Skip to main content
Top
Published in: Neurological Sciences 3/2010

01-06-2010 | Case Report

Insulinoma presenting as idiopathic hypersomnia

Authors: Michelangelo Maestri, Fabio Monzani, Enrica Bonanni, Elisa Di Coscio, Fabio Cignoni, Angela Dardano, Alfonso Iudice, Luigi Murri

Published in: Neurological Sciences | Issue 3/2010

Login to get access

Abstract

We report the case of a 32-year-old woman with a history of increased sleep need and difficulty waking up; the diagnosis of idiopathic hypersomnia was hypothesized. During ambulatory polysomnography (PSG), the patient presented an episode characterized by loss of consciousness and jerking of the four limbs. A video-PSG monitoring was performed and the patient showed unresponsiveness and drowsiness at 7 a.m. During the episode, EEG showed theta–delta diffuse activity, and blood glucose level was 32 mg dl−1. The diagnosis of insulinoma was then assumed; CT scan showed a hypodense mass into the pancreatic tail, and a partial pancreasectomy was performed. The described symptoms disappeared, and 5 years later the findings of a complete clinical and neurophysiological examination were negative. The clinical picture of insulinoma presenting with paroxysmal disorders has been previously described; however, whereas hypersomnia is uncommon, in the current case it represents the main symptom. Clinicians should keep in mind that neuroglycopenia should be considered in the differential diagnosis of patients with hypersomnia, particularly if the clinical scenario does not conform to standard criteria.
Literature
1.
go back to reference Thorpy MJ (2005) Which clinical conditions are responsible for impaired alertness? Sleep Med 6:S13–S20CrossRefPubMed Thorpy MJ (2005) Which clinical conditions are responsible for impaired alertness? Sleep Med 6:S13–S20CrossRefPubMed
2.
go back to reference Dizon AM, Kowalyk S, Hoogwerf BJ (1999) Neuroglycopenic and other symptoms in patient with insulinomas. Am J Med 106:307–310CrossRefPubMed Dizon AM, Kowalyk S, Hoogwerf BJ (1999) Neuroglycopenic and other symptoms in patient with insulinomas. Am J Med 106:307–310CrossRefPubMed
3.
go back to reference Rechtschaffen A, Kales A (eds) (1968) A manual of standardized terminology, techniques and scoring system for sleep stages of human subjects. US Department of Health, Education, and Welfare Public Health Service, NIH publication no. 204, Washington, DC Rechtschaffen A, Kales A (eds) (1968) A manual of standardized terminology, techniques and scoring system for sleep stages of human subjects. US Department of Health, Education, and Welfare Public Health Service, NIH publication no. 204, Washington, DC
4.
go back to reference Salinsky M, Sutula T, Roscoe D (1987) Representation of sleep stages by color density spectral array. Electroencephalogr Clin Neurophysiol 66:579–582CrossRefPubMed Salinsky M, Sutula T, Roscoe D (1987) Representation of sleep stages by color density spectral array. Electroencephalogr Clin Neurophysiol 66:579–582CrossRefPubMed
5.
go back to reference Piovesan A, Pia A, Visconti G et al (2003) Proinsulin-secreting neuroendocrine tumor of the pancreas. J Endocrinol Invest 6:758–761 Piovesan A, Pia A, Visconti G et al (2003) Proinsulin-secreting neuroendocrine tumor of the pancreas. J Endocrinol Invest 6:758–761
6.
go back to reference Bazil CW, Pack A (2001) Insulinoma presenting as seizure disorder. Neurology 56:817–818PubMed Bazil CW, Pack A (2001) Insulinoma presenting as seizure disorder. Neurology 56:817–818PubMed
7.
go back to reference Dion MH, Cossette P, St-Hilaire JM, Rasio E, Nguyen DK (2004) Insulinoma misdiagnosed as intractable epilepsy. Neurology 62:1443–1445PubMed Dion MH, Cossette P, St-Hilaire JM, Rasio E, Nguyen DK (2004) Insulinoma misdiagnosed as intractable epilepsy. Neurology 62:1443–1445PubMed
8.
