Bilateral Involvement of Juvenile Temporal Arteritis Associated with Kimura Disease

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

Concurrent bilateral juvenile temporal arteritis and hypereosinophilic syndrome: a case report and review of the literature

“…These patients complained of painless nodules on their temple, and the pathologic findings revealed nongiant cell inflammation of temporal arteries [4]. From then on until February 2019, according to our literature review through PubMed and Scopus, 18 more cases have been reported [23567891011121314151617181920]. Including the present case, a total of 23 cases of JTA were reported and their mean age was 27 years (range, 7 to 44 years) and there was the male predominance (18 men and 5 women).…”

“…Two of the 18 cases that resolved after excision recurred later on the opposite side [1218]. Of the 3 cases without excision, 2 were treated with steroid because of combined disease such as Kimura disease [20] or HES [12], and 1 was treated with tocopherol nicotinate [8], and these patients reported no recurrence.…”

“…Since the first report of JTA in 1975 [4], a total of 22 cases have been reported so far [234567891011121314151617181920], but its etiology, pathogenesis, and long-term prognosis are still unknown. Although JTA is a localized disease, it is often accompanied by peripheral blood eosinophilia, so some authors have suggested the concept of JTA with eosinophilia [81213].…”