Dentoalveolar Abscesses Not Associated with Caries or Trauma: A Diagnostic Hallmark of Hypophosphatemic Rickets Initiall
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CASE REPORT
Dentoalveolar Abscesses Not Associated with Caries or Trauma: A Diagnostic Hallmark of Hypophosphatemic Rickets Initially Misdiagnosed as Hypochondroplasia Silvia Elena Yacarini Paredes1 · Raquel Assed Bezerra Segato1 · Leila Daher Moreira2 · Alcides Moreira2 · Kranya Victoria Díaz Serrano1 · Clarissa Teles Rodrigues3 · Luciana Yamamoto Almeida4 · Jorge Esquiche León5 Received: 19 September 2017 / Accepted: 27 November 2017 © Springer Science+Business Media, LLC, part of Springer Nature 2017
Abstract Hypophosphatemic rickets is a rare genetic disorder involving the regulation of fibroblast growth factor 23 (FGF23), a phosphaturic agent, clinically showing bowing of the legs, short stature and dentoalveolar abscesses. A 7-year-old boy, with previous hypochondroplasia diagnosis, was referred to our pediatric dentistry clinic presenting short stature, bone deformities and sinus tracts at deciduous teeth apex levels not related with trauma, restorations or dental caries. After deciduous teeth extraction, due to root resorption and mobility, light microscopy exhibited typical hypophosphatemic dentin, and micro-computed tomography revealed tubular clefts and porosities throughout the teeth. Laboratory tests confirmed the HR diagnosis, after which the treatment was initiated. Keywords Dentoalveolar abscesses · Oral manifestations · Hypophosphatemic rickets · Pediatric patient · Oral histopathology · µ-CT
Introduction Rickets is a metabolic disorder frequently affecting children, which presents alteration of calcium and phosphate blood levels, disturbance of bone ossification, defective bone growth and neurological alterations such as tetany. * Jorge Esquiche León [email protected] 1
Department of Pediatric Dentistry, School of Dentistry of Ribeirão Preto, University of Sao Paulo, São Paulo, SP, Brazil
2
Diagnóstico Bucal do Centro Universitário da Grande Dourados (UNIGRAN-MS), Dourados, Brazil
3
Department of Dentistry, Endodontics and Dental Materials, School of Dentistry of Bauru, University of São Paulo, São Paulo, SP, Brazil
4
Hematology Division, Department of Clinical Medicine, Ribeirão Preto Medical School (FMRP/USP), University of São Paulo, São Paulo, SP, Brazil
5
Oral Pathology, Department of Stomatology, Public Oral Health, and Forensic Dentistry, School of Dentistry of Ribeirão Preto, University of São Paulo, São Paulo, SP, Brazil
The treatment includes administration of vitamin D and/ or sufficient sunlight. Besides the classic rickets, there are also hereditary hypophosphatemic rickets (HR) which can be divided into autosomal dominant (ADHR), autosomal recessive (ARHR) and X-linked dominant (XLHR). Among them, the XLHR is the most common form of inherited metabolic rickets. The therapy of rickets-like disorders, different from classic rickets, is more difficult due to their genetic origins [1–4]. Thus, HR is a hereditary disease presenting hypophosphatemia due to renal tubular loss of phosphate into urine, which causes a decrease in the calcium and potassium ion produ
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