Tips for Transient Perivascular Inflammation of the Carotid Artery Syndrome.

BACKGROUND: Idiopathic carotidynia, also known as transient perivascular inflammation of the carotid artery (TIPIC) syndrome, is a rare, self-limited, clinical-radiologic entity. Over the years, the diagnosis of carotidynia has been controversial, but recent pathologic, radiologic, clinical, and laboratory findings support an inflammatory etiology. CASE REPORT: A 61-year-old woman with a history of hypertension, left lower extremity liposarcoma, and right internal jugular port placement 2 weeks prior with initiation of chemotherapy presented to the emergency department with right neck pain and swelling of the lateral neck and lower face for the past 3 days. Computed tomography-neck with IV contrast revealed marked mural thickening of the right common carotid artery, which can be seen with carotidynia (Fay syndrome and TIPIC syndrome) and vasculitis. The patient had elevated inflammatory markers and was treated clinically for carotidynia with ibuprofen, evaluated by vascular surgery, and discharged home. WHY SHOULD AN EMERGENCY PHYSICIAN BE AWARE OF THIS?: The causes of acute neck pain are diverse, ranging from nonemergent to surgically emergent etiologies. As radiologists and emergency physicians, we believe TIPIC syndrome is a rare entity with important clinical impact deserving attention, as it is not typically included in medical training and is usually learned only through years of clinical experience and practice. TIPIC syndrome requires a unique combination of both clinical and radiologic findings to diagnose accurately and appropriately. It is important to be familiar with this diagnosis because treatment is focused on symptomatic relief without the need for invasive procedures. Our goal was to increase awareness of this uncommon diagnosis to improve patient care by preventing unnecessary invasive procedures and aid in timely and accurate diagnosis.

Transient perivascular inflammation of the carotid artery (TIPIC) syndrome - a rare differential for anterior neck pain - Series of 3 cases and review of literature.

We report a series of 3 cases of transient perivascular inflammation of the carotid artery (TIPIC) syndrome in an otherwise healthy individual. We would also like to review this rare entity and compare it with other similar cases reported in the literature. Our first case was a young male with right-sided neck pain of 1-week duration with magnetic resonance imaging (MRI) showing right carotid perivascular inflammation which completely resolved after 2 weeks with anti-inflammatory drugs. In the second case, a young male presented with left-sided neck pain and odynophagia of 5 days duration with an MRI showing left carotid perivascular inflammation which completely resolved after 2 weeks with anti-inflammatory drugs. In the third case a young male presented with right-sided neck pain of 1-day duration with an MRI showing right common carotid perivascular inflammation near the bifurcation with complete resolution in pain but with residual wall thickening. We want to highlight the existence of this new entity by reporting these 3 case series with a brief review of the literature. The cause and pathogenesis of this rare entity remain unknown. It has been hypothesized to be autoimmune or viral-mediated inflammation which requires further understanding.

Carotidynia: Overview of an uncommon identification for unilateral neck pain.

BACKGROUND: Carotidynia, also known as Fay Syndrome, manifests as an atypical facial neuralgia characterized by an unusual neck pain extending towards the head and associated with carotid artery tenderness. Diagnostic criteria include neck tenderness, elevated carotid pulse without anatomical abnormalities, and neck distension. It was initially classified as a vascular headache but later re-evaluated and reclassified as a nonentity-a general condition caused by nonvascular factors. The etiology has not been extensively elucidated. CASE PRESENTATION: We present two cases characterized by dysphagia, intermittent discomfort, and numbness in the throat and cervical region. Although the neurological examinations yielded no abnormalities, the diagnosis of carotidynia was ultimately established among the differential diagnoses upon the identification of wall thickening and inflammatory alterations through neuroimaging. CONCLUSIONS: Carotidynia is consistent with idiopathic vasculitis near the distal common carotid artery. Inflammatory processes trigger sympathetic plexus stimulation, causing discomfort in the head and neck. Neuroimaging resolves ambiguities in idiopathic unilateral neck pain, detecting soft tissue growth near the carotid artery. "Carotidynia" now refers to a diagnostic symptom and clinical entity encompassing a variety of disorders; therefore, approach to definition remains controversial. This report aims to raise healthcare awareness by highlighting two different cases of carotidynia.

