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PubMed1998

Aberrant neutrophil trafficking and metabolic oscillations in severe pyoderma gangrenosum.

Having previously associated metabolic oscillations with cell locomotion, we hypothesized that patients with abnormalities in neutrophil trafficking may display aberrant intracellular oscillations. A pyoderma gangrenosum patient exhibiting aberrant leukocyte trafficking in vivo and skin ulceration without infection was identified. This patient's neutrophils constitutively overexpressed and clustered the leukocyte integrins CR3 and CR4 and failed to display appropriate integrin-to-GPI receptor interactions. Increased levels of tyrosine phosphorylation were observed. NAD(P)H oscillations, which are sinusoidal in normals, were chaotic with multiple frequency components in this patient's neutrophils. Normal cell shape and sinusoidal NAD(P)H oscillations were restored by providing a pulsed electric field to drive metabolic oscillations and by temperature reduction. N-acetyl-D-glucosamine disrupted CR3 clusters and sinusoidal NAD(P)H oscillations returned. Anecdotal reports suggest that local hypothermia is clinically useful for this patient. These data define the first metabolic oscillation-associated disease and suggest that pyoderma gangrenosum can be classified as a dynamical disease at the cellular level.

Acetylglucosamine↗
PubMed1987

[Pyoderma gangrenosum and pelvispondylitis without patent intestinal disease. Apropos of a case].

The authors report a case of pyoderma gangrenosum associated with a positive B27 pelvispondylitis. The search for a hemorrhagic rectocolitis, possible link between the two syndromes, revealed to be negative. Pelvispondylitis is an unusual complication of pyoderma gangrenosum. Review of the literature shows only one case of sacroiliitis, beside seronegative polyarthritis unrelated to HLA B27.

Adult↗
PubMed1987

Pyoderma gangrenosum: a Waikato experience.

Ten cases of pyoderma gangrenosum were seen over a period of nine years at the Waikato Hospital dermatology clinic. Several associated conditions were found. Four cases were associated with inflammatory bowel disease, three cases with arthritis, one case was found in association with myelofibrosis. Four patients were diabetic, an incidence higher than in previous studies. Most patients were treated with oral steroids but additional therapies were used to achieve disease control and for their steroid-sparing effect.

Adult↗
PubMed1995

Pyoderma gangrenosum as a complication of coronary artery bypass grafting.

A 60-year-old male patient developed progressive wound ulcerations, simulating wound sepsis after coronary bypass operation. The condition did not respond to intensive antibiotic therapy. Based on clinical signs and biopsy, the diagnosis of pyoderma gangrenosum was made and successfully treated with cyclosporin A. Pyoderma gangrenosum, although extremely rare, must be considered as a possible diagnosis in wound complications unresponsive to traditional therapy.

Angina Pectoris↗
PubMed1994

Cyclosporine, azathioprine and local therapy for pyoderma gangrenosum.

A markedly obese 54 year old woman with seropositive rheumatoid arthritis, anaemia, dyspepsia, controlled hypothyroidism and depression presented with a seven month history of large pyoderma gangrenosum ulcers on the shins. Routine dressings for the ulcers had been ineffective. Her arthritis was being treated with azathioprine and NSAID's. Initial treatment with clobestasol propionate and disodium cromoglycate under occlusion produced only partial healing. Introduction of Cyclosporin A and continuation of topical therapy, with the addition of triamcinolone acetonide injections, led to progressive healing which was complete after seven months. There has been no relapse to date. Cyclosporine can be combined with azathioprine and local therapy for successful treatment of pyoderma gangrenosum.

Administration, Topical↗
PubMed1997

Pyoderma gangrenosum of the breast treated with low-dose cyclosporin A.

Pyoderma gangrenosum (PG) is a painful chronic ulcerative skin disorder often occurring in association with systemic disease. It typically affects the lower limbs, but may also involve other sites, or sometimes develop after trauma of surgical procedures. We report the case of a woman with rheumatoid arthritis who developed disfiguring and severe PG of the right breast, a rare site, following biopsy for a benign breast lesion, and who was subsequently successfully treated with low-dose cyclosporin A.

