Mycetoma

INTRODUCTION: Pseudoallescheria boydii is a ubiquitous fungus present in soil, sewage and polluted waters. Although the clinical spectrum is wide, the most frequent disease in immunocompetent individuals is a mycetoma. An increasing number of cases of disseminated infections are reported in immunocompromised patients yet a review of the lung transplant literature yields relatively few cases of disseminated infection. The clinical and histopathological presentation of Psedoallescheria boydii infection is similar to Aspergillus spp and Fusarium spp; therefore culture is necessary to make the correct diagnosis. This is especially relevant since infection due to Psedoallescheria boydii is often resistant to antimycotic drugs such as Amphoterecin B and Flucytosine, which are commonly used to treat Aspergillus infections.

CASE PRESENTATION: A 57-year-old female patient, status post a right single lung transplant 14 months ago for COPD, presented to the hospital with complaints of severe left mastalgia. She had bilateral silicone breast implants placed twenty five years ago. She was maintained on routine triple drug therapy consisting of Tacrolimus, Mycophenolate mofetil and Prednisone. She was also on routine opportunistic infection prophylaxis with Itraconazole, Valgancyclovir and Trimethoprim-Sulfamethaoxazole. Recently the patient was diagnosed with Grade A1B0 acute rejection on surveillance bronchoscopy which had not resolved inspite of two treatments with pulse steroids. The patient reported a one week history of severe, continuous left breast pain with concomitant tight sided pleuritic chest pain. In addition she had developed painful skin lesions on both thighs and blurting of vision. On examination the left breast was swollen, warm and exquisitely tender to touch around the implant capsule.

She had multiple skin lesions which were approximately one centimeter in diameter, red, warm, tender and nonblanching. Neurological exam revealed ptosis of the left eyelid and third cranial nerve palsy. CT scan chest showed a loculated right pleural effusion suggestive of empyema and a persistent right lower lobe parenchymal nodule. Skin biopsy from the thigh lesion revealed a fungal abscess with pseudohyphae consistent with Pseudoallescheria spp. An MRI breast revealed marked inflammatory enhancement around the left silicone breast implant. Bilateral breast implants were emergently surgically removed. Microscopic evaluation of the left implant capsule demonstrated evidence of invasion by fungal hyphae. MRI brain revealed multiple ring enhancing lesions in the parenchyma suggestive of a hematogenously disseminated infection. The patient was treated with high dose intravenous Voriconazole. Multiple tissue cultures grew Pseudoallescheria boydii. We suspect that the initial nidus was the pulmonary nodule, with hematogenous dissemination at a time of increased immunosuppression. We were unable to identify a definite exposure inspite of extensive questioning. This makes it difficult to predict any future re-infection.

DISCUSSIONS: P.boydii may cause life-threatening illness in the lung transplant population. Early diagnosis should be done by fungal culture so that adequate therapy may be initiated. In the pre Voriconazlole era, disseminated infection in the lung transplant population was uniformly fatal. However in recent years there have been some treatment successes with the use of Voriconazole. To our knowledge this is the first reported case of breast implant infection with Psedoallescheria boydii in the lung transplant population. This raises questions regarding the safety of these foreign bodies in an immunosuppressed population prone to opportunistic infections. There may be a case here for removal of such foreign bodies prior to transplant. We reviewed our lung transplant database but could not identify any other case of breast implant infection.

CONCLUSION: Cosmetic foreign bodies in a lung transplant recipient have the potential to be infected, due to the immune suppressed nature of the patient. There should be a low threshold to remove such implants during the pre-transplant work up, if agreeable to the patient.

DISCLOSURE: Hina Sahi, None.

Hina Sahi MD * Marie Budev DO Robin Avery MD Cleveland Clinic Foundation, Cleveland, OH

COPYRIGHT 2005 American College of Chest Physicians
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