A 56-year-old postmenopausal female patient was admitted to another hospital for "multiple dark red painful nodules on the limbs for 2 months". Two months prior, the patient developed several dark red papules, nodules and granulomatous changes in the limbs with no obvious cause. A pathological examination indicated mixed cellular granuloma in the whole dermis, and secretion culture yielded white, smooth, yeast-like colonies. Based on the results of the histopathological examination and secretion culture, the patient was diagnosed with sporotrichosis, and itraconazole (200mg/time, BID) was orally administered.
The symptoms did not resolve, and the patient was admitted to our hospital one month later. The number of nodules on her limbs had increased, and some of the nodules appeared to be due to hyperplasia. Ulcerations with purulent secretions were observed, and the patient reported significant pain and fatigue. Specialist examination findings were as follows: swelling and purulent exudation from the wrist (Figure 1A) to the left forearm (Figure 1B); beaded proliferative nodules from the dorsum of the right hand (Figure 1C) to the right forearm (Figure 1D); and scattered nodules on both knees (Figure 2A) and the left lower limb (Figure 2B). The patient's medical history was as follows: diagnosed with right lateral malleolar tuberculosis with synovitis and anemia 8 years previously; diagnosed with hypothyroidism. She reported stiff fingers, pale and blue fingers when cold for 8 years, dry mouth, dry eyes and fatigue for 2 years. The patient denied any history of trauma or mosquito bites. Upon admission to our hospital, histopathological examination and secretion culture were performed again. The histopathological results showed superficial and deep granulomatous dermatitis with suppuration, multinucleated giant cells (Figure 3A); and negative periodic acid Schiff (PAS) staining (Figure 3B). Acid-fast bacilli were seen on skin biopsy stained with Ziehl-Neelsen stain (Figure 3C). White, smooth colonies were observed in potato dextrose agar (PDA) medium after 1 week (Figure 4). Acid-fast staining of the bacterial suspension demonstrated acid-fast bacilli (Figure 5). The auxiliary examination findings were as follows: antinuclear antibody (ANA) detection: nucleoprotein U1RNP antibody (+++), Sjogren's syndrome A (SSA) antibody (+++), and 52-kDa protein antibody (+++); ANA fluorescence results: nuclear particle type (1:1000 positive) and cytoplasmic particle type (1:3200 positive). The diagnosis was atypical cutaneous mycobacterial infection and SS. The following drugs were administered: 5 mg betamethasone (intramuscular injection), 450 mg rifampicin once/d (oral), and 500 mg clarithromycin once/d (oral). The colonies on the PDA medium were sent to Shenggong Bioengineering (Shanghai) Co., Ltd. Genomic DNA was extracted, amplified and sequenced with 16S rRNA primers (upstream 5'-CagagttTGATCCTGGCT-3', downstream 5'-AggaggTGATCCAGCCGCA-3'). A Basic Local Alignment Search Tool (BLAST) comparison was performed in GenBank, and 100% homology with Mycobacterium chelonae was revealed. The patient was diagnosed with cutaneous Mycobacterium chelonae infection and SS. Due to economic reasons, the patient did not attend a follow-up examination at our hospital. The patient was informed by telephone to change the treatment to 500 mg clarithromycin once/d, and 500mg ciprofloxacin twice/d. Additional telephone follow-ups are in progress.