Transient Perivascular Inflammation of the Carotid Artery as a Poorly Recognized Cause of Neck Pain.

Transient perivascular inflammation of the carotid artery (TIPIC) syndrome, historically named idiopathic carotidynia or Fay syndrome, is a rare condition characterized by inflammation and pain in the carotid artery. The diagnosis requires a specific clinical-radiological presentation. We describe a 37-year-old female who presented with headaches and left neck pain and was diagnosed with TIPIC syndrome with temporary perivascular infiltration.

Long-term clinical and ultrasound follow-up after transient perivascular inflammation of the carotid artery (TIPIC) syndrome: a multicenter study.

BACKGROUND: The present study aimed to describe the clinical and ultrasound (US) long-term follow-up of patients with transient perivascular inflammation of the carotid artery (TIPIC) syndrome and the risk of recurrence. METHODS: We enrolled patients with a definitive diagnosis of TIPIC syndrome who were included in a retrospective multicenter study. These patients were recontacted at least six months after the first TIPIC episode for a clinical and imaging follow-up. Each patient underwent a clinical evaluation through a tailored questionnaire as well as US imaging. RESULTS: Twenty-eight patients were enrolled with a median follow-up of 58.7 months (interquartile range = 8-121). Nineteen out of the 28 patients (67.8%) had residual pain, eight (28.6%) had experienced a clinical recurrence and 12 (42.9%) had a thickening of the carotid wall on US. No patients had neurological complication or other associated diseases. CONCLUSIONS: Patients with TIPIC syndrome have often residual pain and recurrence in about one quarter of cases but the long-term follow-up is in favor a benign self-limited pathology.Trial registration: ClinicalTrials.gov (identifier NCT03804112).

Transient perivascular inflammation of the carotid artery-A transient but potentially recurrent disease.

Transient perivascular inflammation of the carotid artery (TIPIC syndrome) is a rare, unclassified vascular syndrome that usually affects the distal part of the common carotid artery and has a favorable prognosis. The disease is often misdiagnosed even by neurologists due to the moderate intensity of clinical symptoms and their transient character. We present a case of a 52-year-old man who experienced two episodes of transient neck pain and moderate local tenderness one and a half years apart. Different imaging modalities, such as ultrasound, CT angiography, and high-resolution 3T MR, were applied to better visualize the perivascular inflammation of the common carotid arteries. Based on the clinical-radiological characteristics of our case and applying the diagnostic criteria, we established the diagnosis of TIPIC syndrome. The patient was treated with nonsteroidal anti-inflammatory drugs for few weeks, and reduction in perivascular changes was observed in both episodes. The case raises questions about the phases of the disease, its duration and the intervals between follow-ups. Our article also increases the awareness of this rare clinical-radiological entity and presents recent data from the literature.

G-CSF-induced TIPIC syndrome and large vessel vasculitis: A case report.

Key Clinical Message: We report a rare adverse event of transient perivascular inflammation of the carotid artery syndrome induced by granulocyte colony-stimulating factor injections. Recognition of this syndrome is important for physicians, to avoid the exposure of the causative medication, rule out differential diagnosis and delay the use of corticosteroids given the spontaneous improvement after discontinuation of the causative medication. Abstract: A 73 year-old Caucasian woman presented with odynophagia, carotidynia, and fever 5 days following a granulocyte colony-stimulating factor (G-CSF) injection for chemotherapy-induced neutropenia in the setting of myelodysplastic syndrome. Examination showed painful swelling of the neck. Lab results showed inflammation with CRP 328 mg/L. A CT-scan revealed tissue infiltration thickening surrounding the left internal carotid artery, the carotid bifurcation, and the common carotid artery, as well as circumferential thickening of the aortic arch. Ultrasound of the left internal carotid artery found isoechoic wall thickening. Symptoms drastically improved without steroids in a short time period. Horton's disease, Takayasu's diseases, and infectious vasculitis were not retained due to the short time delay of symptoms onset, atypical echogenicity, and spontaneous improvement. A diagnosis of G-CSF-induced large vessel vasculitis transient perivascular inflammation of the carotid artery (TIPIC) syndrome was made. Seven days later, ultrasound control showed diminished thickening infiltration. G-CSF TIPIC is a rare adverse event that should be kept in mind in patients under G-CSF.

