Unilateral gluteal myositis as a unique presentation in mesenteric Kikuchi-Fujimoto disease

Author:

Watanabe Kazuhisa1,Yamamoto Masaki1,Matsubayashi Tadashi1ORCID

Affiliation:

1. Department of Pediatrics, Seirei Hamamatsu General Hospital , Shizuoka, Japan

Abstract

ABSTRACT Kikuchi-Fujimoto disease (KFD) is a self-limiting disease, characterised by fever and cervical lymphadenopathy. Lymphadenopathy without cervical lymph node involvement is rare and may mimic lymphoma. Although KFD can be associated with extranodal involvement, muscle involvement has not been reported. Herein, we report a novel case of unilateral gluteal myositis associated with mesenteric KFD in a patient who presented with persistent fever and right hip pain. Radiological imaging revealed an inflammatory lesion on the right gluteal muscle and multiple enlarged abdominal lymph nodes. No cervical lymphadenopathy was observed. A mesenteric lymph node biopsy was performed, and the histopathological findings led to a diagnosis of KFD. By day 29, the patient’s body temperature gradually returned to normal without any therapeutic intervention. Follow-up radiological imaging showed resolution of the gluteal lesion and a significant decrease in abdominal lymph node size. Considering the clinical course, the unilateral myositis may have developed as an extranodal involvement of KFD. Even if the clinical findings appear unrelated to those of KFD, a differential diagnosis that includes KFD should be considered in patients with unknown origin of fever.

Publisher

Oxford University Press (OUP)

Subject

Rheumatology

Reference19 articles.

1. Kikuchi disease in children: case report and review of the literature;Abdu;Pediatr Infect Dis J,2022

2. Arthritis as an unusual manifestation of Kikuchi-Fujimoto disease;Douglas;Rheumatology (Oxford),2003

3. Kikuchi’s disease and the skin: case report and review of the literature;Yasukawa;Br J Dermatol,2001

4. Kikuchi’s disease with hemophagocytic lymphohistiocytosis: a case report and literature review;Duan;Medicine (Baltimore),2020

5. Panuveitis as a possible ophthalmic complication of Kikuchi-Fujimoto disease;Pérez Alvarez;Arch Soc Esp Oftalmol,2005

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