- Email: [email protected]
Pulsatile tinnitus alleviated by contralateral neck compression: a case report
Auris, Nasus, Larynx 30 (2003) 89 /91 www.elsevier.com/locate/anl
Pulsatile tinnitus alleviated by contralateral neck compression: a case report Byung-Hoon Jun a,*, Ick-Soo Choi a, Ghi-Jai Lee b a
Department of Otolaryngology, Seoul Paik Hospital, Inje University College of Medicine, Jur-Dong 2ka, 85 Choong-Ku, Seoul 100-032, South Korea b Department of Radiology, Seoul Paik Hospital, Inje University College of Medicine, Seoul 100-032, South Korea Received 18 May 2002; accepted 20 September 2002
Abstract A case of a 58-year-old man with right pulsatile tinnitus originating from a small dural arteriovenous fistula (DAVF) of the jugular bulb is described. The tinnitus was alleviated by contralateral neck compression. This unusual observation ruled out venous pulsatile tinnitus, although a temporal bone CT scan showed a high jugular bulb. The fistula was confirmed by angiographic study. The patient was treated by transarterial embolization of the fistula and remains free of symptoms 1 year after treatment. # 2002 Elsevier Science Ireland Ltd. All rights reserved. Keywords: Pulsatile tinnitus; Dural arteriovenous fistula; Neck compression
1. Introduction
2. Case report
The causes of vascular pulsatile tinnitus may be divided into two broad groups based on etiology*/ arterial and venous [1]. On physical examination, evaluation of tinnitus changes elicited with neck compression and head rotation is useful for predicting the cause of the tinnitus. In patients with venous etiology tinnitus, light digital compression of the ipsilateral neck and full head rotation toward the side of the tinnitus result in a decrease or complete cessation of tinnitus. Compression of the opposite neck and head rotation toward the contralateral side have the opposite effect; increased tinnitus intensity. In patients with arterial etiology tinnitus, these maneuvers produce no change in tinnitus intensity [2]. In this paper, we describe a case of pulsatile tinnitus arising from a dural arteriovenous fistula that changed intensity with neck compression of the uninvolved side.
A 58-year-old man presented with right-sided pulsatile tinnitus and sleep disturbance for the previous 10 months. He had been diagnosed with pulsatile tinnitus originating from a high jugular bulb after CT scan at another hospital. His sleep disturbance was aggravated after 8 months of clinical observation, so he was referred to our clinic for further evaluation and treatment. The tinnitus was described as constant and exacerbated by exercise. There was no history of head or neck trauma, cardiovascular disease or head surgery. He was otherwise in a good state of health. Otoscopic examination revealed no middle ear abnormalities. Very low intensity sound could be heard on placing a Toynbee tube in the external auditory canal in an audiometric testing booth. Interestingly, the pulsatile tinnitus was eliminated by gentle digital compression of the left (contralateral) jugular vein and head rotation to the left side. Otherwise, the tinnitus was not changed by ipsilateral neck compression. Pure-tone audiometry, tympanometry, fundoscopy, CBC, EKG and thyroid function test were performed. All tests were normal. A high-resolution CT scan of the temporal bones showed bilateral high jugular bulbs without evidence of bony erosion or dehiscence. A brain MRI study revealed no intracranial or intratemporal lesions, or vascular malformations.
* Corresponding author. Tel.: /82-2-2270-0070; fax: /82-2-22700073 E-mail address: [email protected] (B.-H. Jun).
0385-8146/02/$ - see front matter # 2002 Elsevier Science Ireland Ltd. All rights reserved. PII: S 0 3 8 5 - 8 1 4 6 ( 0 2 ) 0 0 1 0 7 - 4
90
B.-H. Jun et al. / Auris, Nasus, Larynx 30 (2003) 89 /91
Fig. 1. Angiographic study. Right CCA injection (lateral projection) revealing a small dural AVF (arrow) draining anteriorly into the jugular bulb.
Fig. 2. Selective angiogram obtained at the site of the occipital artery, revealing the dural AVF (arrow) fed by the stylomastoid branch.
