Pyostomatitis Vegetans Research Articles

Pyostomatitis vegetans. Report of two cases and review of the literature

Pyostomatitis vegetans (PV) is a rare benign chronic mucocutaneous pustular disorder of unknown etiology, though it is usually associated with chronic inflammatory disease of the bowel—particularly ulcerative colitis. At the oral level PV manifests as an exophytic lesion with an erythematous perimeter and a creamy-yellow surface presenting a friable covering membrane which easily disintegrates, giving rise to the development of small ulcers or superficial erosions. The histology shows epithelial acanthosis and superficial ulceration. The underlying connective tissue exhibits neutrophil and eosinophil infiltration, with miliary abscesses in some cases. The present study describes 2 cases of PV associated with ulcerative colitis. The differential diagnosis should be established with Neumann type pemphigus vegetans, since in both cases the lesions are clinically similar and the histology reveals an important eosinophil response, acanthosis, and the formation of intraepithelial miliary microabscesses. The treatment of PV focuses on control of the disease. In the event the orofacial lesions persist, topical oral drugs such as corticoids, with additional antibiotherapy, multivitamin complexes, and nutritional supplements, can be provided.

P0700 PROSPECTIVE EVALUATION OF ORAL CROHNS DISEASE IN CHILDREN

Introduction: Previous reports indicate that oral lesions occur in 6 to 20% of patients with Crohn’s disease (CD). A recent retrospective study at our centre revealed a significantly higher incidence of disease specific oral lesions (48%) in patients undergoing formal evaluation by a paediatric dental surgeon. The aim of this study was to prospectively document the proportion of children with oral lesions at initial evaluation for suspected inflammatory bowel disease. Methods: Data were collected between Jan 1999 and Dec2001. All children suspected of having IBD were included in the study. Systematic dental examinations were carried out on the day of endoscopy and findings documented. Information regarding age, sex, presenting symptoms, oral symptoms, family history, medications, findings on clinical examination, routine laboratory results and barium follow-through studies were recorded. All children underwent upper gastrointestinal endoscopy and colonoscopy. Biopsy specimens were taken from grossly affected tissue and unaffected gastric and colonic mucosa. Oral biopsies were taken as appropriate. Results: Forty-eight of 49 patients with CD were examined by the dentist. Findings consistent with oral CD were found in 20 patients (41.7%). Non-specific changes were found in 8(16.7%) patients, while 20(41.7%) patients had a normal oral examination. Six patients with oral CD were on medications at the time of evaluation. Patients with oral CD were more likely to complain of oral symptoms, 14 /20(70%) versus 9/28 (32.1%) of patients without oral CD (p=0.013). Oral findings included mucogingivitis (12), tags (4), deep ulceration (4), cobblestoning (3), lip swelling (3) and pyostomatitis vegetans (1). Non-caseating granulomas were found in each of the eight oral biopsies that were performed. Two patients with granulomas in oral biopsies had no granulomas in biopsies from elsewhere in the GI tract. There was no difference in the systemic symptoms or laboratory markers in patients with or without oral CD. When comparing disease location between patients with and without oral CD five patients were excluded as they had H. Pylori infection (n=43). Children with oral CD were more likely to have perianal disease 10/20 (50%) compared to 4/23 (17.4%) (p =0.023), but other sites of disease were similarly affected in both groups. In seven patients (36.84%) with oral CD was the mouth found to be abnormal by the gastroenterologist. Only in 25(58.1%) of patients did the consultants findings correlate with those of the dentist. Conclusion: Children with CD commonly have oral CD findings even in the absence of symptoms. These oral changes provide important diagnostic information. Expert dental evaluation is necessary in order to accurately identify these oral lesions.

Pyostomatitis vegetans.

