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Case report of cheilitis granulomatosa and joint complaints as presentation of Crohn's disease.

Hoekman DR, Roelofs JJ, van Schuppen J, Schonenberg-Meinema D, D'Haens GR, Benninga MA - Clin J Gastroenterol (2016)

Bottom Line: Cheilitis granulomatosa is characterized by granulomatous lip swelling.We report a case of a 13-year-old girl who presented with orofacial swelling and arthralgia, who eventually was diagnosed with Crohn's disease, which was successfully treated with infliximab and azathioprine combination therapy.Recurrent or persistent orofacial swelling should prompt consideration of cheilitis granulomatosa, and further diagnostic evaluation to exclude the presence of Crohn's disease seems warranted.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatric Gastroenterology and Nutrition, Academic Medical Center, Meibergdreef 9, 1105 AZ, Amsterdam, The Netherlands. d.r.hoekman@amc.uva.nl.

ABSTRACT
Cheilitis granulomatosa is characterized by granulomatous lip swelling. We report a case of a 13-year-old girl who presented with orofacial swelling and arthralgia, who eventually was diagnosed with Crohn's disease, which was successfully treated with infliximab and azathioprine combination therapy. Recurrent or persistent orofacial swelling should prompt consideration of cheilitis granulomatosa, and further diagnostic evaluation to exclude the presence of Crohn's disease seems warranted.

No MeSH data available.


Related in: MedlinePlus

Low-power view microphotograph of labial biopsy showing non-characteristic signs of chronic inflammation. No granulomas were found. Original magnification 100×
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Fig2: Low-power view microphotograph of labial biopsy showing non-characteristic signs of chronic inflammation. No granulomas were found. Original magnification 100×

Mentions: Because of persistent swelling of the lip (Fig. 1b—February 2013), the patient was evaluated by an expert in oral pathology, who made a clinical diagnosis of orofacial granulomatosis (cheilitis granulomatosa). Histopathological evaluation of a biopsy of a swollen part of the lip showed a mononuclear inflammatory infiltrate and no granulomas (Fig. 2—June 2013). Topical clobetasol was initiated, with little effect. A few months later, the patient developed abdominal pain, accompanied by altered bowel habits and weight loss. Physical examination showed abdominal tenderness, but no other abnormalities; specifically, no perianal abnormalities. Laboratory evaluation revealed elevated levels of CRP (31.0 mg/L) and fecal calprotectin (>1800 µg/g). Subsequent gastroscopy and ileocolonoscopy performed in August 2013 revealed multiple large, longitudinal ulcers in the terminal ileum and multiple aphthous lesions throughout the colon and in the duodenum and stomach (Fig. 3). Biopsies showed inflammatory changes compatible with CD (Fig. 4). Magnetic resonance imaging showed several sites of wall thickening, with pathologic contrast enhancement in the terminal ileum and more proximal ileum, and prestenotic dilatation in one segment (Fig. 5). The Mantoux-test and a chest X-ray showed no signs of tuberculosis. Thus, a diagnosis of CD could be confirmed according to the Porto criteria [1]. A Pediatric Crohn’s Disease Activity Index (PCDAI) score of 32.5 at the time of diagnosis indicated moderate to severe CD [2].Fig. 2


Case report of cheilitis granulomatosa and joint complaints as presentation of Crohn's disease.

Hoekman DR, Roelofs JJ, van Schuppen J, Schonenberg-Meinema D, D'Haens GR, Benninga MA - Clin J Gastroenterol (2016)

Low-power view microphotograph of labial biopsy showing non-characteristic signs of chronic inflammation. No granulomas were found. Original magnification 100×
© Copyright Policy - OpenAccess
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4829618&req=5

Fig2: Low-power view microphotograph of labial biopsy showing non-characteristic signs of chronic inflammation. No granulomas were found. Original magnification 100×
Mentions: Because of persistent swelling of the lip (Fig. 1b—February 2013), the patient was evaluated by an expert in oral pathology, who made a clinical diagnosis of orofacial granulomatosis (cheilitis granulomatosa). Histopathological evaluation of a biopsy of a swollen part of the lip showed a mononuclear inflammatory infiltrate and no granulomas (Fig. 2—June 2013). Topical clobetasol was initiated, with little effect. A few months later, the patient developed abdominal pain, accompanied by altered bowel habits and weight loss. Physical examination showed abdominal tenderness, but no other abnormalities; specifically, no perianal abnormalities. Laboratory evaluation revealed elevated levels of CRP (31.0 mg/L) and fecal calprotectin (>1800 µg/g). Subsequent gastroscopy and ileocolonoscopy performed in August 2013 revealed multiple large, longitudinal ulcers in the terminal ileum and multiple aphthous lesions throughout the colon and in the duodenum and stomach (Fig. 3). Biopsies showed inflammatory changes compatible with CD (Fig. 4). Magnetic resonance imaging showed several sites of wall thickening, with pathologic contrast enhancement in the terminal ileum and more proximal ileum, and prestenotic dilatation in one segment (Fig. 5). The Mantoux-test and a chest X-ray showed no signs of tuberculosis. Thus, a diagnosis of CD could be confirmed according to the Porto criteria [1]. A Pediatric Crohn’s Disease Activity Index (PCDAI) score of 32.5 at the time of diagnosis indicated moderate to severe CD [2].Fig. 2

Bottom Line: Cheilitis granulomatosa is characterized by granulomatous lip swelling.We report a case of a 13-year-old girl who presented with orofacial swelling and arthralgia, who eventually was diagnosed with Crohn's disease, which was successfully treated with infliximab and azathioprine combination therapy.Recurrent or persistent orofacial swelling should prompt consideration of cheilitis granulomatosa, and further diagnostic evaluation to exclude the presence of Crohn's disease seems warranted.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatric Gastroenterology and Nutrition, Academic Medical Center, Meibergdreef 9, 1105 AZ, Amsterdam, The Netherlands. d.r.hoekman@amc.uva.nl.

ABSTRACT
Cheilitis granulomatosa is characterized by granulomatous lip swelling. We report a case of a 13-year-old girl who presented with orofacial swelling and arthralgia, who eventually was diagnosed with Crohn's disease, which was successfully treated with infliximab and azathioprine combination therapy. Recurrent or persistent orofacial swelling should prompt consideration of cheilitis granulomatosa, and further diagnostic evaluation to exclude the presence of Crohn's disease seems warranted.

No MeSH data available.


Related in: MedlinePlus