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PP-089 Atypical harlequin color change between upper and lower halves body case report
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  1. Sarah Abdullah Bedir,
  2. Sian Jenkins,
  3. Isabela Gavor
  1. Glangwili Hospital West UK

Abstract

Material and Method HCC of the newborn, known also as Intractable vascular autonomic dysregulation (Harlequin phenomenon), is a transient unilateral erythema that is seen in up to 10% of healthy newborns and was first described by Neligan and Strang in 1952. HCC is macular and blanchable displaying a sharp midline cutoff. It is often gravity dependent. HCC typically presents between 2ndand 5th day of life, but some infants may present with episodes up to the third week of life. We herein report a case of full term male who had Harlequin Color Change between upper and lower halves of his body.

Results 39+1w singleton pregnancy boy born by spontaneous vaginal delivery without any antenatal, intranatal issues had a bluish lower half of the body and pale upper half which lasted 4 hours and resolved spontaneously without any other symptoms. In 3rdweek of life he presented to the hospital with virus infection, bluish discoloration was noted around the umbilicus and abdomen distension. ECG, ECHO, CSF analysis and abdominal ultrasound were all normal. Patient was discharged home and presented later for troponin level follow-up which was normal. The differential diagnosis includes port-wine stain, nascent hemangioma of infancy, cutis marmorata, cutis marmorata telangiectatica congenita, CHILD syndrome, Acrocyanosis, Drug reaction.

Conclusions The exact etiology of HCC is unknown, but the evidence points to sympathetic autonomic dysfunction in the control of the capillary tone, most likely secondary to hypothalamic immaturity in the newborns. Most cases are idiopathic in healthy newborns. HCC has also been noted in the setting of prematurity, low birth weight, hypoxia, infusion of prostaglandin E1(PGE1), neonatal meningitis. Learning points: Harlequin phenomenon occurs in healthy newborn. Recognition and education of this skin disorder are important as HCC is a surprising and alarming occurrence for parents, so reassurance about the benign nature of the condition, and to avoid additional diagnostic procedures and unnecessary treatments. Beyond the group age the search for an underlying cause should be vigorously pursued.

  • Atypical Harlequin Color change

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