Episodic Spontaneous Hypothermia Potentially Triggered by Hyperinsulinemia

Episodic spontaneous hypothermia is an infrequent disorder, the pathogenic mechanisms of which have not been completely clarified, although alterations in the serotoninergic system have been suggested. We report the history of a girl with episodes of dizziness and shivering associated with a body temperature lower than 35°C since the age of 10 months. At the age of 11 years, she was admitted to a local hospital and an oral glucose tolerance test showed high total insulin levels. Hypoglycemia secondary to hyperinsulinemia was suspected, and a low-carbohydrate (simple) diet was proposed without results. Due to the recurrence of the episodes, episodic spontaneous hypothermia triggered by hyperinsulinemia was suspected, and treatment with flunarizine, a drug considered the first line in the treatment of migraine-related disorders, was started with a resulting reduction in the episodes. A new endocrinological evaluation showed decreased insulin secretion. In our patient, the success of the therapy might be due to the well-known effect of calcium antagonists in inhibiting serotonin uptake and thereby regulating serotonin levels after hyperinsulinism. This case suggests hyperinsulinemia as a potential mechanism for episodic spontaneous hypothermia, probably mediated by an interaction between insulin and the serotoninergic system.

[1]  O. Kolosova,et al.  Effects of calcium antagonists on serotonin-dependent aggregation and serotonin transport in platelets of patients with migraine , 2000, Bulletin of Experimental Biology and Medicine.

[2]  F. Lechin,et al.  Central Nervous System Circuitry Involved in the Hyperinsulinism Syndrome , 2007, Neuroendocrinology.

[3]  J. Serratrice,et al.  Hypothermies spontanées : dix observations, place du syndrome de Shapiro , 2005 .

[4]  S. Raptis,et al.  Serotonin and insulin release in vitro , 1968, Diabetologia.

[5]  J. Serratrice,et al.  [Spontaneous hypothermia: a series of ten cases, place of Shapiro's syndrome]. , 2005, La Revue de medecine interne.

[6]  B. Gener,et al.  Episodic spontaneous hypothermia: a periodic childhood syndrome. , 2003, Pediatric neurology.

[7]  Richard A. Greenberg,et al.  Pediatric nonenvironmental hypothermia presenting to the emergency department: Episodic spontaneous hypothermia with hyperhidrosis , 2003, Pediatric emergency care.

[8]  S. Evers,et al.  [Treatment of idiopathic headache in childhood - recommendations of the German Migraine and Headache Society (DMKG)]. , 2002, Schmerz.

[9]  R. Pothmann,et al.  Therapie idiopathischer Kopfschmerzen im Kindesalter , 2002, Der Schmerz.

[10]  C. Rouch,et al.  Activation of hypothalamic insulin by serotonin is the primary event of the insulin–serotonin interaction involved in the control of feeding , 2000, Brain Research.

[11]  S. Nicolaidis,et al.  Determination, Using Microdialysis, of Hypothalamic Serotonin Variations in Response to Different Macronutrients , 1998, Physiology & Behavior.

[12]  Z. Spirer,et al.  Periodic syndromes of childhood. , 1997, Advances in pediatrics.

[13]  H. Arroyo,et al.  Episodic spontaneous hypothermia with hyperhidrosis. , 1994, Pediatric neurology.

[14]  R. Sheth,et al.  Episodic spontaneous hypothermia with hyperhidrosis: implications for pathogenesis. , 1994, Pediatric neurology.

[15]  M. Raizada,et al.  The cellular and physiological actions of insulin in the central nervous system , 1993, Neurochemistry International.

[16]  F. Lechin,et al.  Doxepin therapy for postprandial symptomatic hypoglycaemic patients: neurochemical, hormonal and metabolic disturbances. , 1991, Clinical science.

[17]  H. Arroyo,et al.  A syndrome of hyperhidrosis, hypothermia, and bradycardia possibly due to central monoaminergic dysfunction , 1990, Neurology.

[18]  F. Micheli,et al.  Movement disorders and depression due to flunarizine and cinnarizine , 1989, Movement disorders : official journal of the Movement Disorder Society.

[19]  D. Capellà,et al.  Parkinsonism, tremor, and depression induced by cinnarizine and flunarizine. , 1988, BMJ.

[20]  F. Plum,et al.  Spontaneous recurrent hypothermia accompanying agenesis of the corpus callosum. , 1969, Brain : a journal of neurology.