Epidemiology of Robin sequence with cleft palate in the East of Scotland between 2004 and 2013

Wright, Marie; Mehendale, Felicity V.; Urquhart, Don S.

doi:10.1002/ppul.24038

Cited by 24 publications

(35 citation statements)

References 28 publications

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“…This population-based case-malformed control study covering almost 4 million births had a novel finding of a strong association between methadone exposure and PRS. These results provide an important confirmation of previous signals 10–12. In addition, we found raised ORs for cleft palate (aOR: 5.0 (95% CI 2.7 to 9.2)) and respiratory anomalies excluding PRS (aOR: 3.4 (95% CI 1.8 to 6.4)).…”

Section: Discussionsupporting

confidence: 91%

“…These independent data allowed a study with sufficient power to examine previously reported associations10–12 between methadone exposure and PRS, considering the rarity of the exposure-outcome combination. EUROCAT registries have previously been validated by examining associations between medications with known teratogenic effects and CA 27…”

Section: Discussionmentioning

confidence: 99%

“…The aetiology is unknown, though some cases may have a genetic basis 8 9. Three reports describe a potential association between methadone and PRS 10–12. An Irish cohort study reported four cases among 618 methadone-exposed babies and eight cases among 60 412 non-exposed 10.…”

Section: Introductionmentioning

confidence: 99%

“…A series of five methadone-exposed PRS cases was previously described 11. A Scottish study noted that 8% of PRS cases were methadone exposed 12. Two further reports describe PRS in neonates with gestational methadone exposure 13 14…”

Section: Introductionmentioning

confidence: 99%

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Methadone, Pierre Robin sequence and other congenital anomalies: case–control study

Cleary

Loane

Addor

et al. 2019

Arch Dis Child Fetal Neonatal Ed

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ObjectiveMethadone is a vital treatment for women with opioid use disorder in pregnancy. Previous reports suggested an association between methadone exposure and Pierre Robin sequence (PRS), a rare craniofacial anomaly. We assessed the association between gestational methadone exposure and PRS.Design/settingThis case-malformed control study used European Surveillance of Congenital Anomalies population-based registries in Ireland, the Netherlands, Italy, Switzerland, Croatia, Malta, Portugal, Germany, Wales, Norway and Spain, 1995–2011.PatientsCases included PRS based on International Classification of Disease (ICD), Ninth Edition-British Paediatric Association (BPA) code 75 603 or ICD, Tenth Edition-BPA code Q8708. Malformed controls were all non-PRS anomalies, excluding genetic conditions, among live births, fetal deaths from 20 weeks’ gestation and terminations of pregnancy for fetal anomalies. An exploratory analysis assessed the association between methadone exposure and other congenital anomalies (CAs) excluding PRS. Methadone exposure was ascertained from medical records and maternal interview.ResultsAmong 87 979 CA registrations, there were 127 methadone-exposed pregnancies and 336 PRS cases. There was an association between methadone exposure and PRS (OR adjusted for registry 12.3, 95% CI 5.7 to 26.8). In absolute terms, this association reflects a risk increase from approximately 1–12 cases per 10 000 births. A raised OR was found for cleft palate (adjusted OR 5.0, 95% CI 2.7 to 9.2).ConclusionsThese findings suggest that gestational methadone exposure is associated with PRS. The association may be explained by unmeasured confounding factors. The small increased risk of PRS in itself does not alter the risk–benefit balance for gestational methadone use. The association with cleft palate, a more common CA, should be assessed with independent data.

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Section: Discussionsupporting

confidence: 91%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Methadone, Pierre Robin sequence and other congenital anomalies: case–control study

Cleary

Loane

Addor

et al. 2019

Arch Dis Child Fetal Neonatal Ed

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“…It is now mostly considered a sequence of three main phenotypes: micrognathia (hypoplastic mandible), glossoptosis and upper airway obstruction (35)(36)(37). Its prevalence varies from 0.5 to 3.7 in 10000 births (8,(38)(39)(40) probably due to various diagnostic criteria applied and heterogeneity of characteristics (41)(42)(43). About 90% of the cases with PRS present also a cleft in the palate (44).…”

Section: Pierre Robin Sequencementioning

confidence: 99%

Minimal incision palatoplasty with or without muscle reconstruction in patients with isolated cleft palate—a cephalometric study at 5 and 10 years

Parikakis

Larson

Karsten

2018

European Journal of Orthodontics

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Summary Objective To compare differences in facial growth in patients with isolated clefts of the hard and/or soft palate treated with the minimal incision technique without (MI) or with muscle reconstruction (MMI). Subjects and method A consecutive series of 170 Caucasian children born with isolated cleft palate were studied. Individuals with other craniofacial malformations, apart from Pierre Robin sequence (PRS), were excluded. The patients were treated surgically with MI (n = 85) or MMI (n = 85) palatoplasty (mean age: 13 months) and divided further into two subgroups: clefts within the soft palate only (small cleft, n = 51) and within the hard and soft palate (big cleft, n = 119). A retrospective evaluation at 5 (mean 5.4) and 10 (mean 10.3) years was performed using lateral cephalograms. Twelve skeletal and one soft tissue measurement was evaluated. Both 95% and 99% confidence intervals were calculated, two-way ANOVA and mixed model analysis was performed including/excluding PRS. Results At 5 years, statistically significant increased inclination of the palatal plane in the big MMI cleft group (P < 0.01), increased posterior upper face height (P < 0.01), and longer mandibular length (P < 0.001) in the small MI cleft group was observed. At 10 years, statistically significant increased inclination of the palatal plane (P < 0.001), decreased posterior upper face height (P < 0.001), and longer palatal length (P < 0.01) was seen in the big MMI group. Limitations Retrospective single centre study, limited sample size, three surgeons. Conclusion Minor differences in craniofacial morphology were found between patients with isolated clefts treated with MI or MMI technique and between small and big cleft lengths.

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Robin Sequence

Saal¹

2020

Cassidy and Allanson's Management of Genetic Syndromes

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Epidemiology of Robin sequence with cleft palate in the East of Scotland between 2004 and 2013

Abstract: RS incidence in EoS is substantially higher than that reported within other populations, and than previously reported in the UK. A possible association with intrauterine Methadone exposure warrants further investigation.

Cited by 24 publications

References 28 publications

Methadone, Pierre Robin sequence and other congenital anomalies: case–control study

Methadone, Pierre Robin sequence and other congenital anomalies: case–control study

Minimal incision palatoplasty with or without muscle reconstruction in patients with isolated cleft palate—a cephalometric study at 5 and 10 years

Robin Sequence

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