Abstract
Castleman-Kojima disease, also known as idiopathic multicentric Castleman disease with TAFRO syndrome (iMCD-TAFRO), is a recently recognized systemic inflammatory disorder with a characteristic series of clinical symptoms, including thrombocytopenia (T), anasarca (A), fever (F), reticulin fibrosis (R), and organomegaly (O). Patients with iMCD-TAFRO often develop severe abdominal pain, elevated alkaline phosphatase levels, and systemic inflammation, but the etiological factors are unknown. To investigate the potential role of bacterial infection in the pathogenesis of iMCD-TAFRO, we performed polymerase chain reaction (PCR) for the bacterial 16S rRNA gene with DNA extracted from liver specimens of three patients with iMCD-TAFRO, four patients with amyotrophic lateral sclerosis, and seven patients with inflammatory conditions. Sequencing of the PCR product showed 99% DNA sequence identity with Campylobacter jejuni in all three patients with iMCD-TAFRO and in two patients with inflammatory conditions. Immunohistochemical and electron microscopy analyses could not identify C. jejuni in patients with iMCD-TAFRO. The findings indicated that C. jejuni infection is not the pathological cause of iMCD-TAFRO; however, this ubiquitous bacterium may play a role in uncontrolled systemic hypercytokinemia, possibly through the development of cross-reactive autoantibodies.
| Original language | English |
|---|---|
| Pages (from-to) | 572-579 |
| Number of pages | 8 |
| Journal | Pathology International |
| Volume | 69 |
| Issue number | 10 |
| DOIs | |
| Publication status | Published - Oct 1 2019 |
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Keywords
- Castleman-Kojima disease
- etiology
- multicentric Castleman disease
- TAFRO syndrome
ASJC Scopus subject areas
- Pathology and Forensic Medicine
Cite this
Hepatic Campylobacter jejuni infection in patients with Castleman-Kojima disease (idiopathic multicentric Castleman disease with thrombocytopenia, anasarca, fever, reticulin fibrosis, and organomegaly (TAFRO) syndrome). / Kageyama, Chihiro; Igawa, Takuro; Gion, Yuka; Iwaki, Noriko; Tabata, Tetsuya; Tanaka, Takehiro; Kondo, Eisei; Sakai, Hajime; Tsuneyama, Koichi; Nomoto, Kazuhiro; Noguchi, Hiroko; Yoshino, Tadashi; Yokota, Kenji; Sato, Yasuharu.
In: Pathology International, Vol. 69, No. 10, 01.10.2019, p. 572-579.Research output: Contribution to journal › Article
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TY - JOUR
T1 - Hepatic Campylobacter jejuni infection in patients with Castleman-Kojima disease (idiopathic multicentric Castleman disease with thrombocytopenia, anasarca, fever, reticulin fibrosis, and organomegaly (TAFRO) syndrome)
AU - Kageyama, Chihiro
AU - Igawa, Takuro
AU - Gion, Yuka
AU - Iwaki, Noriko
AU - Tabata, Tetsuya
AU - Tanaka, Takehiro
AU - Kondo, Eisei
AU - Sakai, Hajime
AU - Tsuneyama, Koichi
AU - Nomoto, Kazuhiro
AU - Noguchi, Hiroko
AU - Yoshino, Tadashi
AU - Yokota, Kenji
AU - Sato, Yasuharu
PY - 2019/10/1
Y1 - 2019/10/1
N2 - Castleman-Kojima disease, also known as idiopathic multicentric Castleman disease with TAFRO syndrome (iMCD-TAFRO), is a recently recognized systemic inflammatory disorder with a characteristic series of clinical symptoms, including thrombocytopenia (T), anasarca (A), fever (F), reticulin fibrosis (R), and organomegaly (O). Patients with iMCD-TAFRO often develop severe abdominal pain, elevated alkaline phosphatase levels, and systemic inflammation, but the etiological factors are unknown. To investigate the potential role of bacterial infection in the pathogenesis of iMCD-TAFRO, we performed polymerase chain reaction (PCR) for the bacterial 16S rRNA gene with DNA extracted from liver specimens of three patients with iMCD-TAFRO, four patients with amyotrophic lateral sclerosis, and seven patients with inflammatory conditions. Sequencing of the PCR product showed 99% DNA sequence identity with Campylobacter jejuni in all three patients with iMCD-TAFRO and in two patients with inflammatory conditions. Immunohistochemical and electron microscopy analyses could not identify C. jejuni in patients with iMCD-TAFRO. The findings indicated that C. jejuni infection is not the pathological cause of iMCD-TAFRO; however, this ubiquitous bacterium may play a role in uncontrolled systemic hypercytokinemia, possibly through the development of cross-reactive autoantibodies.
AB - Castleman-Kojima disease, also known as idiopathic multicentric Castleman disease with TAFRO syndrome (iMCD-TAFRO), is a recently recognized systemic inflammatory disorder with a characteristic series of clinical symptoms, including thrombocytopenia (T), anasarca (A), fever (F), reticulin fibrosis (R), and organomegaly (O). Patients with iMCD-TAFRO often develop severe abdominal pain, elevated alkaline phosphatase levels, and systemic inflammation, but the etiological factors are unknown. To investigate the potential role of bacterial infection in the pathogenesis of iMCD-TAFRO, we performed polymerase chain reaction (PCR) for the bacterial 16S rRNA gene with DNA extracted from liver specimens of three patients with iMCD-TAFRO, four patients with amyotrophic lateral sclerosis, and seven patients with inflammatory conditions. Sequencing of the PCR product showed 99% DNA sequence identity with Campylobacter jejuni in all three patients with iMCD-TAFRO and in two patients with inflammatory conditions. Immunohistochemical and electron microscopy analyses could not identify C. jejuni in patients with iMCD-TAFRO. The findings indicated that C. jejuni infection is not the pathological cause of iMCD-TAFRO; however, this ubiquitous bacterium may play a role in uncontrolled systemic hypercytokinemia, possibly through the development of cross-reactive autoantibodies.
KW - Castleman-Kojima disease
KW - etiology
KW - multicentric Castleman disease
KW - TAFRO syndrome
UR - http://www.scopus.com/inward/record.url?scp=85073632479&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85073632479&partnerID=8YFLogxK
U2 - 10.1111/pin.12856
DO - 10.1111/pin.12856
M3 - Article
C2 - 31631463
AN - SCOPUS:85073632479
VL - 69
SP - 572
EP - 579
JO - Pathology International
JF - Pathology International
SN - 1320-5463
IS - 10
ER -