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Ochroconis Gallopavum Infection in a Lung Transplant Recipient: Report of a Case
Ochroconis Gallopavum Infection in a Lung Transplant Recipient: Report of a Case E.I. Brokalaki, U. Sommerwerck, E.H. von Heinegg, and U. Hillen ABSTRACT Disseminated phaeohyphomycoses are rare infections caused by dematiaceous fungi. Ochroconis gallopavum is a neurotropic dematiaceous mold responsible for life-threatening respiratory and central nervous system infections in domestic poultry and in immunologically compromised humans. The world literature describes only 13 previous O gallopavum infections in solid organ transplant recipients. We report herein an O gallopavum phaeohyphomycosis with involvement of skin in a transplant recipient. A 69-year-old white man with a history of bilateral lung transplantation 6 years ago presented with acute onset of severe respiratory distress. Chest X-ray showed no evidence of pneumonia. Lung function showed bronchiolitis obliterans syndrome. Excision biopsy was performed because of a suspected pigmented basal cell carcinoma. Histopathology revealed dermal granulomatous dermatitis, focally suppurative, dominated by epitheloid cells with brownish round fungi. Further microbiological work-up of the biopsy specimen subsequently revealed O gallopavum as the causative organism. No brain involvement was observed. Antimycotic therapy with voriconazole 200 mg twice a day was immediately initiated and the patient was treated for 3 months. Additional intravenous therapy with tobramycin and imipenem was started upon detection of Enterobacter clocae and Enterococci in the sputum. The patient recovered during the next few weeks and was discharged on day 26. ISSEMINATED phaeohyphomycoses are rare infections caused by dematiaceous fungi. Over the past decades, phaeohyphomycoses have been associated with over 100 species and 60 genera of fungi. Their clinical presentation varies greatly, ranging from keratitis to solitary subcutaneous nodules to fulminant disseminated disease.1 Ochroconis gallopavum is a neurotropic dematiaceous mold responsible for life-threatening respiratory and central nervous system infections in domestic poultry and in immunologically compromised humans.1–3 We report herein an O gallopavum phaeohyphomycosis with involvement of skin in a transplant recipient.
D
CASE REPORT A 69-year-old white man with a history of bilateral lung transplantation (LTX) 6 years ago presented with acute onset of severe respiratory distress. His immunosuppressive regimen consisted of tacrolimus, azathioprine, and prednisolone. On physical examination he was found to be cachectic, with erythematous, firm, ulcerative nodules in the right thigh and left lower abdomen (Fig 1a). Chest X-ray showed no evidence of pneumonia. Lung function showed bronchiolitis obliterans syndrome (BOS). His best forced expiratory volume in 1 second (FEV1) after LTX had been 3.9 L
and decreased over the years to 0.49 L. Biochemical laboratory tests revealed leukocytosis (22,000/nL; normal range, 3500 –10,000/ nL), elevated c-reactive protein levels (141.8 mg/L; normal range, ⬍5 mg/L), and stage II renal insufficiency (glomerular filtration rate 63.6 mL/min; normal range, ⬎90 mL/min). Excision biopsy was performed because of a suspected pigmented basal cell carcinoma. Histopathology revealed dermal granulomatous dermatitis, focally suppurative, dominated by epitheloid cells with brownish round fungi (Fig 1b). Periodic acid and Grocott stain showed additionally numerous septated hyphae (Fig 1c and 1d). Further microbiological work-up of the biopsy specimen subsequently revealed O gallopavum as causative organism (Fig 1e). No brain involvement was observed. Antimycotic therapy with voriconazole 200 mg twice a day was immediately initiated and the patient was treated for 3 months. Additional intravenous therapy with tobramycin and imipenem was started on detection of Enterobacter From the Department of Dermatology (E.I.B., U.H.), and the Institute of Medical Microbiology (E.H.v.H.), University Hospital Essen, and the Center of Pneumonology and Thorax Surgery (U.S.), Ruhrlandklinik, Essen, Germany. Address reprint requests to Dr. Eirini I. Brokalaki, MD, Department of Dermatology, University Hospital Essen, Hufelandstraße 55, 45122 Essen, Germany. E-mail: [email protected]
0041-1345/12/$–see front matter http://dx.doi.org/10.1016/j.transproceed.2012.09.007
© 2012 by Elsevier Inc. All rights reserved. 360 Park Avenue South, New York, NY 10010-1710
2778
Transplantation Proceedings, 44, 2778 –2780 (2012)
OCHROCONIS GALLOPAVUM INFECTION
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Fig 1. (A) Ulcerative nodule on the abdominal wall. (B) Hematoxylin-eosin stain with dermal granulomatous dermatitis, focally suppurative, dominated by epitheloid cells with brownish round fungi. (C) Septated hyphae on periodic acid stain and (D) Grocott stain of skin biopsy specimen. (E) Fungal septated branching hyphae isolated on fungal culture from skin biopsy specimen. clocae and Enterococci in the sputum. The patient recovered over the next few weeks and was discharged on day 26 in a slightly stable condition. FEV1 increased to 0.721 (⫹46%). Oxygen requirement decreased from 15 to 3 L/min. He was readmitted 71 days later with a new progression of his BOS requiring 24-hour noninvasive ventilation soon thereafter. The patient requested no further escalation of therapy, was placed on comfort measures, and died 2 days later with no evidence of infection.
