Kikuchi-Fujimoto disease (KFD) is a form of necrotizing lymphadenitis. Diagnosis is usually confirmed by analysis of samples from an excisional biopsy of the affected nodes. The patient had another 26 days of chemotherapy, and there was no recurrence 17 months after discharge. Management is symptomatic including analgesic (e.g., NSAIDs) and antipyretic. It is known as histiocytic necrotizing lymphadenitis, Kikuchi necrotizing lymphadenitis, phagocytic necrotizing lymphadenitis, subacute necrotizing lymphadenitis and necrotizing lymphadenitis [1-4]. The patient had continued hyperthermia, and his highest body temperature recorded was 40°C. Symptoms and Treatment of Kikuchi Disease. Kikuchi disease, also called histiocytic necrotizing lymphadenitis or Kikuchi-Fujimoto disease, is a disease that affects the lymph nodes, causing lymph node inflammation. The exact cause of the disease is not yet known, although some researchers have suggested it is an infection or autoimmune disorder. Infusion-Related Reactions It is non-cancerous and non-malignant disorder. It is a benign, self-limited disease that predominantly occurs in young women. Histiocytic necrotising lymphadenitis is a benign condition presenting with lymphadenopathy, mild fever, and occasionally a rash or other systemic symptoms [1].. Histiocytic necrotising lymphadenitis is also known as Kikuchi disease, Kikuchi–Fujimoto disease, and histiocytic necrotising lymphadenopathy. The patients condition quickly worsened, and eventually DIC developed. This disorder is often mistaken for malignant lymphoma, especially cervical adenopathy because the symptoms are very similar. Case report and literature review. Lymphadenitis Definition Lymphadenitis is the inflammation of a lymph node. Ubels FL. Kikuchi-Fujimoto disease is a disease of unknown cause. Successful treatment of severe Kikuchi's disease with intravenous immunoglobulin. [6]. Serologic tests for Epstein-Barr virus were negative, and a T-SPOT was negative for tuberculosis. Chemotherapy, continuous HDF and PE may be one of the ways to treat children with severe condition. Arch Pathol Lab Med. It generally affects young individuals, especially females, of Oriental-Asian origin. Awareness about this disorder may help prevent misdiagnosis and inappropriate investigations and treatment. So it is important that the patients with Kikuchi-Fujimoto disease should have a regular follow up with rheumatology for a detailed autoimmune work up. This volume in the Essentials in Cytopathology book series will focus on the cytopathology of lymph nodes. Continuous hemodiafiltration, plasma exchange and chemotherapy should be reserved for those patients who fail to respond to IVIG and corticosteroids. Histiocytoses. The aim of this study was to review the authors' institutional experience with KD in children over a 16-year period. Kikuchi disease usually gets better (resolves) on its own within one to four months (although it may take up to a year), with or without treatment. MeSH Please try after some time. Extranodal manifestations can occur, especially skin lesions and aseptic meningitides. The patient developed acute respiratory failure, DIC, and capillary leak syndrome. Sykes JA, Badizadegan K, Gordon P, et al. We report the case of a 4-year-5-month-old boy who suffered from fever, cervical lymphadenopathy, pancytopenia, hypertriglyceridemia, splenomegaly, low NK cell activity. Highlight selected keywords in the article text. 2018 Oct 3;31:77. doi: 10.11604/pamj.2018.31.77.16569. [Updated 2019 Mar 22]. Mahadeva U, Allport T, Bain B, et al. to maintaining your privacy and will not share your personal information without (adsbygoogle = window.adsbygoogle || []).push({}); (adsbygoogle = window.adsbygoogle || []).push({ HLH can be associated with KD, especially in childhood, and may have an aggressive clinical course. Several other clinical manifestations have been described, However, the possible recurrence rate of 3% to 4% has been reported that can occur even eight to nine years later. [3] The KD and HLH may be parts of a continuum of a single clinical condition, rather than representing separate entities for the etiology and clinical features between KD and HLH are some overlaps.[13]. [32]. Clinically focused chapters take an evidence-based approach to the management of pediatric surgical patients for residents in training and general surgeons in practice Targets the practitioner who is well-versed in the basic tenets of ... The interferon-gamma and IL-6 levels returned to normal during convalescence. 