Carotid angiography revealed a small dural arteriovenous fistula of the right jugular bulb, fed by the stylomastoid branch of the occipital artery (Figs. 1 and 2. The venous outflow was normal, without restriction or retrograde drainage. The fistula was occluded by transarterial coil embolization of the feeding artery (Fig. 3). After embolization, the patient experienced immediate relief of tinnitus. One year later, the patient remained free of symptoms.
3. Discussion
Fig. 3. Right ECA angiogram obtained after transarterial coil embolization (arrow) of the DAVF revealing complete obliteration of the fistula.
Vascular pulsatile tinnitus is the result of blood turbulence generated by increased flow volume, luminal stenosis or high flow shunts [1]. History and physical examination are very important in evaluating patients
B.-H. Jun et al. / Auris, Nasus, Larynx 30 (2003) 89 /91
with complaints of pulsatile tinnitus. Neck compression and head rotation tests, in particular, can provide clues for differentiating between an arterial or venous origin for tinnitus. In cases of venous origin, gentle neck compression and head rotation result in changes of venous volume, so effects on the intensity of the tinnitus are noted. Conversely, it is well known that changes of jugular venous volume have no effects on arterial tinnitus. Consequently, there have been no reported cases of tinnitus symptoms being altered by neck compression in patients with arterial pulsatile tinnitus. In our case, contralateral neck compression completely eliminated the patient’s tinnitus symptoms. We postulate a mechanism whereby increased venous flow of the ipsilateral jugular vein, resulting from contralateral neck compression, reduces shunt flow. Lower shunt flow volume, resulting from a reduced pressure gradient across the fistula, results in an elimination of tinnitus. Paraganglioma, dural arteriovenous fistula, idiopathic venous hum and benign intracranial hypertension are believed to be common causes of vascular tinnitus [1]. Abnormal jugular veins, such as high jugular bulb [3], jugular megabulb [4] and jugular bulb diverticulum [5] have also been reported as causes of pulsatile tinnitus. DAVFs are rare, accounting for only 10 /15% of all intracranial AV malformations. The most frequent sites of involvement include the dura surrounding the sigmoid and transverse sinuses. Uncommonly, the areas of the cavernous sinus and jugular foramen are involved [6]. Pulsatile tinnitus is the most frequent complaint of DAVF patients, being particularly common in sigmoid and transverse sinus fistula cases. Although many authors have reported DAVFs presenting as pulsatile tinnitus, they have not reported that this symptom could be altered or eliminated by neck compression. DAVF is a dynamic disease that can progress from minor venous restriction to severe venous outflow obstruction associated with significant morbidity and death. Failure or delay in the diagnosis, or misdiagnosis such as benign intracranial hypertension or high jugular bulb, can have catastrophic consequences. In our case, if we had not given attention to the unusual finding that the tinnitus intensity changed during contralateral neck compression, we would have ascribed the tinnitus to the high jugular bulb incidentally demonstrated on CT scan. In pulsatile tinnitus patients with normal otoscopy, Shah suggested that early angiography should be demanded because CT scanning is inaccurate in diagnosing DAVF [6].
91
Lalwani graded DAVFs based on the presence or absence of cortical venous drainage and antegrade venous drainage through the ipsilateral sigmoid sinus and the jugular bulb [7]. Our case is consistent with Lalwani’s Grade 1. DAVFs can develop from trauma, infection and intracranial surgery, but are often idiopathic. DAVFs have been managed in many ways, such as clinical observation, manual carotid compression, endovascular therapy and direct surgery [8,9]. Endovascular technique has been widely used as a first treatment of DAVFs via either a transarterial or transvenous route [10]. Grade I DAVFs are considered to have benign behavior, so carotid compression can be attempted as initial therapy. More aggressive treatment, however, is indicated if symptoms are progressive, the patient wishes to be relieved of intolerable tinnitus or compression therapy fails. In conclusion, this case demonstrates that pulsatile tinnitus originating from a small DAVF can be reduced in intensity by compression of the contralateral neck. In such a case, early angiography is recommended for accurate diagnosis and optimal treatment.
