History. Mr. J, a 43-year-old otherwise healthy man (no underlying disease), was admitted to the authors’ facility, a large comprehensive hospital with 2900 beds, with a nonhealing ulcer on his left chest that was discharging purulent drainage. The patient had begun to feel soreness without apparent cause in his upper left chest 15 months earlier, and a focal lump (2 cm × 2 cm) without tenderness could be palpated. Approximately 1 month later, the mass gradually enlarged, and Mr. J experienced intermittent fever and was hospitalized successively in different hospitals where lymph node biopsy and ulcer exudate, as well as smears, microbial culture, acid-fast staining, and tuberculous/nontuberculous mycobacteria nucleic acid testing, were negative for bacterial or fungal infection.
Several pathological examinations of the wound and adjacent lymph nodes involving immunohistochemical staining and biopsy revealed vascular proliferation accompanied by the infiltration of inflammatory cells. After incision and drainage, Mr. J’s chest and neck wounds failed to heal but large amounts of yellowish exudate were present and samples were sent for microbiological cultures to look for possible bacteria or fungi, with negative results. Mr. J was treated successively with antibiotics including cephalosporin, quinolone, and antituberculosis drugs appropriate to the infectious manifestations but he still developed intermittent fever. A sterile gauze sponge was applied for drainage and the wound was dressed with gauze, foam, alginate, and silver dressings in succession; nothing brought about wound healing.
After approximately 14 months of treatment in 5 hospitals, Mr. J was transferred to the authors’ hospital. Mr. J was running an intermittent body temperature of 36.4˚ C to 38.5˚ C, his vital signs were stable, he was conscious, and the wound did not affect activities of daily living. He reported only mild pain from the chest wound according to visual analogue scale assessments, and he had no comorbidities. Wound exudate was sent for bacterial and fungal cultures multiple times and all returned negative. On day 5 of admission, thoracic and abdominal computed tomography (CT) examinations showed a left cervicothoracic abscess, splenic abscess, and right axillary lymph node enlargement. Splenic puncture and abscess drainage were performed under CT guidance. No bacteria or fungi were found in the drained fluid, and only inflammatory cells were detected in the pathological examination.
On day 11 of admission, bone marrow biopsy results showed active hematopoiesis with prominent myeloid hyperplasia and blood tests showed significantly elevated indicators of infection (white blood cell and neutrophil counts [27.42 x 109/L], C-reactive protein [185.59 mg/L], and erythrocyte sedimentation rate [107 mm/hour]). After anti-infection treatments (eg, penicillin, azithromycin, ceftazidime, imipenem, and metronidazole) were provided in succession in the appropriate dosage and according to the manufacturer instructions, the aforementioned indices did not change significantly. The yellowish exudate from the ulcer increased and sinus depth was 6 cm to 9 cm; neither improved after dressing changes (2 to 3 times every day) or use of sterile gauze sponges in the sinuses to drain the exudate (see Figure 1A).
On day 15 of admission, Mr. J was still experiencing intermittent fever and his blood pressure began to drop, indicating septic shock (lowest blood pressure was 79/42 mm Hg). After antishock therapy with noradrenalin, Mr. J’s blood pressure returned to normal. The following day, the wounds were debrided under general anesthesia and intraoperative exploration revealed deep chest sinuses reaching to the ribs and parietal pleura, with a large amount of cheese-like necrotic tissue packed on the sinus wall. Negative pressure wound therapy (NPWT) was initiated in the operating room and continuously applied at a setting of -100 mm Hg for 6 to 7 days. Mr. J had no fever after the surgery, and the infection indexes gradually decreased to normal. Tissue culture showed no growth of bacteria or fungi.
Diagnosis. Pathological examination of sampled tissues suggested lymphohematopoietic malignancies in the necrotic tissue deep inside the chest wall and lymphoid tissues of the left neck (see Figure 2). Taking into account the morphological and immunological phenotypes and a history of long-term skin ulcers, the diagnosis of DLBCL associated with chronic inflammation was confirmed. A positron emission tomography-CT (PET-CT) examination revealed increased fluorodeoxyglucose (FDG) uptake in the cervicothoracic and right axillary lymph nodes and spleen, indicating invasive lymphoma.
Treatment. Mr. J was provided rituximab, vindesine, epirubicin, cyclophosphamide, and prednisolone (R-CHOP) chemotherapy for 7 days, repeated every 3 weeks in the department of hematology. After 3 rounds of chemotherapy, a follow-up PET-CT scan showed FDG uptake in cervicothoracic soft tissues was reduced significantly in both intensity and scope and the nodular FDG uptake in the spleen also decreased significantly. During the period of chemotherapy, Mr. J’s left cervicothoracic wound gradually decreased in size; deep sinuses in the wounds closed after several surgical debridements and NPWT treatments.
Approximately 11 weeks after admission, the left thoracic wound was debrided and a split-skin graft was applied, after which the cervicothoracic wound healed (see Figure 1B). Mr. J was discharged on day 93 after admission.