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Oral Manifestations of Autoimmune Neutropenia: A Case Report
1University Dental Hospital Manchester, University of Manchester, England
*Corresponding Author(s): Carly C Dixon E-mail: Carly.Dixon@manchester.ac.uk
Autoimmune neutropenia is a self-limiting condition characterized by the presence of granulocyte-specific autoantibodies. This case report describes a 2-year-old female who presented to the Department of Pediatric Dentistry with a history of oral ulceration, complicated by high temperature, gingival hypertrophy and gingival hemorrhage. These symptoms had precipitated an admission to a district general hospital and diagnosis of viral tonsillitis. The severity of the presentation to the Pediatric Dental Department prompted a review of recent discharge blood investigations, which revealed a neutrophil count of 0.07×109/L(1.0–8.5 ×109/L) indicating severe neutropenia. Urgent referral to Pediatric Hematology resulted in same-day admission and a diagnosis of autoimmune neutropenia was established. The patient was managed with chlorhexidine mouthwash, prophylactic antimicrobial and antifungal medication and granulocyte colony stimulating factor (G-CSF). Neutropenia may present with features exclusive to the oral cavity. Recognition of atypical presentations such as oral ulceration with associated systemic illness may assist in early diagnosis.
Autoimmune neutropenia; Oral manifestations; Children
Carly C Dixon,Francesca J Thomson,Siobhan M Barry. Oral Manifestations of Autoimmune Neutropenia: A Case Report. Journal of Clinical Pediatric Dentistry. 2019. 43(1);46-50.
1 Wang L, Wang C, Chu C, et al. Primary autoimmune neutropenia in children in Taiwan. Transfusion 49(5):1003-1006, 2009.
2 Bux J, Kissel K, Nowak K, Spengel U, Mueller-Eckhardt C. Autoimmune neutropenia: clinical and laboratory studies in 143 patients. Ann Hematol Oncol 63(5):249-252, 1991.
3. Wintrobe MGreer J. Wintrobe’s Clinical Hematology. 1st ed. Philadelphia: Wolters Kluwer/Lippincott Williams & Wilkins Health 2009.
4. Rindom Schiott C, Loe H. The origin and variation in number of leukocytes in the human saliva. J Periodont Res 5(1):36-41, 1970.
5. Porter S, Scully C, Standen G. Autoimmune neutropenia manifesting as recurrent oral ulceration. Oral Surg, Oral Med, Oral Pathol Oral Radiol Endod 78(2):178-180, 1994.
6. Andrews R, Benjamin S, Shore N, Canter S. Chronic benign neutropenia of childhood with associated oral manifestations. Oral Surg, Oral Med, Oral Pathol Oral Radiol Endod 20(6):719-725, 1965.
7. Deasy M, Vogel R, Macedo-Sobrinho B, Gertzman G, Simon B. Familial benign chronic neutropenia associated with periodontal disease: A Case Report. J Periodont 51(4):206-210, 1980.
8. Farruggia P, Dufour C. Diagnosis and management of primary autoimmune neutropenia in children: insights for clinicians. Ther Adv Hematol 6(1):15-24, 2014.
9. Deas D, Mackey S, McDonnell H. Systemic disease and periodontitis: manifestations of neutrophil dysfunction. Period 32(1):82-104, 2003.
10. Fioredda F, Calvillo M, Burlando O, et al. Infectious complications in children with severe congenital, autoimmune or idiopathic neutropenia. Pediatr Infect Dis J 32(4):410-412, 2013.
11. Otsuka Y, Matsuyama J, Tomizawa M, Noda T, Iwano F. Chronic benign neutropenia diagnosed due to severe stomatitis: a case report. Pediatr Dent J 15(1):134-138, 2005.
12. Hajishengallis G, Moutsopoulos N, Hajishengallis E, Chavakis T. Immune and regulatory functions of neutrophils in inflammatory bone loss. Semin Immunol 28(2):146-158, 2016.
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