Analysis of the clinical manifestations and 18F-FDG PET-CT findings in 40 patients with histiocytic necrotizing lymphadenitis

Zhang, Rui; Liang, Li-Dan; Li, Daoming; Bai, Yuling; Li, Xiangzhou

doi:10.1097/md.0000000000027189

Cited by 3 publications

(3 citation statements)

References 32 publications

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“…A systematic study of blood and bone marrow changes in a series of KFD patients demonstrated that a significant number of patients had single or multiple cytopenias of central origin (particularly those evolving into HLH), while a smaller fraction presented low-degree leukocytosis, thrombocytosis or atypical lymphocytes (for these patients, bone marrow studies were not available) [37]. Contrary to these findings, a recent PET/CT scan study showed that 50% (20/40) of KFD patients had splenomegaly and spleen hypermetabolism, 75% (30/40) had hypermetabolism of the central and peripheral bone marrow and they all exhibited features of myeloid hyperplasia on bone marrow smears [38]. A single study dating back to 2013 demonstrated the upregulation of ISG in the circulating phagocytes of KFD patients [39].…”

Section: Discussionmentioning

confidence: 95%

Increased Expression of CD169 on Monocytes in Adult-Onset Kikuchi–Fujimoto Disease

Malipiero,

Machin,

Ermacora

et al. 2023

Hemato

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Kikuchi–Fujimoto disease (KFD) is a rare, benign lymphoproliferative disease of uncertain origin that can mimic other inflammatory or clonal lymphoproliferative disorders. Given the lack of available blood biomarkers, diagnosis is based on the biopsy of an affected lymph node. In recent years, evidence has been mounting that a dysregulated type I INF innate immune response plays a pivotal role in the pathogenesis of the disease and might be a future therapeutic target. Nonetheless, laboratory assays measuring the expression of interferon alpha (INFα) and INF-stimulated genes (ISGs) are cumbersome and not widely available, limiting their use in clinical and translational research and encouraging the use of more convenient surrogate markers. In this study, a rapid flow cytometry assay detected increased levels of expression of CD169 (Siglec-1), an INFα-induced surface protein involved in innate immunity regulation, on circulating monocytes from two patients with KFD. Our results are in line with previous experiences and set the stage for a more extended investigation into the use of this assay in exploring the pathophysiology of KFD.

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Section: Discussionmentioning

confidence: 95%

Increased Expression of CD169 on Monocytes in Adult-Onset Kikuchi–Fujimoto Disease

Malipiero,

Machin,

Ermacora

et al. 2023

Hemato

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“…Recently, studies have been conducted to understand the role of positron emission tomography scans in identifying KFD and have shown utility in the majority of patients with hypermetabolic lymph nodes [ 25 , 26 ].…”

Section: Discussionmentioning

confidence: 99%

A Rare Case of Kikuchi-Fujimoto Disease

Qasim¹,

Itare²,

Anwar³

et al. 2023

Cureus

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Kikuchi-Fujimoto disease (KFD) is an autoimmune condition that is more common in females and occurs in the third decade of life. The condition is usually benign and self-resolving and is characterized by fever, cervical lymphadenopathy, night sweats, myalgia, and rashes. The disease can be misdiagnosed as reactive follicular hyperplasia, tuberculous lymphadenitis, systemic lupus erythematosus, and malignant lymphoma. The diagnosis of KFD involves the excision of the affected lymph node. Although there is no specific treatment for the disease, usually symptomatic and supportive measures are effective; however, steroids and immunosuppressive therapies are considered in more severe cases. The disease lasts for around one to four months. The neurological complications include cerebellar ataxia, meningoencephalitis, and aseptic meningitis. Here, we describe the case of a 36-year-old male who presented with complaints of fever, malaise chills, anorexia, and fatigue associated with a tender right axillary lymph node. The patient underwent a biopsy which confirmed KFD and responded well to supportive therapy.

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“…Areas of hypointensity in peripheral distribution and clear margins representing necrosis in paracortical areas, and occasional focal non-enhancing areas suggestive of necrosis within the enlarged lymph nodes were observed in T2-weighted images in the same study. Positron emission tomography scanning has been used to assess disease severity and most patients are found to have multiple hypermetabolic lymph nodes but only a few were enlarged[ 135 , 136 ]. Radiating vessels from the central hilum to the periphery of the lymph node and normal or scant hilar vascularity within the whole lesion in 92% of patients with KFD on color Doppler imaging indicates its benign nature[ 137 ].…”

Section: Diagnosismentioning

confidence: 99%

Kikuchi-Fujimoto disease: A comprehensive review

Mahajan¹,

Sharma²,

Sharma³

et al. 2023

World J Clin Cases

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Kikuchi-Fujimoto disease, a rare form of necrotizing lymphadenitis, is an uncommon, benign, self-limiting disorder of obscure etiology. It affects mostly young adults of both genders. Clinically, it presents with fever and lymphadenopathy of a firm to rubbery consistency frequently involving cervical lymph nodes while weight loss, splenomegaly, leucopenia, and elevated erythrocyte sedimentation rate feature in severely affected patients. Cutaneous involvement occurs in about 30%-40% of cases as facial erythema and nonspecific erythematous papules, plaques, acneiform or morbilliform lesions of great histologic heterogeneity. Both Kikuchi-Fujimoto disease and systemic lupus erythematosus share an obscure and complex relationship as systemic lupus erythematosus may occasionally precede, develop subsequently, or sometimes be associated concurrently with Kikuchi-Fujimoto disease. It is often mistaken for non-Hodgkin lymphoma while lupus lymphadenitis, cat-scratch disease, Sweet’s syndrome, Still’s disease, drug eruptions, infectious mononucleosis, and viral or tubercular lymphadenitis are other common differentials. Fine needle aspiration cytology mostly has features of nonspecific reactive lymphadenitis and immunohistochemistry studies usually show variable features of uncertain diagnostic value . Since its diagnosis is exclusively from histopathology, it needs to be evaluated more carefully; an early lymph node biopsy will obviate the need for unnecessary investigations and therapeutic trials. Its treatment with systemic corticosteroids, hydroxychloroquine, or antimicrobial agents mostly remains empirical. The article reviews clinicoepidemiological, diagnostic, and management aspects of KFD from the perspective of practicing clinicians.

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Analysis of the clinical manifestations and 18F-FDG PET-CT findings in 40 patients with histiocytic necrotizing lymphadenitis

Cited by 3 publications

References 32 publications

Increased Expression of CD169 on Monocytes in Adult-Onset Kikuchi–Fujimoto Disease

Increased Expression of CD169 on Monocytes in Adult-Onset Kikuchi–Fujimoto Disease

A Rare Case of Kikuchi-Fujimoto Disease

Kikuchi-Fujimoto disease: A comprehensive review

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