Rutger S. Gunther MD , Thornton S. Mu MD , Paul Clark DO
{"title":"胎儿MRI和产后表现严重的婴儿皮质肥大症:1例产前卡菲病报告并文献复习","authors":"Rutger S. Gunther MD , Thornton S. Mu MD , Paul Clark DO","doi":"10.1016/j.radcr.2025.05.070","DOIUrl":null,"url":null,"abstract":"<div><div>Caffey disease, also known as infantile cortical hyperostosis, is a rare skeletal disorder characterized by self-limited cortical bone hyperostosis and soft tissue swelling, typically presenting within the first 6 months of life. We report a rare case of prenatal Caffey disease, with fetal MRI imaging and postnatal radiographs demonstrating the hallmark findings of severe cortical hyperostosis. A current review of literature details the varied sporadic and familial origins of infantile cortical hyperostosis revealing a genetic link with a mutation of the COL1A1 gene coding for type 1 collagen. Differential diagnostic considerations are also reviewed for potentially similar patterns of bone diseases encountered in the neonatal period. This case presentation aims to review the clinical, genetic, and imaging implications of this unique disease to aid radiologists and clinicians towards accurate diagnosis, prognosis, and management. While supportive care remains the mainstay of treatment of Caffey disease, the prognosis is generally favorable, with spontaneous resolution being the most common outcome.</div></div>","PeriodicalId":53472,"journal":{"name":"Radiology Case Reports","volume":"20 9","pages":"Pages 4530-4535"},"PeriodicalIF":0.0000,"publicationDate":"2025-06-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Fetal MRI and postnatal findings of severe infantile cortical hyperostosis: A case report of prenatal Caffey disease with literature review\",\"authors\":\"Rutger S. Gunther MD , Thornton S. Mu MD , Paul Clark DO\",\"doi\":\"10.1016/j.radcr.2025.05.070\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><div>Caffey disease, also known as infantile cortical hyperostosis, is a rare skeletal disorder characterized by self-limited cortical bone hyperostosis and soft tissue swelling, typically presenting within the first 6 months of life. We report a rare case of prenatal Caffey disease, with fetal MRI imaging and postnatal radiographs demonstrating the hallmark findings of severe cortical hyperostosis. A current review of literature details the varied sporadic and familial origins of infantile cortical hyperostosis revealing a genetic link with a mutation of the COL1A1 gene coding for type 1 collagen. Differential diagnostic considerations are also reviewed for potentially similar patterns of bone diseases encountered in the neonatal period. This case presentation aims to review the clinical, genetic, and imaging implications of this unique disease to aid radiologists and clinicians towards accurate diagnosis, prognosis, and management. While supportive care remains the mainstay of treatment of Caffey disease, the prognosis is generally favorable, with spontaneous resolution being the most common outcome.</div></div>\",\"PeriodicalId\":53472,\"journal\":{\"name\":\"Radiology Case Reports\",\"volume\":\"20 9\",\"pages\":\"Pages 4530-4535\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2025-06-24\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Radiology Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S1930043325005060\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"Medicine\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Radiology Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S1930043325005060","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"Medicine","Score":null,"Total":0}
Fetal MRI and postnatal findings of severe infantile cortical hyperostosis: A case report of prenatal Caffey disease with literature review
Caffey disease, also known as infantile cortical hyperostosis, is a rare skeletal disorder characterized by self-limited cortical bone hyperostosis and soft tissue swelling, typically presenting within the first 6 months of life. We report a rare case of prenatal Caffey disease, with fetal MRI imaging and postnatal radiographs demonstrating the hallmark findings of severe cortical hyperostosis. A current review of literature details the varied sporadic and familial origins of infantile cortical hyperostosis revealing a genetic link with a mutation of the COL1A1 gene coding for type 1 collagen. Differential diagnostic considerations are also reviewed for potentially similar patterns of bone diseases encountered in the neonatal period. This case presentation aims to review the clinical, genetic, and imaging implications of this unique disease to aid radiologists and clinicians towards accurate diagnosis, prognosis, and management. While supportive care remains the mainstay of treatment of Caffey disease, the prognosis is generally favorable, with spontaneous resolution being the most common outcome.
期刊介绍:
The content of this journal is exclusively case reports that feature diagnostic imaging. Categories in which case reports can be placed include the musculoskeletal system, spine, central nervous system, head and neck, cardiovascular, chest, gastrointestinal, genitourinary, multisystem, pediatric, emergency, women''s imaging, oncologic, normal variants, medical devices, foreign bodies, interventional radiology, nuclear medicine, molecular imaging, ultrasonography, imaging artifacts, forensic, anthropological, and medical-legal. Articles must be well-documented and include a review of the appropriate literature.