Rheumatology Advance Access originally published online on January 28, 2008
Rheumatology 2008 47(3):379-380; doi:10.1093/rheumatology/kem347
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Autologous skin grafting—a limb-saving procedure in a patient with diffuse cutaneous systemic sclerosis
1Rheumatic Diseases Centre, University of Manchester, Hope Hospital, Salford and 2South Manchester University Hospitals, Wythenshawe Hospital-Plastic Surgery, Wythenshawe, Manchester, UK
Correspondence to: S. Chitale, Rheumatic Diseases Centre, Hope Hospital, Stott Lane, Salford. M6 8HD, UK. E-mail: sarangchitale{at}hotmail.com
SIR, Systemic sclerosis (SSc) is a connective tissue disorder characterized by interplay between early immunological events and vascular changes, resulting in the generation of a population of activated fibroblasts, generally considered to be the effector cell in the disease [1]. These fibroblasts are responsible for overproduction of collagen, leading to the thickening and tightening of the skin. This skin tightening, together with the characteristic microvascular changes of SSc, can compromise wound healing. For example, skin ulcers do not heal well in patients with SSc and are difficult to treat, due to severe microvascular involvement, concomitant arterio-venous disturbances and superadded infection [2].
Previous experience shows that, when clinically normal skin is grafted to a sclerodermatous bed, it becomes sclerodermatous [3, 4] and if sclerodermatous skin is grafted to normal skin it remains sclerodermatous [3]. We describe a patient with SSc in whom successful closure of an extensive lower limb wound was achieved by split thickness skin grafting, involving sclerodermatous skin grafted to a sclerodermatous wound site. Had the wound not healed then it is likely that amputation would have been required.
In February 2003, a 51-yr-old female with diffuse cutaneous SSc suffered a tonic–clonic seizure and sustained a simple fracture of distal right femur, just above the knee joint. The cause of her seizure was thought to be viral meningitis. She underwent open reduction and internal fixation of the femoral fracture.
She had first presented to the rheumatology clinic in early 2002, with a history of Raynaud's phenomenon since May 2001, recent gradual onset of shortness of breath, swallowing problems and skin thickening of upper and lower limbs extending proximally beyond elbows and knees. Her modified Rodnan skin score at this time was 36 out of 51 [5] reflecting a very widespread skin thickening. Renal function was normal. Gastroscopy revealed oesophagitis. Pulmonary function tests showed a restrictive pattern with forced expiratory ventilation in first second (FEV1) to forced vital capacity (FVC) ratio of 85% and slightly reduced diffusion capacity (DLCO) at 79% predicted. High-resolution computed tomography (HRCT) scan showed minimal basal fibrosis. Echocardiogram was normal. She tested negative for antinuclear antibodies (ANA), anti-centromere and anti-topoisomerase 1 (Scl-70) antibodies. She was commenced on i.v. cyclophosphamide of which she had six infusions at monthly intervals. She was also started on omeprazole for the oesophagitis and nifedipine for Raynaud's phenomenon [1, 6]. The cyclophosphamide was followed by oral mycophenolate mofetil [7]. Her modified Rodnan skin sclerosis score was 40/51 just prior to the seizure. At the time of her seizure, she was on mycophenolate, nifedipine and omeprazole.
As the skin around the wound site (which extended from just above the knee joint to proximal tibia) was thickened and tight, primary closure of the wound was not possible and the wound was left open, following the open reduction and internal fixation of the femoral fracture. If left untreated there was a serious risk of infection and eventual need for amputation. Hence, she was referred to the plastic surgeons. Her wound was grafted with a split thickness skin graft taken from her thigh. The skin at the donor site was also sclerodermatous (Rodnan skin score at that site was 2 out of a possible 3, which means it was possible to move the skin but not pinch it). The wound healed very well with the grafted sclerodermatous skin. The donor site also healed well without complications (Fig. 1).
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To our knowledge, this is the first report of a successful split thickness skin graft to a large surgical wound in a patient with sclerodermatous skin. In this case history, the key point is a clinical one—that wound healing may be uncomplicated even if the graft is taken from affected sclerodermatous skin and transplanted to another affected area. There are reports in the literature of using artificial [8], split thickness [9] and autologous [10] skin grafts to promote ulcer healing in patients with SSc.
In conclusion, in this case, primary closure of the wound was impossible, and there was a real concern that our patient might have required amputation. Grafted skin presented a natural barrier for preventing infection and allowed early wound healing. Our patient thus demonstrates how split thickness skin grafting even from sclerodermatous skin can be successfully used to cover large wounds with a good outcome in patients with SSc.
Disclosure statement: The authors have declared no conflicts of interest.
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- Hafner J, Schneider E, Burg G, Cassina PC. Management of leg ulcers in patients with rheumatoid arthritis or systemic sclerosis: the importance of concomitant arterial and venous disease. J Vasc Surg (2000) 32:322–9.[CrossRef][Web of Science][Medline]
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- Herrick AL, Marcuson R, Freemont AJ, Jayson MIV. Clinically normal skin becomes sclerodermatous when transplanted into a sclerodermatous area. Br J Rheumatol (1992) 31:707–9.
[Abstract/Free Full Text] - Black CM, Silman AJ, Herrick AL, et al. Interferon-alpha does not improve outcome at one year in patients with diffuse cutaneous scleroderma: results of a randomized double-blind placebo controlled trial. Arthritis Rheum (1999) 42:299–305.[CrossRef][Web of Science][Medline]
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- Stratton RJ, Wilson H, Black CM. Pilot study of anti-thymocyte globulin plus mycophenolate mofetil in recent onset diffuse scleroderma. Rheumatology (2001) 40:84–8.
[Abstract/Free Full Text] - Limova M, Mauro T. Treatment of leg ulcers with cultured epithelial autografts: clinical study and case reports. Ostomy Wound Manage (1995) 41:48–60.[Medline]
- Hafner J, Kohler A, Enzler M, Brunner U. Successful treatment of an extended leg ulcer in systemic sclerosis. VASA (1997) 26:302–4.[Web of Science][Medline]
- Giuggioli D, Sebastiani M, Cazzato M, Piaggesi A, Abatangelo G, Ferri C. Autologous skin grafting in the treatment of severe cutaneous ulcers: a case report. Rheumatology (2003) 42:694–6.
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