Antirheumatic Agents↗
PubMed2003

Crohn's disease of the penis masquerading as pyoderma gangrenosum: a case report and review of the literature.

Both pyoderma gangrenosum (PG) and cutaneous (metastatic) Crohn's disease (CCD) may occur in the setting of inflammatory bowel disease (IBD). Clinical distinction between PG and CCD may be difficult because clinical and pathologic features often are similar. Although surgical debridement is therapeutic in CCD, it may lead to increased tissue loss and disease progression (pathergy) in PG. Thus, it is important to determine a definitive diagnosis before surgical debridement, especially in tissue-sensitive sites. We present a patient with chronic ulceration of the penis who ultimately was diagnosed with CCD following an initial misdiagnosis of PG.

Crohn Disease↗
PubMedDate not supplied

Pyoderma gangrenosum with a contiguous erosion of the distal ulna.

BACKGROUND: Pyoderma gangrenosum (PG) has been described in association with sterile neutrophilic infiltration of several organ systems, including the skeleton. The most frequent cause of bony disease in PG has been chronic, recurrent, multifocal osteomyelitis, a sterile inflammatory disease of children and young adults mimicking infectious osteomyelitis. Bony erosions have been only rarely described in direct contiguity to a PG ulcer. OBJECTIVE: We report a 53-year-old woman with recurrent PG who developed an erosion of the distal ulna adjacent to a PG ulcer. The patient responded to high-dose prednisone, and a repeat radiograph of the wrist four months later was normal. CONCLUSION: This case demonstrates another example of cortical osteolysis directly adjacent to a PG ulcer in which the bony changes may be neutrophil-induced.

Bone Diseases↗
PubMed2000

Treatment of pyoderma gangrenosum with oral Tripterygium wilfordii multiglycoside.

Two patients with refractory pyoderma gangrenosum (PG) were treated with oral Tripterygium wilfordii multiglycoside (TWG). TWG is a Chinese medicine extracted from a medicinal herb, Tripterygium wilfordii Hook F, and has potent anti-inflammatory and immunosuppressive effects. The effect of TWG on PG was demonstrated by clinical findings. Improvement of the lesions occurred within two weeks, and the ulcers healed about a month. Mild side effects such as gastrointestinal disturbances were observed in both patients. These side effects were patient-acceptable, and there was no need to stop the treatment. Transient elevation of serum ALT was observed in one patient; the serum ALT returned completely to normal after the discontinuation of TWG. These results suggest that TWG may be an effective alternative for refractory PG and that careful monitoring of liver function during TWG treatment is necessary.

Administration, Oral↗
PubMed1983

Pyoderma gangrenosum. Response to cyclophosphamide therapy.

A 46-year-old woman had a 17-year history of intermittently severe pyoderma gangrenosum without identifiable associated systemic disease. Her condition had become unresponsive to corticosteroid and sulfone therapy given for systemic effect, but responded completely to 150 mg/day of cyclophosphamide. Immunosuppressive therapy should be considered in patients with severe, recalcitrant pyoderma gangrenosum, even in the absence of associated systemic disease.

Adult↗
PubMed2002

Sensorineural hearing loss as an extra-intestinal manifestation of ulcerative colitis in an adolescent girl with pyoderma gangrenosum.

UNLABELLED: Inflammatory bowel disease is becoming increasingly frequent in children of all ages. In addition to the usual gastrointestinal stigmata of weight loss, anaemia, and rectal bleeding, children may exhibit prominent extra-intestinal manifestations such as joint symptoms, skin signs and some other auto-immune manifestations. Here we present a 15-year-old girl with ulcerative colitis in whom pyoderma gangrenosum and acute sensorineural hearing loss developed. CONCLUSION: Although pyoderma gangrenosum is well described with inflammatory bowel disease, sensorineural hearing loss a is very unusual finding, possibly of auto-immune aetiology. We recommend steroid or immunosuppressive therapy in such a patient.