2014 Oct. 32 (10):1298.e1-2. Rheumatology (Oxford). Some patients with Kikuchi-Fujimoto disease have elevated aminotransferases and lactate dehydrogenase (LDH) levels. [35]. Lymphadenitis Lymphadenitis Lymphadenitis is an acute infection of one or more lymph nodes. Nishiwaki M, Hagiya H, Kamiya T. Kikuchi-Fujimoto disease complicated with reactive hemophagocytic lymphohistiocytosis. Kikuchi-Fujimoto disease is a rare clinical entity known as histiocytic necrotizing lymphadenitis characterized most often by several weeks of tender lymphadenopathy (most commonly cervical) and fever in individuals of Asian descent under the age of 40. 2017 Feb 16. In a study of 88 patients with Kikuchi-Fujimoto disease in the United States, 75 percent were Caucasian. In conclusion, although KD usually has a benign course, is self-limiting and, in most cases, administration of IVIG and corticosteroids yield satisfactory results. It was more common in Asian people, especially in Japanese. Pathol Int 2009;59:631–5. Chen[18] reported treatment of a patient with IVIG (2 g/kg) and prednisolone (2 mg/kg/day), led to a good recovery. Acta Haematol 1995;94:182–91. Kikuchi-Fujimoto disease: analysis of 244 cases. no specific treatment and symptomatic treatment Kikuchi disease (histiocytic necrotizing lymphadenitis) in is sufficient in almost all patients. Lim GY, Cho B, Chung NG. Its main presentation is cervical lymphadenopathy. Haemophagocytosis in bone marrow aspirate--a review of the clinical course of 10 cases. However, treatments are available to relieve some of the associated signs and symptoms. Netherlands J Med 2017;75:112–6. However, the absence of monoclonal lymphocyte receptors on the histologic analysis rules out a lymphoma. While the exact cause of this condition is unknown, infectious and autoimmune causes have been suggested. Introduction:Kikuchi-Fujimoto Disease, or histiocytic necrotizing lymphadenitis, is a rare condition with a benign course that mimics malignancy on presentation. It is considered to be autoimmune disorder and is infectious. World J Clin Cases. Atlas of Lymph Node Pathology reviews the histopathology of nodal diseases, illustrating the use of ancillary studies and includes concise discussions of pathogenesis, clinical settings and clinical significance of the pathologic diagnosis. Leukemia Lymphoma 2007;48:2447–51. 2021 Aug 9;13(8):e17021. Case Report: We report a case of young boy with Kikuchi-Fujimoto disease who presented with prolonged fever and … Hyperferritinemia in the critically ill child with secondary hemophagocytic lymphohistiocytosis/sepsis/multiple organ dysfunction syndrome/macrophage activation syndrome: what is the treatment? PMC Eur J Case Rep Intern Med. Kikuchi-Fujimoto disease is a distinctive syndrome of necrotizing lymphadenitis which was formally described in 1972 by two Japanese pathologists, Kikuchi and Fujimoto. Special considerations in continuous hemodiafiltration with critically ill pediatric patients. The course is usually benign with resolution in a few months with the use of antiinflammatory drugs. Kikuchi-Fujimoto disease (KFD) is a rare cause of acute lymphadenitis in pregnancy. The rash became more severe and pruritic and involved the patients entire body. High-volume hemofiltration in critically ill patients with secondary hemophagocytic lymphohistiocytosis/macrophage activation syndrome: a prospective study in the PICU. Would you like email updates of new search results? KD usually has a benign course and is self-limiting, but sometimes it may be fatal with HLH or if these patients develop DIC. J Am Acad Dermatol. Indian J Pediatrics 2011;78:616–9. About a rare disease misdiagnosed as malignant lymphoma or tuberculosis: Kikuchi-Fujimoto's disease. Kikuchi-Fujimoto disease (KFD), or histiocytic necrotizing lymphadenitis, was first described in 1972 simultaneously by Kikuchi1 and Fujimoto and colleagues2 as a lymphadenitis with focal proliferation of histiocytic cells and abundant karyorrhectic debris. HLH-associated KFD has a potentially fatal outcome, and early treatment is required. While the exact cause of Kikuchi-Fujimoto disease is unknown, infectious and autoimmune causes have been suggested. In most cases, KFD has a benign course; there is 2. It … Am J Surg Pathol 1995;19:798–809. Methylprednisolone treatment was initiated, followed by dexamethasone, cyclosporine, etoposide, continuous HDF and PE, and the patient recovered uneventfully. Chmait RH, Meimin DL, Koo CH, et al. JEMPERLI is indicated for the treatment of adult patients with mismatch repair deficient (dMMR) recurrent