Treatment of Pyodermatitis-Pyostomatitis Vegetans: A Systematic Review and Meta-analysis
Main Article Content
Keywords
Pyodermatitis, Pyostomatitis, Inflammatory Bowel Diseases, Extra-intestinal Manifestations, Treatment Outcomes
Abstract
Background: Pyodermatitis-pyostomatitis vegetans (PDV-PSV) is a rare muco-cutaneous disorder characterized by vegetating and pustular plaques and is often associated with inflammatory bowel disease (IBD). The purpose of this study was to systematically identify and analyze reports of PDV-PSV to determine the most effective treatment.
Methods: Reports of PDV-PSV were identified using the OVID-Medline database from inception through November 2019. Publications were excluded if no new patient case was included, if there was not clinical and histological evidence of PDV, PSV, or PDV-PSV, or if no treatment was discussed.
Results: The final sample was comprised of 74 publications plus an additional patient from the authors’ institution, corresponding to 95 total patients. The basis of the review and analysis is limited to case reports and case series, which likely only report the cases with positive outcomes. Statistical analysis revealed that oral corticosteroids (OCS), 6-mercaptopurine/azathioprine, oral calcineurin inhibitors (OCNI), 5-aminosalicylic-acid (5-ASA), and biologics (BIO) were the most effective treatments for PDV-PSV. Topical medications, colchicine, oral dapsone, and other antibiotics were ineffective treatments, with topical medications being the least effective option. When OCS are used, they work best when used as initial treatment to induce remission. 5-ASA and BIO are most effective when used as maintenance therapies after initial remission.
Conclusions: Thus, first line therapy for PDV-PSV should begin with OCS with transition to steroid-sparing agents including OCNI, BIO, and 5-ASA if indicated.
References
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3. Clark LG, Tolkachjov SN, Bridges AG, Camilleri MJ. Pyostomatitis vegetans (PSV)-pyodermatitis vegetans (PDV): A clinicopathologic study of 7 cases at a tertiary referral center. J Am Acad Dermatol. 2016 Sep;75(3):578–84.
4. Bianchi L, Carrozzo AM, Orlandi A, Campione E, Hagman JH, Chimenti S. Pyoderma vegetans and ulcerative colitis. Br J Dermatol. 2001 Jun;144(6):1224–7.
5. Mehravaran M, Kemény L, Husz S, Korom I, Kiss M, Dobozy A. Pyodermatitis-pyostomatitis vegetans. Br J Dermatol. 1997 Aug;137(2):266–9.
6. Brunsting LA, Underwood LJ. Pyoderma vegetans in association with chronic ulcerative colitis. Arch Dermatol Syphilol. 1949 Aug;60(2):161–72.
7. Abellaneda C, Mascaró JM, Vázquez MG, Pablo IM-D, Iranzo P. All that glitters is not pemphigus: Pyodermatitis-pyostomatitis vegetans misdiagnosed as IgA pemphigus for 8 years. Am J Dermatopathol. 2011 Feb;33(1):e1-6.
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9. Al-Rimawi HS, Hammad MM, Raweily EA, Hammad HM. Pyostomatitis vegetans in childhood. Eur J Pediatr. 1998 May;157(5):402–5.
10. Atarbashi-Moghadam S, Lotfi A, Atarbashi-Moghadam F. Pyostomatitis Vegetans: A Clue for Diagnosis of Silent Crohn’s Disease. J Clin Diagn Res JCDR. 2016 Dec;10(12):ZD12–3.
11. Ayangco L, Rogers RS, Sheridan PJ. Pyostomatitis vegetans as an early sign of reactivation of Crohn’s disease: a case report. J Periodontol. 2002 Dec;73(12):1512–6.
12. Ballo FS, Camisa C, Allen CM. Pyostomatitis vegetans. Report of a case and review of the literature. J Am Acad Dermatol. 1989 Aug;21(2 Pt 2):381–7.
13. Bens G, Laharie D, Beylot-Barry M, Vergier B, Noblesse I, Beylot C, et al. Successful treatment with infliximab and methotrexate of pyostomatitis vegetans associated with Crohn’s disease. Br J Dermatol. 2003 Jul;149(1):181–4.
