Ⅰ. INTRODUCTION
Histiocytic necrotizing lymphadenitis (Kikuchi-Fujimoto disease, KFD) was first reported by Kikuchi and Fujimoto in 1972
1,2) and is known to occur mainly in females under 30 years of age. The pathogenesis of histiocytic necrotizing lymphadenitis is still unclear, and there are only a few reports suggesting relevance to viral infection, bacterial infection, and systemic lupus erythematosus (SLE).
3) Clinically, acute or subacute painful lymphadenitis (0.5–4 cm) progresses over 1–3 weeks and occurs predominantly in the posterior cervical triangle.
4) KFD is benign, mostly self-healing, and can
be misdiagnosed as various diseases. Thus, accurate diagnosis is needed. In this study, we present a case of KFD in the right cervical lymph node area, which was successfully treated with prednisolone (PSL).
Ⅱ. CASE REPORT
A 52-year-old female was admitted to our hospital with persistent febrile sense, right cervical swelling, odynophagia, and fatigue. The patient had no other medical history. Right submandibular swelling with fever, fatigue, and chills were observed on clinical examination.
At the time of admission, the initial clinical diagnosis was a right submandibular abscess. There was no specific cause of abscess in the oral cavity. Computed tomography
Korean Journal of Oral and Maxillofacial Pathology 2019;43(4):111-114
ISSN:1225-1577(Print); 2384-0900(Online)
Available online at http://journal.kaomp.org
https://doi.org/10.17779/KAOMP.2019.43.4.002
* Correspondence: So-Young Choi, Department of Oral & Maxillofacial
Surgery, School of Dentistry, Kyungpook National University
Tel: +82-53-600-7561, Fax: +82-53-426-5365
E-mail: [email protected]
ORCID: 0000-0002-2563-3539
Received: Jul. 10. 2019; Revised: Jul. 19. 2019; Accepted: Aug. 2. 2019
우측 경부 림프절에 발생한 키쿠치-후지모토씨 병 : 증례보고 및 문헌고찰
정상환, 장한슬, 장성백, 권대근, 최소영*
경북대학교 치과대학 구강악안면외과학 교실
<Abstract>
Kikuchi-Fujimoto disease in the right cervical lymph node:
a case report & literature review
Sang-Hwan Jung, Han-Seul Jang, Seong-Baek Jang, Tae-Geon Kwon and So-Young Choi
*
Department of Oral & Maxillofacial Surgery, School of Dentistry, Kyungpook National University
Histiocytic necrotizing lymphadenitis (Kikuchi-Fujimoto disease, KFD) is a benign self-limiting lesion that can self-heal when no severe symptoms are present. KFD resembles tuberculous lymphadenitis and malignant lymphoma. Thus, early differential diagnosis will minimize unnecessary evaluation and treatment. Histological examination of a lymph node biopsy or fine needle aspiration could be a reliable method for KFD diagnosis. This study reports a case of histiocytic necrotizing lymphadenitis of the right cervical lymph node in a 52-year-old female patient. According to the fine needle aspiration biopsy result, the patient was diagnosed with Kikuchi-Fujimoto disease and treated with prednisolone (15 mg/day). After treatment, there was no recurrence or adverse event.
Key words : Histiocytic necrotizing lymphadenitis, Kikuchi-Fujimoto disease, Fine needle aspiration biopsy, Prednisolone
112 (CT) showed right cervical lymph node enlargement at level II and no abscess (Fig. 1A, B). Ultrasonography of the right neck showed the presence of multiple conglomerate lymph nodes and the largest node being 2.14 × 1.38 mm at level II (Fig. 1C, D). Abdominal CT was performed to determine the cause of the fever; however, there was no specific finding.
Laboratory tests revealed increased C-reactive protein (CRP) (5.96 mg/dL), erythrocyte sedimentation rate (ESR) (41 mm/h), and ferritin (421.6 ng/mL). The level of lactate dehydrogenase (200.0 U/L) was normal.
The provisional diagnosis was KFD, SLE, or tuberculosis.
The result of real-time PCR for mycobacterium tuberculosis and non-tuberculosis mycobacterium was negative. Antinuclear antibody and antineutrophil cytoplasmic antibody screenings were negative. The serum level of complement C3 was normal; however, the serum level of complement C4 was slightly elevated (48.4 mg/dL). Fine-needle aspiration biopsy (FNAB) was performed for further differential diagnosis.
FNAB revealed scattered histiocytes with oval or crescentic nuclei around reactive lymphocytes, which may indicate KFD (Fig. 2). PSL (Solondo
®) was orally administered (15 mg/day) as a corticosteroid. After oral administration, fever and lymphadenopathy were rapidly reduced in 12 hours.
PSL was maintained at 15 mg/day for 14 days. Fever (normal range), lymphadenopathy, and pain were markedly attenuated.
Thereafter, the PSL dose was tapered to 5 mg/day, and the treatment was terminated after 7 days. Recurrence of KFD or adverse event was not observed for 1 years..
Ⅲ. DISCUSSION
The etiology of KFD has not been clearly elucidated.
KFD has been reported to be associated with infections.
Viral infections include Epstein-Barr virus, human herpes virus 6, human herpes virus 8, cytomegalovirus, and parvovirus B19.
5,6,7) Bacterial infections are also possible causative agents, such as toxoplasma.
8)
The difference in prevalence between males and females varies widely among studies. Nevertheless, it is considered to occur more frequently in young women under 30 years of age.
3) Cervical lymphadenopathy occurs in 80% of the cervical lymph nodes. The involvement of the posterior cervical triangle has been reported to be the most common, Fig. 2.
Fine-needle aspiration biopsy (FNAB) smear showing scattered
histiocytes with crescentic nuclei, plasmacytoid monocytes,
and karyorrhexis around lymphocytes (Papanicolaou-stained
smears, ×400).
Fig. 1.
(A, B) Axial & coronal computed tomography (CT) images
showing right lymph node enlargement at level II. (C, D)
Ultrasonography of the right neck showing the presence
of multiple conglomerate lymph nodes and the largest node
being 2.14 × 1.38 mm at level II.
