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Oral Ulcerations As a Sign of Crohn's Disease in a Pediatric Patient

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Oral Ulcerations As a Sign of Crohn's Disease in a Pediatric Patient Case Report Oral Ulcerations as a Sign of Crohn’s Disease in a Pediatric Patient: A Case Report Franci Stavropoulos, DDS Joseph Katz, DMD Marcio Guelmann, DDS Enrique Bimstein, DDS Dr. Stavropoulos is assistant professor, Dr. Katz is professor, Department of Oral and Maxillofacial Surgery and Diagnostic Sciences, Dr. Guelmann is assistant professor and Dr. Bimstein is visiting professor, Department of Pediatric Dentistry, College of Dentistry, University of Florida, Gainesville, Fla. Correspond with Dr. Stavropoulos at [email protected] Abstract Crohn’s disease is an inflammatory intestinal disease of unknown etiology. The disease primarily affects whites, with both sexes being affected equally. A genetic predisposition exists. Symptoms frequently present in the second to third decades of life, although they may present in the pediatric and/or geriatric populations. Oral lesions are significant as they are frequently reported to precede intestinal symptoms. Treatment of Crohn’s dis- ease is palliative, with a focus on remission. Pediatric and general dentists play a critical role in the early diagnosis of Crohn’s disease. Evaluation of a pediatric patient with com- plaints of oral ulcerations, as well as gastrointestinal symptoms, fatigue, and/or weight loss requires prompt referral to a gastroenterologist for further evaluation for Crohn’s disease. (Pediatr Dent. 2004;26:355-358) KEYWORDS: CROHN’S DISEASE, ORAL ULCERATIONS Received December 12, 2002 Revision Accepted May 8, 2003 rohn’s disease, an inflammatory and likely immu- approximately 20% of patients with Crohn’s disease, al- nologically mediated disease of unknown etiology, though this prevalence is not significantly higher than in falls under the rubric of the diagnosis of inflam- the unaffected population.10 Polystomatitis vegetans or C 1,2 matory bowel disease (IBD). “snail track ulcerations” may also be present, which is a rare IBD no. 2666600 in the On Line Mendelian Inheritance presentation of IBD.1 Secondary changes and symptoms in Man (OMIM) occurs more commonly in whites and include stomatitis, which is present in less than 1% of pa- Ashkenazi Jews.2 It is considered a complex genetic trait, as tients with Crohn’s disease, and may be caused by inheritance may not follow any simple Mendelian model, but Staphyloccus aureus.1 is linked to chromosomes 16p12 to q13.3 Crohn’s disease is a Histological features of oral lesions of Crohn’s disease chronic, inflammatory, intestinal process affecting any site include nonnecrotizing granulomatous inflammation within along the gastrointestinal tract, from the mouth to the anus. the submucosal connective tissue. The oral ulcerations do Symptoms frequently begin in early adult life, and approxi- not differ from the common apthous lesions, clinically or mately 25% of the cases begin before the age of 25.4 Oral histopathologically.1 Therefore, early recognition of the oral lesions are of significance, as they are reported to precede the lesions and their association with Crohn’s disease is intestinal symptoms in 30% to 60% of the cases.1,5-8 Oral essential in obtaining a timely diagnosis, preventing com- manifestations of IBD may precede the onset of intestinal le- plications, and improving the prognosis. sions by as much as a year or more.9 This case report discusses an adolescent male who pre- Oral manifestations of Crohn’s disease include hyper- sented with oral ulcerations, that led to the diagnosis of trophy of the lips and gingival soft tissue swelling Crohn’s disease. resembling fibrous hyperplasia from an ill-fitting denture, a cobblestone appearance of the buccal mucosa and pal- Case report ate, and deep, granulomatous-appearing ulcers. The ulcers A 15-year-old male presented to the Oral Medicine Clinic may appear linear in nature within the vestibule. Polypoid at the University of Florida College of Dentistry with a “tag” lesions may be found on vestibular or retromolar chief complaint of oral ulcerations and consequential dys- mucosa. Apthous ulcerations are also present clinically in phagia. The symptoms began approximately 4 months Pediatric Dentistry – 26:4, 2004 Oral ulcerations in Crohn’s disease Stavropoulos et al. 355 Figure 1. Photomicrograph of oral mucosal biopsy. Arrow points to Figure 2. Photomicrograph of oral mucosal biopsy demonstrating epithiloid granuloma with giant cells. (Hematoxylin and eosin stain, eosinophilia. (Hematoxylin and eosin stain, original magnification original magnification ×10). ×40). earlier with continued ulcerations of about 2-week’s du- commencing drug therapy, the patient’s oral and systemic ration each and associated soreness of his mouth. By symptoms improved. He