The incidence of chronic
rhinosinusitis with
nasal polyps has recently increased in Japan and other East Asian countries, and this disease is called eosinophilic chronic
sinusitis (ECRS) in Japan. ECRS usually occurs in adults and is frequently accompanied by refractory
bronchial asthma. However, its occurrence in children under 10 years of age is rare. Here, we present an unusual case of ECRS complicated by intractable
asthma in an 8-year-old boy.
CASE PRESENTATION:
Oral administration of
prednisolone (10 mg/day) initially relieved the ECRS and
bronchial asthma, but both returned during
prednisolone dose reduction. Because nasal cavity-opening surgery was ineffective,
oral administration prednisolone at 10 mg/day was continued.
Pancytopenia was observed 16 months after the start of treatment, and the patient was admitted to our hospital. He was diagnosed with refractory
cytopenia in childhood, but gradually improved after
cyclosporine treatment. Although the dose of
cyclosporine was therapeutic for
asthma, it did not alleviate the
asthma attacks, and the patient's quality of life markedly decreased. We administered
omalizumab even though its use was contraindicated by negative results in an inhalable
antigen test. After the third administration of
omalizumab, the
asthma was better controlled and respiratory function improved; however, the nasal symptoms of ECRS persisted. Attempts to relieve these symptoms by increasing the therapeutic dose of
omalizumab were only partially successful. We replaced
omalizumab with
mepolizumab; doing so slightly improved the
sinusitis symptoms, but quality of life remained unsatisfactory. We repeated the nasal cavity-opening surgery. After surgery, the
asthma and
sinusitis were unchanged.
CONCLUSIONS:
Omalizumab effectively treated the severe combined
asthma in a young patient, but its effect on
sinusitis was insufficient. More cases and long-term follow-up data are needed to better evaluate the effectiveness of
mepolizumab for treatment of ECRS.