Takayasu arteritis presenting with fever of unknown origin and bilateral carotid artery tenderness: A case report.

A 51-year-old otherwise healthy woman was referred to our hospital with a fever of unknown origin, liver dysfunction, and anemia. One month prior, she had persistent and spontaneous anterior neck pain, with no exacerbation during swallowing or neck movements. Physical examination revealed no pharyngeal or tonsillar abnormalities, heart murmur, arthritis, skin rash, or lymphadenopathy, except for mild bilateral common carotid artery tenderness at the level of the thyroid cartilage. Blood tests showed nonspecific chronic inflammatory findings, anemia, and liver damage, whereas blood cultures, viral antibodies, interferon-γ release assay, and antibodies specific for any collagen disease showed negative results. Echocardiography and computed tomography without contrast of the neck, chest, abdomen, and pelvis showed no apparent abnormalities. She was subsequently diagnosed with Takayasu arteritis using positron emission tomography. Identifying a characteristic history of bilateral carotid artery tenderness and subsequent positron emission tomography can be useful for diagnosing Takayasu arteritis.

Transient Perivascular Inflammation of the Carotid Artery (TIPIC) Syndrome: An Atypical Cause of Neck Pain.

Transient perivascular inflammation of the carotid artery (TIPIC) is an uncommon condition characterized by inflammation of the carotid artery wall, leading to unilateral neck pain. While TIPIC has been acknowledged by the International Classification of Headache Disorders, only a few patient series have been published thus far. The clinical presentation of TIPIC syndrome typically manifests as unilateral neck pain localized specifically over the carotid artery. This pain is accompanied by ipsilateral tenderness and increased arterial pulsation. The condition commonly follows a self-limited course or demonstrates a favorable response to treatment with nonsteroidal anti-inflammatory drugs. When evaluating patients with suspected TIPIC syndrome, conducting a comprehensive assessment of their clinical history is imperative, while utilizing imaging studies to exclude any potential structural abnormalities of the carotid artery effectively. The authors present a case involving a 57-year-old woman who presented with a two-month history of persistent left cervical pain and tenderness. Ultrasonography findings revealed indirect indications of inflammation in the intima-media layer of the carotid artery, suggestive of carotidynia. Notably, other significant differential diagnoses such as aneurysms or carotid dissection were ruled out. Over the course of the evaluation, there was a gradual and spontaneous improvement in both clinical symptoms and radiological findings, indicating the resolution of the inflammatory process as confirmed by imaging follow-up. This case presents a rare and atypical manifestation of transient neck pain attributed to TIPIC.

"TransIent perivascular inflammation of the carotid artery (TIPIC) syndrome" as a rare case of laterocervical pain: Multimodal diagnosis.

''TransIent Perivascular Inflammation of the Carotid artery (TIPIC) syndrome" is an unusual cause of unilateral neck pain, due to a nonspecific inflammation of the carotid artery. This entity has been for long known as "carotidynia" and described as a syndrome rather than a distinct pathologic entity. Recently, the presence of structural abnormalities of the carotid artery wall has been demonstrated, leading to the introduction of radiological criteria which, in the appropriate clinical context, allow to diagnose TIPIC syndrome. TIPIC syndrome is a rather rare disease and, since its first description by Fay in 1927, only a small series of patients have been published. The interest of our case lies in the fact that diagnosis and follow-up were assessed on ultrasound and magnetic resonance imaging, demonstrating that a correlation between clinical evolution and radiological findings does exist. In addition, DWI sequence was performed at the time of diagnosis and at resolution. To our knowledge, such an assessment has never been reported in the previous literature.

