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Salvage of major joint prostheses in aggressive non-specific panniculitis
British Journal of Plastic Surgery (2002) 55, 585–587 © 2002 Published by Elsevier Science Ltd on behalf of The British Association of Plastic Surgeons doi:10.1054/bjps.2002.3934
CASE REPORT Salvage of major joint prostheses in aggressive non-specific panniculitis I. C. Josty, S. Sathappan, M. A. C. S. Cooper and A. Dawson Welsh Centre for Burns and Plastic Surgery, Morriston Hospital, Swansea, UK SUMMARY. Panniculitis is a relatively rare condition, which is usually treated non-operatively. We present a case of panniculitis causing an acute septicaemic episode and threatening the integrity of underlying joint prostheses. The situation was salvaged by radical debridement of the affected areas, and reconstruction using split-skin grafts. Considerations in the management of patients with this condition are discussed. © 2002 The British Association of Plastic Surgeons Keywords: panniculitis, joint prosthesis, rheumatoid arthritis, split-skin graft.
We present the case of a 65-year-old man with rheumatoid arthritis who had recurrent attacks of panniculitis over his right shoulder and right hip at the sites of his joint replacements. Despite extensive investigation, the specific aetiology of the panniculitis remained undiagnosed. However, the panniculitis was effectively treated by excision and split-skin grafting. We believe that this is the first reported case of this treatment modality being used in such multifocal panniculitis.
Histology of the resected specimen revealed an extensive inflammatory process, with scarring extending throughout the skin and into the subcutis (Fig. 2). There was a patchy chronic inflammatory cell infiltrate with numerous plasma cells
Case report A 65-year-old man was referred to the plastic surgical services in 1998 with a history of chronic ulceration of the right deltoid region. He was diagnosed with seropositive rheumatoid arthritis in 1969, and had been treated systemically with steroids, nonsteroidal anti-inflammatory drugs and penicillamine at various stages during his illness. Multiple intra-articular steroid injections did not control his local joint disease, and he progressed to a right total hip replacement in 1987 and a right shoulder replacement in 1995. In 1997, he presented with an insidious onset of skin induration in the right deltoid region. This was followed by breakdown of the skin, forming multiple shallow ulcers with a sloughy base. Biopsy at that time showed perivasculitis and a diffuse chronic inflammatory infiltration involving the subcutaneous tissue and dermis; the inflammatory infiltrate consisted of lymphocytes and exhibited no specific features. The lesions were treated non-operatively with dressings, but did not improve. At the time of transfer to our unit, in 1998, he was stable, having recently recovered from a septic episode requiring ITU admission. The necrotic shoulder wound was enlarging, and there was concern about it communicating with the underlying prosthesis. He was also noted to have induration overlying the right hip, but at this stage there was no ulceration (Fig. 1). There was no radiological evidence of loosening of either prosthesis. The necrotic shoulder wound was debrided, and was found to extend to the muscle and into the intermuscular septa. As the joint was not exposed, the wound was closed with a split-skin graft. Postoperative recovery was uneventful, with 95% graft take.
Figure 1—Preoperative appearance of the necrotic right shoulder wound, and induration over the right hip.
Figure 2—Extensive collagenous scarring of the pannus, with focal lymphoid infiltrate (H&E 10 objective).
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British Journal of Plastic Surgery
Figure 3—Focal vasculitis within specimen (H&E 20 objective).
Figure 5—Appearance of the right shoulder 3 years postoperatively.
Figure 4—Ulcerative panniculitis of the right hip.
Figure 6—Appearance of the right hip 2 years postoperatively.
