Unusual presentation of Kikuchi-Fujimoto disease

Santos, Marcelo Volpon; Gallo, Pasquale; Roked, Fozia; Nicklaus-Wollenteit, Ina; Rodrigues, Desiderio

doi:10.3171/2013.6.peds13118

Cited by 5 publications

(12 citation statements)

References 13 publications

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“…This presentation has been previously described in three case reports, two of them being adult patients [ 3 - 4 ] and one was a pediatric patient [ 1 ]. In these case reports, the patients presented with subdural effusions and aseptic meningitis, and an analysis of the latter typically shows lymphocytic pleocytosis and slightly elevated glucose and protein levels [ 1 , 3 - 4 ]. Our case is unique, as the patient had a recurrent involvement of the CNS and the subdural effusion recurred on the side opposite to where it first presented.…”

Section: Introductionmentioning

confidence: 65%

“…Kikuchi’s syndrome (histiolytic necrotizing lymphadenitis) was first described by Masahiro Kikuchi and Y. Fujimoto et al in 1972. The disease usually affects young adults and the mean age at diagnosis is 25 years [ 1 ] with a male to female ratio of 1.4:1 [ 5 ]. Regarding the etiology, various infections have been postulated to play a role, such as toxoplasma, brucellosis, Bartonella henselae, Yersinia enterocolitica, and viruses such as herpes viruses, human immunodeficiency virus (HIV), Epstein-Barr virus, rubella, paramyxovirus, and parainfluenza virus [ 6 ].…”

Section: Discussionmentioning

confidence: 99%

“…Kikuchi’s syndrome has a good prognosis in which the lymphadenopathy is usually self-limited and almost always resolves in six months [ 1 ]. However, there have been case reports of Kikuchi’s syndrome recurring up to eight years after the initial presentation.…”

Section: Discussionmentioning

confidence: 99%

“…Kikuchi syndrome, also known as Kikuchi-Fujimoto disease, was first described in the literature by Kikuchi and Fujimoto et al It is a rare disorder, and, histopathologically, it is a histiocytic necrotizing lymphadenitis. In this disease, the most common presentation is lymphadenopathy, and the patient may also be having accompanying constitutional symptoms in varying frequency, such as fever, weight loss, and night sweats [ 1 ]. Kikuchi’s syndrome has been described to have an association with the development of systemic lupus erythematosus (SLE) [ 1 ].…”

Section: Introductionmentioning

confidence: 99%

“…In this disease, the most common presentation is lymphadenopathy, and the patient may also be having accompanying constitutional symptoms in varying frequency, such as fever, weight loss, and night sweats [ 1 ]. Kikuchi’s syndrome has been described to have an association with the development of systemic lupus erythematosus (SLE) [ 1 ]. The involvement of the central nervous system (CNS) has also been reported and can be a harbinger of significant morbidity and even mortality, in a disease whose course is otherwise benign in most cases [ 2 ].…”

Section: Introductionmentioning

confidence: 99%

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An Unusual Presentation of Kikuchi-Fujimoto Disease with Recurrent Subdural Effusion

Shahid

Alam

Hadley

2018

Cureus

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A 24-year-old man complained of a right temporal headache for four weeks. The patient denied any trauma or previous anticoagulation use. He also reported tender right facial swelling. His physical exam was unrevealing except for right cranial nerve (CN) VI palsy, right parotid enlargement, and cervical adenopathy. Laboratory findings were significant for mild leukopenia at 3300 cells/uL. The computed tomography (CT) scan obtained showed a chronic left subdural effusion with a 4 mm midline shift and confirmed right parotid enlargement and cervical lymphadenopathy. Surgical burr hole evacuation was done and the fluid was sent for wound culture analysis. The infectious diseases service recommended initiating antibiotics, which were later stopped due to cerebrospinal fluid (CSF) cultures with no growth of any organisms. His CN VI palsy resolved during admission. The patient was discharged with follow-up for biopsy. The patient was lost to follow-up. The patient presented to the emergency department (ED) three months later, with a left-sided frontal headache. A repeat CT scan showed a new, right-sided fluid collection outside the brain parenchyma. Burr hole evacuation was done again and purulent fluid was drained. Antibiotics were held this time, but anti-tuberculous therapy was initiated empirically. The otolaryngology service was consulted and a lymph node biopsy was performed. The pathology showed histiocytic necrotizing lymphadenitis. A dural biopsy was done as well and was consistent with histiocytic necrotizing lymphadenitis involving the dura. Cultures from the subdural fluid did not grow any organism. The patient remained neurologically intact. He improved after surgery was done to drain the fluid and was managed by analgesics. The cultures from the extra-parenchymal fluid collection remained negative for pathogens and tuberculous mycobacteria. The patient was discharged with rheumatology clinic follow-up. He saw the rheumatologist six weeks after the discharge. During his clinic visit, the patient reported no recurrence of headaches, fevers, rash, or joint pain. Our patient had a rare presentation of Kikuchi-Fujimoto disease, in which he had a subdural fluid collection resulting in neurological complications that required surgical intervention.

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Section: Introductionmentioning

confidence: 65%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

An Unusual Presentation of Kikuchi-Fujimoto Disease with Recurrent Subdural Effusion

Shahid

Alam

Hadley

2018

Cureus

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Unique presentation of recurrent subdural effusions and subsequent abdominal lymphadenopathy in a patient diagnosed with Kikuchi-Fujimoto disease

Rana

Awad

Wang

et al. 2022

Radiology Case Reports

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Kikuchi Fujimoto Disease, originally discovered in 1972, is a rare lymphoproliferative disorder traditionally characterized by cervical lymphadenopathy, fevers, parotid gland enlargement, and several other nonspecific manifestations. Differentials include lymphoma, other viral diseases such as Epstein-Bar Virus, as well as other autoimmune conditions such as Systemic Lupus Erythematosus. Central nervous system involvement is exceptionally rare, with manifestations including meningitis as well as subdural effusions, as presented in this case. This review will summarize a case of a 24-year-old man with recurrent subdural effusions requiring intervention, subsequent relapse with abdominal lymphadenopathy, and possible IgG4 related disease. The background epidemiology, radiology, and potential pathophysiology will be reviewed.

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

Watanabe,

Yamamoto,

Matsubayashi

2023

Modern Rheumatology Case Reports

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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.

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Unusual presentation of Kikuchi-Fujimoto disease

Cited by 5 publications

References 13 publications

An Unusual Presentation of Kikuchi-Fujimoto Disease with Recurrent Subdural Effusion

An Unusual Presentation of Kikuchi-Fujimoto Disease with Recurrent Subdural Effusion

Unique presentation of recurrent subdural effusions and subsequent abdominal lymphadenopathy in a patient diagnosed with Kikuchi-Fujimoto disease

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

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