Rare Wound: Extensive Skin Lesions and Poor Wound Healing of the Calf in a Patient With Behçet's Disease

Behçet's disease (BD) is a chronic systemic vasculitic disease characterised by recurrent oral ulcers, genital ulcers, ocular inflammation and skin lesions, with recurrent episodes that can affect multiple systems [1]. This case report presents a 60-year-old male who suffered extensive skin lesions on both lower extremities after trauma. Despite undergoing debridement and targeted anti-infective treatment (initially treated with ceftazidime and subsequently meropenem, a total of 14 days), the wound failed to heal and progressively worsened, complicated by multiple organ dysfunction, leading to a severe condition. After a multidisciplinary consultation, BD was diagnosed. The treatment strategy was revised by introducing glucocorticoids combined with immunosuppressants, anti-infective treatment was adjusted to meropenem combined with linezolid and the patient underwent further debridement, skin grafting and vacuum sealing drainage (VSD). Ultimately, the patient's wound healed, and he was discharged. At present follow-up, the patient's wound has recovered well. Extensive skin lesions are rare in BD patients; therefore, this case aims to enrich clinical data and enhance clinical awareness of the disease. The patient, a 60-year-old male, was admitted to the Department of Orthopedics due to ‘12 days after wound debridement and suturing of the left ankle and right calf, with skin wound exudation for 5 days’ on September 29, 2024. Twelve days prior to admission, the patient accidentally fell and sustained lacerations from glass to his left ankle and right calf, accompanied by pain, bleeding and restricted limb movement. Initial wound debridement and suturing were performed at a local hospital. Five days prior to admission, wound exudation and slight limb swelling were noted during dressing changes and local anti-infective treatments proved ineffective. The patient had a history of hypertension for 2 years. He underwent skin surgery on his right back 2 years earlier, resulting in persistent non-healing similar to the current presentation. He denied histories of diabetes mellitus, renal disease, cardiovascular and cerebrovascular diseases, hepatitis, tuberculosis, syphilis and AIDS. The patient reported a history of smoking but denied alcohol consumption, allergy or long-term medication use other than antihypertensive agents. Physical examination upon admission showed body temperature of 36.7°C, pulse rate of 98 beats/min, respiratory rate of 20 breaths/min and blood pressure of 108/75 mmHg. Localised redness and swelling (approximately 3 × 4 cm) were observed at the left medial ankle with a surgical incision (approximately 2 cm in length) exhibiting exudation, and mildly limited limb movement. Similar findings were present on the anterior tibial region of the middle right calf (approximately 5 × 7 cm), with a surgical incision (approximately 6 cm in length) exhibiting exudation. No other abnormalities were found on physical examination. Upon admission, preliminary laboratory results indicated significantly elevated inflammatory indicators, including white blood cell count, hypersensitive C-reactive protein (CRP), procalcitonin (PCT), interleukin-6 (IL-6) and erythrocyte sedimentation rate (ESR) (Table 1). The patient was initially diagnosed with lower extremity skin infection and treated with ceftazidime 2 g intravenously every 12 h on the day of admission. After excluding contraindications, debridement with VSD was performed on 2 October 2024. During surgery, after removal of necrotic and devitalised tissue, thin, pale grey-brown exudate was noted within the superficial fascia and lateral compartment at the mid-portion of the right lower leg, accompanied by partial necrosis and dissolution of the fascial and muscular tissues, with extensive lateral compartment involvement. Partial superficial fascial necrosis was observed at the left medial malleolus and medial tibialis anterior tendon, without obvious exposure of joint spaces. After thorough removal of necrotic fascia and muscle tissue, the wound was repeatedly irrigated and soaked in iodophor. Upon releasing the tourniquet, punctate bleeding was obvious (Figure 1A–C). Postoperatively, ceftazidime (2 g intravenously every 12 h) was continued as anti-infective treatment. However, follow-up laboratory tests showed persistent elevation and a rising trend of white blood cell count, CRP, PCT, interleukin-6 and ESR (Table 1). Concurrently, liver dysfunction, hypoalbuminemia, anaemia (Table 1), pneumonia and significant pleural effusion developed, prompting discontinuation of ceftazidime and initiation of meropenem 1 g intravenously every 8 h as alternative anti-infective treatment. After 2 weeks, the patient's skin lesions continued to enlarge, accompanied by exudation and bilateral lower extremity swelling. The above inflammatory indicators, hypoproteinemia, anaemia, pneumonia and