ArticleBone reports2016
Case series: Odontohypophosphatasia or missed diagnosis of childhood/adult-onset hypophosphatasia? - Call for a long-term follow-up of premature loss of primary teeth.
Article in Bone reports, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers, 2 of them syntheses that pooled it.
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28 citing papers in PubMed, 2 syntheses or guidelines pooled it, 58 citations in OpenAlex.
- Hypophosphatasia: Canadian update on diagnosis and management.Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA · 2019Guideline
- Frequency and age at occurrence of clinical manifestations of disease in patients with hypophosphatasia: a systematic literature review.Orphanet journal of rare diseases · 2019Pooled it
- Pharmacokinetics of Asfotase Alfa in Adult Patients With Pediatric-Onset Hypophosphatasia.Journal of clinical pharmacology · 2021Trial
- Diagnosis and Treatment of Hypophosphatasia.Calcified tissue international · 2025Review
- Japanese nationwide dental survey of hypophosphatasia reveals novel oral manifestations.Scientific reports · 2025Article
- Decade-long delayed diagnosis of hypophosphatasia until next generation tooth loss: case reports on dental rehabilitation, diagnostic challenges and clinical implications.Frontiers in oral health · 2025Article
- Adult-onset hypophosphatasia diagnosed after consecutive tooth loss during orthodontic treatment: a case report.Journal of medical case reports · 2024Article
- Role of PLP-Level as a predictive marker for oral health status in adult hypophosphatasia.Clinical oral investigations · 2024Article
- The impact of enzyme replacement therapy on the oral health manifestations of hypophosphatasia among children: a scoping review.European archives of paediatric dentistry : official journal of the European Academy of Paediatric Dentistry · 2023Article
- Childhood Hypophosphatasia Associated with a Novel BiallelicInternational journal of molecular sciences · 2022Article
- Proposing a clinical algorithm for better diagnosis of hypophosphatasia in resource-limiting situations.Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA · 2022Review
- Large-Scale Survey of Missing Deciduous Anterior Teeth on Medical Examination at the Age of 3.5 Years.Children (Basel, Switzerland) · 2022Article
- Dental manifestation and management of hypophosphatasia.The Japanese dental science review · 2022Review
- Dental manifestations in adult hypophosphatasia and their correlation with biomarkers.JIMD reports · 2022Article
- Premature Loss of Deciduous Teeth as a Symptom of Systemic Disease: A Narrative Literature Review.International journal of environmental research and public health · 2022Review
- Hypophosphatasia.Journal of clinical medicine · 2021Review
- Biochemical, clinical and genetic characteristics in adults with persistent hypophosphatasaemia; Data from an endocrinological outpatient clinic in Denmark.Bone reports · 2021Article
- Case Report: Variations in theFrontiers in genetics · 2021Article
- Multigenerational case examples of hypophosphatasia: Challenges in genetic counseling and disease management.Molecular genetics and metabolism reports · 2020Article
- Hypophosphatasia: Current Literature for Pathophysiology, Clinical Manifestations, Diagnosis, and Treatment.Cureus · 2020Review
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
introductionHypophosphatasia, a metabolic bone disease caused by a tissue-nonspecific alkaline phosphatase deficiency, leads to undermineralization of bone and/or teeth, impaired vitamin B6 metabolism, and a spectrum of disease presentation. At the mild end of the spectrum, it presents as pathologic fractures in later adulthood. Patients with isolated dental manifestations, typically presenting as premature loss of primary teeth, are classified as having odontohypophosphatasia (odontoHPP). A subset of patients diagnosed with odontoHPP in childhood can later develop extra-dental manifestations that constitute childhood- or adult-onset hypophosphatasia. CASE REPORTS METHODS/
resultsRetrospective data related to onset, detailed clinical course, and method of diagnosis were collected as part of a natural history of adult patients with hypophosphatasia. Of 9 initial patients, all had low serum alkaline phosphatase levels for their age and gender at adult presentation (Table 2). The majority (8/9) demonstrated childhood dental signs of hypophosphatasia as the initial clinical manifestation: premature loss of primary teeth (7/9), absent primary teeth (1/9), and delayed loss of primary teeth (1/9). Despite childhood dental presentation and/or other signs/symptoms, diagnosis of hypophosphatasia was delayed 20-54 years (median = 46) since the primary tooth problems and 8-45 years (median = 27) since the first fracture or onset of a major adult tooth problem.
conclusionPatients with primary tooth loss in childhood were often diagnosed with hypophosphatasia later in life. Pediatric patients classified as having odontoHPP under present practice can manifest significant disease burden later in life.
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