DISCUSSION
O gallopavum infections are usually multiorganic processes with central nervous system involvement in approximately 50% of patients.4 The world literature describes only 13 previous O gallopavum infections in solid organ transplant recipients.4,5 Outcomes are excellent in cases with early diagnosis and no central nervous system involvement. The mortality rate otherwise can be as high as 66%.4 Early detection in conjunction with prompt systemic antifungal treatment contributed to the rapid
recovery of our patient. To the best of our knowledge, this is the second report of systemic O gallopavum infection with skin manifestations in a transplant recipient. As in our case, O gallopavum infection may become first evident in the skin. Clinically cutaneous involvement may be uncharacteristic and can mimic a skin tumor such as pigmented basal cell carcinoma or seborrhoic keratosis. Given the increasing incidence of disseminated phaeohyphomycosis reports in recent years,1 such previously rare systemic fungal infection should not be disregarded in the differential diagnosis of severe posttransplantation infections.
REFERENCES 1. Revankar SG, Patterson JE, Sutton DA, et al: Disseminated phaeohyphomycosis: review of an emerging mycosis. Clin Infect Dis 34:467, 2002
2780 2. Singh N, Chang FY, Gayowski T, et al: Infections due to dematiaceous fungi in organ transplant recipients: case report and review. Clin Infect Dis 24:369, 1997 3. Rossmann SN, Cernoch PL, Davis JR: Dematiaceous fungi are an increasing cause of human disease. Clin Infect Dis 22:73, 1996
BROKALAKI, SOMMERWERCK,
VON
HEINEGG ET AL
4. Shoham S, Pic-Aluas L, Taylor J, et al: Transplant-associated Ochroconis gallopava infections. Transpl Infect Dis 10:442, 2008 5. Wong JS, Schousboe MI, Metcalf SS, et al: Ochroconis gallopava peritonitis in a cardiac transplant patient on continuous ambulatory peritoneal dialysis. Transpl Infect Dis 12:455, 2010
D
CASE REPORT A 69-year-old white man with a history of bilateral lung transplantation (LTX) 6 years ago presented with acute onset of severe respiratory distress. His immunosuppressive regimen consisted of tacrolimus, azathioprine, and prednisolone. On physical examination he was found to be cachectic, with erythematous, firm, ulcerative nodules in the right thigh and left lower abdomen (Fig 1a). Chest X-ray showed no evidence of pneumonia. Lung function showed bronchiolitis obliterans syndrome (BOS). His best forced expiratory volume in 1 second (FEV1) after LTX had been 3.9 L
and decreased over the years to 0.49 L. Biochemical laboratory tests revealed leukocytosis (22,000/nL; normal range, 3500 –10,000/ nL), elevated c-reactive protein levels (141.8 mg/L; normal range, ⬍5 mg/L), and stage II renal insufficiency (glomerular filtration rate 63.6 mL/min; normal range, ⬎90 mL/min). Excision biopsy was performed because of a suspected pigmented basal cell carcinoma. Histopathology revealed dermal granulomatous dermatitis, focally suppurative, dominated by epitheloid cells with brownish round fungi (Fig 1b). Periodic acid and Grocott stain showed additionally numerous septated hyphae (Fig 1c and 1d). Further microbiological work-up of the biopsy specimen subsequently revealed O gallopavum as causative organism (Fig 1e). No brain involvement was observed. Antimycotic therapy with voriconazole 200 mg twice a day was immediately initiated and the patient was treated for 3 months. Additional intravenous therapy with tobramycin and imipenem was started on detection of Enterobacter From the Department of Dermatology (E.I.B., U.H.), and the Institute of Medical Microbiology (E.H.v.H.), University Hospital Essen, and the Center of Pneumonology and Thorax Surgery (U.S.), Ruhrlandklinik, Essen, Germany. Address reprint requests to Dr. Eirini I. Brokalaki, MD, Department of Dermatology, University Hospital Essen, Hufelandstraße 55, 45122 Essen, Germany. E-mail: [email protected]