Mycotic internal carotid artery pseudoaneurysm secondary to Mycobacterium tuberculosis.

Mycobacterium tuberculosis is a rare causative agent for mycotic aneurysms of the extracranial carotid arteries. We describe a case of acute mycotic pseudoaneurysm and abscess in the right proximal internal carotid artery in close proximity to the carotid bifurcation, and subsequent management with antibiotic therapy, surgical debridement and resection with an end-to-end anastomosis.

Transient perivascular inflammation of the carotid artery (TIPIC) syndrome.

Background: The Transient Perivascular Inflammation of the Carotid artery (TIPIC) syndrome is presumably a very rare disease characterized by a local transient inflammation of the tissue around the carotid artery. Its pathophysiology remains unknown. We performed an updated study of TIPIC syndrome cases in the setting of a multinational collaborative study. Methods: This study was conducted as an observational multinational retrospective individual patient level cohort study. Information from all known cases diagnosed with TIPIC syndrome in the literature (2005-2020) was collected after a semi-structured literature search of PubMed and Web of Science. We also collected unpublished information of patients from French, Swiss, and Italian vascular medicine or radiology departments. Results: A total of 72 patients were included and served for data analysis: 42 (58.3%) were women; the mean age was 47.9 (SD=11.4) years. Symptoms were unilateral in 92% of patients and 81.4% required pain killers. At baseline, irrespective of the imaging method used, the median thickness of the carotid lesions was 5 (Q1-Q3: 4-7; range: 2-11) mm and the median length of the lesion was 20 (Q1-Q3: 10-30; range: 3-50) mm. We found a positive linear correlation between thickness and length. At follow-up, the thickness of the carotid lesions decreased to a median of 2 (Q1-Q3: 1-3; range: 0-6) mm; the length decreased to a median 10 (Q1-Q3: 5-15; range: 0-41) mm. A linear correlation between baseline and follow-up values was observed for both thickness and length measurements. Symptoms disappeared after a median of 14 (Q1-Q3: 10-15) days. Thirteen patients experienced a recurrence after a median follow-up of 6 (Q1-Q3: 2-12) months. Conclusions: The present analysis elucidates clinical and sonographic characteristics of TIPIC syndrome, indicating the benign nature of this condition. A future international registry will study the long-term course of the disease.

Multimodal imaging features of transient perivascular inflammation of the carotid artery (TIPIC) syndrome in a patient with Covid-19.

We report the case of a 38-year-old man with transient perivascular inflammation of the carotid artery syndrome that occurred in the course of covid-19. We describe for the first-time multimodal imaging features of the perivascular changes surrounding the carotid artery, and long-term follow-up by ultrasound. The imaging features observed on ultrasound, angiography-CT, MRI and FDG-Pet scan support the hypothesis of the inflammatory nature of the perivascular tissue thickening. The ultrasound follow-up confirmed the spontaneous resolution of the lesion, leaving on site some residual changes as sequelae. The good knowledge of the imaging features reported herein helps to recognize this entity in patients with covid-19.

TIPIC-A Newly Recognized Syndrome: Multimodality Imaging of a Rare Clinicoradiological Entity.

Carotidynia is a controversial clinicopathological entity vastly described in Western literature as neck pain in the region of carotid bifurcation secondary to an underlying inflammatory etiology. Radiologically, this appears as perivascular inflammation and has recently been designated as transient perivascular inflammation of the carotid artery (TIPIC) syndrome. The authors of this report discuss the multimodality imaging features of a rare case of this disease in our country to familiarize radiologists with the imaging findings and to encourage the inclusion of TIPIC syndrome as a differential diagnosis for focal neck pain.

Granulocyte-Colony Stimulating Factor-Induced Vasculitis Successfully Treated With Short-Term Corticosteroid Therapy: A Case Report.

