between the patients with and without cochleovestibular symptoms ( p > 0.05), Table 4.
4. Discussion Although FM patients generally have subjective symptoms, clinical or laboratory assessments usually fail to find out any objective finding related to these subjective symptoms. Likewise, the patients in this study had also subjective cochleovestibular symptoms, but otolaryngologic assessments failed to prove the objective basis to their corresponding complaints. In addition to that, despite abnormal positional test or audiometry results, some patients were not suffering from cochleovestibular symptoms. Therefore, it seems that cochleovestibular symptoms in FMS are not associated with an organic ear disorder, or an FM patient may not be aware of or lack possible cochleovestibular symptoms even in the presence of an underlying ear disease. There was only positional vertigo in 5 patients, which may be a coincidence as the paroxysmal positional vertigo being the most common cause of peripheral vertigo. It was suggested that vestibular test results had been normal in pediatric FMS, and that the common musculoskeletal abnormalities of FM might have affected the proprioceptive orientation, therefore giving a sense of imbalance [6]. Central nervous system dysfunction frequently occurs in patients with FMS, although proprioceptive may be involved in some of the abnormalities observed [7]. It was reported that 72% of FM patients had vertigo and dizziness, while 15% and 30% had sensorineural hearing loss and abnormal ABR results, respectively [7]. These rates are not identical to the rates found in this study. This may be due to the characteristics of FMS in which there is discordance between the patient symptoms and laboratory findings. It was suggested that FM patients had temporomandibular joint dysfunction [8]. It is likely that some cochleovestibular symptoms can be seen in temporomandibular joint disease like Costen syndrome. Therefore, otologic manifestations of FMS can in part be due to the joint involvement. Although we did not get into details of temporomandibular joint disease, there was no evidence of the joint involvement in our otolaryngologic examination. Alternatively, it was proposed that antibodies against phospholipids, serotonin and gangliosides can cause immune or sudden hearing loss, and these antibodies can also be high in FMS which can be the causes
of otologic manifestation in FMS [9]. However, although otologic disease can coexist with FMS, almost all patients in this study had no proven ear disease. In the light of these findings, patients with FMS may have an altered perception of normal and disease states. This contention could be supported by the previous study that showed a generalized disturbance of perceptual thresholds in patients with FM, which was not restricted to the perception of pain [10]. There is an evidence that a central nervous system involvement exists in FMS because there is normal flow level with slight but significant focal flow decreases in dorsolateral frontal cortical areas of both hemispheres, and dysfunction at least at the brainstem level as detected by ABR [11]. That altered perception of the normal and disease states in our patients may be attributed to the involvement of the brain structures. However, there is no detectable brain lesion with magnetic resonance imaging. This may be pointing out some events at the level of mediators or neural integration. In order to assess the neural integrator, we performed ABR which helps record evoked potentials generated by the auditory pathway from ear to the high brainstem in response to auditory stimuli. It can also indirectly show the functional status of the neural integration in the brainstem [12]. There were many ABR abnormalities detected in the FMS patients in this study. Prolonged IPLs in the majority of patients indicate a neural disintegration at the brainstem level. However, no significant difference was found when the ABR results of the symptomatic and asymptomatic patients were compared. This also supports our contention that there is a discordance between the symptoms and signs of or subjective and objective data obtained from these patients. The brainstem pathology causing ABR abnormality might also be causing altered perception as well as the discordance. The brainstem contains the relay stations of the incoming peripheral audiovestibular inputs on the way to the cortical sites where they are interpreted or perceived. An abnormal perception can be due to neural disintegration, which upsets the modification of the incoming stimuli at the brainstem level. A number of otologic manifestations can be seen in FMS, which can bring the patient to otolaryngologist initially. According to the results of this study, almost 15% of FM patients had cochlear symptoms while 40% had vestibular symptoms. In other words, almost 50% of the patients had some sort of cochleovestibular symptoms. On the other hand, the majority of these patients had normal audiovestibular test results. Therefore, an otolaryngologist should be familiar
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Y.A. Bayazıt et al. / Journal of the Neurological Sciences 196 (2002) 77–80
with the FMS in order to handle such patients precisely. Particularly, in the presence of a discordance between the otologic symptoms and clinical and audiovestibular findings as well as the coexistence of some other problems consistent with FMS, it seems necessary to consult other disciplines to make the differential diagnosis of FMS.