or advanced endometrial cancer (EC), as determined by an FDA-approved test, that has progressed on or following prior treatment with a platinum-containing regimen. The affected lymph nodes are described as follows: A = axillary, AB = abdomen, C = cervical, CH = chest, G = generalized, I = inguinal, M = mesenteric, MD = mediastinal, P = para-aortic, PEL = pelvis, R = retroperitoneum. [2] It seems that HLH associated with KD that occurs during childhood may have a less aggressive clinical course and better prognosis than in adults,[3] but sometimes it may still be fatal. Kikuchi’s disease, also known as histiocytic necrotizing lymphadenopathy, is a rare, benign, (noncancerous, nonmalignant) disorder of the lymph nodes of young adults, predominantly of young women. It is also called Histiocytic Necrotizing Lymphadenitis or Kikuchi Fujimoto disease. Initially described in Japan, cases of HNL are being reported in the United States and other western countries with increasing frequency. [15]. Kikuchi disease is a rare, benign condition of lymph nodes. Disseminated intravascular coagulopathy caused by Kikuchi-Fujimoto disease resulting in death: first case report in Turkey. Search for Similar Articles Epstein-Barr virus has been studied extensively in Kikuchi-Fujimoto disease, but no causative link has been demonstrated. Kikuchi-Fujimoto disease (KFD) is a rare entity characterized by subacute necrotizing lymphadenopathy and frequently associated with fever. It typically affects young women. He was admitted to the inpatient floor with persistent fever and cough. Simultaneous acquired self-limited hemophagocytic lymphohistiocytosis and Kikuchi necrotizing lymphadenitis in a 16-year-old teenage girl: a case report and review of the literature. google_ad_client: "ca-pub-9759235379140764", The classical histological findings are of focal proliferation of reticular cells, and cell debris surrounded by histiocytes,2 with a lack of neutrophils or eosinophils. Uslu E, Gurbuz S, Erden A, et al. 800-638-3030 (within USA), 301-223-2300 (international). Pepe F, Disma S, Teodoro C, Pepe P, Magro G. Arch Pathol Lab Med. A case of haemophagocytic syndrome and Kikuchi-Fujimoto disease occurring concurrently in a 17-year-old female. [18]. Among adults, 3 of the 9 cases were fatal. [5] It commonly manifests as a prolonged high fever, cervical lymphadenopathy, and leukopenia. Unable to load your collection due to an error, Unable to load your delegates due to an error. (B) Immunohistochemical stains showing CD163-positive histiocytes (X400). A possible role for interferon-gamma and interleukin (IL)-6 in the pathogenesis of this syndrome is suggested by one study of four men with biopsy-proven Kikuchi-Fujimoto disease. Kikuchi-Fujimoto disease (KFD), also known as histiocytic necrotizing lymphadenitis, is an uncommon condition, typically characterized by lymphadenopathy and fevers. What is histiocytic necrotising lymphadenitis?. Gerritsen A, Lam K, Schneider EM, et al. It is typically a self-limited disease, and spontaneous resolution occurs in one to four months. [27,32] No effective or standard unified treatment has been established for this disease. Data is temporarily unavailable. Introduction. Less common symptoms include weight loss, nausea, vomiting, and sore throat. The dis-ease was described in 1972 by Kikuchi (1) and independently by Fujimoto et al (2). Initiate treatment with insulin as clinically indicated. The only significant lab abnormalities are leukopenia, elevated erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP). Several infectious agents have been suggested as possible causative agents, for example, herpes simplex virus type 1 and 2, varicella-zoster virus, cytomegalovirus, Epstein-Barr virus (EBV), human herpesvirus (HHV 6, 7, 8), parvovirus B19, human papillomavirus, hepatitis B virus, human T-lymphotropic virus 1, Brucella, toxoplasma, and Yersinia. Registered users can save articles, searches, and manage email alerts. On the third day of hospitalization, the patients body temperature returned to normal. [26]. Careers. Kikuchi-Fujimoto disease is a rare benign self-limiting inflammatory lymphadenitis that develops in young patients, initially described in Asian populations in the 1970s. The patient was treated with intravenous immunoglobulin (IVIG, 2 g/kg) without improvement. J Clin Pathol 2000;53:636–8. Kikuchi-Fujimoto disease. Most patients are younger than 40 years of age, but this condition has been reported in patients ranging in age from six to 80 years, most of whom were previously well. Found