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Is Carotidynia Syndrome a Subset of Vasculitis? To the Editor: Carotidynia is the syndrome characterized by unilateral neck pain initially described by Fay1 in 1927. The clinical finding is tenderness over the carotid bifurcation and pain aggravated by movement of the neck without structural abnormality2. The symptom has a short duration of about 2 to 4 weeks, suggesting self-limited disease. The occurrence and cause of carotidynia are unknown. We describe the case of a 74-year-old woman with diabetes mellitus, hyperlipidemia, and hypothyroidism who developed carotidynia. She consulted us complaining of acute pain and swelling at the left side of her neck and stiffness from the left side of her neck to shoulder in October 2007. She had appeared a few days earlier without any previous infectious symptoms or trauma. The physical findings showed tenderness on palpation from her left carotid bifurcation to internal carotid artery, which was swollen about 2 cm. Her body temperature was 36.3°C. The presence of a carotid bruit was unclear. Laboratory findings showed normal peripheral blood cell counts. High-sensitivity C-reactive protein (hs-CRP) was slightly elevated at 0.367 mg/dl (0.046 mg/dl at 6 mos previously) and serum amyloid A protein (SAA) was slightly elevated at 10.5 µg/ml (normal range is less than 8.0 µg/ml). Hepatitis B surface-antigen and hepatitis C virus-antibody tests were negative. ELISA for myeloperoxidase-antineutrophil cytoplasmic antibodies (ANCA) and PR3-ANCA were negative. Ultrasonography (US) of the carotid artery showed a hypoechoic wall thickening of the proximal left internal carotid artery (Figure 1A). Wall thickening was found in 2 different layers of the vessel wall with slight narrowing of the affected vessel. Magnetic resonance imaging (MRI) of the carotid artery showed the following findings: (1) mildly increased intensity in internal carotid sheath was seen on T2-weighted images, (2) fat-suppressed T2-weighted MRI and gadolinium-enhanced T1-weighted MRI showed homogeneous enhancement of the left internal carotid wall including soft tissue around arterial wall (Figures 2A, B), and (3) MR angiogram showed no significant carotid lumen stenosis (image not shown). According to the criteria for carotidynia as defined by the International Headache Society (IHS) classification of 19882, we diagnosed her condition as carotidynia. She was treated with a nonsteroidal antiinflammatory agent for 2 weeks. Her neck pain gradually diminished and was resolved 2 weeks after the onset. Four weeks later, her ultrasonography test showed significantly fewer abnormal findings (Figure 1B). Further, the levels of hs-CRP and SAA were also reduced to 0.020 mg/dl and 2.5 µg/ml, respectively.
Carotidynia indicates not only unilateral neck pain but also various symptoms including headache, sore throat, jaw claudication, dysphagia, malaise, nasal congestion, and lacrimation. We should consider the following diseases and syndromes as differential diagnoses: arterial dissection, cervical disk disease, cluster headaches, myofascial pain syndrome, giant cell arteritis, acute pharyngitis, peritonsillar abscess, acute sinusitis, thyroiditis, sialolithiasis, mononucleosis, and cervical adenitis. In our case, these diseases were ruled out. On the other hand, the second International Headache Society classification in 2004 no longer refers to carotidynia as an independent pathological entity3. That is, they concluded that carotidynia was a not validated entity, but a syndrome of a unilateral neck pain. Currently, the view prevails that the conventional concept of carotidynia as having no structural abnormality should be removed from the previous classification system4. These findings of carotidynia on US, computerized tomography (CT), and MRI examinations have recently been reported4-7. In our case, the findings on US and MRI were compatible with the findings of previous reports. The positive inflammatory findings have not been previously described in carotidynia. However, nonspecific vascular inflammation has been demonstrated histologically in carotidynia8 and both hs-CRP and SAA levels were slightly elevated in the far-advanced stage of carotidynia in our case, indicating the existence of acute inflammation. Indeed, the measurement of hs-CRP is beginning to be considered as a trivial inflammatory marker of arteriosclerosis, and relative risk of cardiovascular event is associated with increase of hs-CRP. That is, hs-CRP > 0.2 mg/dl is a high risk factor of cardiovascular event9. Our most important finding was that the changes of symptoms and images and those of inflammatory findings of hs-CRP and SAA were parallel. Recently, a new disorder concept of "idiopathic carotiditis" was reported7. These authors proposed new criteria and our case fulfilled their criteria. Considering the above, our findings support the hypothesis that carotidynia could be a distinct disease entity, caused by inflammation. In other words, this means that carotidynia may be a subset of vasculitis. We emphasize the importance of measuring hs-CRP and SAA as characteristics of disease activity in patients with carotidynia. Carotidynia is an age-old disease entity and may be more prevalent than previously thought. We must investigate further cases of carotidynia in order to elucidate its pathophysiology and mechanisms of development, and the association between carotidynia and vasculitis. YOSHINORI TANIGUCHI, MD; TARO HORINO, MD, PhD; KOZO HASHIMOTO, MD, PhD, Department of Endocrinology, Metabolism and Nephrology, Kochi Medical School, Kochi University, Kohasu, Oko-cho, Nankoku, Kochi 783-8505, Japan. Address reprint requests to Dr. Taniguchi. E-mail: jm-yoshii@kochi-u.ac.jp 2. Headache Classification Committee of the International Headache Society. Classification and diagnosis criteria for headache disorders, cranial neuralgias and facial pain. Cephalalgia 1988;8 Suppl:48-9. 3. Second Headache Classification SubCommittee of the International Headache Society. Classification and diagnosis criteria for headache disorders, cranial neuralgias and facial pain. Cephalalgia 2004;24 Suppl:70-1. 4. Kuhn J, Harzheim A, Horz R, Bewermeyer H. MRI and ultrasonographic imaging of a patient with carotidynia. Cephalalgia 2006;26:483-5. [MEDLINE] 5. Arning C. Ultrasonography of carotidynia. AJNR 2005;26:201-2. [MEDLINE] 6. Michael PB, Mark CD. Carotidynia. AJR 2001;177:947. [MEDLINE] 7. Tardy J, Pariente J, Nasr N, et al. Carotidynia: a new case for an old controversy. Eur J Neurol 2007;14:704-5. [MEDLINE] 8. Upton PD, Smith JG, Charnock DR. Histologic confirmaion of carotidynia. Otolaryngol Head Neck Surg 2003;129:443-4. [MEDLINE] 9. Ridker PM. High-sensitivity C-reactive protein. Circulation 2001;103:1813-8. [MEDLINE]
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