(with Russell bodies), but also a focal neutrophil infiltrate with abscess formation. Old endarteritis and focal vasculitis were present (Fig. 3). The ulceration appeared to be secondary to the extensive scarring in the subcutis and dermis. The appearances suggested a chronic active panniculitis/fasciitis. No granulomata or features of rheumatoid nodule were seen. He was investigated for other known causes of panniculitis, which were negative: levels of alpha 1 antitrypsin and thyroid stimulating hormone were normal, and tests for tuberculosis and treponema were negative. During the following year the panniculitis on his right hip progressed and ulcerated to form a defect measuring 10 cm 6 cm with surrounding induration (Fig. 4). Wide resection of all clinically involved tissue, measuring 26 cm18 cm, was performed. The lesion extended to the deeper muscles. Histologically, the specimen appeared similar to that excised from the shoulder. The wound was covered with a split-skin graft. The postoperative recovery was complicated by a wound infection, and 50% of the graft was lost. Wound swabs grew Enterococcus feacalis, Coliforms and Corynebacterium sp. He was treated with the appropriate antibiotics, and the wound was left to heal by secondary intention. Two years later his wounds remained healed, with a stable appearance and no evidence of recurrence of the panniculitis (Figs 5 and 6). In 1999 he had a left total hip replacement, and currently has no skin problems at this site.
Discussion This patient presents an interesting diagnostic problem. Clinical and histological findings confirmed the diagnosis of panniculitis, but the aetiology remains uncertain. Panniculitis can be classified clinically as either primary or secondary and histologically as septal, lobular or mixed. In many cases the diagnosis can be difficult to establish. Table 1 gives a list of recognised secondary causes. Although primary panniculitis cannot be excluded in this patient, it is more probable that he had a secondary panniculitis due to one or more of the following: rheumatoid arthritis; a foreign-body reaction; steroid injections; immunosuppressive therapy; and/or surgical trauma. Various treatments for panniculitis have been proposed. The first strategy should be the withdrawal or elimination of any aetiological factor, if possible. Unfortunately, this alone is usually inadequate, and further therapies, with corticosteroids, methotrexate, D-penicillamine, chloroquine and azathioprine, have been reported, with limited success.3 This patient was treated with some of the above agents, but his disease continued to progress, threatening his joint prostheses. He had large necrotic ulcers that were infected and had resulted in a septicaemic episode
Salvage of major joint prostheses
587
Table 1 Aetiology of secondary panniculitis Aetiological category
Disease process
connective tissue disease infective
rheumatoid arthritis1 tuberculosis2 erysipelas3 streptococcus brucellosis3 tertiary syphilis3 chronic hepatitis3 lymphoma3 alpha 1 antitrypsin deficiency4–6 haematological malignancy3,7 pancreatic cancer6 metastatic disease3 arterial occlusion venous or lymphatic stasis gout1 pancreatic insufficiency6 renal insufficiency1 parathyroid disease1 steroid withdrawal1 trauma7 post-infective1 factitious6 collagen injection8 cold panniculitis1,6 insect bites
haematological neoplastic vascular metabolic
pharmacological physical
necessitating ITU care. He needed treatment of the infected areas, and this was accomplished effectively by surgical excision. The surgical management of this patient was relatively straightforward, and consisted of wide local excision down to healthy tissue, and closure using a split-skin graft. Although relatively simple, we were unable to find any previous reports in the literature of this treatment being used in such an aggressive panniculitis. A previous report cautioned against surgical debridement, as the authors claimed that the trauma of surgery could exacerbate the disease process.6 This was not our experience. In fact, surgical debridement and reconstruction prevented exposure and secondary infection of our patient’s joint prostheses. In conclusion, we report a curious case of progressive ulcerating areas of panniculitis, overlying the sites of joint prostheses in a patient with rheumatoid arthritis, which were successfully treated by excision and splitskin grafting. This report emphasises that the diagnosis of panniculitis is both clinical and histological. A search for the