5. Conclusion Dizziness is the most frequent otologic symptom in FMS, and a positive positional test result is the almost only detectable otologic finding. Patients with FM can complain of otologic symptoms even when they do not have any clinically or audiologically detectable ear disease. However, a neural disintegration can exist in the brainstem as noted on ABR testing. This neural disintegration or some other events related to neural mediators in the brainstem relay stations may be the mechanisms involved in the pathogenesis of otoneurologic as well as other systemic manifestations of the disease that possibly leads to abnormal perception of the stimuli coming from internal or external environment.
References [1] Bennet RM. The fibromyalgia syndrome. Text of rheumatology. Philadelphia: Saunders, 1997. pp. 515 – 8.
[2] Yunus BM. Fibromyalgia syndrome current concepts of biophysiological mechanism and management. Tibbi Rehabil Derg 1997;8:120. [3] Freundlıich B, Leventhal L. Diffuse pain syndromes in primer on the rheumatoid disease. Primer on the rheumatic disease. Atlanta: Arthritis and Foundation, 1997. pp. 123 – 7. [4] Burckhardt DS, Clark SR, Bennett RM. The fibromyalgia impact questionnaire development and validation. J Rheumatol 1991;18: 728 – 33. [5] Bayazit Y, Yilmaz M, Kepekci Y, Mumbucß S, Kanlikama M. Use of the auditory brainstem response testing in the clinical evaluation of the patients with diabetes mellitus. J Neurol Sci 2000;181:29 – 32. [6] Rusy LM, Harvey SA, Beste DJ. Pediatric fibromyalgia and dizziness: evaluation of vestibular function. J Dev Behav Pediatr 1999;20:211 – 5. [7] Rosenhall U, Johansson G, Orndahl G. Otoneurologic and audiologic findings in fibromyalgia. Scand J Rehabil Med 1996;28:225 – 32. [8] Waylonis GW, Heck W. Fibromyalgia syndrome. New associations. Am J Phys Med Rehabil 1992;71:343 – 8. [9] Heller U, Becker EW, Zenner HP, Berg PA. Incidence and clinical relevance of antibodies to phospholipids, serotonin and ganglioside in patients with sudden deafness and progressive inner ear hearing loss. HNO 1998;46:583 – 6. [10] Dohrenbusch R, Sodhi H, Lamprecht J, Genth E. Fibromyalgia as a disorder of perceptual organization? An analysis of acoustic stimulus processing in patients with widespread pain. Z Rheumatol 1997;56: 334 – 41. [11] Johansson G, Risberg J, Rosenhall U, Orndahl G, Svennerholm L, Nystrom S. Cerebral dysfunction in fibromyalgia: evidence from regional cerebral blood flow measurements, otoneurological tests and cerebrospinal fluid analysis. Acta Psychiatr Scand 1995;91:86 – 94. [12] Bayazit Y, Bekir N, Gungor K, Kepekci Y, Mumbuc S, Kanlikama M. The predictive value of auditory brainstem responses for diabetic retinopathy. Auris, Nasus, Larynx 2000;27:219 – 22.