References [1] Sismanis A, Smoker WRK. Pulsatile tinnitus: recent advances in diagnosis. Laryngoscope 1994;104:681 /8. [2] Jastreboff PJ, Gray WC, Mattox DE. Tinnitus and hyperacusis. In: Cummings CW, Fredrickson JM, Harker LA, Krause CJ, Richardson MA, Schuller DE, editors. Otolayngology head and neck surgery. St. Louis, MI: Mosby, 1998:3198 /222. [3] Adler JR, Ropper AH. Self-audible venous bruit and high jugular bulb. Arch Neurol 1986;43:257 /9. [4] Buckwalter JA, Sasaki CT, Virapongse C, Kier EL, Bauman N. Pulsatile tinnitus arising from jugular megabulb deformity: a treatment rationale. Laryngoscope 1983;93:1534 /9. [5] Pappas DG, Jr, Hoffman RA, Cohen NL. Petrous jugular malposition (diverticulum). Otolaryngol Head Neck Surg 1993;109:847 /52. [6] Shaf SB, Lalwani AK, Dowd CF. Transverse/sigmoid sinus dural arteriovenous fistula presenting as pulsatile tinnitus. Laryngoscope 1999;109:54 /8. [7] Lalwani AK, Dowd CF, Halbach VV. Grading venous restrictive disease in patients with dural arteriovenous fistulas of the transverse/sigmoid sinus. J Neurosurg 1993;79:11 /5. [8] Landman JA, Braun IF. Spontaneous closure of a dural arteriovenous fistula associated with acute hearing loss. AJNF 1985;6:448 /9. [9] Halbach VV, Higashida RT, Heishima GB, Goto K, Norman K, Newton T. Dural fistulas involving the transverse and sigmoid sinuses: results of treatment in 28 patients. Radiology 1987;163:443 /7. [10] Ohtakara K, Murao K, Kawaguchi K, Kuga Y, Kojima T, Taki W, Waga S. Selective transvenous liquid embolization of a type 1 dural arteriovenous fistula at the junction of the transverse and sigmoid sinuses. J Neurosurg 2000;92:1045 /9.
Pulsatile tinnitus alleviated by contralateral neck compression: a case report Byung-Hoon Jun a,*, Ick-Soo Choi a, Ghi-Jai Lee b a
Department of Otolaryngology, Seoul Paik Hospital, Inje University College of Medicine, Jur-Dong 2ka, 85 Choong-Ku, Seoul 100-032, South Korea b Department of Radiology, Seoul Paik Hospital, Inje University College of Medicine, Seoul 100-032, South Korea Received 18 May 2002; accepted 20 September 2002
Abstract A case of a 58-year-old man with right pulsatile tinnitus originating from a small dural arteriovenous fistula (DAVF) of the jugular bulb is described. The tinnitus was alleviated by contralateral neck compression. This unusual observation ruled out venous pulsatile tinnitus, although a temporal bone CT scan showed a high jugular bulb. The fistula was confirmed by angiographic study. The patient was treated by transarterial embolization of the fistula and remains free of symptoms 1 year after treatment. # 2002 Elsevier Science Ireland Ltd. All rights reserved. Keywords: Pulsatile tinnitus; Dural arteriovenous fistula; Neck compression
1. Introduction
2. Case report
The causes of vascular pulsatile tinnitus may be divided into two broad groups based on etiology*/ arterial and venous [1]. On physical examination, evaluation of tinnitus changes elicited with neck compression and head rotation is useful for predicting the cause of the tinnitus. In patients with venous etiology tinnitus, light digital compression of the ipsilateral neck and full head rotation toward the side of the tinnitus result in a decrease or complete cessation of tinnitus. Compression of the opposite neck and head rotation toward the contralateral side have the opposite effect; increased tinnitus intensity. In patients with arterial etiology tinnitus, these maneuvers produce no change in tinnitus intensity [2]. In this paper, we describe a case of pulsatile tinnitus arising from a dural arteriovenous fistula that changed intensity with neck compression of the uninvolved side.