causative agent(s) must be made, and, if at all possible, these must be eliminated. Wide local excision and split-skin grafting gives immediate relief and may prevent the pathological process from worsening. Surgery should be limited and focussed, as these patients may not be able to tolerate more complex reconstructions. We believe that this is the first reported case of wide excision and split-skin grafting of ulcerating panniculitis. Surgery should be considered when medical management has failed or when clinical urgency dictates the need for debridement to prevent sepsis or extension into the underlying joints. References 1. Peters MS, Su WPD. Panniculitis. Dermatol Clin 1992; 10: 37–57. 2. Rademaker M, Lowe DG, Munro DD. Erythema induratum (Bazin’s disease). J Am Acad Dermatol 1989; 21: 740–5. 3. Naschitz JE, Boss JH, Misselevich I, Yeshurun D, Rosner I. The fasciitis–panniculitis syndromes – clinical and pathological features. Medicine 1996; 75: 6–16. 4. Smith KC, Su WPD, Pittelkow MR, Winkelmann RK. Clinical and pathological correlations in 96 patients with panniculitis, including 15 patients with deficient levels of alpha-1 antitrypsin. J Am Acad Dermatol 1989; 21: 1192–6. 5. Hendrick SJ, Silverman AK, Solomon AR, Headington JT. Alpha-1 antitrypsin deficiency associated with panniculitis. J Am Acad Dermatol 1988; 18: 684–92. 6. Cohen LM. Panniculitis: diagnosis and management. Dermatol Nurs 1996; 8: 405–16. 7. White JR, Winkelmann RK. Weber–Christian panniculitis: a review of 30 cases with this diagnosis. J Am Acad Dermatol 1998; 39: 56–62. 8. Biasi D, Caramaschi P, Carletto A, Colombari R, Bambara LM. Panniculitis induced by collagen and plexiglas microsphere injections used for aesthetic treatment. Clin Rheumatol 1999; 18: 328–9.
The Authors Ian C. Josty BSc, MBBS, FRCS(Plast), Specialist Registrar M. A. C. S. Cooper FRCS(Plast), Consultant Plastic Surgeon A Dawson FRCPath, Consultant Pathologist Welsh Centre for Burns and Plastic Surgery, Morriston Hospital, Swansea SA6 6NL, UK. S. Sathappan FRCS, Consultant Plastic Surgeon Department of Plastic Surgery, Hospital Universiti Kebangsaan Malaysia, 56000 Kuala Lumpur, Malaysia. Correspondence to Mr Ian Josty. Paper received 17 April 2002. Accepted 25 July 2002, after revision.
CASE REPORT Salvage of major joint prostheses in aggressive non-specific panniculitis I. C. Josty, S. Sathappan, M. A. C. S. Cooper and A. Dawson Welsh Centre for Burns and Plastic Surgery, Morriston Hospital, Swansea, UK SUMMARY. Panniculitis is a relatively rare condition, which is usually treated non-operatively. We present a case of panniculitis causing an acute septicaemic episode and threatening the integrity of underlying joint prostheses. The situation was salvaged by radical debridement of the affected areas, and reconstruction using split-skin grafts. Considerations in the management of patients with this condition are discussed. © 2002 The British Association of Plastic Surgeons Keywords: panniculitis, joint prosthesis, rheumatoid arthritis, split-skin graft.
We present the case of a 65-year-old man with rheumatoid arthritis who had recurrent attacks of panniculitis over his right shoulder and right hip at the sites of his joint replacements. Despite extensive investigation, the specific aetiology of the panniculitis remained undiagnosed. However, the panniculitis was effectively treated by excision and split-skin grafting. We believe that this is the first reported case of this treatment modality being used in such multifocal panniculitis.
