PMID- 31852109
OWN - NLM
STAT- MEDLINE
DCOM- 20191225
LR  - 20221005
IS  - 1536-5964 (Electronic)
IS  - 0025-7974 (Print)
IS  - 0025-7974 (Linking)
VI  - 98
IP  - 50
DP  - 2019 Dec
TI  - Tissue-infiltrating immune cells contribute to understanding the pathogenesis of 
      Kimura disease: A case report.
PG  - e18300
LID - 10.1097/MD.0000000000018300 [doi]
LID - e18300
AB  - RATIONALE: Kimura disease (KD) is a rare, chronic inflammatory disorder 
      characterized by subcutaneous granuloma in the head and neck region, as well as 
      increased eosinophil counts and high serum immunoglobulin E (IgE) levels. Kimura 
      disease is suspected to be an IgE-mediated disease, associated with an allergic 
      response, in which antigen-specific B cells are stimulated to undergo specific 
      IgE class switching with disease-specific CD4+ T (Th) cells help. Thus, 
      exploration of the Th cells in affected tissues with KD is a highly promising 
      field of the investigation. However, there have been no reports with direct 
      evidence to implicate Th cells in affected lesions with KD. Here we 
      quantitatively demonstrate that CD4+ GATA3+ T cells and interleukin (IL)-4+ IgE+ 
      c-kit+ mast cells prominently infiltrate in affected lesion with KD. PATIENT 
      CONCERNS: A 56-year-old Japanese man who exhibited painless swelling in the left 
      parotid region. DIAGNOSES: Diagnosis of KD was made based on characteristic 
      histopathologic findings, in conjunction with peripheral eosinophilia and 
      elevated serum IgE levels. INTERVENTIONS: The patient underwent corticosteroid 
      therapy and had been followed for 2 years. OUTCOMES: We report a rare case of KD 
      of the parotid region in a 56-year-old man, followed by corticosteroid therapy 
      for 2 years. The mass decreased in size and skin itchiness decreased after 
      therapy. He was discharged without any complications. Furthermore, we 
      quantitatively demonstrate the dominance of CD4+ GATA3+ T cells in affected 
      tissues of KD and detect IL-4+ IgE+ c-kit+ mast cells in lesions by multicolor 
      staining approaches. LESSONS: The findings from this case suggest that peripheral 
      blood eosinophilia might serve as a marker of recurrent disease, long-term 
      follow-up is necessary due to the possibility of recurrent. Interactions among 
      expanded IgE+ B cells, CD4+ GATA3+ T cells, eosinophils, and activated mast cells 
      might play a critical role in the pathogenesis of KD.
FAU - Maehara, Takashi
AU  - Maehara T
AD  - Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic 
      and Surgical Sciences.
FAU - Munemura, Ryusuke
AU  - Munemura R
AD  - Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic 
      and Surgical Sciences.
FAU - Shimizu, Mayumi
AU  - Shimizu M
AD  - Department of Oral and Maxillofacial Radiology, Faculty of Dental Science, Kyushu 
      University, Fukuoka, Japan.
FAU - Kakizoe, Noriko
AU  - Kakizoe N
AD  - Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic 
      and Surgical Sciences.
FAU - Kaneko, Naoki
AU  - Kaneko N
AD  - Ragon Institute of MGH, MIT and Harvard, Massachusetts General Hospital, Harvard 
      Medical School, Boston, MA.
FAU - Murakami, Yuka
AU  - Murakami Y
AD  - Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic 
      and Surgical Sciences.
FAU - Masafumi, Moriyama
AU  - Masafumi M
AD  - Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic 
      and Surgical Sciences.
FAU - Kiyoshima, Tamotsu
AU  - Kiyoshima T
AD  - Laboratory of Oral Pathology, Division of Maxillofacial Diagnostic and Surgical 
      Sciences, Faculty of Dental Science, Kyushu University, Fukuoka, Japan.
FAU - Kawano, Shintaro
AU  - Kawano S
FAU - Nakamura, Seiji
AU  - Nakamura S
AD  - Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic 
      and Surgical Sciences.
LA  - eng
PT  - Case Reports
PT  - Journal Article
PL  - United States
TA  - Medicine (Baltimore)
JT  - Medicine
JID - 2985248R
RN  - 37341-29-0 (Immunoglobulin E)
SB  - IM
MH  - Angiolymphoid Hyperplasia with Eosinophilia/blood/diagnosis/*immunology
MH  - B-Lymphocytes/*immunology/pathology
MH  - Biopsy
MH  - Eosinophils/*immunology/pathology
MH  - Humans
MH  - Immunoglobulin E/*blood
MH  - Magnetic Resonance Imaging
MH  - Male
MH  - Mast Cells/*immunology/pathology
MH  - Middle Aged
PMC - PMC6922356
COIS- The authors have no conflicts of interest to disclose.
EDAT- 2019/12/20 06:00
MHDA- 2019/12/26 06:00
PMCR- 2019/12/16
CRDT- 2019/12/20 06:00
PHST- 2019/12/20 06:00 [entrez]
PHST- 2019/12/20 06:00 [pubmed]
PHST- 2019/12/26 06:00 [medline]
PHST- 2019/12/16 00:00 [pmc-release]
AID - 00005792-201912130-00049 [pii]
AID - MD-D-19-02181 [pii]
AID - 10.1097/MD.0000000000018300 [doi]
PST - ppublish
SO  - Medicine (Baltimore). 2019 Dec;98(50):e18300. doi: 10.1097/MD.0000000000018300.