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Features of sequential CT and US-guided biopsy in recurrent Kikuchi disease of the neck: a case report.

Abstract

A 26-year-old woman presented with recurrent Kikuchi disease 7 years after her initial episode. Computed tomography (CT) and ultrasonography demonstrated enlarged lymph nodes with extensive necrosis at the same site as the initial episode. Cytologic and histologic examinations were not conclusive. CT performed 1 month later demonstrated a complete resolution of the lymphadenopathy, which confirmed the diagnosis of recurrent Kikuchi disease. Care must be taken to avoid misdiagnosis of recurrent Kikuchi disease as tuberculous lymphadenitis.

Introduction

Kikuchi disease is a self-limited illness characterized by lymphadenitis, fever, and leukopenia. It most commonly affects women younger than 30 years of age, and it often resolves spontaneously. It is believed to be caused by an exaggerated T-cell response to external antigens, probably as a result of viral infection. (1) Some authors have proposed the possibility that Epstein-Barr virus or human herpesvirus 6 are the causative pathogens. (2,3) Because no specific laboratory tests are available to identify this disease, imaging is important for diagnosis and follow-up. Imaging studies have shown that lymph nodes affected by Kikuchi disease exhibit homogeneous enhancement without necrosis. (4,5) On the other hand, Kwon et al noticed that a minority of cases featured lymph nodes with gross necrotic loci. (5) The diagnosis is confirmed by histologic findings of apoptotic necrosis with histiocytic and lymphocytic infiltration of the lymph nodes. (7)

Cases of recurrent Kikuchi disease have been reported, (8-10) but to the best of our knowledge, the features of sequential imaging in recurrent disease have not appeared in the literature. We report a case of recurrent Kikuchi disease of the neck, and we discuss its features on sequential computed tomography (CT) and ultrasound (US)-guided biopsy.

Case report

A 26-year-old Korean woman presented for further evaluation of palpable lymph nodes on the left side of her neck. Approximately 7 days earlier, she had experienced fever and chills. The next day, she developed swelling of the left side of her neck, which had been managed elsewhere with intravenous hydration and antibiotics. When her symptoms persisted, she was referred to our medical center.

Physical examination revealed the presence of tender, palpable lymph nodes on the left side of the neck. The patient's body temperature was 39.0[degrees]C. Laboratory tests revealed leukopenia, with a white blood cell count of 3,070/[micro]l (normal: 5,200 to 12,400); thrombocytopenia, with a platelet count of 96,000/[micro]l (normal: 130,000 to 400,000); and an increased C-reactive protein level of 8.45 mg/dl (normal: 0 to 0.5).

Since the patient had been seen at our institution during her initial episode, the CT images taken 7 years earlier were still available to us. Those CTs showed multiple enlarged lymph nodes with extensive necrosis and perinodal inflammation on the left side of the neck, predominantly in the posterior cervical space (figure 1).

CT obtained on the day of her most recent presentation revealed multiple enlarged necrotic lymph nodes at the same site as the initial episode (figure 2, A and B). These findings suggested a diagnosis of recurrent Kikuchi disease or tuberculous lymphadenitis. On gray-scale and power Doppler US, the enlarged lymph nodes exhibited either preserved hilar fat or a necrotic center, and they were hypervascular except in areas of necrosis (figure 3, A and B). Perinodal inflammation was suggested by increased echogenicity around the enlarged lymph nodes.

US-guided fine-needle aspiration biopsy (US-FNAB) of an enlarged lymph node, which had been performed during the initial episode, was repeated (figure 4, A); both biopsies exhibited nonspecific lymphadenitis, and neither was diagnostic. A polymerase chain reaction assay for Mycobacterium tuberculosis of the US-FNAB specimen was negative.

The patient then underwent a US-guided core-needle biopsy (US-CNB) because both she and the referring physician were concerned about the possibility that she had tuberculous lymphadenitis. US-CNB was performed on an enlarged lymph node on the left at level VA with a 16-gauge needle. Findings at US-CNB were suspicious for, but not definitive of, Kikuchi disease (figure 4, B).

