Asymmetric sweating and flushing in infants with esophageal atresia☆
Section snippets
Patients and methods
Between 1967 and 2007, 136 patients with esophageal atresia and/or tracheoesophageal fistula were treated at our institution. We reviewed the hospital records of patients consecutively admitted between January 2003 and December 2007 to update the database used in a previous study [2]. In our database, flushing was not coded. We then selected all patients who presented with hyperhidrosis and reviewed their original written notes to select the patients who presented with unilateral sweating. The
Results
Of 136 patients with esophageal atresia and/or tracheoesophageal fistula, 40 patients presented hyperhidrosis. Of these, cases 2 and 3 presented unilateral sweating, and case 1 presented an hemifacial flushing associated with a symmetrical hyperhidrosis. All the 3 infants had a right thoracotomy to repair an esophageal atresia with a lower tracheoesophageal fistula. Case 2 had a right-sided aortic arch which required also a left thoracotomy for excision of a severe esophageal stricture at the
Discussion
Hemifacial flushing without body involvement (Case 1) is most likely to be the first reported in an infant with repaired esophageal atresia. Redness of the skin of one half of the body was first described by Neligan and Strang [4] in premature babies or in full-term babies with clinical evidence of cerebral damage. They called this condition “Harlequin color change.” The unilateral distribution of color change suggested a dysfunction of the autonomic centers which control the tone of the skin
Acknowledgment
We thank Dr Francesco Morini for signalizing that the hemifacial flushing of case 1 had been represented in the literature as Harlequin syndrome.
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Harlequin syndrome in a melanoderm patient
2016, Revue de Stomatologie, de Chirurgie Maxillo-faciale et de Chirurgie OraleHarlequin Syndrome
2018, Indian Journal of Pediatrics
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No grant or other form of payment was given to anyone to produce the manuscript. There is no potential conflict of interest, real or perceived.