Pulmonary Pseudallescheria Boydii Infection After Near Drowning
Pulmonary Pseudallescheria Boydii Infection After Near Drowning
Pseudallescheria boydii is a ubiquitously occurring fungus. While rarely causing opportunistic infection in humans, it is the most common cause of fungal pneumonia in cases of near drowning, and is associated with high mortality. P boydii typically causes cutaneous mycetomas but may invade the lungs or brain. P boydii infections are difficult to treat due to amphotericin B resistance and frequent need for surgical resection. Zygomycetous infections, often referred to as mucormycoses, usually occur in immunocompromised hosts, trauma or burn victims. Like P boydii, these organisms are found on decaying vegetation and in soil. Zygomycetous infections generally require debridement and prolonged amphotericin B. We report a case of P boydii pneumonia with a simultaneous brain lesion and cutaneous mucormycosis in a near drowning patient. The pneumonia responded to treatment with voriconazole and the brain lesion resolved without surgery. The cutaneous mucormycosis responded to surgery and amphotericin B. This is the first documented case of simultaneous invasive P boydii and cutaneous mucormycosis successfully treated with dual systemic antifungal therapy and resection.
A 42-year-old male with underlying hepatitis C drove into a polluted Louisiana bayou in late May. Witnessed submersion time was 8 minutes. His initial Glascow Coma Score (GCS) was three. Tracheal suction showed mud and blood before intubation.
Physical examination revealed a slender male, sedated and intubated but responsive with a GCS of 12. The physical examination was unremarkable except for diffuse wheezing. Initial hypoxemia required a fractional inspiratory oxygen of 100%. His white blood cell count was 4,300/mm, while hemoglobin, hematocrit, and platelet counts were normal. Urine toxicology detected cocaine and cannabinoids.
The chest radiograph (Fig. 1) showed diffuse reticulonodular opacities. Intravenous steroids were administered for presumed asthma. The patient was extubated on hospital day 3, but became febrile. He was treated empirically with antibiotics and fluconazole. When the sputum culture yielded Pseudomonas aeruginosa, the antibiotic regimen was changed to ciprofloxacin and piperacillin/tazobactam. On day 8, a chest radiograph demonstrated new cavitary lesions throughout the lung fields (Fig. 2). A transthoracic echocardiogram was negative as were sputum cultures for acid-fast bacilli. New cutaneous nodules over the abdomen, left chest wall and left upper arm were observed soon thereafter (Fig. 3a). These were incised and the drainage sent for stains and cultures, which were nondiagnostic.
(Enlarge Image)
Initial chest radiograph showing diffuse reticulonodular opacities.
(Enlarge Image)
Chest radiograph on hospital day 8 showing multiple ill-defined nodules of varying sizes scattered throughout both lungs.
(Enlarge Image)
A, left chest wall lesion one week after incision and drainage. B, left chest wall several months after wide excision and amphotericin B treatment.
On day 10, consultation was obtained for persistent fevers. Repeated blood and urine cultures remained negative. Antibiotics were discontinued except ciprofloxacin. Continued fevers prompted adding amphotericin B to cover fungal pathogens.
Axial computed tomography (CT) confirmed multiple cavitary lesions throughout both lungs (Fig. 4a) but a transesophageal echocardiogram was negative for vegetations. The previously drained nodules did not heal and a punch biopsy revealed broad, aseptate hyphae consistent with a zygomycete (Fig. 5). Because the most common fungal etiology of near drowning pneumonia is P boydii, an organism resistant to amphotericin B, bronchoscopy was performed to assess for a common pulmonary and cutaneous pathogen. Sputum cultures finally yielded heavy growth of P boydii (Fig. 6). Itraconazole was initiated while compassionate-use voriconazole was requested. The known predilection of P boydii to invade the central nervous system (CNS) prompted brain magnetic resonance imaging (MRI), revealing an 0.8 cm lesion in the right parietal white matter (Fig. 7a). An MRI one week later revealed a 50% increase in the size of the lesion (Fig. 7b). Neurosurgical resection was unnecessary, as MRI revealed resolution of the lesion following several weeks of voriconazole. Follow-up CT scans documented improvement of pulmonary lesions several weeks into therapy (Fig. 4b). Fungal cultures from the cutaneous lesions repeatedly failed to yield the suspected zygomycete, but an antibody test performed by the Centers for Disease Control confirmed a Rhizomucor species.
(Enlarge Image)
A, axial CT scan of the chest revealing multiple cavitary lesions with irregular walls. B, axial CT scan revealing near-resolution of the cavitary lesions after three weeks of voriconazole.
(Enlarge Image)
Broad aseptate hyphae of Rhizomucor from punch biopsy of left arm lesion. (A, hematoxylin and eosin stain, B, Gomori-Methenamine silver stain; original magnification × 400.)
(Enlarge Image)
Wet mount of sputum culture revealing narrow, septated hyphae of P boydii. (Lactophenol cotton blue dye; original magnification × 200.)
(Enlarge Image)
A, MRI scan of the brain showing an 0.8 cm enhancing lesion in the white matter of the right frontal lobe. B, brain MRI scan one week later showing an increase in the size of brain lesion to 1.2 cm.
The patient was discharged on day 68 on oral voriconazole. He declined further therapy with amphotericin B and was re-admitted two months later with recurrent lesions of the groin, thigh and gluteal region, which were successfully excised. He has remained well with regular follow-up after one year of voriconazole.
