J Rheum Dis.  2018 Jan;25(1):65-68. 10.4078/jrd.2018.25.1.65.

Bilateral Involvement of Juvenile Temporal Arteritis Associated with Kimura Disease

Affiliations
  • 1Division of Rheumatology, Department of Internal Medicine, Kyungpook National University School of Medicine, Daegu, Korea. ymkang@knu.ac.kr
  • 2Division of Rheumatology, Department of Internal Medicine, Andong Medical Group Hospital, Andong, Korea.
  • 3Department of Pathology, Kyungpook National University School of Medicine, Daegu, Korea.

Abstract

Juvenile temporal arteritis (JTA) is a localized nodular arteritis confined to the temporal artery without evidence of systemic inflammation, and it occurs mainly in patients younger than 50 years. From the first case report, the pathological features of JTA have been suspected to be the morphological equivalent of Kimura disease (KD), which has been supported further by the concurrent cases of JTA with KD. We present the first case of bilateral JTA accompanying KD, which was confirmed by histological and ultrasound evaluations and supports the hypothesis that JTA is a manifestation of KD. The un-excised JTA lesion was resolved completely after corticosteroid therapy with no recurrence.

Keyword

Juvenile temporal arteritis; Kimura disease; Corticosteroids

MeSH Terms

Adrenal Cortex Hormones
Angiolymphoid Hyperplasia with Eosinophilia*
Arteritis
Giant Cell Arteritis*
Humans
Inflammation
Recurrence
Temporal Arteries
Ultrasonography
Adrenal Cortex Hormones

Figure

  • Figure 1. Gross appearance (A: left, B: right), computed tomographic angiography (C) and Doppler ultrasound images (D: short axis, E: long axis) of the nodular lesion on the left superficial temporal artery.

  • Figure 2. Inguinal and antecubital lymph nodes showed markedly increased lymphoid follicles with germinal centers and exuber-ant eosinophilic infiltrations, which is compatible with Kimura disease (H&E, A: ×40; B: ×200; C: ×400).

  • Figure 3. Biopsy of the superficial temporal artery lesions (H&E, A: ×40; B: ×100) showed intravascular proliferating structures, with eosinophilic infiltrations covered by endothelial linings (C: CD 34, ×200) and intramural and perivascular inflammatory lesions.


Cited by  1 articles

Concurrent bilateral juvenile temporal arteritis and hypereosinophilic syndrome: a case report and review of the literature
Ji-Su Shim, Ji Hyun Oh, Soo Jie Chung, Byung-Keun Kim, Yoon-Seok Chang, Sae-Hoon Kim
Asia Pac Allergy. 2019;9(3):.    doi: 10.5415/apallergy.2019.9.e23.


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