Histology of the resected specimen revealed an extensive inflammatory process, with scarring extending throughout the skin and into the subcutis (Fig. 2). There was a patchy chronic inflammatory cell infiltrate with numerous plasma cells
Case report A 65-year-old man was referred to the plastic surgical services in 1998 with a history of chronic ulceration of the right deltoid region. He was diagnosed with seropositive rheumatoid arthritis in 1969, and had been treated systemically with steroids, nonsteroidal anti-inflammatory drugs and penicillamine at various stages during his illness. Multiple intra-articular steroid injections did not control his local joint disease, and he progressed to a right total hip replacement in 1987 and a right shoulder replacement in 1995. In 1997, he presented with an insidious onset of skin induration in the right deltoid region. This was followed by breakdown of the skin, forming multiple shallow ulcers with a sloughy base. Biopsy at that time showed perivasculitis and a diffuse chronic inflammatory infiltration involving the subcutaneous tissue and dermis; the inflammatory infiltrate consisted of lymphocytes and exhibited no specific features. The lesions were treated non-operatively with dressings, but did not improve. At the time of transfer to our unit, in 1998, he was stable, having recently recovered from a septic episode requiring ITU admission. The necrotic shoulder wound was enlarging, and there was concern about it communicating with the underlying prosthesis. He was also noted to have induration overlying the right hip, but at this stage there was no ulceration (Fig. 1). There was no radiological evidence of loosening of either prosthesis. The necrotic shoulder wound was debrided, and was found to extend to the muscle and into the intermuscular septa. As the joint was not exposed, the wound was closed with a split-skin graft. Postoperative recovery was uneventful, with 95% graft take.
Figure 1—Preoperative appearance of the necrotic right shoulder wound, and induration over the right hip.
Figure 2—Extensive collagenous scarring of the pannus, with focal lymphoid infiltrate (H&E 10 objective).
585
586
British Journal of Plastic Surgery
Figure 3—Focal vasculitis within specimen (H&E 20 objective).
Figure 5—Appearance of the right shoulder 3 years postoperatively.
Figure 4—Ulcerative panniculitis of the right hip.
Figure 6—Appearance of the right hip 2 years postoperatively.
(with Russell bodies), but also a focal neutrophil infiltrate with abscess formation. Old endarteritis and focal vasculitis were present (Fig. 3). The ulceration appeared to be secondary to the extensive scarring in the subcutis and dermis. The appearances suggested a chronic active panniculitis/fasciitis. No granulomata or features of rheumatoid nodule were seen. He was investigated for other known causes of panniculitis, which were negative: levels of alpha 1 antitrypsin and thyroid stimulating hormone were normal, and tests for tuberculosis and treponema were negative. During the following year the panniculitis on his right hip progressed and ulcerated to form a defect measuring 10 cm 6 cm with surrounding induration (Fig. 4). Wide resection of all clinically involved tissue, measuring 26 cm18 cm, was performed. The lesion extended to the deeper muscles. Histologically, the specimen appeared similar to that excised from the shoulder. The wound was covered with a split-skin graft. The postoperative recovery was complicated by a wound infection, and 50% of the graft was lost. Wound swabs grew Enterococcus feacalis, Coliforms and Corynebacterium sp. He was treated with the appropriate antibiotics, and the wound was left to heal by secondary intention. Two years later his wounds remained healed, with a stable appearance and no evidence of recurrence of the panniculitis (Figs 5 and 6). In 1999 he had a left total hip replacement, and currently has no skin problems at this site.
Discussion This patient presents an interesting diagnostic problem. Clinical and histological findings confirmed the diagnosis of panniculitis, but the aetiology remains uncertain. Panniculitis can be classified clinically as either primary or secondary and histologically as septal, lobular or mixed. In many cases the diagnosis can be difficult to establish. Table 1 gives a list of recognised secondary causes. Although primary panniculitis cannot be excluded in this patient, it is more probable that he had a secondary panniculitis due to one or more of the following: rheumatoid arthritis; a foreign-body reaction; steroid injections; immunosuppressive therapy; and/or surgical trauma. Various treatments for panniculitis have been proposed. The first strategy should be the withdrawal or elimination of any aetiological factor, if possible. Unfortunately, this alone is usually inadequate, and further therapies, with corticosteroids, methotrexate, D-penicillamine, chloroquine and azathioprine, have been reported, with limited success.3 This patient was treated with some of the above agents, but his disease continued to progress, threatening his joint prostheses. He had large necrotic ulcers that were infected and had resulted in a septicaemic episode
