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Atypical presentation of an uncommon pathogen
oleh: Amada Sanchez-Gonzalez, Hannah Fox, Manjusha Narayanan, Julie Samuel, Ulrich Schwab, Christopher Duncan
| Format: | Article |
|---|---|
| Diterbitkan: | Elsevier 2022-01-01 |
Deskripsi
Establishing a definitive diagnosis of Nocardia infection can be challenging due to its rarity, its diverse clinical presentation and the limitations in cultivating this microorganism. Only two cases of laryngeal nocardiosis have previously been reported, both in immunocompromised patients. We describe the case of a 67-year-old seemingly immunocompetent wildlife photographer, with an extensive travel history to Asia and Central America, who presented with an 8 month history of hoarse voice and odynophagia. An initial CT had revealed the presence of a laryngeal “mass” suggestive of malignancy, but several biopsies showed granulomatous inflammation. These were not sent for microbiological investigation. Empirical high dose steroids -started due to concerns of airway compromise- did not significantly improve his symptoms. Further laryngeal biopsies led to the isolation of a fungal mold (Chaetomium spp) of uncertain relevance. Voriconazole was initiated but not tolerated. A repeat biopsy was arranged: Fungal cultures were negative, however histological examination revealed branching gram positive bacteria. Nocardia spp was isolated on mycobacterial culture media and confirmed by PCR. Speciation was inconclusive. Co-trimoxazole was continued for 6 months with good clinical and radiological response. Treatment was discontinued at the patient's request, but within two months he relapsed with worsening dysphagia and odynophagia accompanied by weight loss. He was found to have developed a further post-cricoid collection with associated necrosis. Co-trimoxazole was restarted for 12 months followed by life-long secondary prophylaxis. He remains well with no further recrudescence.This case illustrates the many challenges in the diagnosis and treatment of nocardiosis.Abstract without diagnosisEstablishing a definitive diagnosis of infection with this microorganism can be challenging due to its rarity, its diverse clinical presentation and the limitations in cultivating it. Only two cases of laryngeal infection have previously been reported, both in immunocompromised patients. We describe the case of a 67-year-old seemingly immunocompetent wildlife photographer, with an extensive travel history to Asia and Central America, who presented with an 8 month history of hoarse voice and odynophagia. An initial CT had revealed the presence of a laryngeal “mass” suggestive of malignancy, but several biopsies showed granulomatous inflammation. These were not sent for microbiological investigation. Empirical high dose steroids -started due to concerns of airway compromise- did not significantly improve his symptoms. Further laryngeal biopsies led to the isolation of a fungal mold (Chaetomium spp) of uncertain relevance. Voriconazole was initiated but not tolerated. A repeat biopsy was arranged: Fungal cultures were negative, however histological examination revealed a pathogen through Gram stain. This microorganism was further isolated on mycobacterial culture media and confirmed by PCR. Speciation was inconclusive. Tailored treatment was continued for 6 months with good clinical and radiological response. Treatment was discontinued at the patient's request, but within two months he relapsed with worsening dysphagia and odynophagia accompanied by weight loss. He was found to have developed a further post-cricoid collection with associated necrosis. Once he completed 12 months of treatment, life-long secondary prophylaxis ensued. He remains well with no further recrudescence.