Pyostomatitis vegetans is a rare oral disorder often associated with gastrointestinal and/or cutaneous diseases and sometimes with other disorders. The association of pyostomatitis vegetans with inflammatory bowel disease, particularly ulcerative colitis, is well known and in most cases bowel disease precedes the onset of oral lesions by months or years. A peripheral eosinophilia has been observed in most reported cases. Pyostomatitis vegetans is characterized by erythematous, thickened oral mucosa with multiple pustules and superficial erosions. Histopathologically, it is characterized by intraepithelial and/or subepithelial abscesses containing large numbers of eosinophils. The pathogenesis is as yet unknown although immunological and microbial factors have been suggested as possible aetiological factors. Management of pyostomatitis vegetans consists mainly of immunosuppression using topical or systemic corticosteroids with medical and/or surgical treatment of any underlying inflammatory bowel disease.

Salivary duct fistula and recurrent buccal space infection: a complication of Crohn’s disease

Crohn’s disease is a chronic granulomatous inflammatory bowel disorder of unknown etiology. It can affect any part of the gastrointestinal tract including the oral cavity, but it most commonly occurs in the terminal ileum. The orofacial features of Crohn’s disease have previously been described in a number of reports and include cobblestoning of the oral mucosa, oral ulceration, diffuse labial buccal or gingival swellings, epithelial tags and folds, angular chelitis, lip fissuring, gingivitis, pyostomatitis vegetans, and persistent lymphadenopathy.1,2

Pyodermatitis—Pyostomatitis Vegetans: Two Cases and a Review of the Literature

Background: Pyodermatitis–pyostomatitis vegetans (PD–PSV) is a rare vegetating, pustular, eosinophilic, mucocutaneous dermatosis characterized by mucocutaneous lesions of the genital, axillary, and oral regions, as well as on the scalp. Objective: We report two patients who were diagnosed with PD–PSV. The published cases of this rare disorder are reviewed. Results: The first patient presented with vegetating pustular plaques of the scalp, vulva, and mouth. The second patient initially showed pustules on fingers and then on toes. Vulva and mouth were later involved. There was no gastrointestinal involvement in either case. In both cases, histology revealed eosinophilic spongiosis with eosinophilic microabscesses and pseudoepitheliomatous hyperplasia. Direct immunofluorescence was negative. Conclusion: Pyodermatitis–pyostomatitis vegetans is a rare disorder of unknown cause. It bears similarities to pemphigus vegetans but histology, direct immunofluorescence, and clinical history show the differences. Response to treatment is generally rapid. In several reported cases, an inflammatory gastrointestinal disorder is found in association with the mucocutaneous signs.

潰よう性大腸炎に合併した増殖性化膿性口内炎（ｐｙｏｓｔｏｍａｔｉｔｉｓ ｖｅｇｅｔａｎｓ）の１例

A case of pyostomatitis vegetans associated with ulcerative colitis is reported. A 57-year-old man with a history of ulcerative colitis was referred to our hospital because of pain in both sides of the buccal mucosa and malnutrition. A snail-track-shaped ulcer and miliary pustules with a cobblestone appearance were found in both sides of the buccal mucosa and soft palate. Treatment with topical corticosteroids had no effect on this lesion. The presence of abdominal pain and hematochezia led us to suspect a recurrence of ulcerative colitis. Immediately after the systemic administration of corticosteroids was started, the oral mucosal lesions resolved. This case suggests that treatment of ulcerative colitis with systemic corticosteroids is required to cure pyostomatitis vegetans.

Pyostomatitis vegetans associated with asymptomatic ulcerative colitisA case report

Pyostomatitis vegetans, a rare pustular disorder of the oral mucosa, is a highly specific marker for inflammatory bowel disease and may be difficult to treat. A case of pyostomatitis vegetans in a patient with long-standing asymptomatic ulcerative colitis is presented. Complete remission was achieved with topical steroids; no systemic treatment was required. ( Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999;87:327-30)

Pyostomatitis vegetans associated with ulcerative colitis: Temporary clearance with fluocinonide gel and complete remission after colectomy

Pyostomatitis vegetans is a rare condition characterized by pustules that affect the oral mucosa. It is consistently associated with inflammatory bowel disease and is usually resistant to treatment. We present the case of a 65-year-old white man with pyostomatitis vegetans that was associated with ulcerative colitis and adenocarcinoma of the colon. Fluocinonide gel resulted in a complete but temporary clearance of the lesions. Complete remission was achieved immediately after a total colectomy.