A 58-year-old man presented with right-sided pulsatile tinnitus and sleep disturbance for the previous 10 months. He had been diagnosed with pulsatile tinnitus originating from a high jugular bulb after CT scan at another hospital. His sleep disturbance was aggravated after 8 months of clinical observation, so he was referred to our clinic for further evaluation and treatment. The tinnitus was described as constant and exacerbated by exercise. There was no history of head or neck trauma, cardiovascular disease or head surgery. He was otherwise in a good state of health. Otoscopic examination revealed no middle ear abnormalities. Very low intensity sound could be heard on placing a Toynbee tube in the external auditory canal in an audiometric testing booth. Interestingly, the pulsatile tinnitus was eliminated by gentle digital compression of the left (contralateral) jugular vein and head rotation to the left side. Otherwise, the tinnitus was not changed by ipsilateral neck compression. Pure-tone audiometry, tympanometry, fundoscopy, CBC, EKG and thyroid function test were performed. All tests were normal. A high-resolution CT scan of the temporal bones showed bilateral high jugular bulbs without evidence of bony erosion or dehiscence. A brain MRI study revealed no intracranial or intratemporal lesions, or vascular malformations.
* Corresponding author. Tel.: /82-2-2270-0070; fax: /82-2-22700073 E-mail address: [email protected] (B.-H. Jun).
0385-8146/02/$ - see front matter # 2002 Elsevier Science Ireland Ltd. All rights reserved. PII: S 0 3 8 5 - 8 1 4 6 ( 0 2 ) 0 0 1 0 7 - 4
90
B.-H. Jun et al. / Auris, Nasus, Larynx 30 (2003) 89 /91
Fig. 1. Angiographic study. Right CCA injection (lateral projection) revealing a small dural AVF (arrow) draining anteriorly into the jugular bulb.
Fig. 2. Selective angiogram obtained at the site of the occipital artery, revealing the dural AVF (arrow) fed by the stylomastoid branch.
Carotid angiography revealed a small dural arteriovenous fistula of the right jugular bulb, fed by the stylomastoid branch of the occipital artery (Figs. 1 and 2. The venous outflow was normal, without restriction or retrograde drainage. The fistula was occluded by transarterial coil embolization of the feeding artery (Fig. 3). After embolization, the patient experienced immediate relief of tinnitus. One year later, the patient remained free of symptoms.
3. Discussion
Fig. 3. Right ECA angiogram obtained after transarterial coil embolization (arrow) of the DAVF revealing complete obliteration of the fistula.
Vascular pulsatile tinnitus is the result of blood turbulence generated by increased flow volume, luminal stenosis or high flow shunts [1]. History and physical examination are very important in evaluating patients
B.-H. Jun et al. / Auris, Nasus, Larynx 30 (2003) 89 /91
with complaints of pulsatile tinnitus. Neck compression and head rotation tests, in particular, can provide clues for differentiating between an arterial or venous origin for tinnitus. In cases of venous origin, gentle neck compression and head rotation result in changes of venous volume, so effects on the intensity of the tinnitus are noted. Conversely, it is well known that changes of jugular venous volume have no effects on arterial tinnitus. Consequently, there have been no reported cases of tinnitus symptoms being altered by neck compression in patients with arterial pulsatile tinnitus. In our case, contralateral neck compression completely eliminated the patient’s tinnitus symptoms. We postulate a mechanism whereby increased venous flow of the ipsilateral jugular vein, resulting from contralateral neck compression, reduces shunt flow. Lower shunt flow volume, resulting from a reduced pressure gradient across the fistula, results in an elimination of tinnitus. Paraganglioma, dural arteriovenous fistula, idiopathic venous hum and benign intracranial hypertension are believed to be common causes of vascular tinnitus [1]. Abnormal jugular veins, such as high jugular bulb [3], jugular megabulb [4] and jugular bulb diverticulum [5] have also been reported as causes of pulsatile tinnitus. DAVFs are rare, accounting for only 10 /15% of all intracranial AV malformations. The most frequent sites of involvement include the dura surrounding the sigmoid and transverse sinuses. Uncommonly, the areas of the cavernous sinus and jugular foramen are involved [6]. Pulsatile tinnitus is the most frequent complaint of DAVF patients, being particularly common in sigmoid and transverse sinus fistula cases. Although many authors have reported DAVFs presenting as pulsatile tinnitus, they have not reported that this symptom could be altered or eliminated by neck compression. DAVF is a dynamic disease that can progress from minor venous restriction to severe venous outflow obstruction associated with significant morbidity and death. Failure or delay in the diagnosis, or misdiagnosis such as benign intracranial hypertension or high jugular bulb, can have catastrophic consequences. In our case, if we had not given attention to the unusual finding that the tinnitus intensity changed during contralateral neck compression, we would have ascribed the tinnitus to the high jugular bulb incidentally demonstrated on CT scan. In pulsatile tinnitus patients with normal otoscopy, Shah suggested that early angiography should be demanded because CT scanning is inaccurate in diagnosing DAVF [6].