significant pleural effusion showed no significant improvement and complications including generalised oedema, anuria, superior limb venous thrombosis in both upper extremities and hypokalaemia emerged (Table 1). Nevertheless, multiple cultures of wound secretions and blood yielded negative results, with hepatitis, syphilis and HIV tests all negative, suggesting inconsistency with a systemic infection. A multidisciplinary consultation was conducted, during which the rheumatology and immunology specialist carefully reviewed the patient's history, performed physical examinations and assessed auxiliary investigations. A strongly positive acupuncture test (marked redness and swelling at multiple puncture sites, despite strict aseptic technique, with subsequent limb swelling) was identified. The patient also presented with recurrent oral ulcers. Based on the 2014 ICBD criteria [2], the patient scored 1 point for strongly positive acupuncture test, 2 points for recurrent oral ulcers and 1 point for skin lesions, totalling 4 points, fulfilling the diagnostic/classification criteria for Behçet's disease (BD). After excluding vasculitis, the patient was diagnosed with Behçet's disease, pneumonia and so forth. A multi-drug combination therapy was administered, consisting of intravenous methylprednisolone (40 mg once daily, gradually decreased), thalidomide (50 mg once nightly), colchicine (1 mg once daily, later discontinued), hydroxychloroquine (0.1 g twice daily), rivaroxaban (10 mg twice daily). Anti-infective treatment was meropenem 1 g intravenously every 8 h combined with linezolid 0.6 g intravenously every 12 h. During hospitalisation, four rounds of surgical debridement and suturing were performed in total, including the fourth surgery combined with skin grafting. Following this treatment regimen, clinical symptoms gradually improved, wounds essentially healed and the patient was discharged following suture removal. After discharge, regular outpatient follow-up was conducted. Currently, the patient receives methylprednisolone tablets 4 mg once daily, thalidomide 50 mg nightly, hydroxychloroquine 0.1 g twice daily and rivaroxaban 10 mg once daily. The lower-limb skin wounds have healed well (Figure 1D–F). Behçet's disease (BD), is a chronic, recurrent inflammatory or autoimmune disorder characterised by vasculitis. More than 70% of patients with Behçet's disease (BD) experience skin lesions, including acne-like eruptions, pustular rashes, pseudofolliculitis, nodular erythema, thrombophlebitis and gangrenous pyoderma [3-5]. However, extensive skin lesions accompanied by poor wound healing and significant exudation, as observed in the current patient, is extremely rare. This clinical scenario can easily be misdiagnosed as bacterial or fungal skin infections, especially in the early stage. Moreover, inflammatory symptoms of BD may mimic infections, making diagnosis and treatment more difficult [6]. Repeated wound secretion cultures from our patient were negative. Despite thorough debridement and successive antibiotic treatments with cephalosporins, meropenem and linezolid, the patient's poor response did not support a diagnosis of isolated skin wound infection. Symptoms significantly improved after treatment with methylprednisolone injections combined with thalidomide, colchicine and hydroxychloroquine. In summary, based on clinical symptoms, signs and auxiliary examinations, the final diagnosis was BD. Currently, BD lacks specific biomarkers or autoantibodies, and its diagnosis primarily relies on clinical features and expert consensus [7]. And the pathogenesis of BD remains unclear but may involve environmental factors and genetic susceptibility [8]. TNF-α plays a key role in BD pathogenesis [9]. Therefore, when traditional DMARD treatments fail, TNF-α inhibitors may offer significant therapeutic benefits. In the present case, satisfactory therapeutic outcomes were achieved with combined corticosteroid and immunosuppressive therapies without using biologics. Although biologic agents might yield superior therapeutic effects, their use in primary healthcare settings is limited. Prompt diagnosis and early initiation of corticosteroids and immunosuppressants can significantly improve outcomes in critically ill patients with extensive skin lesions. In summary, for wounds unresponsive to conventional anti-infective treatments, clinicians should broaden diagnostic considerations, obtain detailed medical histories, Emphasise multidisciplinary consultation. Early diagnosis and intervention can significantly improve patient prognosis. Furthermore, wounds with concurrent infections should undergo prudent debridement, along with systemic inflammation control, to prevent excessive skin irritation and disease exacerbation. The authors declare no conflicts of interest. The data that support the findings of this study are available on request from the corresponding author. The data are not publicly available due to privacy or ethical restrictions.