Pyostomatitis vegetans and associated systemic disease: A review and two case reports

In this article two case reports of pyostomatitis vegetans are presented. Both cases were associated with inflammatory bowel disease and one case was also associated with liver dysfunction. It has recently been reported that there may be a link between liver dysfunction and pyostomatitis vegetans. The management of the condition is illustrated and the literature on the subject is reviewed.

Oral Crohn's disease and pyostomatitis vegetans: An unusual association

Oral features of Crohn's disease include ulcers, lip fissuring, cobblestone plaques, angular cheilitis, polypoid lesions, and perioral erythema. Pyostomatitis vegetans is a rare eruption of the oral mucosa characterized by tiny yellow pustules. It is considered a marker for inflammatory bowel disease. We describe a 45-year-old woman with a 6-month history of painful sores in her mouth, diarrhea, weight loss, and cutaneous lesions. Oral examination revealed cobblestone plaques and indentation on the tongue and friable vegetating pustules on the labial commissures. Staphylococcus simulans was isolated from the pustules. Laboratory studies revealed leucocytosis, eosinophilia, and low hemoglobin and zinc levels. Histologic study of the labial lesions revealed hyperplastic epithelium with intraepithelial clefts that contain eosinophils and neutrophils. Tongue lesions showed chronic inflammation with noncaseating granulomas. Later, colonoscopy and biopsy demonstrated Crohn's disease of the anorectal region. Pyostomatitis vegetans lesions regressed after oral zinc supplementation. Prednisone treatment resulted in healing of the tongue lesions. In our patient, pyostomatitis vegetans appeared to be related to zinc deficiency that may have been caused by malabsorption. The pathogenetic interrelationship between pyostomatitis vegetans and Crohn's disease is discussed.

Pyostomatitis vegetans and primary sclerosing cholangitis: Markers of inflammatory bowel disease

The case of a 34-year-old woman with both pyostomatitis vegetans and primary sclerosing cholangitis is reported and the literature reviewed. Pyostomatitis vegetans is a rare disorder characterized by friable pustules on the buccal mucosa and often peripheral blood eosinophilia. In this review, the frequent association of pyostomatitis vegetans with inflammatory bowel disease as well as the potential for pyostomatitis vegetans to present with sclerosing cholangitis and liver disease before any other manifestations of inflammatory bowel disease is discussed. Patients with pyostomatitis vegetans may need long-term care for gastroenterological disease in addition to treatment for oral lesions.

Pyostomatitis vegetans: Oral manifestation of ulcerative colitis

Two cases of pyostomatitis vegetans, an uncommon oral manifestation of ulcerative colitis, are reported, and the literature is reviewed.

口腔内に症状を呈したクローン病の１例

Crohn's disease is defined as a granulomatous inflammatory condition affecting sectionsfrom the oral cavity to anus. A 57-year-old female suffering from Crohn's disease with oralmanifestations is reported in this paper. She had ulcerative lesions in the oral cavity, in theesophagus and in the anus. She also suffered from a Sjogren's syndrome like condition. Theoral manifestations of the patient were aphthous ulcer, mucosal tag, pyostomatitis vegetans, nodular swelling on the dorsum of the tongue and gingivitis.

