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Oral Manifestations As the First Presenting Sign of Crohn's Disease

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Oral Manifestations As the First Presenting Sign of Crohn's Disease J Clin Exp Dent. 2017;9(7):e934-8. Oral manifestations of pediatric Crohn’s disease Journal section: Oral Medicine and Pathology d oi:10.4317/jced. 53914 Publication Types: Case Report http://dx.doi.org/10.4317/jced.53914 Oral manifestations as the first presenting sign of Crohn’s disease in a pediatric patient Ashley Eckel 1, Dale Lee 2, Gail Deutsch 3, Anthony Maxin 4, Dolphine Oda 5 1 MD, PhD, University of Washington, Department of Laboratory Medicine, Seattle, WA 2 MD, MSCE, Seattle Children’s Hospital, Division of Gastroenterology and Hepatology, Seattle, WA 3 MD, Seattle Children’s Hospital, Department of Pathology, Seattle, WA 4 Seattle Children’s Hospital, Seattle, WA 5 BDS, MSc, University of Washington School of Dentistry, Department of Oral & Maxillofacial Surgery, Seattle, WA Correspondence: 1959 NE Pacific Street, NW120 Seattle, WA 98195-7110 [email protected] Eckel A, Lee D, Deutsch G, Maxin A, Oda D. Oral manifestations as the first presenting sign of Crohn’s disease in a pediatric patient. J Clin Exp Dent. 2017;9(7):e934-8. Received: 05/04/2017 http://www.medicinaoral.com/odo/volumenes/v9i7/jcedv9i7p934.pdf Accepted: 20/05/2017 Article Number: 53914 http://www.medicinaoral.com/odo/indice.htm © Medicina Oral S. L. C.I.F. B 96689336 - eISSN: 1989-5488 eMail: [email protected] Indexed in: Pubmed Pubmed Central® (PMC) Scopus DOI® System Abstract Crohn’s disease (CD) is a chronic inflammatory disorder affecting the gastrointestinal (GI) tract. Although the GI tract is the primary site of involvement, many patients, particularly in pediatric cases, first present with non-intesti- nal manifestations, including oral lesions. Oral manifestations of CD in children occur in around 50-80% of cases, and about 30% of CD cases in children occur first in the mouth. Recognizing such oral lesions in the pediatric po- pulation, and requesting a biopsy, may expedite the diagnosis of CD. We describe a 15 year old male who presented with oral findings of multiple aphthous ulcers and plaques of pink papules of the buccal vestibule. We highlight the initial pathology findings, including non-caseating granulomas, sialadenitis, and a notable plasmacytosis, from biopsy of the left retromolar pad area, which triggered further testing for CD. We provide discussion of how CD was eventually diagnosed and treated and highlight the significance of the pathological findings in this case as they relate to the pathogenesis of CD. Key words: Crohn’s disease, Inflammatory bowel disease, Oral manifestations, Pediatric, Granulomatous inflam- mation, Monotypic plasma cells. Introduction pediatric patients with CD have oral changes including Inflammatory Bowel Diseases (IBD) which include multiple superficial ulcers (aphthous ulcers) and cob- Crohn’s disease (CD) and ulcerative colitis (UC) are blestone appearing pink papules on the buccal mucosa chronic and disabling diseases with a high and increa- and vestibule (3). Of those, in more than 30%-60%, the sing incidence across continents (1). CD is an immune oral findings may be the first manifestation of CD (3,4). mediated chronic inflammatory disorder characterized In the pediatric population, failure to consider CD on the by granulomatous inflammation that can occur from the differential diagnosis of persistent multiple oral ulcers oral cavity to the anus (2). Approximately 50-80% of and other oral findings can delay necessary care for pa- e934 J Clin Exp Dent. 2017;9(7):e934-8. Oral manifestations of pediatric Crohn’s disease tients (3). We describe a 15 year old male who presented biopsy. Following results of the incisional biopsy of the with multiple persistent oral ulcers and papules which left retromolar pad, the patient was next referred to Gas- were found to be the first manifestation of CD. We pre- troenterology and underwent endoscopy and colonosco- sent this case with review of current literature pertaining py. Additional history obtained by the Gastroenterologist to oral CD in pediatric patients. This case also offers an prior to endoscopic evaluation was significant for a ten interesting pathology finding, highlighting the immuno- pound weight loss over approximately one year associa- logic nature of the disease. Consistent with almost all ted with intermittent right-sided abdominal discomfort cases of CD, non-caseating granulomas were found and and episodes of blood on the toilet paper. The patient’s were the trigger for further testing for CD; in this case, family history is notable for Crohn’s disease in a pater- however, there was also a collection of monotypic plas- nal grandmother, lymphocytic colitis in a paternal aunt, ma cells in the oral biopsy. and ulcerative colitis in a