{"id":56783,"date":"2024-03-20T10:04:47","date_gmt":"2024-03-20T10:04:47","guid":{"rendered":"https:\/\/biomedpharmajournal.org\/?p=56783"},"modified":"2024-04-03T05:09:31","modified_gmt":"2024-04-03T05:09:31","slug":"effectively-using-infliximab-to-treat-pyoderma-gangrenosum-in-a-woman-with-ulcerative-colitis-case-report","status":"publish","type":"post","link":"https:\/\/biomedpharmajournal.org\/staging\/vol17no1\/effectively-using-infliximab-to-treat-pyoderma-gangrenosum-in-a-woman-with-ulcerative-colitis-case-report\/","title":{"rendered":"Effectively Using Infliximab to Treat Pyoderma Gangrenosum in a Woman With Ulcerative Colitis – Case Report"},"content":{"rendered":"\n

Introduction <\/strong><\/p>\n\n\n\n

Pyoderma gangrenosum (PG) is considered a reactive inflammatory dermatosis and part of the spectrum of neutrophilic dermatosis. The disease was first described by Brocq in 1916 as a \u201cphagedenisme geometrique\u201d. 1<\/sup> It is usually related to inflammatory bowel disease (IBD), myeloproliferative disorders or various arthropathies such as spondylitis and rheumatoid arthritis, but it can be idiopathic in about 50% of cases. 2,3, 4,5<\/sup> Skin lesions could follow, precede or appear at the same time as the disease they follow. Its diagnosis is usually a diagnosis of exclusion and it is made based on Delphi criteria \u2013 including major and minor criteria. 6<\/sup><\/p>\n\n\n\n

The incidence of PG in patients with inflammatory bowel disease (IBD) in individual studies ranged from 0.4 to 2.6%.7 <\/sup>Infliximab is a chimeric monoclonal antibody to tumour necrosis factor-alpha (TNF-\u03b1) used to treat moderate to severe IBD.<\/p>\n\n\n\n

Using TNF-\u03b1 antagonists in IBD patients has been successful both in treating intestinal changes and extra-intestinal manifestations, including PG. 4,8<\/sup><\/p>\n\n\n\n

Here we will\npresent the case of a patient with ulcerative colitis (UC) complicated by PG\ninvolving the left hand and right lower leg, successfully treated with\ninfliximab. This is the first case report from Kosovo of a successful treatment\nof PG in UC using infliximab.<\/p>\n\n\n\n

Case Report <\/strong><\/p>\n\n\n\n

Our\npatient is a 28-year-old female with a history of UC who was under treatment with\nsulfasalazine and prednisone when she developed chronic bloody diarrhoea,\nulcerative lesions on her left wrist, right foot (Figure 1,2) and pain in both\nknees that began one month after the diarrhoea. \nShe was diagnosed with UC 2 years ago. <\/p>\n\n\n\n

The\npatient denied any family history of IBD in the family. She does not smoke or\nuse alcohol. No significant past surgical history.  <\/p>\n\n\n\n\n\n
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Figure 1:<\/em> L hand ulceration of pyoderma gangrenosum.<\/strong><\/p>\n

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Figure 2: R foot ulceration of pyoderma gangrenosum<\/strong><\/p>\n

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Laboratory\nstudies were significant for 100 mm\/hour erythrocyte sedimentation, C-reactive\nprotein 79.5 mg\/L, red blood cells 3,08 10 6<\/sup>\/\u00b5L, haemoglobin 96 g\/L,\nMCV 96 fL, platelets 500,000 x 109<\/sup>\/L, leukocytes 13, 6 x 109<\/sup>\/L,\niron 6.5 \u03bcmol\/L, ferritin 1178 \u00b5g\/L, vitamin D 19.7 ng\/mL. Faecal calprotectin\nwas 1426.1 \u00b5g\/mg. <\/p>\n\n\n\n

Antibiotic\ntherapy was initiated, and cultures were collected from the wounds. E. coli and\nStaphylococcus spp. sensitive-based antibiotic therapy was initiated without\nany significant improvement in the subsequent visits. No skin biopsy was\ncollected on the initial visit.<\/p>\n\n\n\n

Plastic\nsurgeons were consulted and recommendations for daily dressings were followed by\nthe patient.<\/p>\n\n\n\n

She\nwas treated with corticosteroids, azathioprine, sulfasalazine tablets, mesalazine\nsuppositories and antibiotics according to the antibiogram but there was only a\nsmall improvement in the general condition and skin changes.<\/p>\n\n\n\n

Diarrhoea\nworkup for Clostridium difficile toxins A and B were negative. Further\nInfectious disease workup for PPD, and QuantiFERON-TB Gold, HLA-B27 were all\nnegative. <\/p>\n\n\n\n

Since\nthere were no improvements with sulfasalazine and prednisone, we decided to\nstart with infliximab 5 mg\/kg at weeks 0, 2, and 6 and every 8\nweeks subsequently. At the same time, azathioprine 2,5 mg\/kg\/day was continued. The patient also received mesalazine suppositories of 1 g\nbefore sleeping. <\/p>\n\n\n\n

After\nthe fifth cycle with infliximab, the changes in the skin were withdrawn, but\nthe scars remained (Figure 3). At the same time, intestinal symptoms such as bloody\ndiarrhoea and abdominal pain significantly improved. <\/p>\n\n\n\n