かいよう性大腸炎に合併した増殖性化のう性口内炎の１症例

A case of pyostomatitis vegetans associated with ulcerative colitis of a 22-year old woman has been reported, and clinical appearances, etiology and immunological mechanisms of development were discussed. Ulcerative colitis, as well as Crolm's disease and proctitis, is one of the inflammatory bowel diseases which have many local and systemic complications. Oral manifestations of ulcerative colitis arc as follows; 1) recurrent aphthous ulceration, 2) pyostomatitis vegetans, 3) ulcers analogous to pyoderma gangrenosum of the skin and 4) hemorrhagic ulcers of the oral mucosa and skin.The present case showed clinical appearances of above pyostomatitis vegetans, indicating numerous small papillary projections that contained miliary abscesses occurring in an area of intense erythema on the gingiva. The mucosa had been thrown into folds along gingivobuccal sulucus. Bacteriologic studies revealed no specific findings except normal oral microorganisms. Little subjective discomfort was associated with this condition. The histopathologic appearances were also compatible with pyostomatitis vegetans in which acanthosis and hyperkeratosis could be seen with elongation of the rete pegs. Small pustules existed in and under the epithelium of papillary projections. A dense lymphocytic and plasms cellular infiltration was found in the lamina propria, and cellular infiltration was reconfirmed by direct immunofluorescence technique using antihuman IgA antibodies, etc.The oral manifestation became worse and regressed according to the conditions of recurrence and covalescence of ulcerative colitis. The lesion responded well to topical application of a gluco-corticoid steroid hormone.The present case indicates that, in findings this kind of lesion on the oral mucosa, a possibility of gastrointestinal disturbance due to inflammatory bowel disease should be considered.

Pyostomatitis Vegetans

• Pyostomatitis vegetans is a rarely occurring disorder of the oral mucosa, which has a dramatic clinical picture and a striking histopathologic pattern. Moreover, the condition is a highly specific oral mucosal marker for inflammatory bowel disease, particularly ulcerative colitis. Our findings in two cases and a review of the literature emphasize the relationship of pyostomatitis vegetans to inflammatory bowel disease and the vegetating dermatoses. ( Arch Dermatol 1985;121:94-98)

The differential diagnosis of pyostomatitis vegetans and its relation to bowel disease

Pyostomatitis vegetans is a rare oral disorder that is usually associated with bowel and/or skin disease. The clinical appearance is of soft hyperplastic folds of mucosa characterized by small miliary abscesses with superficial erosions. Upon analysis of the clinical, histologic, and immunofluorescent findings, cases of pyostomatitis vegetans may be classified as the oral manifestation of either pyoderma vegetans of Hallopeau, pemphigus vegetans of Hallopeau, or pemphigus vegetans of Neumann, although the exact definitions and even the existence of these diseases as separate entities are still a matter of controversy.

A case of ulcerative colitis and pyostomatitis vegetans in an African.

A case of pyostomatitis vegetans and ulcerative colitis in a 26-year-old African is described. The colitis, confirmed clinically and radiologically, antedated the development of pyostomatitis. Systemic corticosteroids apparently cured the pyostomatitis but only caused an improvement in the ulcerative colitis. The rarity of ulcerative colitis amongst the Nigerian population and of pyostomatitis vegetans as a complication makes the occurrence of these two diseases in a Nigerian an extraordinary event.

A SYNDROME OF PYOSTOMATITIS VEGETANS AND ULCERATIVE COLITIS.

A syndrome of pyostomatitis and colitis has been described and a series of six cases presented. In studying purulent vegetating lesions of the oral mucosa, a history of gastrointestinal disturbance should be considered. In view of the high incidence of colitis, it may be of value to carry out more careful oral and skin examinations in these patients. Because of the significance of psychogenic factors in the etiology of ulcerative colitis, 10,11 the importance of psychological background must not be overlooked in the consideration of the possible etiology of this rare syndrome.

Pyostomatitis vegetans: Report of a case

The case history of a patient with pyostomatitis vegetans has been presented. The condition was characterized in its early stage by painless minute miliary abscesses; later, the lesions became verrucous in character. Microscopically, there was a marked acanthosis and keratosis. Microscopic abscesses were present in the epithelium. There was a dense cellular infiltration of the tunica propria, with the cosinophil predominating.

Pyostomatitis vegetans. Report of three cases: Francis P. McCarthy. Arch. Dermat. & Syph. 60: 750, 1949

Pyostomatitis vegetans. Report of three cases: Francis P. McCarthy. Arch. Dermat. & Syph. 60: 750, 1949