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Focal Epithelial Hyperplasia in a Child with Hereditary Sensory and Autonomic Neuropathy type IV: A Rare Co-Occurrence

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Hereditary sensory and autonomic neuropathies (HSAN) are a set of rare neurological conditions with five differing phenotypes. Type IV HSAN is notable for congenital insensitivity to pain. Focal epithelial hyperplasia (Heck's disease), a variant of human papillomavirus (HPV), presents as intraoral papules. The purpose of this report is to present the case of a five-year-old patient with HSAN type IV who was referred by her pediatric dentist to evaluate a tongue mass. Her medical history included recurrent urinary tract infections and verrucae on her hands and self-mutilation of the tongue and hands. Her dental history revealed that she had self-extracted 14 primary teeth. A soft tissue exam revealed flat surface papules on the tongue and a raised yellow, white and pink lesion on the right lateral tongue, which was diagnosed as focal epithelial hyperplasia. The co-occurrence of HSAN type IV and focal epithelial hyperplasia is a rare case that demonstrates the need for coordination among medical and dental professionals to achieve the optimal outcome. (J Dent Child 2019;86(3):154-7)
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Keywords: FOCAL EPITHELIAL HYPERPLASIA; HEREDITARY SENSORY AND AUTONOMIC NEUROPATHIES; ORAL PATHOLOGY; PEDIATRIC DENTISTRY; TONGUE

Document Type: Case Report

Affiliations: 1: Dr. Calvo is a resident, Department of Orofacial Sciences, Division of Pediatric Dentistry, School of Dentistry, University of California San Francisco, San Francisco, Calif., USA;, Email: Jean.Cal[email protected] 2: Dr. Stewart is a professor, Department of Orofacial Sciences, Division of Pediatric Dentistry, School of Dentistry, University of California San Francisco, San Francisco, Calif., USA 3: Dr. Ramos is a professor, Department of Orofacial Sciences, Division of Oral Medicine, School of Dentistry, University of California San Francisco, San Francisco, Calif., USA

Publication date: September 1, 2019

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  • Acquired after the merger between the American Society of Dentistry for Children and the American Academy of Pediatric Dentistry in 2002, the Journal of Dentistry for Children (JDC) is an internationally renowned journal whose publishing dates back to 1934. Published three times a year, JDC promotes the practice, education and research specifically related to the specialty of pediatric dentistry. It covers a wide range of topics related to the clinical care of children, from clinical techniques of daily importance to the practitioner, to studies on child behavior and growth and development. JDC also provides information on the physical, psychological and emotional conditions of children as they relate to and affect their dental health.
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