The combination of thermal dysregulation and agenesis of corpus callosum: Shapiro's or/and reverse Shapiro's syndrome

Topcu Y., Bayram E., KARAOĞLU P., YİŞ U., Kurul S. H.

ANNALS OF INDIAN ACADEMY OF NEUROLOGY, cilt.16, sa.4, ss.716-719, 2013 (SCI-Expanded) identifier identifier identifier

  • Yayın Türü: Makale / Tam Makale
  • Cilt numarası: 16 Sayı: 4
  • Basım Tarihi: 2013
  • Doi Numarası: 10.4103/0972-2327.120451
  • Dergi Adı: ANNALS OF INDIAN ACADEMY OF NEUROLOGY
  • Derginin Tarandığı İndeksler: Science Citation Index Expanded (SCI-EXPANDED), Scopus
  • Sayfa Sayıları: ss.716-719
  • Anahtar Kelimeler: Agenesis of corpus callosum, Shapiro's syndrome, vomiting, CORTICOTROPIN-RELEASING-FACTOR, SPONTANEOUS RECURRENT HYPOTHERMIA, SPONTANEOUS PERIODIC HYPOTHERMIA, CYCLIC VOMITING SYNDROME, HYPERHIDROSIS, VARIANT, STRESS
  • Dokuz Eylül Üniversitesi Adresli: Evet

Özet

Shapiro syndrome is an extremely rare condition consisting the clinical triad of recurrent hypothermia, hyperhydrosis and agenesis of the corpus callosum. On the other hand, reverse Shapiro's sydrome is characterized periodic hyperthermia and agenesis of the corpus callosum. Here, we describe a 3.5-year-old girl with complete agenesis of corpus callosum presenting with recurrent fever and vomiting. She also had hypothermia attacks with accompanying diaphoresis. To the best of our knowledge, there is no described case with episodes of hyperthermia, hypothermia, and vomiting associated with agenesis of the corpus callosum. Recurrent vomiting may be a newly defined symptom associated with these syndromes.