14. Bertlich I, Gauss A, Schäkel K, Enk A, Hoffmann JHO. Pyodermatitis-pyostomatitis vegetans with histological and immunohistological aspects of autoimmune blistering disease treated with infliximab. J Dtsch Dermatol Ges J Ger Soc Dermatol JDDG. 2019;17(5):540–2.
15. Brinkmeier T, Frosch PJ. Pyodermatitis-pyostomatitis vegetans: a clinical course of two decades with response to cyclosporine and low-dose prednisolone. Acta Derm Venereol. 2001 May;81(2):134–6.
16. Calobrisi SD, Mutasim DF, McDonald JS. Pyostomatitis vegetans associated with ulcerative colitis. Temporary clearance with fluocinonide gel and complete remission after colectomy. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1995 Apr;79(4):452–4.
17. Canpolat F, Cemil BÇ, Yılmazer D, Yeşilli O, Eskioğlu F. Pyoderma vegetans associated with ulcerative colitis: a case with good response to steroids. Case Rep Dermatol. 2011 Mar 26;3(1):80–4.
18. Carvalho S, Sanches M, Alves R, Selores M. Pyodermatitis vegetans of the vulva. Dermatol Online J. 2016 Jun 16;22(6).
19. Cataldo E, Covino MC, Tesone PE. Pyostomatitis vegetans. Oral Surg Oral Med Oral Pathol. 1981 Aug;52(2):172–7.
20. Chan SW, Scully C, Prime SS, Eveson J. Pyostomatitis vegetans: oral manifestation of ulcerative colitis. Oral Surg Oral Med Oral Pathol. 1991 Dec;72(6):689–92.
21. Chaudhry SI, Philpot NS, Odell EW, Challacombe SJ, Shirlaw PJ. Pyostomatitis vegetans associated with asymptomatic ulcerative colitis: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1999 Mar;87(3):327–30.
22. Dodd EM, Howard JR, Dulaney ED, Rosenthal SI, Wanna MR, Farah RS. Pyodermatitis-pyostomatitis vegetans associated with asymptomatic inflammatory bowel disease. Int J Dermatol. 2017;56(12):1457–9.
23. Dupuis EC, Haber RM, Robertson LH. Pyoblepharitis Vegetans in Association With Pyodermatitis-Pyostomatitis Vegetans: Expanding the Spectrum of a Rare, Multisystem Disorder. J Cutan Med Surg. 2016 Apr;20(2):163–5.
24. Ficarra G, Cicchi P, Amorosi A, Piluso S. Oral Crohn’s disease and pyostomatitis vegetans. An unusual association. Oral Surg Oral Med Oral Pathol. 1993 Feb;75(2):220–4.
25. Forman L. TWO CASES OF PYODERMITE VEGETANTE (HALLOPEAU): AN EOSINOPHILIC PUSTULAR AND VEGETATING DERMATITIS WITH CONJUNCTIVAL, ORAL AND COLONIC INVOLVEMENT. Proc R Soc Med. 1965 Apr;58:244–9.
26. Gonzalez-Moles MA, Gil-Montoya JA, Ruiz-Avila I, Esteban F, Bascones-Martinez A. Pyostomatitis vegetans: dramatic clinical response to clobetasol propionate treatment in aqueous solution. J Eur Acad Dermatol Venereol JEADV. 2008 Feb;22(2):252–3.
27. Hansen LS, Silverman S, Daniels TE. The differential diagnosis of pyostomatitis vegetans and its relation to bowel disease. Oral Surg Oral Med Oral Pathol. 1983 Apr;55(4):363–73.
28. Harish K, Varghese T, Najeeba R, Harikumar R. Pyoderma vegetans and ulcerative colitis. J Postgrad Med. 2006 Dec;52(4):302–3.
29. Healy CM, Farthing PM, Williams DM, Thornhill MH. Pyostomatitis vegetans and associated systemic disease. A review and two case reports. Oral Surg Oral Med Oral Pathol. 1994 Sep;78(3):323–8.
30. Kajihara I, Ichihara A, Higo J, Kidou M, Ihn H. Pyodermatitis vegetans associated with multiple myeloma. J Dermatol. 2013 Mar;40(3):222–3.
