A case of subacute necrotizing lymphadenitis with recurrent aseptic meningitis 11 years after the first episode

Abstract

We report a 29-year-old man with subacute necrotizing lymphadenitis (SNL) associated with recurrent aseptic meningitis following an 11-year remission period. In both episodes, headache and fever were followed by lymphadenopathy, with increased serum IgE level. Although pleocytosis in cerebrospinal fluid was confirmed at admission in the first episode, it appeared at one week after admission in the second episode. Administration of glucocorticoid was effective for treating meningitis. The present case suggests a pathomechanism for SNL that involves both an immunological background and an acute viral infection as triggers of exacerbation of aseptic meningitis.

Recurrent aseptic meningitis in association with Kikuchi-Fujimoto disease: case report and literature review

“…Because IgE was not elevated in the CSF, his high serum IgE titre did not appear to play a pathogenic role in aseptic meningitis. A prior case with an elevation of serum IgE in recurrent aseptic meningitis with KFD has been reported [9]. In this study, we speculate that IgE elevation may be reflected the immunostimulatory condition that was activated upon KFD in a patient with atopic dermatitis.…”

Recurrent Aseptic Meningitis Associated with Kikuchi's Disease (Histiocytic Necrotizing Lymphadenitis): A Case Report and Literature Review

“…In September 2020, we searched MEDLINE, Web of Science, and Google Scholar for previous literature on aseptic meningitis associated with Kikuchi's disease using the terms “Kikuchi's disease,” “Kikuchi disease,” “Kikuchi-Fujimoto disease,” “Kikuchi-Fujimoto's disease” or “necrotizing lymphadenitis,” and “meningitis.” We found 30 cases reported in 23 English- or Japanese-language articles that were available in full text ( 7 , 9 - 30 ) ( Table 2 ). Only four of these cases were found to have recurred after the complete resolution of aseptic meningitis.…”

Aseptic meningitis revealing isolated Kikuchi-Fujimoto disease

“…The majority of cases are reported separately as sporadic cases and the review of the literature finds only about 30 observations of aseptic meningitis associated to KFD [7][8][9][13][14][15][16][17][18][19][20][21]]. Aseptic meningitis is therefore described as unusual [13] and atypical [14] during this disease.…”

“…Aseptic meningitis is therefore described as unusual [13] and atypical [14] during this disease. It can be acute [15] or chronic and recurrent [9,16], isolated or associated with other neurological signs [17][18][19] or other severe visceral manifestations of the disease [14,20], be seen during the evolution of already known KFD or more exceptionally be the first sign revealing it [14,17,21]. Evolution is usually favorable under systemic corticosteroids [7-9,13-21] and recurrence remains rare and the prerogative of undiagnosed forms of the disease [9,16].…”