Spontaneous endogenous hypermelatoninemia: a new disease?
Melatonin, a major photoperiod-dependent hormone, regulates circadian rhythms and biological rhythms and acts as a prominent sleep promoter. Symptoms related to hypermelatoninemia have been reported in individuals supplemented with melatonin. However, spontaneous endogenous hypermelatoninemia has not been reported previously. A 6-year-old girl previously diagnosed with Shapiro's syndrome was admitted to our hospital on several occasions during a 1-year period with complaints of altered consciousness, syncope, hypothermia and episodes of sweating. The episodes occurred daily and during sleep and lasted for 1-6 h. During these episodes, she sweated profusely and felt faint and her skin was pale and cool. Other complaints included recurrent abdominal pain, urge incontinence and myopia. She was shown to have hypermelatoninemia (>1,000 pg/ml, normal range 0-150 pg/ml) during these episodes. The duration of her attacks decreased with phototherapy and she was successfully treated with propranolol. To our knowledge, this is the first case of hypermelatoninemia without any detectable organic pathology. We did not determine the exact mechanism of hypermelatoninemia in this patient; however, it might have been related to irregular control of pinealocytes by the suprachiasmatic nucleus or related pathways. Hypermelatoninemia should be considered in patients with spontaneous periodic hypothermia and hyperhidrosis, and also in patients with Shapiro's syndrome.
Department of Pediatric Neurology, Faculty of Medicine, Akdeniz University, Antalya, Turkey.
Ozgur
O
Duman
Erdem
E
Durmaz
Sema
S
Akcurin
Mustafa
M
Serteser
Senay
S
Haspolat
D055673:Acrocallosal Syndrome / Q000097:blood / Q000188:drug therapy; D000319:Adrenergic beta-Antagonists / Q000627:therapeutic use; D061085:Agenesis of Corpus Callosum; D002648:Child; D005260:Female; D006801:Humans; D006945:Hyperhidrosis / Q000097:blood / Q000188:drug therapy; D007035:Hypothermia / Q000097:blood* / Q000188:drug therapy; D008550:Melatonin / Q000097:blood*; D010789:Phototherapy; D011433:Propranolol / Q000627:therapeutic use
D000319:Adrenergic beta-Antagonists; D011433:Propranolol; D008550:Melatonin