A 52-year-old female from a remote village in southern India, who is a known case of uncontrolled type 2 diabetes and hypothyroidism for the past 5 years, presented with a 1-year history of pain over her left shoulder and 2 months of increasing swelling of the same shoulder after a trivial fall. She had undergone an arthroscopic rotator cuff repair for the same side in 2019, with metal and bioabsorbable suture anchors from elsewhere. After the surgery, she was doing well with her shoulder, except for the occasional pain, that aggravated to the present status in 2 years. Owing to the COVID-19 pandemic, she did not have regular post-operative follow-ups. In March 2021, she had a trivial fall over the same shoulder, and since then, she noticed an increasing swelling around the same shoulder, which grew from the size of a lemon to a half-cut watermelon spanning in between her neck and affected shoulder (Figure 1).
Owing to the raging COVID situations in India, she could not attend a specialist hospital and took treatment at a local bonesetter with herbal massages until she presented here. She also had a few courses of broad-spectrum antibiotics from local hospitals with no resolution of her symptoms. The swelling looked fiery red at the time of presentation and had a local rise in temperature, and her shoulder movements were painful. There was no distal neurovascular deficit. There was no history of fever, chills or rigour, loss of weight or loss of appetite, or evening rise of temperature, or involvement of any other joints. There was no past or contact history of Tuberculosis (TB) nor any history of recent COVID infection. The pre-operative evaluation revealed normal leukocyte and neutrophil counts but raised C-reactive protein (CRP) - 67.6 and a normal erythrocyte sedimentation rate (ESR) - 06 mm. Her Hba1c was 10 depicting her uncontrolled diabetic status. Her blood culture was negative. An X-ray revealed bony destruction at the lateral end of the clavicle with the metal anchor in situ in the shoulder (Figure 2). Magnetic resonance imaging (MRI) revealed a large fluid collection with few septations in the subcutaneous tissue in the supraclavicular region communicating to the acromioclavicular (AC) joint, which was widened and eroded. Moderate glenohumeral joint effusion extending into the subacromial and subdeltoid spaces were also reported suggestive of corresponding bursitis (Figure 3). Our differential diagnosis where pyogenic/tubercular/fungal septic arthritis, which are the usual afflictions.
The patient underwent swelling excision in toto and debridement of the left acromioclavicular and shoulder joint (Figures 4, 5, and 7). Surprisingly the whole sac of swelling was traced from the subcutaneous plane through the AC joint to the subacromial and subdeltoid bursae up to the humeral head entry site for the metal anchor. Around 500 mL of straw yellow pus was drained out of the excised sac and from the shoulder joint (Figure 6). The deep-seated metal anchor in the humeral head was surrounded by a nidus of osteomyelitis sequestrum, which was debrided and retrieved along with the metal and bio absorbable anchors (Figure 8).
Multiple specimens of infected tissue and pus were sent for histopathology, aerobic and anaerobic bacterial cultures, acid-fast bacteria (AFB), gram staining, TB GeneXpert, and Fungal DNA-PCR. The bacterial and tubercular reports were all negative and eventually, the fungal DNA detection by PCR was positive for Debaryomyces subglobosus, an extremely rare fungi to infect the shoulder joint, even in immunocompromised patients. HPE was consistent with chronic granulomatous infection (Figures 9 and 10). The patient was started on Voriconazole, the specific anti-fungal for the same and got a complete resolution of her symptoms within 6 weeks of initiation of treatment. The serum markers for infection, CRP and ESR, returned to normal within this period, and her surgical wound healed completely.
However, her functional recovery was lagging behind, even at two months follow-up, owing to the chronicity of the infection and the residual damages it has already made to the rotator cuff muscles and the joint.