The case was managed conservatively, and the patient's neck swelling resolved. Because she and the referring physician wanted to confirm the diagnosis of Kikuchi disease, follow-up CT was performed 1 month later, and it showed a complete resolution of lymphadenopathy (figure 5).

Discussion

To the best of our knowledge, this is the first report of recurrent Kikuchi disease that includes sequential CT features during the initial episode, the recurrence, and the recovery. Our case presented a diagnostic dilemma because most enlarged lymph nodes during the initial and recurrent episodes showed extensive necrosis, which suggested tuberculous lymphadenitis, particularly in a younger person who lives in a region where the prevalence of tuberculosis is still high.

Fortunately, clinical data and CT images that had been obtained at our medical center 7 years earlier were still available to us for review. A comparison of the CT features of the initial episode and the second presentation suggested that the latter represented recurrent Kikuchi disease. However, we could not confidently exclude the possibility of tuberculous lymphadenitis.

Microscopic examination of the samples obtained by US-FNAB performed during the initial and recurrent episodes of the illness were reported as nonspecific lymphadenitis and were not diagnostic. Histologic examinations of the specimens obtained by US-CNB were reported as suspicious for, but not definitive of, Kikuchi disease. Thus, this case illustrates the difficulties of making a diagnosis of Kikuchi disease on the basis of cytologic and histologic examinations. (7) Accordingly, sequential imaging features may be important for the diagnosis of this illness. Comparisons of previous imaging if available with follow-up imaging may facilitate the correct diagnosis of recurrent Kikuchi disease. In our case, the diagnosis was confirmed by follow-up CT performed 1 month after the recurrent episode resolved.

Most studies have demonstrated that lymph nodes affected by Kikuchi disease show homogeneous enhancement. The rarity of extensive necrosis is well known in Kikuchi disease, and it is often mistaken for lymphoma, clinically and histologically. (4,5) In the investigation by Kwon et al, the authors studied 96 patients and found that 80 of them (83.3%) had homogeneous nodal enhancement, while the remaining 16 (16.7%) had lymph nodes with gross necrotic loci. (6) When nodal necrosis is present, Kikuchi disease can mimic tuberculous lymphadenitis or metastatic lymphadenopathy. (11)

Recurrent Kikuchi disease has been reported in various sites, including the axillas and the mesentery and retroperitoneum, (9) as well as the neck. (10) According to Cheng et al, 14 of 96 patients (14.6%) with Kikuchi disease who were followed for more than 6 months developed a clinical recurrence. (12) The longest reported interval between an initial onset and a recurrence is 12 years, (8)

Researchers have evaluated the possible predictive factors for recurrence among patients with Kikuchi disease, and they have found that patients with a recurrence were more likely to have fever, fatigue, and extranodal involvement and to have remained symptomatic for a rather longer time. (13) Also, the rate of positivity for the fluorescence antinuclear antibody test was significantly higher among patients who experienced a recurrence than in those who did not. (13) However, according to our review of the English-language literature, common imaging features that might reliably predict recurrence have not yet been identified.

Some studies of recurrent Kikuchi disease have found extensively necrotic lymph nodes on histologic examination. (8) We speculate that extensive areas of necrosis histologically (8) or radiologically, as in our case, might predispose patients to a recurrence of this illness.

Kwon et al reported that in most patients with Kikuchi disease, lymphadenopathy either resolved completely or improved in 1 to 8 months without medical treatment, as determined by follow-up imaging studies in many patients. (6) In this way, Kikuchi disease is different from other nodal diseases, such as tuberculosis, lymphoma, and metastasis. Therefore, follow-up CT may be helpful in evaluating patients with unusual CT findings mimicking those of nodal diseases other than Kikuchi disease.