Neurotologic manifestations of the fibromyalgia syndrome ¨ zer b, Gu¨nhan Karakurum c, Ercan Madenci a Yıldırım A. Bayazıt b,*, Savasß Gu¨rsoy a, Enver O a
Department of Physical Medicine and Rehabilitation, Faculty of Medicine, Gaziantep University, Kolejtepe, Gaziantep, Turkey b Department of Otolaryngology, Faculty of Medicine, Gaziantep University, Kolejtepe, Gaziantep, Turkey c Department of Orthopedics, Faculty of Medicine, Gaziantep University, Kolejtepe, Gaziantep, Turkey Received 14 November 2001; accepted 28 January 2002
Abstract The purpose was to assess otologic symptoms, and audiologic and vestibular findings in fibromyalgia (FM) syndrome. Twenty-four female patients with FM syndrome (FMS) were included in the study. The assessments were based on history, physical examination, audiometry, bithermal caloric testing and auditory brainstem response (ABR) testing. Dizziness was the most common complaint of the patients, and was followed by tinnitus, hearing loss and vertigo. Almost 50% of the patients had some sort of otologic symptoms. Dix – Halpike maneuver proved positional rotary vertigo in 5 (20.8%) patients. The audiometry results of 23 patients were normal. None of the patients had abnormal bithermal caloric testing. Although there were a variety of ABR abnormalities, the ABR results of the patients with and without cochleovestibular symptoms were not significantly different ( p < 0.05). In conclusion, FM patients can complain otologic symptoms even though they do not have any clinically or audiologically detectable ear disease. A neural disintegration or some other events related to neural mediators may be the mechanisms involved in the pathogenesis of otoneurologic as well as systemic manifestations of the disease that possibly leads to abnormal perception of the stimuli coming from internal or external environment. D 2002 Elsevier Science B.V. All rights reserved. Keywords: Fibromyalgia syndrome; Vertigo; Dizziness; Tinnitus; Hearing loss
1. Introduction Fibromyalgia (FM) is a syndrome of unknown etiology, and is characterized by chronic widespread pain, increased tenderness on palpation, and some additional symptoms like disrupted sleep, stiffness, fatigue, psychological disease and cold intolerance. This syndrome is mostly seen in females. Some other disorders like irritable bowel syndrome, migraine and premenstrual syndrome are also related to this specific syndrome [1]. Recent research has shown some sort of neuroendocrine dysfunctions in FM syndrome (FMS), including serotonin deficiency, increased cerebrospinal fluid substance P, disturbance of non-REM sleep, and involvement of the caudate nuclei as well as thalami as detected by brain SPECT [2]. It would not be surprising to encounter otologic symptoms in FMS in which the patients could suffer from multisystem symptoms. However, little is known about the otologic
*
Corresponding author. Tel.: +90-342-336-5400; fax: +90-342-3365505. E-mail address: [email protected] (Y.A. Bayazıt).
manifestations of the disease. For the purpose of this study, otologic symptoms were investigated in patients with FMS.
2. Materials and methods Twenty-four female patients who were diagnosed as having FMS were included in the study. The ages ranged between 21 and 46 years (mean 30.6). The diagnosis of FMS was made on the basis of the criteria of the American College of Rheumatology, 1990 [3]. Briefly, the criteria were diffuse aches and stiffness in the muscle and tendon insertions on digital palpation with an approximate force of 4 kg (the amount of pressure required to blanch a thumbnail) lasting for at least 3 months. To meet the diagnostic criteria, pain must be in 11 or more out of the 18 specified tender point sites. The patients with the objective sign of articular or periarticular disease, erythrocyte sedimentation rate less than 10 mm/h (Westergren), negative latex fixation test, normal ceratine phosphokinase values, and obvious underlying disease such as diabetes mellitus, chronic renal insufficiency, epilepsy, multiple sclerosis, or hypothyroidism were not admitted to the study.