Salvage of major joint prostheses
587
Table 1 Aetiology of secondary panniculitis Aetiological category
Disease process
connective tissue disease infective
rheumatoid arthritis1 tuberculosis2 erysipelas3 streptococcus brucellosis3 tertiary syphilis3 chronic hepatitis3 lymphoma3 alpha 1 antitrypsin deficiency4–6 haematological malignancy3,7 pancreatic cancer6 metastatic disease3 arterial occlusion venous or lymphatic stasis gout1 pancreatic insufficiency6 renal insufficiency1 parathyroid disease1 steroid withdrawal1 trauma7 post-infective1 factitious6 collagen injection8 cold panniculitis1,6 insect bites
haematological neoplastic vascular metabolic
pharmacological physical
necessitating ITU care. He needed treatment of the infected areas, and this was accomplished effectively by surgical excision. The surgical management of this patient was relatively straightforward, and consisted of wide local excision down to healthy tissue, and closure using a split-skin graft. Although relatively simple, we were unable to find any previous reports in the literature of this treatment being used in such an aggressive panniculitis. A previous report cautioned against surgical debridement, as the authors claimed that the trauma of surgery could exacerbate the disease process.6 This was not our experience. In fact, surgical debridement and reconstruction prevented exposure and secondary infection of our patient’s joint prostheses. In conclusion, we report a curious case of progressive ulcerating areas of panniculitis, overlying the sites of joint prostheses in a patient with rheumatoid arthritis, which were successfully treated by excision and splitskin grafting. This report emphasises that the diagnosis of panniculitis is both clinical and histological. A search for the
causative agent(s) must be made, and, if at all possible, these must be eliminated. Wide local excision and split-skin grafting gives immediate relief and may prevent the pathological process from worsening. Surgery should be limited and focussed, as these patients may not be able to tolerate more complex reconstructions. We believe that this is the first reported case of wide excision and split-skin grafting of ulcerating panniculitis. Surgery should be considered when medical management has failed or when clinical urgency dictates the need for debridement to prevent sepsis or extension into the underlying joints. References 1. Peters MS, Su WPD. Panniculitis. Dermatol Clin 1992; 10: 37–57. 2. Rademaker M, Lowe DG, Munro DD. Erythema induratum (Bazin’s disease). J Am Acad Dermatol 1989; 21: 740–5. 3. Naschitz JE, Boss JH, Misselevich I, Yeshurun D, Rosner I. The fasciitis–panniculitis syndromes – clinical and pathological features. Medicine 1996; 75: 6–16. 4. Smith KC, Su WPD, Pittelkow MR, Winkelmann RK. Clinical and pathological correlations in 96 patients with panniculitis, including 15 patients with deficient levels of alpha-1 antitrypsin. J Am Acad Dermatol 1989; 21: 1192–6. 5. Hendrick SJ, Silverman AK, Solomon AR, Headington JT. Alpha-1 antitrypsin deficiency associated with panniculitis. J Am Acad Dermatol 1988; 18: 684–92. 6. Cohen LM. Panniculitis: diagnosis and management. Dermatol Nurs 1996; 8: 405–16. 7. White JR, Winkelmann RK. Weber–Christian panniculitis: a review of 30 cases with this diagnosis. J Am Acad Dermatol 1998; 39: 56–62. 8. Biasi D, Caramaschi P, Carletto A, Colombari R, Bambara LM. Panniculitis induced by collagen and plexiglas microsphere injections used for aesthetic treatment. Clin Rheumatol 1999; 18: 328–9.
The Authors Ian C. Josty BSc, MBBS, FRCS(Plast), Specialist Registrar M. A. C. S. Cooper FRCS(Plast), Consultant Plastic Surgeon A Dawson FRCPath, Consultant Pathologist Welsh Centre for Burns and Plastic Surgery, Morriston Hospital, Swansea SA6 6NL, UK. S. Sathappan FRCS, Consultant Plastic Surgeon Department of Plastic Surgery, Hospital Universiti Kebangsaan Malaysia, 56000 Kuala Lumpur, Malaysia. Correspondence to Mr Ian Josty. Paper received 17 April 2002. Accepted 25 July 2002, after revision.