Kikuchi-Fujimoto disease in the regional lymph nodes with node metastasis in a patient with tongue cancer: A case report and literature review

Abstract

Kikuchi-Fujimoto disease (KFD), also known as histiocytic necrotizing lymphadenitis, is a rare self-limiting disorder typically affecting the cervical lymph nodes (LNs), which is often misdiagnosed as other LN-associated diseases. KFD frequently presents with necrotic lesions and recurrences, which are also features of metastatic LNs. Clinicians may thus suspect LN metastasis when they encounter ipsilateral cervical lymphadenopathy in a patient with head and neck cancer. The present study reports the case of a 48-year-old man with tongue cancer and KFD affecting the right edge of his tongue and ipsilateral cervical LNs. LN metastasis was initially suspected, but pathological examination of the dissected LNs revealed one necrotic metastatic lesion and two necrotic KFD lesions. Ipsilateral cervical lymphadenopathy recurred 6 years after the initial surgery, and it was not possible to differentiate clinically between a second primary tumor and recurrent KFD prior to treatment. To the best of our knowledge, this is the first reported case of simultaneous tongue cancer, regional LN metastasis and KFD. This highlights the requirement to consider KFD in the event of LNs with necrotic lesions but no cancerous cells. A combination of clinical and pathological approaches may aid in the diagnosis of KFD, in addition to ruling out LN metastasis in initial and recurrent lymphadenopathies. The present study indicate that a diagnosis of KFD should be considered in patients with head and neck cancer that exhibit necrotic LNs lacking cancerous cells. This is important, as misdiagnosis of KFD as LN metastasis may lead to unnecessary adjuvant therapy.

A rare co-existence of histiocytic necrotizing lymphadenitis with metastatic papillary thyroid carcinoma and review of the literature

“…At present, 10 cases of HNL combined with other tumors have been reported (Table 1 ), most of which occurred in women (7/11), predominantly in Asia (6/11), aged 27–66 years. The tumors combined with HNL were PTC [ 6 , 11 ] (2 cases), gastric carcinoma [ 12 ] (1 case), breast carcinoma [ 13 ] (1 case), squamous cell carcinoma of the tongue [ 14 ] (1 case), malignant melanoma [ 15 ] (1 case), malignant fibrous histiocytoma [ 16 ] (1 case), multiple myeloma [ 17 ] (1 case), and diffuse large B lymphoma in remission [ 18 ] (2 cases).…”

“…Review the literature on the coexistence of HNL with other tumors: PTC [ 6 , 11 ], gastric carcinoma [ 12 ], breast carcinoma [ 13 ], squamous cell carcinoma of the tongue [ 14 ], and malignant melanoma [ 15 ], all of which occurred on the same side of the tumor as the present case, may indicate that HNL can be induced by tumor-associated local antigens and raise the possibility of specific immune responses to antigenic stimulation in HNL. Dequante et al reported a possible correlation between increased cytotoxic activity of T cells stimulated by the tumor and disease transformation [ 17 ].…”

Self-limiting COVID‐19-associated Kikuchi‐Fujimoto disease with heart involvement: case-based review

“…Infectious background is the most suggested one, with various viral, bacterial, and parasitic pathogens suspected as causative agents for KFD [12]. There are also reports of KFD induction after breast implants [21] and with association with solid tumors [22]. SARS-CoV-2 virus may be another potential trigger of KFD.…”

Follicular Thyroid Carcinoma Concurrent with Kikuchi-Fujimoto Disease: A Case Report and Literature Review

“…KFD mimicking lymph node metastasis in patients with prior thyroid cancer has likewise only been reported in a few cases [20][21][22]. But differential diagnosis of the lymph node metastasis versus KFD is still limited when examined by neck ultrasound, CT scan or pos-itron emission tomography (PET) scan [23]. Since the sensitivity and specificity of neck ultrasound to detect lymph node metastasis is not good enough, LN-FNA-Tg combined with lymph node aspirate cytology may offer an accurate way to distinguish lymph node metastasis from differentiated thyroid cancers [13,14].…”