Co-existence of cutaneous and presumptive pulmonary alternariosis

A 71-year-old man presented with an asymptomatic lesion on the arm. He was born in rural West Virginia, spent several years in military service in the South Pacific, and is a long-term resident of Maine. He has chronic obstructive pulmonary disease, coronary artery disease, and noninsulin-dependent diabetes melitus. He was evaluated for episodes of hemoptysis by bronchoscopy in 1980 and 1984, but no cause was established. His usual medications included prednisone 10 mg daily. One month prior to the dermatology consultation the patient had been admitted to another hospital because of a flare-up of his lung disease. He was treated for 10 days with intravenous steroids and bronchodilators, resulting in improvement of his respiratory status. A cardiac pacemaker was inserted because of arrythmias. By the time of discharge, 1 week later, the prednisone dose had been tapered to his usual 10 mg daily. One week prior to the dermatology consultation, he was admitted to this institution for evaluation of recurrent atrial fibrillation. On admission, his chest X-ray revealed a pulmonary nodule which had not been present 1 month previously (Fig. la). The dermatology service was asked to evaluate an enlarging lesion on the right arm. The arm lesion began 2 weeks earlier as a group of papules which grew and coalesced. Two days prior to evaluation, the lesion had spontaneously drained purulent material. There was no pruritus or pain. The patient did not recall any similar skin problem in the past and had no history of trauma to the area. He reported a 30 Ib weight loss in the 2 months prior to admission despite a good appetite. There was no history of fever or malaise. On examination, a 3.3 × 1.5 cm plaque was noted on the right extensor forearm just above the wrist (Fig. 2). The plaque was erythematous with a shiny atrophic epidermis and fine scale. One portion of the plaque had a red-yellow hue and was indurated while the other had a grey-blue pigmented appearance. The remainder of his skin examination was unremarkable. There was no adenopathy or hepatosplenomegaly. Routine laboratory tests were significant for hyperglycemia, a mildly elevated white blood cell count of 11.5 K/UL with a normal differential; lactate dehydrogenase was 325 U/ L (normal, 88–196) and alkaline phosphatase 113 U/L (normal, 16–95). CT scan of the chest revealed emphysema and multiple bilateral pulmonary nodules with cavitation (Fig. 1b). A low attenuation region in the spleen was interpreted as representing an inflammatory or infectious focus, or, less likely, a cyst. CT scan of the abdomen revealed a normal liver and no abdominal adenopathy. Bone scan and ophthalmologic examination revealed no evidence of bony or ocular involvement. A 4-mm punch biopsy from the plaque on the wrist revealed suppurative granulomatous inflammation extending from the lower papillary dermis down to the deep dermis at the base of the biopsy specimen (Fig. 3a). Many multinucleated giant cells were observed. The overlying epidermis showed slight acanthosis with compaction and focal parakeratosis of the stratum corneum. In the dermis, numerous rounded structures and septate hyphae, measuring 5–9 microns in diameter, were present both within giant cells and extracellularly (Fig. 3b). These structures were stained by a Grocott methenamine silver stain, consistent with fungi (Fig. 3c). These findings, and the lack of encapsulation typically seen in subcutaneous phaeohyphomycosis, suggest a systemic fungal disease rather than direct cutaneous inoculation.8 A second specimen was sent for culture to the University of Texas Health Science Center in San Antonio and grew Alternaria alternata (no further description is available). In retrospect, the pigment seen clinically may have resulted either from the fungus, which is typically pigmented, or from an ecchymosis. Sensitivity testing revealed that the fungus was susceptible to amphotericin 0.29 μg/mL, itraconazole 0.07 μg/mL, and ketoconazole 0.1 μg/mL. Serum titers for complement-fixing antibodies to aspergillus and blastomyces were negative. Although no specific antifungal therapy was started, follow-up chest X-rays showed a dramatic decrease in the size of the pulmonary nodules (Fig. 1c). During the same week, the plaque on the arm also decreased in size to 2.7 × 1.4 cm. The alkaline phosphatase decreased to normal and the lactate dehydrogenase decreased to 261. The patient was discharged home without antifungal therapy, and prednisone was discontinued 10 days after discharge. The skin lesion resolved spontaneously, in the 2 years since presentation there has not been a recurrence of skin or lung lesions.