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A « tipical » but rare cause of neck pain

  • Quentin Richier
    Correspondence
    Corresponding author at: Felix Guyon University Hospital Center, 75012 Paris, France.
    Affiliations
    Internal medicine department, Felix Guyon University hospital, Saint-Denis, La Réunion, France
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  • Jean-Yves Travers
    Affiliations
    Radiology department, Felix Guyon University hospital, Saint-Denis, La Réunion, France
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  • Loïc Raffray
    Affiliations
    Internal medicine department, Felix Guyon University hospital, Saint-Denis, La Réunion, France

    Université de La Réunion, CNRS 9192, INSERM U1187, IRD 249, Unité Mixte Processus Infectieux en Milieu Insulaire Tropical (PIMIT), Plateforme Technologique CYROI, Sainte-Clotilde, La Réunion, France
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Published:August 16, 2022DOI:https://doi.org/10.1016/j.ejim.2022.08.016

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      1. Case description

      A 34-year-old man with medical history of familial deafness presented to the emergency department for an acute left cervicalgia and painful jaw for one week. He reported a context of recent physical effort because of house moving. He had already performed a cervical echography, which showed a thickening of the wall of the left common carotid artery (panel A) without disturbance of the vascular flow on Doppler (panel B). At presentation, vital parameters were normal with symmetrical blood pressure measurement, and no fever. Clinical examination, in particular neurological examination, was normal. Electrocardiogram was unremarkable. Biological tests showed a normal C-reactive protein value (4.4 mg/L), normal troponin, and search was negative for anti-neutrophil cytoplasmic antibodies, anti-nuclear antibodies (titer at 1:80) and anti-DNA antibodies. Finally, cervical angio-CT-scan eliminated a carotid dissection and revealed a perivascular infiltration surrounding the left common carotid artery with a thickened carotid wall (panel C). All these elements were suggestive of transient perivascular inflammation of the carotid artery (TIPIC) syndrome, also known as carotidynia or Fay syndrome (Image).
      Image
      ImageThickened wall (white two-way arrows) of the left communal carotid artery compared to normal carotid thickness (blacks arrows) on echography (A). No disturbance of the carotid vascular flow on Doppler (B). Fat peri-vascular infiltration surrounding the left communal carotid on cervical angio-computed tomography scan (white one-way arrows, C).

      2. Discussion

      Fay first described carotidynia in 1927 as an atypical neuralgia. Historically characterized by the association of unspecific clinical signs like tenderness and pain at the level of the carotid bifurcation, this entity was controversial and it is still unrecognized as a distinct pathology [
      • Lecler A
      • Obadia M
      • Savatovsky J
      • Picard H
      • Charbonneau F
      • Menjot de Champfleur N
      • et al.
      TIPIC syndrome: beyond the myth of carotidynia, a new distinct unclassified entity.
      ]. A recent multi-centric cases series of 47 patients proposed a new definition based on four criteria: a) presence of acute pain overlying the carotid artery, which may or may not radiate to the head, b) eccentric perivascular infiltration on imaging, c) exclusion of another vascular or non-vascular diagnosis with imaging, d) improvement within 14 days either spontaneously or with anti-inflammatory treatment [
      • Lecler A
      • Obadia M
      • Savatovsky J
      • Picard H
      • Charbonneau F
      • Menjot de Champfleur N
      • et al.
      TIPIC syndrome: beyond the myth of carotidynia, a new distinct unclassified entity.
      ]. The pathogenesis of the TIPIC syndrome remains unclear, cardiovascular, inflammatory or autoimmune origins are suspected [
      • Taniguchi Y.
      • Horino T.
      • Hashimoto K.
      Is carotidynia syndrome a subset of vasculitis?.
      ]. The two major differential diagnosis for carotid involvement are carotid dissection and vasculitis, notably Takayasu's arteritis or giant cell arteritis. Only one study reported a histologically evaluation and found proliferation of small blood vessels, low-grade of lymphocytic infiltration and early fibrosis. These histological finding were not specific but were different from the histological aspect of abscess wall, granulation tissue or vasculitis like giant cells arteritis [
      • Upton P.D.
      • Smith J.G.
      • Charnock D.R.
      Histologic confirmation of carotidynia.
      ]. Our patient did not receive any treatment and he recovered in seven days. TIPIC syndrome is a rare but not serious condition, which usually improves spontaneously. However, it is important to recognize in order to avoid over-medicalization.

      References

        • Lecler A
        • Obadia M
        • Savatovsky J
        • Picard H
        • Charbonneau F
        • Menjot de Champfleur N
        • et al.
        TIPIC syndrome: beyond the myth of carotidynia, a new distinct unclassified entity.
        Am J Neuroradiol. 2017; 38: 1391-1398
        • Taniguchi Y.
        • Horino T.
        • Hashimoto K.
        Is carotidynia syndrome a subset of vasculitis?.
        J Rheumatol. 2008; 35: 1901-1902
        • Upton P.D.
        • Smith J.G.
        • Charnock D.R.
        Histologic confirmation of carotidynia.
        Otolaryngol Head Neck Surg. 2003; 129: 443-444