Unique Dental and Craniofacial Manifestations of Hypoplastic Amelogenesis Imperfecta in a Patient With Prune Belly Syndrome: A Rare Case Report

<b>Introduction:</b> Prune belly syndrome (PBS) is a rare congenital disease characterized by hypoplastic abdominal wall muscles, urological abnormalities, and bilateral cryptorchidism. This report describes a rare case of aberrant hypoplastic amelogenesis imperfecta in a patient with PBS. <b>Case Presentation:</b> A 10-year-old boy with PBS presented with difficulties in speech and mastication. Oral and radiological examinations revealed aberrant hypoplastic enamel defects, mandibular hypoplasia and retrusion, maxillary constriction, and anterior open bite. <b>Conclusion:</b> Although dental manifestations are not frequent in PBS, rare cases may present with aberrant hypoplastic amelogenesis imperfecta.

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Publication Details

Journal
Case Reports in Dentistry
Published
2025-01-01
DOI
https://doi.org/10.1155/crid/1724807
Primary Topic
Urological Disorders and Treatments
Type
article
Field-Weighted Citation Impact
0.00

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article

Unique Dental and Craniofacial Manifestations of Hypoplastic Amelogenesis Imperfecta in a Patient With Prune Belly Syndrome: A Rare Case Report

Jan C.‐C. Hu, Jung‐Wook Kim
Case Reports in Dentistry
Urological Disorders and Treatments
article

Unique Dental and Craniofacial Manifestations of Hypoplastic Amelogenesis Imperfecta in a Patient With Prune Belly Syndrome: A Rare Case Report

Jan C.‐C. Hu, Jung‐Wook Kim
article en

Abstract

<b>Introduction:</b> Prune belly syndrome (PBS) is a rare congenital disease characterized by hypoplastic abdominal wall muscles, urological abnormalities, and bilateral cryptorchidism. This report describes a rare case of aberrant hypoplastic amelogenesis imperfecta in a patient with PBS. <b>Case Presentation:</b> A 10-year-old boy with PBS presented with difficulties in speech and mastication. Oral and radiological examinations revealed aberrant hypoplastic enamel defects, mandibular hypoplasia and retrusion, maxillary constriction, and anterior open bite. <b>Conclusion:</b> Although dental manifestations are not frequent in PBS, rare cases may present with aberrant hypoplastic amelogenesis imperfecta.

Case Reports in DentistryVol. 2025(1)
Seoul National University (KR), University of Michigan (US)
National Research Foundation, National Research Foundation of Korea
Openalex Percentile: Top 63%
Urological Disorders and Treatments
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Unique Dental and Craniofacial Manifestations of Hypoplastic Amelogenesis Imperfecta in a Patient With Prune Belly Syndrome: A Rare Case Report — Jan C.‐C. Hu, Jung‐Wook Kim · Case Reports in Dentistry (2025) | TGRS Research Map | TGRS