31. Kalman RS, Gjede JM, Farraye FA. Pyostomatitis vegetans in a patient with fistulizing Crohn’s disease. Clin Gastroenterol Hepatol Off Clin Pract J Am Gastroenterol Assoc. 2013 Dec;11(12):A24.
32. Khader A, Ambooken B, Muhammed K, Binitha MP, Sureshan DN, Manakkad SP. Pyodermatitis-pyostomatitis Vegetans with Penile Ulceration Complicated by Acute Glomerulonephritis. Indian J Dermatol. 2016 Apr;61(2):239.
33. Kim TH, Kim S-C. Pyodermatitis-Pyostomatitis Vegetans Associated with Crohn’s Disease. Ann Dermatol. 2015 Oct;27(5):624–5.
34. Kitayama A, Misago N, Okawa T, Iwakiri R, Narisawa Y. Pyodermatitis-pyostomatitis vegetans after subtotal colectomy for ulcerative colitis: Pyodermatitis-pyostomatitis vegetans. J Dermatol. 2010 Jul 22;37(8):714–7.
35. Knapp N, Khan Z, Albuquerque R, Richards A, Brown RM. Pyostomatitis Vegetans in Ulcerative Colitis; Management
with Topical Tacrolimus and Systemic Azathioprine in a 10-year-old Boy (Case Report and Review of the Literature). J Int Oral Health. 2015 Aug 15;8(1):132–6.
36. Ko H-C, Jung D-S, Jwa S-W, Cho H-H, Kim B-S, Kwon K-S, et al. Two cases of pyodermatitis-pyostomatitis vegetans. J Dermatol. 2009 May;36(5):293–7.
37. Konstantopoulou M, O’Dwyer EM, Steele JC, Field EA, Lewis M a. O, Macfarlane AW. Pyodermatitis–pyostomatitis vegetans complicated by methicillin-resistant Staphylococcus aureus infection. Clin Exp Dermatol. 2005;30(6):666–8.
38. Leibovitch I, Ooi C, Huilgol SC, Reid C, James CL, Selva D. Pyodermatitis-pyostomatitis vegetans of the eyelids case report and review of the literature. Ophthalmology. 2005 Oct;112(10):1809–13.
39. Li S, Li Z, Feng S. Low-dose sulfasalazine in a case of Pyodermatitis-pyostomatitis Vegetans. J Am Acad Dermatol. 2018 Oct 20;
40. Lopez-Jornet P, Gomez-Garcia F, Camacho-Alonso F. Pyostomatitis vegetans. Clinical marker of ulcerative colitis. N Y State Dent J. 2012 Mar;78(2):36–7.
41. Lourenço SV, Hussein TP, Bologna SB, Sipahi AM, Nico MMS. Oral manifestations of inflammatory bowel disease: a review based on the observation of six cases. J Eur Acad Dermatol Venereol JEADV. 2010 Feb;24(2):204–7.
42. Maseda R, Rodriquez AI, Feito M, De Lucas R. A case of pyoderma vegetans associated with Crohn’s disease under adalimumab. J Am Acad Dermatol. 2017 Jun;76(6):AB8.
43. Markiewicz M, Suresh L, Margarone J, Aguirre A, Brass C. Pyostomatitis Vegetans: A Clinical Marker of Silent Ulcerative Colitis. J Oral Maxillofac Surg. 2007 Feb;65(2):346–8.
44. Gold S. Ulcerative colitis with pyodermite vegetante. Proc R Soc Med. 1962 Dec;55:1072–3.
45. Matias F de AT, Rosa DJ de F, Carvalho MTF de, Castañon MCMN. Pyodermatitis-pyostomatitis vegetans: case report and review of medical literature. An Bras Dermatol. 2011 Aug;86(4 Suppl 1):S137-140.
46. Merkourea SS, Tosios KI, Merkoureas S, Sklavounou-Andrikopoulou A. Pyostomatitis vegetans leading to Crohn’s disease diagnosis. Ann Gastroenterol. 2013;26(2):187.
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Jul 9, 2021
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Hobbs, L. K., Zufall, A., Khalil, S., & Flowers, R. (2021). Treatment of Pyodermatitis-Pyostomatitis Vegetans: A Systematic Review and Meta-analysis . SKIN The Journal of Cutaneous Medicine, 5(4), 333–346. https://doi.org/10.25251/skin.5.4.1
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