Bilateral Involvement of Juvenile Temporal Arteritis Associated with Kimura Disease

Na Ri Kim; Churl Hyun Im; Jong Wan Kang; Ji Hun Kim; Tae-In Park; Han-Ik Bae; Eon Jeong Nam; Young Mo Kang

doi:10.4078/jrd.2018.25.1.65

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Kim, Im, Kang, Kim, Park, Bae, Nam, and Kang: Bilateral Involvement of Juvenile Temporal Arteritis Associated with Kimura Disease

Case Report

J Rheum Dis 2018;25(1):65-68.

Published online: 31 October 2018

DOI: https://doi.org/10.4078/jrd.2018.25.1.65

Bilateral Involvement of Juvenile Temporal Arteritis Associated with Kimura Disease

Na Ri Kim¹, Churl Hyun Im¹, Jong Wan Kang¹, Ji Hun Kim², Tae-In Park³, Han-Ik Bae³, Eon Jeong Nam¹, Young Mo Kang¹

¹Division of Rheumatology, Department of Internal Medicine, Kyungpook National University School of Medicine, Daegu, Korea

²Division of Rheumatology, Department of Internal Medicine, Andong Medical Group Hospital, Andong, Korea

³Department of Pathology, Kyungpook National University School of Medicine, Daegu, Korea

Corresponding to: Young Mo Kang, Division of Rheumatology, Department of Internal Medicine, Kyungpook National University Hospital, 130 Dongdeok-ro, Jung-gu, Daegu 41944, Korea. E-mail: ymkang@knu.ac.kr

Received 10 April 2017 Revised 13 May 2017 Accepted 29 May 2017

This is a Open Access article, which permits unrestricted non-commerical use, distribution, and reproduction in any medium, provided the original work is properly cited.

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.

Keywords: Juvenile temporal arteritis, Kimura disease, Corticosteroids

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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.

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