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Buccal localization of Crohn's disease with long-term infliximab therapy: a case report.

Ciacci C, Bucci C, Zingone F, Iovino P, Amato M - J Med Case Rep (2014)

Bottom Line: Treatment of Crohn's disease with an anti-tumor necrosis factor alpha agent (infliximab) successfully induced remission of both the gastrointestinal disease and the oral lesion.Our recommendation is that physicians should be able to recognize cheilitis granulomatosa as a possible marker of a more complex systemic disease and proceed first with an accurate physical examination, and further suggest investigations of the bowel.In cases of Crohn's disease, a therapy with biological agents can be successful.

View Article: PubMed Central - PubMed

Affiliation: Gastroenterology Unit, Department of Medicine and Surgery, University of Salerno, Baronissi Campus, via S, Allende, 84081 Baronissi, Salerno, Italy. cciacci@unisa.it.

ABSTRACT

Introduction: Cheilitis granulomatosa causes persistent idiopathic lip swelling and ulceration and it can sometimes be recognized as a unique or early manifestation of Crohn's disease. Spontaneous remission is rare and with the lack of controlled trials, different therapeutic approaches have been used. Some cases have been treated with an exclusion diet in the attempt to rule out diet allergens, while the most popular treatments include antibiotics such as tetracycline and clofazimine tranilast, benzocaine topical or intralesional steroids, and cheiloplasty, with different outcomes.

Case presentation: We describe the case of a 23-year-old Caucasian man, primarily diagnosed with cheilitis granulomatosa for a severe lower lip swelling, and then with Crohn's disease of the terminal ileum and anus. Treatment of Crohn's disease with an anti-tumor necrosis factor alpha agent (infliximab) successfully induced remission of both the gastrointestinal disease and the oral lesion.

Conclusions: Our recommendation is that physicians should be able to recognize cheilitis granulomatosa as a possible marker of a more complex systemic disease and proceed first with an accurate physical examination, and further suggest investigations of the bowel. In cases of Crohn's disease, a therapy with biological agents can be successful.

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Related in: MedlinePlus

Colonoscopy. Panel A shows the rectal inflamed mucosa and panel B (arrow) the opening of an anal fistula.
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Fig1: Colonoscopy. Panel A shows the rectal inflamed mucosa and panel B (arrow) the opening of an anal fistula.

Mentions: We present the case of a 23-year old-Caucasian man who was initially diagnosed with CG of the lower lip in 2009. He underwent a number of first topic and then systemic treatments with antibiotics and steroids with little or no improvement in the lower lip. In the same period, he underwent psychotherapy for low self-esteem and bad school performance due to his mouth appearance. In January 2011, to confirm the diagnosis of CG, a lower lip biopsy was taken. The pathologist described normal keratinizing squamous epithelium overlying inflammatory tissue with non-caseating granulomatous inflammation in the deeper subcutaneous and parafollicular tissues, consistent with cheilitis granulomatosa. His Ziehl-Neelsen, silver, periodic acid-Schiff, and Warthin–Starry staining results were negative for acid-fast (Mycobacteria and Actinomyces, specifically), fungal, and spirochetal organisms. In February 2011, he complained of pain during defecation and underwent an evaluation. His rectal examination showed a diffuse, severe, perianal disease characterized by perianal fissures, fistulae, and abscesses. After an in-depth interview, he revealed that in 2009 he had an over-the-counter topic preparation prescribed by his general practitioner (GP) for anal fissuration and had had a moderate discomfort at evacuation since then. The severity of the anal disease and the previous diagnosis of CG alerted us to investigate the possibility of Crohn’s disease by colonoscopy. His endoscopy examination showed a diffuse aphthosis in a very limited region of the rectal ampulla and terminal ileum, and ileal and rectal biopsies were suggestive of a diffuse granulomatous inflammation, compatible with Crohn’s disease (Figure 1). Intestinal ultrasound and magnetic resonance of the intestine confirmed the diagnosis of terminal ileal and perianal Crohn’s disease. After surgical drainage of perianal disease, he was started on infliximab (given as intravenous infusions at dosage of 5mg/kg at 0, 2 and 6 weeks, and at maintenance schedule of 5mg/kg every 8 weeks). He also underwent regular follow-ups that included an endoscopy, histology, and intestinal ultrasound, as per our protocol.


