Evidence map›Paper›PMID 39669238›Full record

ArticleGenetics in medicine open2023

A novel syndrome associated with prenatal fentanyl exposure.

Erin Wadman, Erica Fernandes, Candace Muss, Nina Powell-Hamilton, Monica H Wojcik, Jill A Madden, Chrystalle Katte Carreon, Robin D Clark, Annie Stenftenagel, Kamal Chikalard and 4 more

Abstract read
In one paragraph

Article in Genetics in medicine open, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
17citing papers in PubMed, 2 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

17 citing papers in PubMed, 2 syntheses or guidelines pooled it.

  1. Pooled it
  2. Pooled it
  3. Embryodysgenesis of the Lids and Orbit.Eye (London, England) · 2026
    Review
  4. Sterol biosynthesis, brain development, and disease.The Journal of clinical investigation · 2026
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

14 authors.

Erin WadmanDivision of Medical Genetics, Nemours Children's Hospital, Wilmington, DE.
Erica FernandesDivision of Medical Genetics, Nemours Children's Hospital, Wilmington, DE.
Candace MussDivision of Medical Genetics, Nemours Children's Hospital, Wilmington, DE.
Nina Powell-HamiltonDivision of Medical Genetics, Nemours Children's Hospital, Wilmington, DE.
Monica H WojcikDivision of Newborn Medicine, Boston Children's Hospital, Boston, MA.
Jill A MaddenThe Manton Center for Orphan Disease Research and Division of Genetics and Genomics, Boston Children's Hospital, Boston, MA.
Chrystalle Katte CarreonDepartment of Pathology, Boston Children's Hospital and Harvard Medical School, Boston, MA.
Robin D ClarkDivision of Pediatric Genetics, Loma Linda University Children's Hospital, Loma Linda, CA.
Annie StenftenagelDivision of Genetics and Genomic Medicine, Department of Pediatrics, University of California-Irvine Medical Center, Irvine, CA.
Kamal ChikalardDivision of Genetics and Genomic Medicine, Department of Pediatrics, University of California-Irvine Medical Center, Irvine, CA.
Virginia KimonisDivision of Genetics and Genomic Medicine, Department of Pediatrics, University of California-Irvine Medical Center, Irvine, CA.
William BruckerDivision of Medical Genetics, Hasbro Children's Hospital, Providence, RI.
Carolina AlvesFDNA Inc., Sunrise, FL.
Karen W GrippDivision of Medical Genetics, Nemours Children's Hospital, Wilmington, DE.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

A novel syndrome was suspected in individuals sharing short stature, microcephaly, distinctive facial features, and congenital anomalies. We enrolled 6 patients in an institutional review board approved study and evaluated medical history, findings, facial photographs, and test results across this original cohort. Four additional cases with similar findings were contributed by clinicians from outside institutions, bringing the number of reported cases to 10 and supporting the existence of this novel syndrome. The 6 individuals enrolled into the institutional review board approved study shared microcephaly, short stature, and distinctive facial features. Congenital malformations included cleft palate, talipes equinovarus or rocker bottom feet, and chordee or hypospadias. Short, broad thumbs, single palmar crease, and mild 2,3 toe syndactyly were present. A hypoplastic corpus callosum was noted in 3 of 5 with appropriate evaluation. Their growth and physical findings were suggestive of Smith-Lemli-Opitz syndrome. Biochemical studies shortly after delivery indicated abnormalities in the cholesterol metabolism pathway that subsequently resolved. No shared genomic or genetic cause was identified. All individuals were born after a pregnancy complicated by prenatal exposure to nonprescription opioids, particularly fentanyl, suggesting fentanyl as a teratogen. Prenatal fentanyl exposure possibly interfered with cholesterol metabolism, giving rise to findings resembling Smith-Lemli-Opitz syndrome. This novel syndrome is clinically recognizable. Four additional cases contributed clinically shared similar findings, increasing the number of cases to 10 and supporting a novel syndrome associated with prenatal fentanyl exposure. Assessment of Shepard and Bradford Hill criteria could be consistent with fentanyl as teratogen, though caution is necessary before assigning causality and data replication is needed.

Indexed as

EmbryopathyGestaltMatcherOpioid use disorderPrenatal fentanyl exposureSmith-Lemli-Opitz syndrome

Identifiers

PMID39669238
PMCPMC11613603

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.