Adolescent↗
PubMed1999

[Pyoderma gangrenosum with an atypical location and a rapid response to cyclosporin A].

Pyoderma gangrenosum is an unusual neutrophilic dermatosis associated to different rheumatologic, haematologic and inflammatory bowel diseases which generally responds to the therapy of the underlying disease. We report a case of pyoderma in a 44-years-old woman with paucisymptomatic, distal, ulcerative colitis which appeared simultaneously in her forehead and hands. We think it of general interest because of its localization, its importance, the failure of response to steroids and the need of cyclosporine A for regression.

Administration, Oral↗
PubMed1998

Corneal melting and scleromalacia perforans in a patient with pyoderma gangrenosum and acute myeloid leukemia.

Postoperative endophthalmitis may present in an atypical fashion (absent or minimal anterior chamber reaction) in the presence of underlying immunosuppressive disorder. The authors describe an apparently healthy 58-year-old man who displayed endophthalmitis with minimal anterior chamber reaction following penetrating keratoplasty for granular corneal dystrophy with underlying acute myeloid leukemia. Scleromalacia perforans in association with pyoderma gangrenosum subsequently developed, leading to ciliary staphyloma and corneal melting. Pyoderma gangrenosum is an uncommon, idiopathic skin disease that may also have ocular manifestations.

Anti-Bacterial Agents↗
PubMedDate not supplied

Chronic recurrent multifocal osteomyelitis preceding pyoderma gangrenosum and occult ulcerative colitis in a pediatric patient.

Chronic recurrent multifocal osteomyelitis (CRMO) is a clinicopathologic entity of unknown origin mainly affecting children and young adults. It shares many clinical features with pyoderma gangrenosum (PG), an uncommon ulcerating inflammatory disorder of the dermis and underlying subcutaneous fat. Both conditions are chronic and relapsing, and have been associated with inflammatory bowel disease (IBD). The diagnoses are made by exclusion. Histology is nonspecific and cultures are negative. There has been only one previous report of both conditions occurring in the same patient. Here we describe the second case, a 12-year-old girl who developed pyoderma gangrenosum in direct continuity to an underlying CRMO lesion of the right anterior tibia. Occult ulcerative colitis (UC) was subsequently discovered.

Child↗
PubMed2004

Pouchitis-associated pyoderma gangrenosum following restorative proctocolectomy for ulcerative colitis.

A 43-year-old female with ulcerative colitis treated by proctocolectomy and ileal pouch--anal anastomosis developed acute pouchitis. Although no extra-intestinal manifestations were present before the surgical procedure, pyoderma gangrenosum developed concomitantly with the appearance of acute pouchitis. Both conditions completely resolved with oral metronidazole only. This is the first report of acute pouchitis-related pyoderma gangrenosum. This association suggests that pouchitis may represent a recurrent ulcerative colitis-like syndrome within the novel environment of the pouch.

Acute Disease↗
PubMed1999

[Therapy-resistant pyoderma gangrenosum--treatment with mycophenolate mofetil and cyclosporine A].

A 68-year old woman presented with ulcerations on the calves that had occurred spontaneously. The very painful lesions both clinically and histologically showed the characteristics of pyoderma gangrenosum. During hospitalization she was treated with corticosteroids (oral, i.v., topically), clofazimine, cyclophosphamide, intravenous immune globulin, cyclosporine (oral, local), dapsone, thalidomide and sodium cromoglycate (topically) without any benefit. Finally, when treated with mycophenolate mofetil (CellCept) (oral) and cyclosporine (oral), her skin lesions showed continuous improvement. The topical application of thrombocytic growth factors (cytokines) probably accelerated the granulation. Eight weeks after initiating this treatment the lesions could be covered with split thickness skin grafts. Our observation suggests that mycophenolate mofetil, a novel immunosuppressive agent which has thus far been used almost exclusively in transplantation medicine, may be an effective therapeutic modality in combination with cyclosporine A for the treatment of pyoderma gangrenosum.

Aged↗