Histiocytic necrotizing lymphadenitis (Kikuchi-Fujimoto disease) with CNS involvement in a child

Gonçalves, Luís F.; Debelenko, Larisa V.; Bhambhani, Kanta; Scheid, Andrea; Altınok, Deniz

doi:10.1007/s00247-013-2786-y

Cited by 11 publications

(4 citation statements)

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“…Nineteen pediatric patients were identified, including two patients we presented (Table 1). 2,4–13 Eight patients were males and eleven patients were females. The onset age ranges from 6 to 18 years (the median age was 14 and the interquartile range was 9–16).…”

Section: Resultsmentioning

confidence: 99%

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Kikuchi–Fujimoto disease associated with the central nervous system in children

Huang

Wang

et al. 2023

Pediatric Discovery

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The clinical data of patients with Kikuchi–Fujimoto disease (KFD) combined with the involvement of the central system is limited, particularly for children. This study aimed to investigate the clinical features and prognosis of KFD associated with the central nervous system in children. We collected the clinical data of patients diagnosed with KFD combined with the involvement of the central system, hospitalized at Children's Hospital of Chongqing Medical University (CHCMU). Furthermore, we summarized the clinical features and outcomes in children by reviewing the published literature. Together with the two patients from CHMCU enrolled in this study, we identified 19 children diagnosed with KFD combined with the involvement of the central nervous system, including eight males and 11 females. The onset age ranges from 6 to 18 years (the median age was 14 and the interquartile range was 9–16). Sixteen patients were diagnosed with aseptic meningitis. Headache (78.9%, 15/19) was the most common symptom. Cerebrospinal fluid pressure was increased in 3 patients (60.0%, 3/5), cell count was elevated in 14 patients (73.7%, 14/19), glucose was decreased in 4 patients (21.0%, 4/19), and protein was elevated in 12 patients (63.1%, 12/19). A total of 9 patients had cranial imaging changes. Electroencephalogram (EEG) was performed on seven patients and 4 patients presented abnormal EEG (57.1%, 4/7). A total of 12 patients received steroid therapy; two were combined with antiepileptic treatment. In children, KFD complicated with the involvement of the central nervous system was rare and aseptic meningitis was the primary manifestation. Most patients had a good prognosis and a few had residual sequelae of neurological damage.

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Section: Resultsmentioning

confidence: 99%

“…2,[4][5][6][7][8][9][10][11][12][13] Eight patients were males and eleven patients were females. The onset age ranges from 6 to 18 years (the median age was 14 and the interquartile range was 9-16).…”

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confidence: 99%

Kikuchi–Fujimoto disease associated with the central nervous system in children

Huang

Wang

et al. 2023

Pediatric Discovery

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“…After diagnosing KFD based on results of the cervical lymph node biopsy, we could not explain the central nervous system (CNS) involvement in this case. Neurological involvement, including aseptic meningitis and encephalitis, is rare, and abnormal MRI findings in KFD usually show symmetrical, hyperintense T2 and FLAIR signals in the temporal lobes, pons, and periaqueductal gray matter [4][5][6][7]. Therefore, we considered the possibility of co-morbid ADEM.…”

Section: Discussionmentioning

confidence: 99%

“…Neurological complications occur in about 11% of cases, as revealed by a review of 244 cases in which a minority of cases evolved to developing encephalitis [3]. Brain magnetic resonance imaging (MRI) includes symmetrical hyperintense T2-weighted and FLAIR (fluidattenuated inversion recovery) signals in the temporal lobes, pons, and periaqueductal gray matter [4][5][6][7]. This is the first report of KFD occurring with testitis and pathologically proven asymmetrical brain regions.…”

Section: Introductionmentioning

confidence: 99%

Kikuchi-Fujimoto disease (histiocytic necrotizing lymphadenitis) with atypical encephalitis and painful testitis: a case report

et al. 2017

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BackgroundKikuchi-Fujimoto disease is a self-limited clinicopathologic entity that is increasingly recognized worldwide. Kikuchi-Fujimoto disease is characterized by cervical lymphadenopathy occurring in young adults. Neurologic involvement is rare, and testitis directly caused by Kikuchi-Fujimoto disease has not yet been reported.Case presentationA 19-year-old man was brought to our clinic with complaints of fever, headache, fatigue, and left lower quadrant pain that had persisted for 3 weeks. On physical examination, painful cervical lymphadenopathies were observed. Meningitis was suspected based on a cerebrospinal fluid examination, and left-sided orchitis was diagnosed based on findings from magnetic resonance imaging and ultrasonography. However, neither antibiotics nor antiviral drugs were effective in treating the patient’s symptoms. On the 20th day of hospitalization, the patient experienced a loss of consciousness, and brain T2-weighted magnetic resonance imaging showed asymmetrical, high-signal intensities in both basal nuclei and the left temporal lobe. Encephalitis was suspected, and the patient was treated with intravenous prednisolone pulse therapy (1 g/day) for 3 days and intravenous immunoglobulin therapy for 5 days. A left cervical lymph node biopsy showed apoptotic necrosis in paracortical and cortical areas with an abundance of macrophages and large lymphoid cells, which had irregular nuclei suggestive of Kikuchi-Fujimoto disease; the pathological findings from a brain biopsy were the same as those of the cervical lymph node biopsy. The encephalitis and cervical lymphadenopathies followed a benign course, as did the testitis.ConclusionsThis is the first report of Kikuchi-Fujimoto disease involving painful testitis and pathologically proven asymmetrical brain regions. Kikuchi-Fujimoto disease should be included in the differential diagnosis when a patient presents with encephalitis, testitis, and fever of unknown origin.

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Encephalitis in Kikuchi-Fujimoto disease being immune-mediated

Chen

Liang

et al. 2022

Neurol Sci

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Histiocytic necrotizing lymphadenitis (Kikuchi-Fujimoto disease) with CNS involvement in a child

Cited by 11 publications

References 4 publications

Kikuchi–Fujimoto disease associated with the central nervous system in children

Kikuchi–Fujimoto disease associated with the central nervous system in children

Kikuchi-Fujimoto disease (histiocytic necrotizing lymphadenitis) with atypical encephalitis and painful testitis: a case report

Encephalitis in Kikuchi-Fujimoto disease being immune-mediated

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