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http://dx.doi.org/10.5933/JKAPD.2014.41.1.80

Oral Symptoms of Intractable Ulcerating Enterocolitis of Infancy and Differential Diagnosis: A Case Report  

Min, Hyoseon (Department of Pediatric Dentistry, College of Dentistry, Yonsei University)
Choi, Hyungjun (Department of Pediatric Dentistry, College of Dentistry, Yonsei University)
Lee, Jaeho (Department of Pediatric Dentistry, College of Dentistry, Yonsei University)
Choi, Byungjai (Department of Pediatric Dentistry, College of Dentistry, Yonsei University)
Lee, Hyoseol (Department of Pediatric Dentistry, College of Dentistry, Yonsei University)
Publication Information
Journal of the korean academy of Pediatric Dentistry / v.41, no.1, 2014 , pp. 80-84 More about this Journal
Abstract
Intractable ulcerating enterocolitis is an uncommon inflammatory bowel disease syndrome of neonatal onset first described in 1991. Intractable ulcerating enterocolitis usually presents in the neonate with a mouth ulceration and the subsequent development of perianal disease and colitis. In this case report, an infant, 18 days from birth, with ulcerative lesion on hard palate for systemic differential diagnosis about oral lesion is referred from the department depiatrics. At that time, there is no abnormality, except oral lesion-like Aphthous ulcer. The patient was discharged from pediatrics, but returned to the hospital 3weeks later with blood diarrhea. As a result of endoscopy, there were large ulcerating lesions and the patient was diagnosed intractable ulcerating enterocolitis. Early recognition of Intractable ulcerating enterocolitis appears to be beneficial because colectomy, as opposed to immunosuppression, appears to be effective in controlling disease symptoms and progression. Most of the infants who were affected intractable ulcerating enterocolitis were normal at birth and oral manifestation appeared earlier than others. So, it is very meaningful for dentists to know about Intractable ulcerating enterocolitis.
Keywords
Intractable ulcerating enterocolitis; Aphthous ulcer; Behcet's disease; Crohn's disease;
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1 Chong S, Wright V, Nishigame T, et al.: Infantile colitis: a manifestation of intestinal Behcet's syndrome. Journal of pediatric gastroenterology and nutrition, 7:622-627, 1988.   DOI
2 Sanderson I, Risdon R, Walker-Smith J: Intractable ulcerating enterocolitis of infancy. Archives of disease in childhood, 66:295-299, 1991.   DOI
3 Jeong JY, Seo JK, KW P, Chi JG: A Case of "Intractable Ulcerating Enterocolitis"of Infants. Korean journal of Pediatrics, 38:264-270, 1995.
4 Avery GB, Villavicencio O, Lilly JR, Randolph JG: Intractable diarrhea in early infancy. Pediatrics, 41:712-722, 1968.
5 Davidson G, Cutz E, Hamilton J, Gall D: Familial enteropathy: a syndrome of protracted diarrhea from birth, failure to thrive, and hypoplastic villus atrophy. Gastroenterology, 75:783-790, 1978.
6 Pittock S, Drumm B, Fleming P, et al.: The oral cavity in Crohn's disease. J Pediatr, 138:767-771,2001.   DOI
7 Kasahara Y, Tanaka S, Nishino M, et al.: Intestinal involvement in Behcet's disease: review of 136 surgical cases in the Japanese literature. Diseases of the Colon & Rectum, 24:103-106, 1981.   DOI   ScienceOn
8 Smith GE, MAJ LRK: The colitis of Behcet's disease: A separate entity? The American journal of digestive diseases, 18:987-1000, 1973.   DOI   ScienceOn
9 Engelhardt KR, Shah N, Faizura-Yeop I, et al.: Clinical outcome in IL-10- and IL-10 receptor-deficient patients with or without hematopoietic stem cell transplantation. J Allergy Clin Immunol, 131:825-830, 2013.   DOI   ScienceOn
10 Thapar N, Lindley KJ, Kiparissi F, et al.:Treatment of intractable ulcerating enterocolitis of infancy by allogeneic bone marrow transplantation. Clinical Gastroenterology and Hepatology, 6:248-250, 2008.   DOI
11 Shim JO, Hwang S, Yang HR, et al.: Interleukin-10 receptor mutations in children with neonatal-onset Crohn's disease and intractable ulcerating enterocolitis. Eur J Gastroenterol Hepatol, 25:1235-1240,2013.
12 Thapar N, Shah N, Ramsay AD, et al.: Long-term outcome of intractable ulcerating enterocolitis of infancy. Journal of pediatric gastroenterology and nutrition, 40:582-588, 2005.   DOI
13 Loftus Jr EV, Tremaine WJ, Habermann TM, et al.: Risk of lymphoma in inflammatory bowel disease. The American journal of gastroenterology, 95:2308-2312, 2000.   DOI   ScienceOn
14 Lewis JD, Bilker WB, Brensinger C, et al.: Inflammatory bowel disease is not associated with an increased risk of lymphoma. Gastroenterology,121:1080-1087, 2001.   DOI   ScienceOn
15 Kim SH, Song JS, Choi HJ, et al.: Early detection of behcet's disease. J Korean Acad Pediatr Dent, 36:575-579, 2009.
16 Rogers RS: Recurrent aphthous stomatitis: clinical characteristics and associated systemic disorders. Seminars in cutaneous medicine and surgery. Elsevier, 278-283, 1997.