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JYMS : Journal of Yeungnam Medical Science

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Hemophagocytic lymphohistiocytosis with recurrent Kikuchi-Fujimoto disease
Sang Min Lee, Young Tae Lim, Kyung Mi Jang, Mi Jin Gu, Jong Ho Lee, Jae Min Lee
Yeungnam Univ J Med. 2021;38(3):245-250.   Published online November 11, 2020
DOI: https://doi.org/10.12701/yujm.2020.00654
  • 6,291 View
  • 108 Download
  • 4 Crossref
AbstractAbstract PDF
Kikuchi-Fujimoto disease (KFD), also known as histiocytic necrotizing lymphadenitis, is a self-limiting lymphadenitis. It is a benign disease mainly characterized by high fever, lymph node swelling, and leukopenia. Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening disease with clinical symptoms similar to those of KFD, but it requires a significantly more aggressive treatment. A 19-year-old Korean male patient was hospitalized for fever and cervical lymphadenopathy. Variable-sized lymph node enlargements with slightly necrotic lesions were detected on computed tomography. Biopsy specimen from a cervical lymph node showed necrotizing lymphadenitis with HLH. Bone marrow aspiration showed hemophagocytic histiocytosis. The clinical symptoms and the results of the laboratory test and bone marrow aspiration met the diagnostic criteria for HLH. The patient was diagnosed with macrophage activation syndrome—HLH, a secondary HLH associated with KFD. He was treated with dexamethasone (10 mg/m2/day) without immunosuppressive therapy or etoposide-based chemotherapy. The fever disappeared within a day, and other symptoms such as lymphadenopathy, ascites, and pleural effusion improved. Dexamethasone was reduced from day 2 of hospitalization and was tapered over 8 weeks. The patient was discharged on day 6 with continuation of dexamethasone. The patient had no recurrence at the 18-month follow-up.

Citations

Citations to this article as recorded by  
  • Histiocytic necrotizing lymphadenitis with hemophagocytic lymphohistiocytosis in adults: A single‐center analysis of 5 cases
    Qingqing Chen, Jing Zhang, Huijun Huang, Tonglu Qiu, Ze Jin, Yu Shi, Huayuan Zhu, Lei Fan, Jianyong Li, Wenyu Shi, Yi Miao
    Immunity, Inflammation and Disease.2024;[Epub]     CrossRef
  • A young Saudi female with combined hemophagocytic lympho-histiocytosis and Kikuchi’s disease: A case report
    Kamal Al-Zahrani, Batol Gasmelseed, Hesham Waaer Shadi, Rehab Y AL-Ansari
    SAGE Open Medical Case Reports.2023; 11: 2050313X2311543.     CrossRef
  • Cefalea y fiebre: no todo es lo que parece
    María Pilar Iranzo-Alcolea, Carmen Ariño-Palao, Grisell Starita-Fajardo, Andrés González-García, Cecilia Suárez-Carantoña
    Revista Española de Casos Clínicos en Medicina Interna.2023; 8(2): 105.     CrossRef
  • Kikuchi–Fujimoto disease: literature review and report of four cases
    V. G. Potapenko, V. V. Baykov, А. Yu. Markova, N. B. Mikhailova, A. S. Ter‑Grigoryan, Yu. А. Krivolapov
    Oncohematology.2022; 17(4): 48.     CrossRef
Delayed treatment-free response after romiplostim discontinuation in pediatric chronic immune thrombocytopenia
Hyun Ji Lim, Young Tae Lim, Jeong Ok Hah, Jae Min Lee
Yeungnam Univ J Med. 2021;38(2):165-168.   Published online August 7, 2020
DOI: https://doi.org/10.12701/yujm.2020.00493
  • 5,406 View
  • 136 Download
  • 3 Crossref
AbstractAbstract PDF
We report the case of a 16-month-old patient with chronic immune thrombocytopenia (ITP) patient who experienced delayed treatment-free response (TFR) after romiplostim treatment. He received intravenous immunoglobulin every month to maintain a platelet count above 20,000/μL for 2 years. Thereafter, he received rituximab and cyclosporine as second-line therapy, with no response, followed by romiplostim. After 4 weeks of treatment, the platelet count was maintained above 50,000/μL. Following 7 months of treatment, he discontinued romiplostim, and the platelet count decreased. His platelet counts remained above 50,000/μL, without any bleeding symptoms, 2 years after romiplostim discontinuation. This is the first report of TFR after romiplostim treatment in pediatric chronic ITP.

Citations

Citations to this article as recorded by  
  • A cost–utility analysis of thrombopoietin receptor agonists for treating pediatric immune thrombocytopenia purpura after failure of first‐line therapies
    Huimin Du, Jiamin Wang, Joel Livingston, Ziyad Alrajhi, Melanie Kirby‐Allen, Brian Chan, Rebecca Hancock‐Howard, Peter C. Coyte
    Pediatric Blood & Cancer.2023;[Epub]     CrossRef
  • Generic romiplostim for children with persistent or chronic immune thrombocytopenia: Experience from a tertiary care centre in North India
    Chandana Mareddy, Manas Kalra, Anupam Sachdeva
    British Journal of Haematology.2022; 197(5): 618.     CrossRef
  • Tapering of the thrombopoietin receptor agonist in paediatric patients with chronic immune thrombocytopenia: Is it possible?
    María Solsona, Rubén Berrueco, Elena Sebastián, Áurea Cervera, Ana Sastre, Itziar Astigarraga, Bienvenida Argilés, María Ángeles Dasí, José Luís Dapena, Emilio Monteagudo
    British Journal of Clinical Pharmacology.2022; 88(9): 4220.     CrossRef

JYMS : Journal of Yeungnam Medical Science