Cory M Resnick1, Judy A Estroff2, Tessa D Kooiman3, Carly E Calabrese4, Maarten J Koudstaal5, Bonnie L Padwa6. 1. Assistant Professor of Oral and Maxillofacial Surgery, Harvard School of Dental Medicine and Harvard Medical School, Boston; Oral and Maxillofacial Surgeon, Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, MA. Electronic address: Cory.Resnick@childrens.harvard.edu. 2. Associate Professor of Radiology, Harvard Medical School, Boston; Radiologist, Division Chief, Fetal-Neonatal Imaging, Department of Radiology, Advanced Fetal Care Center, Boston Children's Hospital, Boston, MA. 3. Medical Student, Erasmus University Medical Center, Rotterdam, The Netherlands. 4. Clinical Research Specialist, Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, MA. 5. Department of Oral and Maxillofacial Surgery, Erasmus Medical Center, Rotterdam, The Netherlands; Research Associate, Harvard School of Dental Medicine and Harvard Medical School, Boston, MA; Oral and Maxillofacial Surgeon, Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, MA. 6. Associate Professor, Harvard School of Dental Medicine and Harvard Medical School, Boston; Oral Surgeon-in-Chief, Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, MA.
Abstract
PURPOSE: The etiology of the palatal cleft in Robin sequence (RS) is unknown. The purpose of this study was to assess the position of the fetal tongue at prenatal magnetic resonance imaging (MRI) and to suggest a potential relation between tongue position and development of the cleft palate seen in most patients with RS. MATERIALS AND METHODS: This is a retrospective case-and-control study including fetuses with prenatal MRIs performed in the authors' center from 2002 to 2017. Inclusion criteria were 1) prenatal MRI of adequate quality, 2) liveborn infant, and 3) postnatal diagnosis of RS (Robin group) or cleft lip and palate (CLP group). Patients with postnatal RS without a palatal cleft were excluded. A control group with normal facial morphology was matched by gestational age. The outcome variable was tongue position at fetal MRI, described as within the cleft, along the floor of the mouth (normal), other, or indeterminate. RESULTS: One hundred twenty-two patients with mean gestational age at MRI of 25.8 ± 4.9 weeks were included (Robin, n = 21 [17%]; CLP, n = 47 [39%]; control, n = 54 [44%]). The tongue was visualized within the palatal cleft in 76.2% of the Robin group and 4.3% of the CLP group. The tongue was found along the floor of the mouth (normal) in the remainder of the Robin and CLP groups and in 100% of the control group. CONCLUSION: These findings suggest a relation between in utero tongue position and the development of cleft palate in RS.
PURPOSE: The etiology of the palatal cleft in Robin sequence (RS) is unknown. The purpose of this study was to assess the position of the fetal tongue at prenatal magnetic resonance imaging (MRI) and to suggest a potential relation between tongue position and development of the cleft palate seen in most patients with RS. MATERIALS AND METHODS: This is a retrospective case-and-control study including fetuses with prenatal MRIs performed in the authors' center from 2002 to 2017. Inclusion criteria were 1) prenatal MRI of adequate quality, 2) liveborn infant, and 3) postnatal diagnosis of RS (Robin group) or cleft lip and palate (CLP group). Patients with postnatal RS without a palatal cleft were excluded. A control group with normal facial morphology was matched by gestational age. The outcome variable was tongue position at fetal MRI, described as within the cleft, along the floor of the mouth (normal), other, or indeterminate. RESULTS: One hundred twenty-two patients with mean gestational age at MRI of 25.8 ± 4.9 weeks were included (Robin, n = 21 [17%]; CLP, n = 47 [39%]; control, n = 54 [44%]). The tongue was visualized within the palatal cleft in 76.2% of the Robin group and 4.3% of the CLP group. The tongue was found along the floor of the mouth (normal) in the remainder of the Robin and CLP groups and in 100% of the control group. CONCLUSION: These findings suggest a relation between in utero tongue position and the development of cleft palate in RS.