Granulocyte-colony stimulating factor (G-CSF) is widely used for preventing neutropenia, and large vessel vasculitis has been recognized as one of its severe adverse events. We report a case of diffuse large B-cell lymphoma in a 78-year-old woman in whom fever and right cervical pain developed after administration of filgrastim. Computed tomography and cervical artery ultrasound imaging revealed wall thickening in the right common carotid artery. We diagnosed her with G-CSF-induced vasculitis and administered prednisolone of 50 mg/day (1 mg/kg/day) to her. Her symptoms disappeared in a few days, and prednisolone was discontinued six weeks after initiation. G-CSF-induced vasculitis may be improved with short-term high-dose corticosteroids with rapid tapering.

Myocarditis and carotidynia caused by Granulocyte-Colony stimulating factor administration.

A 59 year-old woman was treated with adjuvant chemotherapy for triple negative breast cancer (TNBC) stage IB. She received pegfilgrastrim as secondary prophylaxis of neutropenia. After administration of pegfilgrastrim on day 11, she was hospitalised because of carotidynia and myocarditis that improved with antibiotics and steroids as an infection was suspected. Once she was recovered, another cycle of chemotherapy with pegfilgrastrim was administrated. At this time, the patient presented to our hospital with fever, odynophagia and chest pain, with diagnosis of myocarditis coupled with cardiogenic shock. She received antibiotics and steroids, advanced life support and also a pericardial window was done, with recovery of her condition. After a complete evaluation and exclusion of other possible aetiologies, we concluded that pegfilgrastrim was responsible for inducing carotidynia and myocarditis. Few cases have been published about Granulocyte-Colony stimulating factor (G-CSF) induced carotidynia and aortitis. However, this is the first reported case about G-CSF induced myocarditis and carotidynia.

Imaging Spectrum of TIPIC Syndrome : Validation of a new Entity with Vessel Wall Imaging.

PURPOSE: Acute neck pain can have non-vascular and vascular causes. Some patients present with distinct vascular and perivascular changes on imaging at the site of tenderness. This study aimed to evaluate the imaging findings of transient perivascular inflammation of the carotid artery (TIPIC) syndrome with an emphasis on vessel wall imaging using 3­Tesla (3-T) high-resolution (HR) magnetic resonance imaging (MRI). METHODS: Clinical data along with diagnostic and follow-up imaging of patients presenting to these hospitals with acute neck pain/tenderness and at least 1 imaging study using color Doppler ultrasound (CDU) and/or MRI including vessel wall imaging from September 2013 through September 2017 were retrospectively evaluated. A total of 15 patients with no other underlying cause of pain, findings meeting the imaging criteria for TIPIC syndrome and clinical recovery (spontaneous or with treatment) were included in the study. RESULTS: The mean patient age was 43.2 years. With CDU and precontrast MRI, perivascular inflammation (PVI) of the involved artery segment was evident in all patients. Contrast enhancement of the adventitia and PVI were noted on postcontrast HR vessel wall MRI in all patients. Of the patients five had co-existing plaques at the site of tenderness. Follow-up imaging demonstrated pronounced regression or complete resolution of the findings. CONCLUSION: Imaging is useful for the establishment of TIPIC syndrome diagnosis and to rule out other conditions. The use of CDU is usually sufficient for diagnosis and follow-up but in clinically doubtful and complicated cases, vessel wall imaging with HR-MRI is very valuable. Thorough knowledge of this entity among radiologists enables a prompt diagnosis, which accelerates the clinical management.

Pathology of the carotid space.

The complex anatomy of the carotid space within a small confined area is unique to the head and neck and allows for a vast array of pathology. This paper will review the anatomy of the carotid space from the skull base to the thorax, defining its borders at multiple levels, as well as its contents. The paper will also describe the myriad of mass lesions and vascular pathologies that may occur within the carotid space. The discussion will include anatomic considerations in differential diagnoses, imaging features, and lesion characteristics across multiple imaging modalities including CT, MRI, ultrasound, and conventional angiography. Entities discussed include paragangliomas, nerve sheath tumors, meningioma, fibromuscular dysplasia, carotidynia, thrombus, dissection, pseudoaneurysm, and pathology of the deep cervical chain lymph nodes. Understanding the complex and unique anatomy of the carotid space, as well as the nuances of navigating a broad differential, will empower the reader to make an accurate diagnosis.