0041-1345/12/$–see front matter http://dx.doi.org/10.1016/j.transproceed.2012.09.007
© 2012 by Elsevier Inc. All rights reserved. 360 Park Avenue South, New York, NY 10010-1710
2778
Transplantation Proceedings, 44, 2778 –2780 (2012)
OCHROCONIS GALLOPAVUM INFECTION
2779
Fig 1. (A) Ulcerative nodule on the abdominal wall. (B) Hematoxylin-eosin stain with dermal granulomatous dermatitis, focally suppurative, dominated by epitheloid cells with brownish round fungi. (C) Septated hyphae on periodic acid stain and (D) Grocott stain of skin biopsy specimen. (E) Fungal septated branching hyphae isolated on fungal culture from skin biopsy specimen. clocae and Enterococci in the sputum. The patient recovered over the next few weeks and was discharged on day 26 in a slightly stable condition. FEV1 increased to 0.721 (⫹46%). Oxygen requirement decreased from 15 to 3 L/min. He was readmitted 71 days later with a new progression of his BOS requiring 24-hour noninvasive ventilation soon thereafter. The patient requested no further escalation of therapy, was placed on comfort measures, and died 2 days later with no evidence of infection.
DISCUSSION
O gallopavum infections are usually multiorganic processes with central nervous system involvement in approximately 50% of patients.4 The world literature describes only 13 previous O gallopavum infections in solid organ transplant recipients.4,5 Outcomes are excellent in cases with early diagnosis and no central nervous system involvement. The mortality rate otherwise can be as high as 66%.4 Early detection in conjunction with prompt systemic antifungal treatment contributed to the rapid
recovery of our patient. To the best of our knowledge, this is the second report of systemic O gallopavum infection with skin manifestations in a transplant recipient. As in our case, O gallopavum infection may become first evident in the skin. Clinically cutaneous involvement may be uncharacteristic and can mimic a skin tumor such as pigmented basal cell carcinoma or seborrhoic keratosis. Given the increasing incidence of disseminated phaeohyphomycosis reports in recent years,1 such previously rare systemic fungal infection should not be disregarded in the differential diagnosis of severe posttransplantation infections.
REFERENCES 1. Revankar SG, Patterson JE, Sutton DA, et al: Disseminated phaeohyphomycosis: review of an emerging mycosis. Clin Infect Dis 34:467, 2002
2780 2. Singh N, Chang FY, Gayowski T, et al: Infections due to dematiaceous fungi in organ transplant recipients: case report and review. Clin Infect Dis 24:369, 1997 3. Rossmann SN, Cernoch PL, Davis JR: Dematiaceous fungi are an increasing cause of human disease. Clin Infect Dis 22:73, 1996
BROKALAKI, SOMMERWERCK,
VON
HEINEGG ET AL
4. Shoham S, Pic-Aluas L, Taylor J, et al: Transplant-associated Ochroconis gallopava infections. Transpl Infect Dis 10:442, 2008 5. Wong JS, Schousboe MI, Metcalf SS, et al: Ochroconis gallopava peritonitis in a cardiac transplant patient on continuous ambulatory peritoneal dialysis. Transpl Infect Dis 12:455, 2010