Pyostomatitis Vegetans in a Young Woman with Gastrointestinal Symptoms: a case report and review of literature

Document Type : Case Review

Authors

1 Associate Professor of Oral Medicine, Faculty of Dentistry, Tehran University of Medical Sciences, Tehran, Iran

2 Professor, Fellowship of Research Biology, Dental Research Center, Dental Research Institute & Department of Oral and Maxillofacial Medicine, Faculty of Dentistry, Tehran University of Medical Sciences, the Academy of Medical Sciences, Tehran, Iran

3 Assistant Professor of Oral Medicine, Faculty of Dentistry, Arak University of Medical Sciences, Arak, Iran

4 Associate Professor of Oral Medicine, Faculty of Dentistry, Aja University of Medical Sciences, Tehran, Iran

10.22062/jkmu.2021.91567

Abstract

Pyodermatitis-pyostomatitis vegetans (PD-PSV) is an uncommon inflammatory mucocutaneous disease with unknown etiology. It is characterized by pustular and vegetating plaques on the skin and oral mucosa. When the oral mucosa is involved alone, it is called pyostomatitis vegetans. This disease is considered as an oral manifestation of inflammatory bowel diseases.
In the present paper, we report a case of pyostomatitis vegetans in a 23-year-old female patient with swelling of the lips and oral lesions. Our patient had simultaneously three important clinical symptoms, including the presence of intra-oral lesions, lip swelling, and gastrointestinal manifestations that may be suggestive of inflammatory bowel disease. Her oral manifestations improved with topical antifungal drug and local and systemic corticosteroids.
In addition, the results of our extensive search in reliable scientific databases including Google and PubMed for articles (case reports and literature reviews) containing the keywords: ”pyostomatitis vegetans” and "pemphigus vegetans” in recent years are presented.

Keywords


Mesquita Kde C, Costa IM. Case for diagnosis. An Bras Dermatol 2012; 87(6):929-31.
Matias Fde A, Rosa DJ, Carvalho MT, Castañon MC. Pyodermatitis-pyostomatitis vegetans: case report and review of medical literature. An Bras Dermatol 2011; 86(4 Suppl 1):S137-40.
Sollecito TP, Stoopler ET, Rangarajan S, Pinto A. Pyostomatitis vegetans and orofacial granulomatosis: a case report and review of the literature. The Internet Journal of Anesthesiology 2003; 14(2).
Saghafi S, Falaki F, Bashardoost N. Pyostomatitis vegetans: report of a rare case. Pol J Pathol 2011; 62(2):125-8.
Mijandrusić-Sincić B, Licul V, Gorup L, Brncić N, Glazar I, Lucin K. Pyostomatitis vegetans associated with inflammatory bowel disease--report of two cases. Coll Antropol 2010; 34(Suppl 2):279-82.
Lopez-Jornet P, Gomez-Garcia F, Camacho-Alonso F. Pyostomatitis vegetans. Clinical marker of ulcerative colitis. N Y State Dent J 2012; 78(2):36-7.
Basirat M, Pakfetrat A, Kakaei N, Rohani B, Bashardoust N. Crohn’s disease with oral onset - a case report. J Res Dentomaxillofac Sci 2016; 1(4):26-32.
Lankarani KB, Sivandzadeh GR, Hassanpour S. Oral manifestation in inflammatory bowel disease: a review. World J Gastroenterol 2013; 19(46):8571-9.
Pereira MS, Munerato MC. Oral manifestations of inflammatory bowel diseases: two case reports. Clin Med Res 2016; 14(1):46-52.
Dhamija A, D'souza P, Meherda A, Kothiwala RK. Pemphigus vegetans: an unusual presentation. Indian Dermatol Online J 2012; 3(3):193-5.
Mendes-Bastos P, Amaro C, Fernandes C. Cobblestone mouth: an exuberant oral presentation of pemphigus vegetans. Actas Dermosifiliogr 2015; 106(1):72-3.
Markopoulos AK, Antoniades DZ, Zaraboukas T. Pemphigus vegetans of the oral cavity. Int J Dermatol 2006; 45(4):425-8.
Augusto de Oliveira M, Martins E Martins F, Lourenço S, Gallottini M, Ortega KL. Oral pemphigus vegetans: a case report. Dermatol Online J 2012; 18(10):10.
von Köckritz A, Ständer S, Zeidler C, Metze D, Luger T, Bonsmann G. Successful monotherapy of pemphigus vegetans with minocycline and nicotinamide. J Eur Acad Dermatol Venereol 2017; 31(1):85-8.
Gholizadeh N, Taghavi Zenouz A, Eslami H. Pemphigus vulgaris associated with rheumatoid arthritis in a patient not taking penicillamine. J Dent Res Dent Clin Dent Prospects 2012; 6(1):33-5.
Femiano F, Lanza A, Buonaiuto C, Perillo L, Dell'Ermo A, Cirillo N. Pyostomatitis vegetans: a review of the literature. Med Oral Patol Oral Cir Bucal 2009; 14(3):E114-7.
Liu MJ, Shyur SD, Chuang HH, Yang PH. Hereditary angioedema and Graves' disease: the first case report. J Formos Med Assoc2017; 116(10):819-20.
Glick M. Burket’s Oral Medicine. 12th ed. USA: People’s Medical Publishing House; 2015. p.57-89.
Shephard M, Venda Nova C, Hodgson TA. Eosinophilia associated with disease exacerbations in a patient with pyostomatitisvegetans. Br J Dermatol 2015; 173(6):1556-7.
Nico MM, Hussein TP, Aoki V, Lourenço SV. Pyostomatitis vegetans and its relation to inflammatory bowel disease, pyoderma gangrenosum, pyodermatitis vegetans, and pemphigus. J Oral Pathol Med 2012; 41(8):584-8.
Markiewicz M, Suresh L, Margarone J 3rd, Aguirre A, Brass C. Pyostomatitis vegetans: a clinical marker of silent ulcerative colitis. J Oral Maxillofac Surg 2007; 65(2):346-8.
Tursi A. Concomitant hidradenitis suppurativa and pyostomatitis vegetans in silent ulcerative colitis successfully treated with golimumab. Dig Liver Dis 2016; 48(12):1511-2.
Ruiz-Roca JA, Berini-Aytés L, Gay-Escoda C. Pyostomatitis vegetans. Report of two cases and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005; 99(4):447-54.
Wu YH, Chang JY, Chen HM, Wang YP. Pyostomatitis vegetans: an oral manifestation of inflammatory bowel disease. J Formos Med Assoc 2015; 114(7):672-3.
Kitayama A, Misago N, Okawa T, Iwakiri R, Narisawa Y. Pyodermatitis-pyostomatitis vegetans after subtotal colectomy for ulcerative colitis. J Dermatol 2010; 37(8):714-7.
Mehravaran M, Kemény L, Husz S, Korom I, Kiss M, Dobozy A. Pyodermatitis-pyostomatitis vegetans. Br J Dermatol 1997; 137(2):266-9.