91
Lalwani graded DAVFs based on the presence or absence of cortical venous drainage and antegrade venous drainage through the ipsilateral sigmoid sinus and the jugular bulb [7]. Our case is consistent with Lalwani’s Grade 1. DAVFs can develop from trauma, infection and intracranial surgery, but are often idiopathic. DAVFs have been managed in many ways, such as clinical observation, manual carotid compression, endovascular therapy and direct surgery [8,9]. Endovascular technique has been widely used as a first treatment of DAVFs via either a transarterial or transvenous route [10]. Grade I DAVFs are considered to have benign behavior, so carotid compression can be attempted as initial therapy. More aggressive treatment, however, is indicated if symptoms are progressive, the patient wishes to be relieved of intolerable tinnitus or compression therapy fails. In conclusion, this case demonstrates that pulsatile tinnitus originating from a small DAVF can be reduced in intensity by compression of the contralateral neck. In such a case, early angiography is recommended for accurate diagnosis and optimal treatment.
References [1] Sismanis A, Smoker WRK. Pulsatile tinnitus: recent advances in diagnosis. Laryngoscope 1994;104:681 /8. [2] Jastreboff PJ, Gray WC, Mattox DE. Tinnitus and hyperacusis. In: Cummings CW, Fredrickson JM, Harker LA, Krause CJ, Richardson MA, Schuller DE, editors. Otolayngology head and neck surgery. St. Louis, MI: Mosby, 1998:3198 /222. [3] Adler JR, Ropper AH. Self-audible venous bruit and high jugular bulb. Arch Neurol 1986;43:257 /9. [4] Buckwalter JA, Sasaki CT, Virapongse C, Kier EL, Bauman N. Pulsatile tinnitus arising from jugular megabulb deformity: a treatment rationale. Laryngoscope 1983;93:1534 /9. [5] Pappas DG, Jr, Hoffman RA, Cohen NL. Petrous jugular malposition (diverticulum). Otolaryngol Head Neck Surg 1993;109:847 /52. [6] Shaf SB, Lalwani AK, Dowd CF. Transverse/sigmoid sinus dural arteriovenous fistula presenting as pulsatile tinnitus. Laryngoscope 1999;109:54 /8. [7] Lalwani AK, Dowd CF, Halbach VV. Grading venous restrictive disease in patients with dural arteriovenous fistulas of the transverse/sigmoid sinus. J Neurosurg 1993;79:11 /5. [8] Landman JA, Braun IF. Spontaneous closure of a dural arteriovenous fistula associated with acute hearing loss. AJNF 1985;6:448 /9. [9] Halbach VV, Higashida RT, Heishima GB, Goto K, Norman K, Newton T. Dural fistulas involving the transverse and sigmoid sinuses: results of treatment in 28 patients. Radiology 1987;163:443 /7. [10] Ohtakara K, Murao K, Kawaguchi K, Kuga Y, Kojima T, Taki W, Waga S. Selective transvenous liquid embolization of a type 1 dural arteriovenous fistula at the junction of the transverse and sigmoid sinuses. J Neurosurg 2000;92:1045 /9.