Unusual Radicular Cyst Formation Derived From Primary Teeth in Hyper Immunoglobulin E Syndrome

Unusual Radicular Cyst Formation Derived From Primary Teeth in Hyper Immunoglobulin E Syndrome

Abstract Introduction Hyperimmunoglobulin E syndrome (HIES) is a rare genetic based multi-system disorder. It is characterized by high serum levels of IgE, pulmonary and recurrent skin infection except for these conditions abnormalities of the dentition, bones and connective tissue could be seen. The etiology of HIES is considered to be dominant-negative alterations in signal transducer and activator of transcription 3 (STAT3). STAT3 is integral to signal transduction for multiple cytokines. STAT3 is well expressed across tissue types. Case Report In this paper, we report a 7-year-old boy with HIES and a rare clinical manifestation regarding radicular cyst which is related to the deciduous tooth. Conclusion Chronic usage of antibiotics in the HIES patients can cause that sup-pressed serious lesion so that radicular cysts may be missed. Therefore, dentists should be alert on clinical and radiologic examination.

___

  • 1. Sowerwine KJ, Holland SM, Freeman AF. Hyper-IgE syndrome update. Ann N Y Acad Sci. 2012;1250:25.
  • 2. Davis SD, Schaller J, Wedgwood RJ. Job’s syndrome: recurrent, “cold”, staphylococcal abscesses. Lancet 1966;1:1013- 15.
  • 3. Grimbacher B, Holland SM, Gallin JI, et al: Hyper-IgE syndrome with recurrent infections—An autosomal dominant multisystem disorder. N Engl J Med. 1999;340:692.
  • 4. DeWitt CA, Bishop AB, Buescher LS, et al: Hyperimmunoglobulin E syndrome: Two cases and a review of the literature. J Am Acad Dermatol. 2006;54:855.
  • 5. Freeman AF, Domingo DL, Holland SM. Hyper IgE (Job’s) syndrome: A primary deficiency with oral manifestations. Oral Dis. 2009;15:2.
  • 6. Donabedian H, Gallin, JI. The hyperimmunoglobulin E recurrent- infection (Job’s) syndrome: A review of the NIH experience and the literature. Medicine.1983;62:195.
  • 7. Shyur SD, Hill HR: Job’s syndrome of hyperimmunoglobulin E and recurrent infections, in Lichtenstein LM, Fauci AS, (eds). Current Therapy in Allergy, Immunology, and Rheumatology. St Louis, MO: Mosby-Year Book, 1992. p 322
  • 8. O’Connell AC, Puck JM, Grimbacher B, et al. Delayed eruption of permanent teeth in hyperimmunoglobulinemia E recurrent infection syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2000;89:177-85.
  • 9. Domingo DL, Freeman AF, Davis J, et al. Novel intraoral phenotypes in hyperimmunoglobulin E syndrome. Oral Dis. 2008;14:73-81.
  • 10. Mass E, Kaplan I, Hirshberg A. A clinical and histopathological study of radicular cysts associated with primary molars. J Oral Pathol Med. 1995; 24(10):458–61 11. Shetty S, Angadi PV, Rekha K. Radicular cyst in deciduous maxillary molars: a rarity. Head Neck Pathol. 2010;4(1):27– 30.
  • 12. Nair PN. New perspectives on radicular cysts: do they heal?. Int Endod J. 1998;31(3):155–60.
  • 13. Nagata T, Nomura J, Matsumura Y, Yanase S, Fujii T, Oka T, et al. Radicular cyst in a deciduous tooth: a case report and review of literature. J Dent Child. 2008;75:80–4.
  • 14. Behrman RE, Kliegman RM, Jenson HB: Nelson Textbook of Pediatrics. Philadelphia, PA, Saunders, 2000
  • 15. Burg FD, Ingelfinger JR, Wald ER, et al. Gellis and Kagan’s Current Pediatric Therapy. Philadelphia, PA, Saunders, 1999
  • 16. Vigliante CE, Costello BJ, Quinn PD. Life- Threatening Cervicofacial Infection in a Child With Hyperimmunoglobulin-E Syndrome. J Oral Maxillofac Surg. 2001;59:561-565