The\npatient now regularly follows up and is currently on a maintenance dose of\ninfliximab, azathioprine, and mesalazine.<\/p>\n\n\n\n\n\n
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Figure 3: Healed ulceration of pyoderma gangrenosum<\/em><\/strong><\/p>\n

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Discussion <\/strong><\/p>\n\n\n\n

There are no definitive guidelines for the treatment of PG. Oral corticosteroids, cyclosporine, and biological agents such as infliximab and adalimumab have traditionally been used for months, even years. 2,6<\/sup><\/p>\n\n\n\n

Late\ndiagnosis results in the appearance of rough scars causing a significantly poor\nquality of life. Usually, the ulcerative variant is seen on the legs, whereas\natypical pyoderma gangrenosum which is a more\nsuperficial form, is more likely to appear on hands.\nOur patient had changes in the left hand and the right foot.<\/p>\n\n\n\n

In\nsome IBD patients, treating the bowel disease leads to treatment of PG, however, this is not often the case. Fortunately,\nin our case, controlling the underlying disease led to the withdrawal of skin\nchanges. To our knowledge, this is the first case report from Kosovo with a\nsuccessful treatment of PG-associated UC with infliximab.<\/p>\n\n\n\n

The diagnosis of PG in our patient was a diagnosis of exclusion and was made with Delhi criteria.6<\/sup> Treatment of PG remains a challenging subject. TNF-\u03b1, a proinflammatory cytokine, stimulates the synthesis and release of other cytokines, promoting the recruitment of inflammatory cells in the skin through increased expression of adhesion molecules. Inhibiting TNF-\u03b1 may mitigate the inflammatory response. In 2005, the Food and Drug Administration (FDA) approved the utilisation of infliximab for adult patients with moderate to severe active UC who have shown an inadequate response to conventional therapy. 9<\/sup> As we said above, there is no FDA-approved therapy for PG. At our department, we use infliximab only for IBD. After the second cycle of infliximab treatment, there was a significant improvement in stool frequency and bleeding and the skin changes started to improve. At the same time, we continued treatment with azathioprine 150 mg\/day and a mesalazine suppository 1000 mg every night before going to bed. This is consistent with other cases in the literature, where improvements were noticed in the induction phase. Regueiro’s study assessed the reaction of medically intractable PG to infliximab. 10<\/sup> The study of Brooklyn with collaborators demonstrated that infliximab is more effective in the treatment of PG than placebo. 11<\/sup> There is more and more evidence for the use of TNF-\u03b1 inhibitors as first-line therapy, mainly infliximab and adalimumab. 12,13,14<\/sup> Agarwal et al. in their systematic review of 49 publications have identified 60 patients with PG-associated IBD. More than half of them (36 patients) received an anti-TNF-\u03b1. Thirty-four patients received infliximab, four received adalimumab and two patients received both. Thirty-three (92%) responded to one or the other. 15<\/sup><\/p>\n\n\n\n

A semi-systematic review of Ben Abdallah et al. evaluated and compared the clinical effect of TNF-\u03b1 inhibitors in adults with PG-associated IBD. There was no significant statistical difference in response rate and complete response rate between infliximab, adalimumab, and etanercept. 16<\/sup> Typically, patients with PG can expect a favourable prognosis, however, recurrences may happen and residual scarring is possible. In our case, infliximab has been observed to be effective in the treatment of PG caused by UC. However, some cases in the literature show that infliximab alone was not sufficient. In two cases reported, ustekinumab was successful in treating patients refractory to anti-TNF-a. 17,18<\/sup><\/p>\n\n\n\n

Conclusion<\/strong><\/p>\n\n\n\n

PG is a rare\ncondition in Kosovo, therefore it is prone to misdiagnosis and it might take\ntime to diagnose. This case being the first one reported from Kosovo, confirms\nthat the usage of infliximab\nin the treatment of PG associated with UC is effective in suppressing symptoms and stimulating\nclinical remission of skin lesions. Most lesions improve leaving visible scars\non the affected sites. For this reason, early recognition and treatment of PG\nis very important to prevent secondary infections and poor quality of life. A\nmultidisciplinary approach is essential in treating PG.<\/p>\n\n\n\n

Acknowledgements<\/strong><\/p>\n\n\n\n

We thank, Dr. Loran Rakovica , for the\nEnglish language editing of the paper.<\/p>\n\n\n\n

Conflict\nof interest<\/strong><\/p>\n\n\n\n

The authors declare no conflict of interest.<\/p>\n\n\n\n

Funding Sources<\/strong><\/p>\n\n\n\n

None<\/p>\n\n\n\n

Authors\u2019 Contribution<\/strong><\/p>\n\n\n\n

ST, FA, AV, AP (Conceptualization, formal analysis, data curation,\nsupervision, writing original draft, review, and editing). FH, RB, ST, FA\n(Investigation, patient administration, software, visualization, validation,\nreview, and editing). All authors declared that they contributed to this\narticle and that they have read and approved the final manuscript. <\/p>\n\n\n\n

Data Availability <\/strong><\/p>\n\n\n\n

The authors confirm that\nthe data supporting the findings of this case report are available within the\narticle.<\/p>\n\n\n\n

Statement of Ethics<\/strong><\/p>\n\n\n\n

Informed consent was\nobtained from the patient for the publication of this case report and any\naccompanying images.<\/p>\n\n\n\n

References<\/strong><\/p>\n\n\n\n

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     CrossRef <\/a><\/li><\/ol>\n","protected":false},"excerpt":{"rendered":"

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