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 2

In 2011, a 19-year-old white man from Ohio, USA, who had X-linked CGD was examined for right neck swelling and tenderness (2 weeks' duration), a yellow ulcerated lesion on the right side of the hard palate, and an enlarged right tonsil with copious exudate. He had been receiving prophylactic trimethoprim-sulfamethoxazole and posaconazole. Erythrocyte sedimentation rate (ESR) was 28 mm/h, and C-reactive protein (CRP) concentration was 82 mg/L.

CGD had been diagnosed at birth on the basis of a positive family history. The patient had had hydrocephalus, catheter-associated fungal meningitis, and Aspergillus fumigatus pneumonia. When he was 15 years of age, CGD proctitis developed. Fourteen months before hospital admission, he had undergone right neck dissection for Rothia aeria infection, which was successfully treated with β-lactams.[8]

CT images showed new bulky lymphadenopathy in the right neck, involving all nodal planes, and increased thickening and asymmetry of the right oropharynx with hypoattenuation of the right palatine tonsils. Culture of the right tonsillar exudate and empirical treatment with meropenem were not helpful. Antimicrobial therapy was switched to ceftriaxone and high-dose penicillin for empirical coverage of G. bethesdensis and Actinomyces spp. Right neck dissection with tonsillectomy yielded Staphylococcus epidermidis, and full 16S rRNA gene sequencing (≈1,500 bp) of 1 colony of a gram-negative bacillus showed a 99.8% match to the G. bethesdensis type strain. Nine weeks of vancomycin and ceftriaxone followed by 8 weeks of cefdinir, doxycycline, and rifampin led to complete resolution of the lymphadenitis.