inside – Page 29771Short - term treatment with proton pump inhibitors , H2 - receptor ... Kikuchi's disease ( histiocytic necrotizing lymphadenitis ) : report of one case . [1]. Fever is the first symptom in 30% to 50% of the cases. Prepared by a preeminent breast imaging expert, this case-based teaching file atlas presents a clinically oriented approach to screening, diagnostic evaluation, and management of patients with breast conditions encountered by radiologists. (1.1) for the adjuvant treatment of patients with melanoma with involvement of lymph node(s) following complete resection. Am J Clin Pathol. Kikuchi-Fujimoto disease was first described in Japan in 1972, by Japanese Pathologists Kikuchi and Fujimoto, is benign and self-limiting disease that mainly affects young women of Asian origin. [30]. ACV = acyclovir, ALF = acute liver failure, AORF = acute olguric renal, ARDS = acute respiratory distress syndrome, ARF = acute respiratory failure, ARNF = acute renal failure, CyA = cyclosporine A, DEX = dexamethazone, EBV = Epstein-Barr virus, IVIG = intravenous immunoglobulin, JIA = systemic juvenile idiopathic arthritis, JMML = juvenile myelomonocytic leukemia, LCN = liver cell necrosis, LN = lymph node, mPDN = methylprednisolone, MTX = methotrexate, NA = not available, PDN = prednisolone, PPE = proximal pulmonary embolus, PVB19 = Parvovirus B19, RSV = respiratory syncytial virus, SLE = Systemic lupus erythematosus, SPTL = subcutaneous panniculitis-like T-cell lymphoma, SS = septic shock, VP16 = Etoposide. What is Kikuchi disease. Initially described in Japan, cases of HNL are being reported in the This site needs JavaScript to work properly. Jump to Citations Citations of article over time Article citations Acute Tonsillitis With Concurrent Kikuchi's Disease as a Cause of Persistent Lymphadenopathy. Kim TY, Ha KS, Kim Y, et al. The median age at diagnosis was 13 years for children and 31 years for adult respectively, and the male-to-female ratio was 1: 1 and 5:4 among children and adults respectively. Kikuchi-Fujimoto Disease (KFD) is a benign, self-limited disease; it was first described in 1972 by Kikuchi and Fujimoto in Japan1,2. 2020;99:51(e23500). Histopathology and immunohistochemical results were suggestive of necrotizing lymphadenitis. An observation that is consistent with a viral etiology is increased levels of interferon-alpha and some other proteins stimulated by interferon-alpha including 2′,5′-oligoadenylate synthetase and tubuloreticular structures in the cytoplasm of stimulated lymphocytes, histiocytes, and vascular endothelium. Please enable scripts and reload this page. An illustrated guide to the pathologic diagnosis of Hodgkin's disease, non-Hodgkin's lymphomas, and other diseases appearing in lymph node biopsies. Chen JS, Chang KC, Cheng CN, et al. [24]. Kikuchi disease is characterized histopathologically by A biopsy of the right cervical lymph node performed on the 11th day showed scattered fibrin deposition, a large amount of nuclear debris and large mononuclear cell aggregates in the necrotic area, with no evidence of malignancy. He was discharged on day 45 after admission due to his good recovery status. He was treated with antibiotics at a local hospital with no clinical improvement. Cervical lymphadenopathy can often be confused with cervical lymphadenitis. Principles and practice of pediatric oncology. Some patients have non-specific presentation including weight loss, nausea, vomiting, night sweats, generalized lymphadenopathy, and hepatosplenomegaly. Cui Y, Zhang YC, Kang YL, et al. Mortality/Morbidity. The course of Kikuchi disease is generally benign and self-limited. Lymphadenopathy most often resolves over several weeks to 6 months, although the disease occasionally persists longer. The disease recurs in about 3% of cases. The item(s) has been successfully added to ", This article has been saved into your User Account, in the Favorites area, under the new folder. Kikuchi–Fujimoto disease (KFD), also known as histiocytic necrotizing lymphadenitis, is a rare, benign, self-limiting disease characterized by local lymphadenopathy. It is an … Al-Allaf AW, Yahia YM. Koga T, Takano K, Horai Y, et al. Diagnosis of Kikuchi Fujimoto disease is based on histopathological studies of the involved lymph nodes. eCollection 2018. Other histological differential diagnoses of Kikuchi-Fujimoto disease include reactive lymphadenitis associated with systemic lupus erythematosus, infectious mononucleosis, tuberculosis, and metastatic cancer. [21]. Mahadeva U[21] reported that treatment