Table. Summary of methylotroph infections in patients with chronic granulomatous disease*.
Patient no., reference | Patient age, y/sex | CGD genetics† | Clinical findings | Microbiological findings | Treatment‡ | Outcome |
---|---|---|---|---|---|---|
1 (this study) | 1/F | Autosomal recessive (NCF2 c.304C>T; p.R102X) | Pneumonia, cervical lymphadenitis | Methylobacterium lusitanum (lung), Serratia marcescens (lymph node) | CRO/CLI/ITZ, HSCT | Recovered |
2 (this study) | 19/M | X-linked (CYBB intragenic deletion) | Necrotizing cervical lymphadenitis | Granulibacter bethesdensis; Staphylococcus epidermidis | VAN/CRO, CFD/DOX/RIF | Recovered |
3 (this study) | 16/M | X-linked (CYBB 13-exon deletion) | Meningitis | G. bethesdensis | MEM/CIP/AMK/DOX/TEC/VCZ, MEM/CIP/AMK/DOX/CSP/LAMB/LZD/RIF/INH/CLR | Died |
4 (this study) | 9/M | X-linked (CYBB point mutation; exon 10) | Cervical abscess, lymphadenitis | Acidomonas methanolica | TZP/VAN/LAMB/CIP, MEM/VAN/LAMB/CIP, CIP/VCZ/TMPSMX/ IFN-γ, HSCT | Recovered |
5 (this study) | 36/M | X-linked (CYBB c.1139 G>A; p.W380X) | Multifocal lymphadenitis | G. bethesdensis; S. epidermidis | VAN/CRO, CRO/DOX/TMPSMX/ITZ | Still receiving treatment as of 2015 |
6 (4) | 10/M | X-linked (CYBB p.Arg226X) | Necrotizing cervical lymphadenitis | A. methanolica | TMPSMX/CRO/DOX, TMPSMX/RFB/GEN | Recovered |
7 (5) | 10/M | X-linked | Bacteremia | G. bethesdensis | TMPSMX/CAZ/MTZ/LZD/VCZ | Died |
8 (6) | 39/M | X-linked | Necrotizing cervical, mediastinal, axillary lymphadenitis | G. bethesdensis | MEM/DOX, CRO/DOX | |
9 (6) | 36/M | X-linked | Multifocal necrotizing lymphadenitis, splenic lesions, ascites | G. bethesdensis | MEM/TMPSMX/ITZ, MEM/TMPSMX/VCZ/TOB, CRO/TMPSMX/IFN-γ, CPD/DOX/TMPSMX/IFN-γ, splenectomy/TGC | Recovered |
10 (6) | 13/M | X-linked | Necrotizing thoracic lymphadenitis | G. bethesdensis, S. epidermidis, Candida glabrata, Streptococcus mitis group | MEM/VCZ, CRO/TOB/DOX/VCZ, CRO, DOX, CRO, CFD | Recovered |
11 (6) | 17/M | X-linked | Necrotizing cervical and mediastinal lymphadenitis | G. bethesdensis | LVX, DOX, lymph node excision | Recovered |
12 (6) | 37/M | X-linked | Necrotizing supraclavicular lymphadenitis, splenic and liver lesions | G. bethesdensis | CRO/GEN/VAN, CRO/DOX/TMPSMX, CFD/ DOX/TMPSMX, DOX/TMPSMX | Recovered |
*AMK, amikacin; CAZ, ceftazidime; CFD, cefdinir; CGD, chronic granulomatous disease; CLI, clindamycin; CLR, clarithromycin; CIP, ciprofloxacin; CPD, cefpodoxime; CRO, ceftriaxone; CSP, caspofungin; CYBB, cytochrome b-245, β polypeptide; DOX, doxycycline; GEN, gentamicin; HSCT, hematopoietic stem cell transplant; IFN-γ, interferon- γ; INH, isoniazid; ITZ, itraconazole; LAMB, liposomal amphotericin B; LVX, levofloxacin; LZD, linezolid; MEM, meropenem; MTZ, metronidazole; NCF2, neutrophil cytosolic factor 2; RFB, rifabutin; RIF, rifampin; TEC, teicoplanin; TGC, tigecycline; TMPSMX, trimethoprim/sulfamethoxazole; TOB, tobramycin; VAN, vancomycin; VRZ, voriconazole. †Information in parentheses indicates, when known, the mutation that led to CGD. ‡Slashes separate drugs in the same regimen; commas separate regimens.