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 1

In 2008, a 1-year-old girl from Mexico who had p67 phox -deficient CGD was examined for fever, weight loss, and enlarged cervical lymph nodes; she had been receiving ceftriaxone, clindamycin, itraconazole, and interferon-γ for treatment of CGD. CGD was diagnosed when she was 6 months of age, at which time she had Penicillium sp. pneumonia and an abnormal dihydrorhodamine oxidation assay result. At 3 weeks of age, she had had a methicillin-sensitive S. aureus labial abscess, followed at 8 months of age by 3 episodes of pneumonia and an S. marcescens buttock abscess.

A computed tomographic (CT) scan performed at the time of admission showed cervical, mediastinal, and mesenteric lymphadenopathy and a right middle lung lobe infiltrate and hepatosplenomegaly. Excisional cervical lymph node and lung biopsy samples were processed for bacterial, nocardial, fungal, and mycobacterial cultures and staining. Direct Gram staining revealed moderate mononuclear cells but no organisms. After 11 days of incubation on chocolate agar at 37°C, the lung biopsy culture grew 1 pink colony. Species level identification conducted by full 16S rRNA gene sequencing (≈1,500 bp) showed a 99.8% match to the M. lusitanum type strain.[7] Etest (bioMérieux Diagnostics, Marcy l'Etoile, France) showed the following MICs (in μg/mL): amikacin (MIC = 4), cefepime (MIC = 8), ceftriaxone (MIC = 2), ciprofloxacin (MIC = 16), piperacillin-tazobactam (MIC = 2), imipenem (MIC = 2), meropenem (MIC ≥32), trimethoprim/sulfamethoxazole (MIC >32), and aztreonam (MIC >256). Culture of the cervical lymph node biopsy sample grew S. marcescens.

After 7 months of treatment with ceftriaxone, clindamycin, and itraconazole, the patient completely recovered from the pneumonia and cervical lymphadenitis. She subsequently underwent successful hematopoietic stem cell transplant.