paternal great-grandfather. Fecal calprotectin prior to endoscopy was 477 mcg/g. Case Report Rheumatoid factor was normal. Erythrocyte sedimentation A 15-year old Caucasian male initially presented to his rate was 18 mm/hr. C-reactive protein was 13 mg/L. Serum family practice physician with palpable bilateral cer- protein electrophoresis demonstrated a broad gamma in- vical lymph nodes. The enlargement of the nodes was crease, with a non-specific pattern and no monoclonal pro- interpreted to be of viral association and was treated teins. Quantiferon gold test was negative for tuberculosis. conservatively. Approximately one month later, the pa- As requested by the oral surgeon, biopsy of the left retro- tient developed episodic oral pain while eating which molar pad area was performed. evolved into gingival bleeding, cheek swelling, and was Histologic sections demonstrated mucosal ulceration associated with development of generalized superficial with discrete non-caseating granulomas, sialadenitis, ulcers with clustering in the posterior soft palate and the and plasmacytosis with patchy infiltrates of monotypic vestibules. The patient also developed confluent flesh- kappa restricted light chain plasma cells (Fig. 1C,D). colored papules in the bilateral posterior vestibule, buc- Testing for Epstein-Barr Virus was negative as well as cal mucosa, and retromolar pad areas of his mouth (Fig. fungal, mycobacterial, and spirochete special stains. A 1 A,B). He was referred to an Otolaryngologist who diagnosis of non-caseating granulomatous inflammation started the patient on a 10 day course of antibiotics and was rendered with a comment mentioning Crohn’s di- referred the patient to an oral surgeon for an incisional sease as a possible etiology. Fig. 1: (A,B) Multiple small and superficial ulcers (yellow arrows) with white centers and red halos in the posterior soft palate of the patient’s oral cavity. Plaques of pink papules are present in the posterior buccal mucosa and bilat- eral retromolar areas. (C,D) Biopsy of the left retromolar pad area demonstrates mucosal ulceration with discrete non-caseating granulomas and plasmacytosis with patchy infiltrates of monotypic kappa light chain plasma cells. e935 J Clin Exp Dent. 2017;9(7):e934-8. Oral manifestations of pediatric Crohn’s disease On subsequent examination by endoscopy, the upper gas- CD, although those etiologies often present more acu- trointestinal tract was notable for mild patchy erythema tely. Granulomas can occur in infectious diseases inclu- in the duodenum. The entire colon demonstrated scatte- ding tuberculosis and Yersinia pseudotuberculosis. red areas of mildly erythematous mucosa and multiple Based on results of the oral biopsy, endoscopic evalua- aphthous ulcers similar to those noted in the oral cavity. tion and associated biopsies, and laboratory values, a Multiple biopsies were taken. The terminal ileum contai- diagnosis of Crohn’s disease was rendered. After discus- ned a patchy area of mucosa that was mildly erythema- sion with the patient and family, the decision was made tous. Scattered white exudates and mucosal hemorrha- to start both corticosteroids and enteral nutritional the- ges were appreciated and, again, multiple biopsies were rapy, with 80% of daily calories from an intact protein taken. The perianal and digital rectal examinations were formula (5). The patient and family were interested in normal (Fig. 2 A,B). MRI enterography demonstrated no considering nutritional therapy long-term. The patient radiographic evidence of bowel inflammation. began a steroid wean and has not had recurrence of Biopsies taken at the time of endoscopy were remarka- oral lesions or gastrointestinal symptoms. The decision ble for mild to moderate chronic active inflammation to start maintenance immunosuppressive therapy was with multifocal discrete non-caseating granulomas in made six weeks after diagnosis as the family decided both the upper and lower gastrointestinal tract similar that primary nutritional therapy would be difficult to to those of the oral cavity lesions. The stomach and co- maintain long-term. lon had multifocal discrete granulomas (Fig. 2C,D). The esophagus and duodenum were more mildly affected. Discussion The terminal ileum had focal prominent surface infiltra- Crohn’s disease is a chronic inflammatory bowel disease tion by neutrophils with underlying granulation changes believed to involve interplay of genetic, immunologic, and subtotal villous architecture atrophy. and environmental factors. It commonly begins gradua- The differential diagnosis for this patient’s clinical lly, but may also start abruptly. Two age peaks in occu- presentation included CD, infectious diseases (fungal, rrence are described: one among early adulthood and the mycobacterial, spirochetes), drug-induced reactions, other in patients over 60 years of age (6,7).
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