Pseudallescheria boydii is a ubiquitously occurring fungus. While rarely causing opportunistic infection in humans, it is the most common cause of fungal pneumonia in cases of near drowning, and is associated with high mortality. P boydii typically causes cutaneous mycetomas but may invade the lungs or brain. P boydii infections are difficult to treat due to amphotericin B resistance and frequent need for surgical resection. Zygomycetous infections, often referred to as mucormycoses, usually occur in immunocompromised hosts, trauma or burn victims. Like P boydii, these organisms are found on decaying vegetation and in soil. Zygomycetous infections generally require debridement and prolonged amphotericin B. We report a case of P boydii pneumonia with a simultaneous brain lesion and cutaneous mucormycosis in a near drowning patient. The pneumonia responded to treatment with voriconazole and the brain lesion resolved without surgery. The cutaneous mucormycosis responded to surgery and amphotericin B. This is the first documented case of simultaneous invasive P boydii and cutaneous mucormycosis successfully treated with dual systemic antifungal therapy and resection.
A 42-year-old male with underlying hepatitis C drove into a polluted Louisiana bayou in late May. Witnessed submersion time was 8 minutes. His initial Glascow Coma Score (GCS) was three. Tracheal suction showed mud and blood before intubation.
Physical examination revealed a slender male, sedated and intubated but responsive with a GCS of 12. The physical examination was unremarkable except for diffuse wheezing. Initial hypoxemia required a fractional inspiratory oxygen of 100%. His white blood cell count was 4,300/mm, while hemoglobin, hematocrit, and platelet counts were normal. Urine toxicology detected cocaine and cannabinoids.
The chest radiograph (Fig. 1) showed diffuse reticulonodular opacities. Intravenous steroids were administered for presumed asthma. The patient was extubated on hospital day 3, but became febrile. He was treated empirically with antibiotics and fluconazole. When the sputum culture yielded Pseudomonas aeruginosa, the antibiotic regimen was changed to ciprofloxacin and piperacillin/tazobactam. On day 8, a chest radiograph demonstrated new cavitary lesions throughout the lung fields (Fig. 2). A transthoracic echocardiogram was negative as were sputum cultures for acid-fast bacilli. New cutaneous nodules over the abdomen, left chest wall and left upper arm were observed soon thereafter (Fig. 3a). These were incised and the drainage sent for stains and cultures, which were nondiagnostic.
(Enlarge Image)
Initial chest radiograph showing diffuse reticulonodular opacities.
(Enlarge Image)
Chest radiograph on hospital day 8 showing multiple ill-defined nodules of varying sizes scattered throughout both lungs.
(Enlarge Image)
A, left chest wall lesion one week after incision and drainage. B, left chest wall several months after wide excision and amphotericin B treatment.
On day 10, consultation was obtained for persistent fevers. Repeated blood and urine cultures remained negative. Antibiotics were discontinued except ciprofloxacin. Continued fevers prompted adding amphotericin B to cover fungal pathogens.
Axial computed tomography (CT) confirmed multiple cavitary lesions throughout both lungs (Fig. 4a) but a transesophageal echocardiogram was negative for vegetations. The previously drained nodules did not heal and a punch biopsy revealed broad, aseptate hyphae consistent with a zygomycete (Fig. 5). Because the most common fungal etiology of near drowning pneumonia is P boydii, an organism resistant to amphotericin B, bronchoscopy was performed to assess for a common pulmonary and cutaneous pathogen. Sputum cultures finally yielded heavy growth of P boydii (Fig. 6). Itraconazole was initiated while compassionate-use voriconazole was requested. The known predilection of P boydii to invade the central nervous system (CNS) prompted brain magnetic resonance imaging (MRI), revealing an 0.8 cm lesion in the right parietal white matter (Fig. 7a). An MRI one week later revealed a 50% increase in the size of the lesion (Fig. 7b). Neurosurgical resection was unnecessary, as MRI revealed resolution of the lesion following several weeks of voriconazole. Follow-up CT scans documented improvement of pulmonary lesions several weeks into therapy (Fig. 4b). Fungal cultures from the cutaneous lesions repeatedly failed to yield the suspected zygomycete, but an antibody test performed by the Centers for Disease Control confirmed a Rhizomucor species.
(Enlarge Image)
A, axial CT scan of the chest revealing multiple cavitary lesions with irregular walls. B, axial CT scan revealing near-resolution of the cavitary lesions after three weeks of voriconazole.
(Enlarge Image)
Broad aseptate hyphae of Rhizomucor from punch biopsy of left arm lesion. (A, hematoxylin and eosin stain, B, Gomori-Methenamine silver stain; original magnification × 400.)
(Enlarge Image)
Wet mount of sputum culture revealing narrow, septated hyphae of P boydii. (Lactophenol cotton blue dye; original magnification × 200.)
(Enlarge Image)
A, MRI scan of the brain showing an 0.8 cm enhancing lesion in the white matter of the right frontal lobe. B, brain MRI scan one week later showing an increase in the size of brain lesion to 1.2 cm.
The patient was discharged on day 68 on oral voriconazole. He declined further therapy with amphotericin B and was re-admitted two months later with recurrent lesions of the groin, thigh and gluteal region, which were successfully excised. He has remained well with regular follow-up after one year of voriconazole.