start-ver=1.4
cd-journal=joma
no-vol=20
cd-vols=
no-issue=1
article-no=
start-page=
end-page=
dt-received=
dt-revised=
dt-accepted=
dt-pub-year=2020
dt-pub=20200113
dt-online=
en-article=
kn-article=
en-subject=
kn-subject=
en-title=
kn-title=Hemophagocytic lymphohistiocytosis complicating invasive pneumococcal disease: a pediatric case report
en-subtitle=
kn-subtitle=
en-abstract=
kn-abstract=Background<br/>
Hemophagocytic lymphohistiocytosis (HLH) is an infrequent but life-threatening disease due to excessive immune activation. Secondary HLH can be triggered by infections, autoimmune diseases, and malignant diseases. Streptococcus pneumoniae is a pathogenic bacterium responsible for invasive pneumococcal disease (IPD) such as meningitis and bacteremia. Although the pneumococcal conjugate vaccine (PCV) has led to reductions in IPD incidence, cases of IPD caused by serotypes not included in PCV are increasing. There are few reports of secondary HLH caused by IPD in previously healthy children. We herein report a rare case of a previously healthy boy with secondary HLH complicating IPD of serotype 23A, which is not included in the pneumococcal 13-valent conjugate vaccine (PCV-13).<br/>
Case presentation<br/>
An 11-month-old boy who had received three doses of PCV-13 was hospitalized with prolonged fever, bilateral otitis media, neutropenia and elevated C-reactive protein (CRP) levels. Blood culture on admission revealed S. pneumoniae, leading to a diagnosis of IPD. HLH was diagnosed based on a prolonged fever, neutropenia, anemia, hepatosplenomegaly, hemophagocytosis in the bone marrow, and elevated serum levels of triglycerides, ferritin, and soluble interleukin-2 receptor. He received broad-spectrum antibiotics and intravenous immunoglobulins for IPD and high-dose steroid pulse therapy and cyclosporine A for HLH; thereafter, his fever resolved, and laboratory findings improved. The serotype of the isolated S. pneumoniae was 23A, which is not included in PCV-13.<br/>
Conclusions<br/>
It is important to consider secondary HLH as a complication of IPD cases with febrile cytopenia or hepatosplenomegaly, and appropriate treatment for HLH should be started without delay.
en-copyright=
kn-copyright=

en-aut-name=TsugeMitsuru
en-aut-sei=Tsuge
en-aut-mei=Mitsuru
kn-aut-name=
kn-aut-sei=
kn-aut-mei=
aut-affil-num=1
ORCID=

en-aut-name=MiyamotoMachiko
en-aut-sei=Miyamoto
en-aut-mei=Machiko
kn-aut-name=
kn-aut-sei=
kn-aut-mei=
aut-affil-num=2
ORCID=

en-aut-name=MiyawakiReiji
en-aut-sei=Miyawaki
en-aut-mei=Reiji
kn-aut-name=
kn-aut-sei=
kn-aut-mei=
aut-affil-num=3
ORCID=

en-aut-name=KondoYoichi
en-aut-sei=Kondo
en-aut-mei=Yoichi
kn-aut-name=
kn-aut-sei=
kn-aut-mei=
aut-affil-num=4
ORCID=

en-aut-name=TsukaharaHirokazu
en-aut-sei=Tsukahara
en-aut-mei=Hirokazu
kn-aut-name=
kn-aut-sei=
kn-aut-mei=
aut-affil-num=5
ORCID=

affil-num=1
en-affil=Department of Pediatrics, Okayama University Graduate School of Medicine,Dentistry, and Pharmaceutical Sciences
kn-affil=

affil-num=2
en-affil=Department of Pediatrics, Matsuyama Red Cross Hospital
kn-affil=

affil-num=3
en-affil=Department of Pediatrics,Ehime University Graduate School of Medicine
kn-affil=

affil-num=4
en-affil=Department of Pediatrics, Matsuyama Red Cross Hospital
kn-affil=

affil-num=5
en-affil=Department of Pediatrics, Matsuyama Red Cross Hospital
kn-affil=
en-keyword=Child
kn-keyword=Child
en-keyword=Hemophagocytic lymphohistiocytosis
kn-keyword=Hemophagocytic lymphohistiocytosis
en-keyword=Invasive pneumococcal disease
kn-keyword=Invasive pneumococcal disease
en-keyword=Pneumococcal conjugate vaccine
kn-keyword=Pneumococcal conjugate vaccine
en-keyword=Serotype replacement
kn-keyword=Serotype replacement
END