reported decreasing episodes of report, the ulcerations were numerous, small, and healed loose stools and improvement in his symptom of dysphagia. over a 2-week period without scarring. In addition, he reported loose stools on a daily basis, a slightly elevated Discussion temperature, and weight loss of approximately 10 pounds. The oral manifestations included recurrent oral ulcerations, The patient had been seen by an otolaryngologist who had tissue tags, and diffuse erythema over a 4- to 5-month pe- initially diagnosed the patient’s signs and symptoms as a riod of time. Clinically, no mucosal scarring was evident. viral infection. His medical history was noncontributory, Unlike other case reports, periodontal involvement and and he denied the use of medication. No allergies were geographic tongue did not exist in this patient nor did he reported. The clinical examination was significant for a complain of halitosis.6,8 pale, thin male of normal stature. Vital signs were nor- The incidence of Crohn’s disease in the United States mal. The head and neck examination was negative for has increased over the past 30 years from 1 per 100,000 to masses, swelling, or lymphadenopathy. Intraorally, mul- as high as 10 per 100,000 individuals. Crohn’s disease typi- tiple minor apthous-like lesions were present in the left cally affects whites and individuals of Northern European mandibular buccal vestibule. Tissue tags and diffuse descent. In the United States, whites are affected more fre- erythema were present on the frenum. The remaining in- quently than blacks, Hispanics, and Asians, respectively. traoral examination was unremarkable without evidence The cause of Crohn’s disease is unknown. However, a of periodontal involvement. myriad of causative factors has been suggested including The patient was referred for a routine serology work- infectious, immunological, nutritional, and environmen- up, which was normal except for an elevated C-reactive tal. A genetic susceptibility exists based on elevated twin protein, an elevated erythrocyte sedimentation rate, and concordance rates and an increased frequency of Crohn’s slight microcytic anemia. A working diagnosis of Crohn’s disease among first-degree relatives of affected patients.6 disease was established. The patient was then referred to Both sexes are affected equally, and the disease may mani- an oral and maxillofacial surgeon for a soft tissue biopsy fest itself at any age, although it is usually diagnosed by age and a pediatric gastroenterologist for an evaluation. The 30. The elderly may also develop Crohn’s disease.2 oral biopsy was remarkable for chronic granulomatous in- Signs and symptoms of Crohn’s disease are variable and flammation, acanthosis, eosinophilia, and intraepithelial may exist in a subclinical state, making the diagnosis more abscesses (Figures 1 and 2). A chronic inflammatory infil- difficult. Intermittent diarrhea, abdominal pain, cramping, trate was present in the area surrounding the noncaseating nausea, fever, weight loss, and fatigue are common clinical granuloma, probably representing a host response. A findings. Abdominal pain associated with cramps is the most colonoscopy was performed; endoscopic biopsy specimens common initial complaint. The abdominal pain is described were taken from the terminal ileum, cecum, and colon, as periumbilical as opposed to lower abdominal pain. The which showed severe chronic active colitis. latter is the location for the pain of ulcerative colitis, an in- A definitive diagnosis of Crohn’s disease was obtained. testinal disorder also described under the rubric of IBD.3 The patient was started on oral mesalamine 800mg tid daily, Malnutrition may occur, a consequence of similar intesti- an anti-inflammatory drug acting directly on the colon. He nal malabsorption processes, leading to anemia, decreased was also started on predinsone 50 mg daily for 4 weeks fol- growth, and a short stature.1 lowed by a tapering dose to 20 mg daily. Within a week of 356 Stavropoulos et al. Oral ulcerations in Crohn’s disease Pediatric Dentistry – 26:4, 2004 The inflammatory process consists of noncaseating and colonic and perianal disease are reported to be the most granulomas within the intestines, and is transmural in na- common presenting clinical findings in young children di- ture. It is more frequently located in the terminal ileum. agnosed with Crohn’s disease. In addition, common The lesions are distinct from those seen in ulcerative coli- presenting symptoms include diarrhea, abdominal pain, tis in that they are discontinuous, with areas of uninvolved vomiting, and chronic fever.17,18 Therefore, any adolescent normal bowel between diseased bowel segments.2 or teenager presenting with complaints of chronic fatigue, Extra-intestinal manifestations are reported to include persisting growth problems, fever, and oral
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