Fatal Meningitis in Patient with X-Linked Chronic Granulomatous Disease Caused by Virulent Granulibacter bethesdensis
Year: 2019
Type: article
Study design: Case Report
Abstract: Granulibacter bethesdensis is a pathogen reported to cause recurrent lymphadenitis exclusively in persons with chronic granulomatous disease.We report a case of fatal meningitis caused by a highly virulent G. bethesdensis strain in an adolescent in Europe who had chronic granulomatous disease. The StudyThe patient was a 16-year-old boy whose X-linked CGD (CYBB exon 13 deletion) was diagnosed when he was 2 years old.His disease had been well-controlled with cotrimoxazole, itraconazole, and interferon-γ.In September 2014, he was hospitalized with a deep cervical abscess (Figure 1, panel A) and received a 5-week course of intravenous ciprofloxacin, doxycycline, and ceftriaxone that resulted in complete clinical and radiologic resolution, followed by 6 weeks of oral amoxicillin/clavulanic acid, doxycycline, and ciprofloxacin along with his usual prophylaxis.No pathogen was identified despite blood cultures, bronchoalveolar lavage, and lymph node biopsy cultures and broad-range bacterial PCR.After this regimen was completed, the boy was readmitted for 8 weeks with pneumonia with pleural effusion (Figure 1, panel B).Full 16S rRNA gene sequencing (≈1,500 bp) identified Cupriavidus spp. in pleural fluid.He received meropenem, amikacin, ciprofloxacin, teicoplanin, doxycycline, and voriconazole, and his condition improved.However, 2 weeks later, fever returned, along with splenomegaly, hemodynamic instability, pancytopenia, Fatal Meningitis in Patient with X-Linked Chronic Granulomatous Disease Caused byVirulent Granulibacter bethesdensis
Source: Emerging infectious diseases
Language: English
Subfield: Endocrinology
Domain: Life Sciences
Open Access status: diamond
OpenAlex ID: W2935340926