0022-510X/02/$ - see front matter D 2002 Elsevier Science B.V. All rights reserved. PII: S 0 0 2 2 - 5 1 0 X ( 0 2 ) 0 0 0 3 2 - 1
78
Y.A. Bayazıt et al. / Journal of the Neurological Sciences 196 (2002) 77–80
FM impact questionnaire (FIQ) [4] was applied to all patients. There were two forms of questions. Some of the questions were as follows: ‘‘how bad has your pain been?’’, or ‘‘how bad has your stiffness been?’’. The tenderness sites of the patients are shown in Table 1. The otolaryngologist assessed all the FM patients as well. The assessments were based on the patient’s history, otolaryngological examination, and some audiologic and vestibular tests. In the patient’s history, questions were asked for the presence or absence of hearing loss, tinnitus, vertigo and dizziness. The diagnosis of tinnitus was made by the patient’s history (subjective tinnitus). None of the patients had objective tinnitus. The patients were asked whether they had a hallucination of motion or light headedness for diagnosing vertigo or dizziness, respectively. Each patient underwent standard otolaryngological examination including otoscopy, rhinoscopy and laryngoscopy, and positional testing (Dix – Halpike maneuver) as well as endoscopy when indicated. In the audiological assessment, pure tone and speech audiometry, tympanometry and auditory brainstem response testing (ABR) were performed. Bithermal caloric testing was performed for vestibular assessment, and 20 diopter Frenzel glasses were used. An interear difference greater than 20% was considered unilateral weakness. On audiometry, pure tone averages (PTA) and speech discrimination scores (SDSs) were evaluated. The PTA was calculated at 0.5-, 1-, 2- and 3-kHz frequencies. Tympanometry was made to assess the acoustic reflex arc. ABR results were interpreted for absolute wave latency of the waves, I, III and V, and for interpeak latency of the waves I –III, III –V and I– V. The active ABR electrode was placed on the mastoid being tested, and the electrode placed on the other mastoid was used as a ground, and the vertex or reference electrode was placed in the midline on the forehead at the level of hairline. Each ear was tested separately. ABR (Intelligent Hearing Sytem, multichannel evoked potential system, Miami, FL) parameters were filters 100– 3000 Hz, stimulus intensity of 80 dB HL and stimulation rate of 21.7 rarefaction clicks per second. ABR results were considered normal if interaural difference was 0.2 ms or less for the waves I –III, III – V and I –V IPLs. Results were considered
Table 1 Body sites and rates of the tenderness points Site
Tenderness point
Trapezius Supraspinatus Low cervical Occiput Gluteal Lateral epicondyl Greater trochanter Second rib Knee
21 20 20 18 10 12 0 0 1
(84%) (80%) (80%) (72%) (40%) (48%) (0%) (0%) (4%)
Table 2 Overview of the audiovestibular symptoms of the patients Symptom
Hearing loss, number of patients (%)
Tinnitus, number of patients (%)
Dizziness, number of patients (%)
Vertigo, number of patients (%)
Present Absent
3 (12.5) 21 (87.5)
4 (16.7) 20 (83.3)
9 (37.5) 15 (62.5)
2 (8.3) 22 (91.7)
borderline if the interaural difference for the waves I –III, III –V and I –V IPLs was between 0.2 and 0.3 ms. If the interaural difference was 0.3 ms or greater for the waves I– III, III –V or I– V IPLs, results were considered abnormal. Results were also considered abnormal if the IPLs or absolute latencies were more than 2 standard deviations (2 S.D.) from the equipment norms [5]. The ABR results of the patients with and without cochlear and/or vestibular symptoms were compared with each other using Mann – Whitney U-test.
3. Results Dizziness was the most common symptom of the patients, and was followed by tinnitus, hearing loss and vertigo in the decreasing order (Table 2). Two (8.3%) patients were suffering from both tinnitus and dizziness. One patient had all of the symptoms, and one had both hearing loss and vertigo. The remaining 20 patients described only one of the symptoms. The otolaryngological examination was normal in all patients except one who had type III adhesive otitis media on the left ear. This patient was not suffering from any cochleovestibular symptom, and had her audiovestibular tests within the normal limits, and underwent a tympanoplasty operation for the ear disease. Dix– Halpike maneuver proved positional vertigo, which was rotary, in 5 (20.8%) patients. The test result was normal in other patients. Audiometry revealed that 23 patients had PTAs and SDSs within the normal limits. The PTA was 31 and 28 dB for the right and left ears only in one patient who had a mild bilateral sensorineural hearing loss. SDS of this patient was also diminished (right, 68%; left, 72%). None of the patients had abnormal bithermal caloric testing result or acoustic reflex arc disorder, (Table 3). On ABR, absolute latencies of the waves I, III and V were found abnormal in 1 (4.2%), 2 (8.3%) and 18 (75%) patients, respectively. IPL of the waves I– III, III – V and I– V was prolonged in 15 (62.5%), 11 (45.8%) and 20 (83.3%) Table 3 Audiovestibular test results of the patients Assessment Dix – Halpike, Audiometry, Caloric testing, Acoustic reflex number of number of number of arc, number of patients (%) patients (%) patients (%) patients (%) Abnormal Normal
5 (20.8) 19 (79.2)
1 (4.2) 23 (95.8)
– 24 (100)
– 24 (100)
Y.A. Bayazıt et al. / Journal of the Neurological Sciences 196 (2002) 77–80
79
Table 4 ABR results of FMS patients FMS patient
Absolute latency, mean F S.D. (ms)
Interpeak latency, mean F S.D. (ms)
Amplitude, mean (Av)
I
III
V
I – III
III – V
I–V
I
III
V
Symptom (+) Symptom ( )
1.5 F 0.2 1.6 F 1.7
3.61 F 0.18 3.7 F 0.21
5.82 F 0.11 5.69 F 0.12
2.28 F 0.23 2.5 F 12
2.34 F 0.14 2.55 F 0.9
4.8 F 0.13 4.92 F 0.22
0.41 0.5
0.36 0.45
0.52 0.61
ABR, auditory brainstem response; FMS, fibromyalgia symptom; S.D., standard deviation.