with corticosteroids, VP16, and cyclosporine for a young boy with KD and HLH led to rapid resolution of symptoms. The patient was treated with corticosteroids, cyclosporine, etoposide, continuous hemodiafiltration (HDF), and plasma exchange (PE). Familial cases of KFD in the literature are rare. It is a rare cause of lymphadenopathy, commonly seen in individuals of Asian descent and frequently associated with fever … Kikuchi's disease, also known as histiocytic necrotizing lymphadenopathy, is a Kwon et al. Cancer 1976;38:209–18. }); Masab M, Farooq H. Kikuchi Disease. Kucukardali Y, Solmazgul E, Kunter E, et al. Less common symptoms include weight loss, nausea, vomiting, and sore throat. Wolters Kluwer Health Kikuchi’s disease: case report and systematic review of cutaneous and histopathologic presentations. Atwater AR, Longley BJ, Aughenbaugh WD. Providers. 2018 Dec 19, Zuckerman R, Damiani L, Ayyad HA, Alpert DR. An essential pocket manual for anyone who treats children "This is a unique and novel approach to a pediatric handbook. Department of Pediatric Emergency Center, Hunan Children's Hospital, Changsha, Hunan,China. Careful examination and histologic confirmation of the diagnosis is therefore critical. This book provides the reader with a complete and concise introduction to rheumatic illness. Kim YM, Lee YJ, Nam SO, et al. eCollection 2021 Aug. Lahma J, Arkoubi Z, Hejjouji R, Nitassi S, El Ayoubi A, Bencheikh R, Benbouzid MA, Oujilal A, Essakalli L. Pan Afr Med J. Br J Haematol 1997;96:868–71. Internal Med 2013;52:1839–43. Disease, which has no speci… Enigmatic Kikuchi-Fujimoto disease: a comprehensive review. Lymphadenopathy most often resolves over several weeks to 6 months, although the disease occasionally persists longer. Echocardiography and chest computed tomography revealed no obvious abnormalities. Clin Rheumatol 2007;26:50–4. Proc (Bayl Univ Med Cent) 2018;31:350–1. Found insideWritten and edited by leading international experts in the field, this is an essential resource for trainee pediatric pathologists, as well as general pathologists who may encounter pediatric cases. On the 16th day, the patient developed capillary leak syndrome. Clinically, KFD is characterized by lymphadenitis of one or more lym … [8] Only about 2% splenomegaly and 3% hepatomegaly are seen in KD,[18] but splenomegaly was observed in 7 (38.9%) and 4 (44.4%) cases, hepatomegaly was observed in 6 (33.3%) and 1 (11.1%) cases for children and adults of HLH-associated KD, respectively. This updated edition remains the essential text for pathologists seeking to make accurate diagnoses from the vast number of differentials. Pediatr Crit Care Med 2016;17:e437–43. Primary epstein-barr virus infection associated with Kikuchi's disease and hemophagocytic lymphohistiocytosis: a case report and review of the literature. Bookshelf For more information, please refer to our Privacy Policy. Characteristics of lymphadenopathy are as follows: Other signs and symptoms may include a flulike prodrome with fever is present in 50% of cases 5): Routine laboratory investigations do not help much in making a diagnosis of Kikuchi-Fujimoto disease. [23]. During the acute phase of illness, these patients had elevated serum levels of interferon-gamma and IL-6 but not interferon-alpha, tumor necrosis factor, or IL-2. LIBTAYO is indicated for the first-line treatment of patients with non–small cell lung cancer (NSCLC) whose tumors have high PD-L1 expression (tumor proportion score [TPS] ≥50%) as determined by an FDA-approved test, with no EGFR, ALK, or ROS1 aberrations, and is locally advanced where patients are … This book will help cytopathologists to conduct these tasks in various organs and clinical contexts. [29]. Hemophagocytic syndrome in pregnancy. Differentiating this disease from common lymphatic disorder is extremely important from the pathologist’s point of view, which is highlighted in the article. It usually occurs in young adults and has a female predilection. The cause of KFD is unknown: a viral infection has been suggested, but not demonstrated, possibly involving human herpes virus 8 or Epstein-Barr virus. Treatment is usually empiric.... read more is lymphadenopathy with pain and/or signs of inflammation (eg, redness, tenderness). 'S disease with intravenous immunoglobulin ( IVIG, 2 ] presentation in a Korean report of 20 individuals with disease! The absence of monoclonal lymphocyte receptors on the presence of OPDIVO or YERVOY in human milk, kikuchi lymphadenitis treatment... 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