Methylotroph Infections and Chronic Granulomatous Disease

E. Liana Falcone; Jennifer R. Petts; Mary Beth Fasano; Bradley Ford; William M. Nauseef; João Farela Neves; Maria João Simões; Millard L. Tierce IV; M. Teresa de la Morena; David E. Greenberg; Christa S. Zerbe; Adrian M. Zelazny; Steven M. Holland


Emerging Infectious Diseases. 2016;22(3):404-409. 

In This Article

Patient 5

In 2014, a 36-year-old white man from Georgia, USA, who had X-linked CGD was hospitalized with cervical and abdominal lymphadenopathy; he had 1-year history of fever, malaise, and weight loss. X-linked CGD had been diagnosed when the patient was 6 months of age, when he had had hepatosplenomegaly, cervical lymphadenitis, and recurrent S. aureus infections. Aspergillus spp. pneumonia developed when he was 2 years of age, a liver abscess required incision and drainage when he was 4 years of age, and multiple abdominal abscesses required surgery when he was 6 years of age. Prophylactic trimethoprim/sulfamethoxazole for CGD had been effective until the hospitalization reported here.

A year before admission, the patient had experienced sudden onset of fever, cough, and shortness of breath. Right-sided pleural effusion was treated with antimicrobial drugs and thoracentesis. Cough and dyspnea improved, but fever, fatigue, malaise, myalgia, and weight loss of >15 kg were refractory to hydroxychloroquine and interferon-γ. At 2 months before admission, he had had cervical and abdominal lymphadenopathy with ascites. Peritoneal fluid examination, esophagogastroduodenoscopy, and colonoscopy were not informative. Excisional biopsy of a right posterior cervical lymph node, tuberculin skin testing, and QuantiFERON–TB Gold (QIAGEN, Valencia, CA, USA) testing produced negative results. Ciprofloxacin and metronidazole did not abate symptoms and fever.

At admission, the patient had right supraclavicular and left axillary lymphadenopathy and hepatosplenomegaly. ESR was 88 mm/h, and CRP concentration was 131 mg/L. CT images showed lung scarring, splenomegaly, pericardial effusion, and multifocal adenopathy of the left axilla, mediastinum, celiac, periaortic, retroperitoneal, and mesenteric regions. Culture of excised axillary lymph node grew S. epidermidis, and G. bethesdensis (100% match to the G. bethesdensis type strain by full 16S rRNA gene sequencing, ≈1,500 bp). Etest of G. bethesdensis isolate showed the following MICs in μg/mL: tobramycin (MIC = 4), ceftriaxone (MIC = 32), doxycycline (MIC = 8), trimethoprim/sulfamethoxazole (MIC = 2), and tigecycline (MIC = 16).

The patient was empirically administered vancomycin and ceftriaxone and was discharged with ceftriaxone, doxycycline, prophylactic trimethoprim/sulfamethoxazole, and itraconazole. His medication was eventually switched to cefdinir along with CGD prophylaxis. After 15 months, inflammatory markers and left neck and supraclavicular lymphadenopathy had improved but had not yet normalized.