A 37-year-old male of Filipino descent who had lived in Bakersfield, California, for three years before presentation, with a history of Lynch syndrome, glucose 6-phosphate deficiency, hypertriglyceridemia, and essential hypertension, presents with a chief complaint of a perianal lesion that had started one month prior. He described the lesion as enlarging, extending to the scrotum with a "C" shape, and associated with rectal pain with poor response to analgesics. He denied having constitutional, neurologic, pulmonary, musculoskeletal, or urinary symptoms. Before this visit, he had a colonoscopy six years prior for his Lynch syndrome without abnormal findings. He denied the use of tobacco, alcohol, or illicit drugs. He was not sexually active, identified as heterosexual, denied a history of anal intercourse, and denied any trauma to the anal and perineal areas. His travel history included going to Pennsylvania one year before the visit, and he was studying for pilot school. He denied known exposures to tuberculosis.
His physical exam demonstrated vital signs within normal limits and a semicircular right firm perianal lesion extending into the perineal and scrotal area; it was erythematous, edematous, and tender to palpation. No masses on digital rectal examination (DRE) or bleeding were noted, with normal sphincter tone. The initial laboratories showed: a complete blood count, comprehensive metabolic panel, hemoglobin A1C within normal limits, and a 4th generation human immunodeficiency virus (HIV) test that was negative.
During the initial visit, he was prescribed seven days of ciprofloxacin. In his follow-up visit, he reported that the mass was growing, and an MRI was performed to evaluate it in detail. A pelvic MRI with contrast showed an extensive 8.6 cm multiloculated thick-walled fluid collection (Fig. 1) within the right perineum consistent with an abscess with inflammatory changes extending towards the left peritoneum, right ischio-anal fossa, and prostate. Focal fluid collections within the prostate were suggestive of abscesses. Several small anal fissures did not appear to communicate with the fluid accumulation. After those results returned, he was scheduled for a new appointment, although due to worsening pain, he decided to come to the emergency department (approximately 3 weeks later after the initial visit), and an emergent surgery was performed. The surgery team performed an incision and drainage. The right perianal abscess cavity tracked anteriorly with induration surrounding and within the anal canal. The bacterial culture grew Klebsiella pneumoniae (susceptible to all tested antibiotics in the antibiogram, including ciprofloxacin). Pathology studies from the abscess cavity demonstrated skin and subcutaneous tissue with marked acute and chronic inflammation and reactive changes. The primary team did not order fungal stains in the tissue sample. He was discharged home on ciprofloxacin 500 mg orally twice daily and metronidazole 500 mg thrice daily. After his discharge, he started to have generalized abdominal pain and increased (3 times a day) watery bowel movements without blood or mucus; the symptoms resolved without any new medication after two weeks. Fifteen days later, fungal cultures of the drained material grew Coccidioides immitis.
Coccidioides serologies resulted in a positive immunodiffusion test and complement fixation (CF) titer of > 1:4096. A chest tomography showed a right lung cluster of nodular opacities. A bone scan did not show any bone lesions; however, there was a pleural-based soft-tissue density in the medial right upper lung (16 x15 mm) adjacent to the posterior right 6th rib. Based on his clinical presentation, serologic studies, and culture results, we presumed Coccidioides infection as the source of the pulmonary, pleural, and prostatic lesions. We started fluconazole 800 mg daily for disseminated coccidioidomycosis. After the incision and drainage, the patient reported immediate improvement in the pain and sensation of fullness. During his initial follow-up, he denied any pulmonary, constitutional, urinary, or recurrent perianal symptoms while on antifungal therapy. In the physical exam, the erythema and edematous appearance of the perineal skin resolved.
After starting fluconazole, he reported persistent nausea, abdominal pain, and dry oral mucosa. Due to this, the patient was changed to posaconazole delayed-release tablets 300 mg daily. After one week (and approximately three months after initial perianal abscess drainage), the patient came to the emergency department with fevers, chills, conjunctival injection, and headaches and tested positive for SARS-CoV-2. After two weeks, most of the symptoms disappeared. Although the headache persisted, it was frontal and bilateral, not associated with changes in mental status, neck pain, neck stiffness, photophobia, changes in vision, focal motor/sensory deficits, rhinorrhea, or sinus pain. CT of the head without contrast did not show any abnormalities.
A lumbar puncture was performed and showed opening pressure, cell count, protein, and glucose within normal limits, with negative bacterial and fungal cultures. Serum Coccidioides complement fixation titer decreased to 1:1. One month later, the patient reported high blood pressure (170/80 mmHg) despite taking his medications appropriately. The posaconazole level was elevated (5.78 mcg/mL). We suspected posaconazole-induced pseudo hyperaldosteronism (PIPH) because of the elevated posaconazole level, hypertension, undetectable aldosterone blood levels, blood renin levels close to the inferior limit (0.6 ng/mL/hour), and potassium serum levels in the inferior border of the range (3.2 mEq/L).
We decreased the dose to 200 mg PO daily and his blood pressure subsequently normalized. The patient lost to follow-up after that visit. The patient was scheduled for a follow-up pulmonary study. However, it is unclear if he will come back.
If he returns to our institution, we will place the patient on 12-24 months of therapy. If his symptoms are absent and there is radiological resolution of the lesions after that interval, we would discontinue antifungal therapy and place him in a period of close observation to evaluate symptoms and CF titers. We would avoid indefinite therapy in the absence of CNS disease. If the patient requires immunosuppressive therapy or acquired/developed a cellular immunodeficiency, we will recommend secondary prophylaxis.