Buccal localization of Crohn's disease with long-term infliximab therapy: a case report.

Ciacci C, Bucci C, Zingone F, Iovino P, Amato M - J Med Case Rep (2014)

Colonoscopy. Panel A shows the rectal inflamed mucosa and panel B (arrow) the opening of an anal fistula.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4265509&req=5

Fig1: Colonoscopy. Panel A shows the rectal inflamed mucosa and panel B (arrow) the opening of an anal fistula.
Mentions: We present the case of a 23-year old-Caucasian man who was initially diagnosed with CG of the lower lip in 2009. He underwent a number of first topic and then systemic treatments with antibiotics and steroids with little or no improvement in the lower lip. In the same period, he underwent psychotherapy for low self-esteem and bad school performance due to his mouth appearance. In January 2011, to confirm the diagnosis of CG, a lower lip biopsy was taken. The pathologist described normal keratinizing squamous epithelium overlying inflammatory tissue with non-caseating granulomatous inflammation in the deeper subcutaneous and parafollicular tissues, consistent with cheilitis granulomatosa. His Ziehl-Neelsen, silver, periodic acid-Schiff, and Warthin–Starry staining results were negative for acid-fast (Mycobacteria and Actinomyces, specifically), fungal, and spirochetal organisms. In February 2011, he complained of pain during defecation and underwent an evaluation. His rectal examination showed a diffuse, severe, perianal disease characterized by perianal fissures, fistulae, and abscesses. After an in-depth interview, he revealed that in 2009 he had an over-the-counter topic preparation prescribed by his general practitioner (GP) for anal fissuration and had had a moderate discomfort at evacuation since then. The severity of the anal disease and the previous diagnosis of CG alerted us to investigate the possibility of Crohn’s disease by colonoscopy. His endoscopy examination showed a diffuse aphthosis in a very limited region of the rectal ampulla and terminal ileum, and ileal and rectal biopsies were suggestive of a diffuse granulomatous inflammation, compatible with Crohn’s disease (Figure 1). Intestinal ultrasound and magnetic resonance of the intestine confirmed the diagnosis of terminal ileal and perianal Crohn’s disease. After surgical drainage of perianal disease, he was started on infliximab (given as intravenous infusions at dosage of 5mg/kg at 0, 2 and 6 weeks, and at maintenance schedule of 5mg/kg every 8 weeks). He also underwent regular follow-ups that included an endoscopy, histology, and intestinal ultrasound, as per our protocol.

Bottom Line: Treatment of Crohn's disease with an anti-tumor necrosis factor alpha agent (infliximab) successfully induced remission of both the gastrointestinal disease and the oral lesion.Our recommendation is that physicians should be able to recognize cheilitis granulomatosa as a possible marker of a more complex systemic disease and proceed first with an accurate physical examination, and further suggest investigations of the bowel.In cases of Crohn's disease, a therapy with biological agents can be successful.

View Article: PubMed Central - PubMed

Affiliation: Gastroenterology Unit, Department of Medicine and Surgery, University of Salerno, Baronissi Campus, via S, Allende, 84081 Baronissi, Salerno, Italy. cciacci@unisa.it.

ABSTRACT

Introduction: Cheilitis granulomatosa causes persistent idiopathic lip swelling and ulceration and it can sometimes be recognized as a unique or early manifestation of Crohn's disease. Spontaneous remission is rare and with the lack of controlled trials, different therapeutic approaches have been used. Some cases have been treated with an exclusion diet in the attempt to rule out diet allergens, while the most popular treatments include antibiotics such as tetracycline and clofazimine tranilast, benzocaine topical or intralesional steroids, and cheiloplasty, with different outcomes.

Case presentation: We describe the case of a 23-year-old Caucasian man, primarily diagnosed with cheilitis granulomatosa for a severe lower lip swelling, and then with Crohn's disease of the terminal ileum and anus. Treatment of Crohn's disease with an anti-tumor necrosis factor alpha agent (infliximab) successfully induced remission of both the gastrointestinal disease and the oral lesion.

Conclusions: Our recommendation is that physicians should be able to recognize cheilitis granulomatosa as a possible marker of a more complex systemic disease and proceed first with an accurate physical examination, and further suggest investigations of the bowel. In cases of Crohn's disease, a therapy with biological agents can be successful.

Show MeSH
Related in: MedlinePlus