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Wagner R, Lacher M, Merkenschlager A, et al. Harlequin Syndrome after Thoracoscopic Repair of a Child with Tracheoesophageal Fistula (TEF). European J Pediatr Surg Rep. 2019;7(1):e63-e65doi: 10.1055/s-0039-1697667.
Wagner, R., Lacher, M., Merkenschlager, A., & Markel, M. (2019). Harlequin Syndrome after Thoracoscopic Repair of a Child with Tracheoesophageal Fistula (TEF). European journal of pediatric surgery reports, 7(1), e63-e65. https://doi.org/10.1055/s-0039-1697667
Wagner, Richard, et al. "Harlequin Syndrome after Thoracoscopic Repair of a Child with Tracheoesophageal Fistula (TEF)." European journal of pediatric surgery reports vol. 7,1 (2019): e63-e65. doi: https://doi.org/10.1055/s-0039-1697667
Wagner R, Lacher M, Merkenschlager A, Markel M. Harlequin Syndrome after Thoracoscopic Repair of a Child with Tracheoesophageal Fistula (TEF). European J Pediatr Surg Rep. 2019 Jan;7(1):e63-e65. doi: 10.1055/s-0039-1697667. Epub 2019 Sep 26. PMID: 31579642; PMCID: PMC6763501.
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European J Pediatr Surg Rep. 2019 Jan;7(1):e63-e65. doi: 10.1055/s-0039-1697667. Epub 2019 Sep 26.

Harlequin Syndrome after Thoracoscopic Repair of a Child with Tracheoesophageal Fistula (TEF).

European journal of pediatric surgery reports

Richard Wagner, Martin Lacher, Andreas Merkenschlager, Moritz Markel

Affiliations

  1. Klinik und Poliklinik für Kinderchirurgie, Universitätsklinikum Leipzig, Leipzig, Sachsen, Germany.
  2. Klinik für Neuropädiatrie, Universitätsklinikum Leipzig Klinik und Poliklinik für Kinder- und Jugendmedizin, Leipzig, Sachsen, Germany.

PMID: 31579642 PMCID: PMC6763501 DOI: 10.1055/s-0039-1697667

Abstract

Harlequin syndrome (HS) is a rare dysautonomia of the sympathetic nervous system leading to asymmetric facial flushing and sweating. In the literature, only a few cases of HS after thoracoscopic tracheoesophageal fistula (TEF) repair are reported. We report on a newborn with TEF who developed HS after thoracoscopic repair. On the first day of life, the girl (3,480 g, gestation age: 41 week) underwent thoracoscopic repair of a type C esophageal atresia (TEF; OR time 105 minute) without complications. The postoperative course was uneventful, the patient swallowed and thrived well and did not require esophageal dilatations. At 2 years of age, missing facial flushing, transpiration, and warming on the right side of her face during agitation were noticed. As no further intervention was required, the girl and her parents adapted well to the symptoms. Our report shows that the late onset of HS after the surgical procedure is unlikely a direct causal relation to the thoracoscopic operation but rather a shared embryological pathogenesis, like a neurocristopathy.

Keywords: Harlequin syndrome; esophageal atresia; neurocristopathy; thoracoscopic repair

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