RESUMEN
We report a case with Clavispora lusitaniae-induced purulent thrombophlebitis. The patient had multiple risk factors for the development of fungal thrombophlebitis including surgical procedure, mechanical ventilation, admission to intensive care unit, total parenteral nutrition and long-term antimicrobial therapy in addition to the insertion of central venous catheter. The symptoms finally improved by a combination therapy of micafungin and flucytosine, but the therapy did not rapidly resolve candidemia. The appropriate antifungal therapy for C. lusitaniae-induced purulent thrombophlebitis is uncertain. Further study is desired to seek the appropriate therapy for the disease.
Asunto(s)
Saccharomycetales , Tromboflebitis , Humanos , Antifúngicos/uso terapéutico , Antifúngicos/farmacología , Micafungina , Tromboflebitis/tratamiento farmacológico , Tromboflebitis/diagnósticoRESUMEN
Muckle-Wells syndrome (MWS) is an autosomal dominant autoinflammatory disease recognized as the intermediate phenotype of cryopyrin-associated periodic syndrome (CAPS) caused by NLRP3 gene mutation. It often takes a long time before the diagnosis is made because the clinical presentation of MWS is variable. We report a pediatric case who had had persistently elevated serum C-reactive protein (CRP) level since infancy and was diagnosed with MWS by the development of sensorineural hearing loss in school age. The patient had no periodic symptoms of MWS until the development of sensorineural hearing loss. It is important to differentiate MWS in patients with persistent serum CRP elevation, even if no periodic symptoms, including fever, arthralgia, myalgia and rash, are observed. Furthermore, in this patient, lipopolysaccharide (LPS)-induced monocytic cell death occurred, but to a lesser degree than has been reported in patients with chronic infantile neurological cutaneous, and articular syndrome (CINCA). Because CINCA and MWS are phenotypic variants on the same clinical spectrum, this suggests that a further large-scale study is desired to investigate the association between degree of monocytic cell death and disease severity in CAPS patients.