In conclusion, Kikuchi disease may recur long after the initial episode, and it can mimic other diseases that are characterized by lymphadenopathy, particularly tuberculous lymphadenitis. Comparisons of features of sequential imaging obtained during the initial and recurrent episodes of illness and during follow-up may facilitate correct diagnosis. Extensive necrosis may indicate a recurrence, although further prospective studies with large numbers of patients are required.

References

(1.) Felgar RE, Furth EE, Wasik MA, et al. Histiocytic necrotizing lymphadenitis (Kikuchi's disease): In situ end-labeling, immunohistochemical, and serologic evidence supporting cytotoxic lymphocyte-mediated apoptotic cell death. Mod Pathol 1997; 10(3):231-41.

(2.) Dominguez DC, Torres ML, Antony S. Is human herpesvirus 6 linked to Kikuchi-Fujimoto disease? The importance of consistent molecular and serologic analysis. South Med J 2003;96(3):226-33.

(3.) Hudnall SD. Kikuchi-Fujimoto disease. Is Epstein-Barr virus the culprit? Am J Clin Pathol 2000;113(6):761-4.

(4.) Fulcher AS. Cervical lymphadenopathy due to Kikuchi disease: US and CT appearance. J Comput Assist Tomogr 1993; 17(1): 131-3.

(5.) Kim TA, Lupetin AR, Graham C. CT appearance of Kikuchi-Fujimoto disease. Clin Imaging 1995;19(1):1-3.

(6.) Kwon SY, Kim TK, Kim YS, et al. CT findings in Kikuchi disease: Analysis of 96 cases. AJNR Am J Neuroradiol 2004;25(6): 1099-1102.

(7.) Menasce LP, Banerjee SS, Edmondson D, Harris M. Histiocytic necrotizing lymphadenitis (Kikuchi-Fujimoto disease): Continuing diagnostic difficulties. Histopathology 1998;33(3):248-54.

(8.) Blewitt RW, Kumar SN, Abraham JS. Recurrence of Kikuchi's lymphadenitis after 12 years. J Clin Pathol 2000; 53(2):157-8.

(9.) Alijotas-Reig J, Casellas-Caro M, Ferrer-Oliveras R, et al. Recurrent Kikuchi-Fujimoto disease during pregnancy: Report of case evolving into systemic lupus erythematosus and review of published work. J Obstet Gynaecol Res 2008;34(4 Pt 2):595-8.

(10.) Sah SK, Pant R, Piper K, et al. Recurrent Kikuchi-Fujimoto disease: Case report. Br J Oral Maxillofac Surg 2007;45(3):231-3.

(11.) Moon WK, Han MH, Chang KH, et al. CT and MR imaging of head and neck tuberculosis. Radiographics 1997;17(2):391-402.

(12.) Cheng CY, Sheng WH, Loy C, et al. Clinical presentations, laboratory results and outcomes of patients with Kikuchi's disease: Emphasis on the association between recurrent Kikuchi's disease and autoimmune diseases. J Microbiol Immunol Infect 2010;43(5):366-71.

(13.) Song IY, Lee l, Park DW, et al. Clinical outcome and predictive factors of recurrence among patients with Kikuchi's disease. Int J Infect Dis 2009;13(3):322-6.

Sang Kwon Lee, MD; Young Eun Bahn, MD; Dong Eun Kim, MD

From the Department of Radiology (Dr. Lee and Dr. Bahn) and the Department of Otolaryngotogy (Dr. Kim), Dongsan Medical Center, Keimyung University School of Medicine, Daegu, Republic of Korea.

Corresponding author: Sang Kwon Lee, MD, Department of Radiology, Dongsan Medical Center, Keimyung University School of Medicine, 56 Dalseong-ro, Jung-gu, Daegu, 700-712, Republic of Korea. Email: sklee@dsmc.or.kr
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Title Annotation:ORIGINAL ARTICLE[
Author:Lee, Sang Kwon; Bahn, Young Eun; Kim, Dong Eun
Publication:Ear, Nose and Throat Journal
Article Type:Case study
Date:Sep 1, 2013
Words:1794
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