patients, respectively. The ABR latencies were not significantly different between the patients with and without cochleovestibular symptoms ( p > 0.05), Table 4.
4. Discussion Although FM patients generally have subjective symptoms, clinical or laboratory assessments usually fail to find out any objective finding related to these subjective symptoms. Likewise, the patients in this study had also subjective cochleovestibular symptoms, but otolaryngologic assessments failed to prove the objective basis to their corresponding complaints. In addition to that, despite abnormal positional test or audiometry results, some patients were not suffering from cochleovestibular symptoms. Therefore, it seems that cochleovestibular symptoms in FMS are not associated with an organic ear disorder, or an FM patient may not be aware of or lack possible cochleovestibular symptoms even in the presence of an underlying ear disease. There was only positional vertigo in 5 patients, which may be a coincidence as the paroxysmal positional vertigo being the most common cause of peripheral vertigo. It was suggested that vestibular test results had been normal in pediatric FMS, and that the common musculoskeletal abnormalities of FM might have affected the proprioceptive orientation, therefore giving a sense of imbalance [6]. Central nervous system dysfunction frequently occurs in patients with FMS, although proprioceptive may be involved in some of the abnormalities observed [7]. It was reported that 72% of FM patients had vertigo and dizziness, while 15% and 30% had sensorineural hearing loss and abnormal ABR results, respectively [7]. These rates are not identical to the rates found in this study. This may be due to the characteristics of FMS in which there is discordance between the patient symptoms and laboratory findings. It was suggested that FM patients had temporomandibular joint dysfunction [8]. It is likely that some cochleovestibular symptoms can be seen in temporomandibular joint disease like Costen syndrome. Therefore, otologic manifestations of FMS can in part be due to the joint involvement. Although we did not get into details of temporomandibular joint disease, there was no evidence of the joint involvement in our otolaryngologic examination. Alternatively, it was proposed that antibodies against phospholipids, serotonin and gangliosides can cause immune or sudden hearing loss, and these antibodies can also be high in FMS which can be the causes
of otologic manifestation in FMS [9]. However, although otologic disease can coexist with FMS, almost all patients in this study had no proven ear disease. In the light of these findings, patients with FMS may have an altered perception of normal and disease states. This contention could be supported by the previous study that showed a generalized disturbance of perceptual thresholds in patients with FM, which was not restricted to the perception of pain [10]. There is an evidence that a central nervous system involvement exists in FMS because there is normal flow level with slight but significant focal flow decreases in dorsolateral frontal cortical areas of both hemispheres, and dysfunction at least at the brainstem level as detected by ABR [11]. That altered perception of the normal and disease states in our patients may be attributed to the involvement of the brain structures. However, there is no detectable brain lesion with magnetic resonance imaging. This may be pointing out some events at the level of mediators or neural integration. In order to assess the neural integrator, we performed ABR which helps record evoked potentials generated by the auditory pathway from ear to the high brainstem in response to auditory stimuli. It can also indirectly show the functional status of the neural integration in the brainstem [12]. There were many ABR abnormalities detected in the FMS patients in this study. Prolonged IPLs in the majority of patients indicate a neural disintegration at the brainstem level. However, no significant difference was found when the ABR results of the symptomatic and asymptomatic patients were compared. This also supports our contention that there is a discordance between the symptoms and signs of or subjective and objective data obtained from these patients. The brainstem pathology causing ABR abnormality might also be causing altered perception as well as the discordance. The brainstem contains the relay stations of the incoming peripheral audiovestibular inputs on the way to the cortical sites where they are interpreted or perceived. An abnormal perception can be due to neural disintegration, which upsets the modification of the incoming stimuli at the brainstem level. A number of otologic manifestations can be seen in FMS, which can bring the patient to otolaryngologist initially. According to the results of this study, almost 15% of FM patients had cochlear symptoms while 40% had vestibular symptoms. In other words, almost 50% of the patients had some sort of cochleovestibular symptoms. On the other hand, the majority of these patients had normal audiovestibular test results. Therefore, an otolaryngologist should be familiar
80
Y.A. Bayazıt et al. / Journal of the Neurological Sciences 196 (2002) 77–80
with the FMS in order to handle such patients precisely. Particularly, in the presence of a discordance between the otologic symptoms and clinical and audiovestibular findings as well as the coexistence of some other problems consistent with FMS, it seems necessary to consult other disciplines to make the differential diagnosis of FMS.
5. Conclusion Dizziness is the most frequent otologic symptom in FMS, and a positive positional test result is the almost only detectable otologic finding. Patients with FM can complain of otologic symptoms even when they do not have any clinically or audiologically detectable ear disease. However, a neural disintegration can exist in the brainstem as noted on ABR testing. This neural disintegration or some other events related to neural mediators in the brainstem relay stations may be the mechanisms involved in the pathogenesis of otoneurologic as well as other systemic manifestations of the disease that possibly leads to abnormal perception of the stimuli coming from internal or external environment.
References [1] Bennet RM. The fibromyalgia syndrome. Text of rheumatology. Philadelphia: Saunders, 1997. pp. 515 – 8.
[2] Yunus BM. Fibromyalgia syndrome current concepts of biophysiological mechanism and management. Tibbi Rehabil Derg 1997;8:120. [3] Freundlıich B, Leventhal L. Diffuse pain syndromes in primer on the rheumatoid disease. Primer on the rheumatic disease. Atlanta: Arthritis and Foundation, 1997. pp. 123 – 7. [4] Burckhardt DS, Clark SR, Bennett RM. The fibromyalgia impact questionnaire development and validation. J Rheumatol 1991;18: 728 – 33. [5] Bayazit Y, Yilmaz M, Kepekci Y, Mumbucß S, Kanlikama M. Use of the auditory brainstem response testing in the clinical evaluation of the patients with diabetes mellitus. J Neurol Sci 2000;181:29 – 32. [6] Rusy LM, Harvey SA, Beste DJ. Pediatric fibromyalgia and dizziness: evaluation of vestibular function. J Dev Behav Pediatr 1999;20:211 – 5. [7] Rosenhall U, Johansson G, Orndahl G. Otoneurologic and audiologic findings in fibromyalgia. Scand J Rehabil Med 1996;28:225 – 32. [8] Waylonis GW, Heck W. Fibromyalgia syndrome. New associations. Am J Phys Med Rehabil 1992;71:343 – 8. [9] Heller U, Becker EW, Zenner HP, Berg PA. Incidence and clinical relevance of antibodies to phospholipids, serotonin and ganglioside in patients with sudden deafness and progressive inner ear hearing loss. HNO 1998;46:583 – 6. [10] Dohrenbusch R, Sodhi H, Lamprecht J, Genth E. Fibromyalgia as a disorder of perceptual organization? An analysis of acoustic stimulus processing in patients with widespread pain. Z Rheumatol 1997;56: 334 – 41. [11] Johansson G, Risberg J, Rosenhall U, Orndahl G, Svennerholm L, Nystrom S. Cerebral dysfunction in fibromyalgia: evidence from regional cerebral blood flow measurements, otoneurological tests and cerebrospinal fluid analysis. Acta Psychiatr Scand 1995;91:86 – 94. [12] Bayazit Y, Bekir N, Gungor K, Kepekci Y, Mumbuc S, Kanlikama M. The predictive value of auditory brainstem responses for diabetic retinopathy. Auris, Nasus, Larynx 2000;27:219 – 22.