go back to reference Graves TD, Gandhi S, Smith SJM, Sisodiya SM, Conway GS (2004) Misdiagnosis of seizures: insulinoma presenting as adult-onset seizure disorder. J Neurol Neurosurg Psychiatry 75:1091–1092CrossRefPubMed Graves TD, Gandhi S, Smith SJM, Sisodiya SM, Conway GS (2004) Misdiagnosis of seizures: insulinoma presenting as adult-onset seizure disorder. J Neurol Neurosurg Psychiatry 75:1091–1092CrossRefPubMed
9.
go back to reference O’Sullivan SS, Redmond J (2005) Insulinoma presenting as refractory late-onset epilepsy. Epilepsia 46:1690–1691CrossRefPubMed O’Sullivan SS, Redmond J (2005) Insulinoma presenting as refractory late-onset epilepsy. Epilepsia 46:1690–1691CrossRefPubMed
10.
go back to reference Jaladyan V, Darbinyan V (2007) Insulinoma misdiagnosed as juvenile myoclonic epilepsy. Eur J Pediatr 166:485–487CrossRefPubMed Jaladyan V, Darbinyan V (2007) Insulinoma misdiagnosed as juvenile myoclonic epilepsy. Eur J Pediatr 166:485–487CrossRefPubMed
11.
go back to reference Groselj LD, Butinar D (2008) Insulinoma presenting itself as a night paroxysmal disorder with spontaneous recovery. Lijec Vjesn 130:104–105PubMed Groselj LD, Butinar D (2008) Insulinoma presenting itself as a night paroxysmal disorder with spontaneous recovery. Lijec Vjesn 130:104–105PubMed
12.
go back to reference Striano S, Striano P, Manganelli F, Boccella P, Bruno R, Santoro L, Percopo V (2003) Distal hypoglycemic neuropathy. An insulinoma-associated case, misdiagnosed as temporal lobe epilepsy. Neurophysiol Clin 33:223–227CrossRefPubMed Striano S, Striano P, Manganelli F, Boccella P, Bruno R, Santoro L, Percopo V (2003) Distal hypoglycemic neuropathy. An insulinoma-associated case, misdiagnosed as temporal lobe epilepsy. Neurophysiol Clin 33:223–227CrossRefPubMed
13.
go back to reference Dhume VM, Amarapurkar AD, Rege JD, Kandalkar BM, Varthakavi P (2004) Insulinoma—a case report. Indian J Pathol Microbiol 47:540–541PubMed Dhume VM, Amarapurkar AD, Rege JD, Kandalkar BM, Varthakavi P (2004) Insulinoma—a case report. Indian J Pathol Microbiol 47:540–541PubMed
14.
go back to reference Cryer PE (2005) Mechanisms of hypoglycemia-associated autonomic failure and its component syndromes in diabetes. Diabetes 54:3592–3601CrossRefPubMed Cryer PE (2005) Mechanisms of hypoglycemia-associated autonomic failure and its component syndromes in diabetes. Diabetes 54:3592–3601CrossRefPubMed
15.
go back to reference Jones TW, Porter P, Sherwin RS et al (1998) Decreased epinephrine responses to hypoglycemia during sleep. N Engl J Med 338:1657–1662CrossRefPubMed Jones TW, Porter P, Sherwin RS et al (1998) Decreased epinephrine responses to hypoglycemia during sleep. N Engl J Med 338:1657–1662CrossRefPubMed
16.
go back to reference Bendtson I, Gade J, Thomsen CE, Rosenfalck A, Wildschiødtz G (1992) Sleep disturbances in IDDM patients with nocturnal hypoglycemia. Sleep 15(1):74–81PubMed Bendtson I, Gade J, Thomsen CE, Rosenfalck A, Wildschiødtz G (1992) Sleep disturbances in IDDM patients with nocturnal hypoglycemia. Sleep 15(1):74–81PubMed
17.
go back to reference Tkacs NC, Pan Y, Sawhney G, Mann GL, Morrison AR (2007) Hypoglycemia activates arousal-related neurons and increases wake time in adult rats. Physiol Behav 91:240–249CrossRefPubMed Tkacs NC, Pan Y, Sawhney G, Mann GL, Morrison AR (2007) Hypoglycemia activates arousal-related neurons and increases wake time in adult rats. Physiol Behav 91:240–249CrossRefPubMed
18.
go back to reference American Academy of Sleep Medicine (2005) International classification of sleep disorders: diagnostic and coding manual, 2nd edn. Westchester, Illinois American Academy of Sleep Medicine (2005) International classification of sleep disorders: diagnostic and coding manual, 2nd edn. Westchester, Illinois
19.
go back to reference Wang S, Hu HT, Wen SQ, Wang ZJ, Zhang BR, Ding MP (2008) An insulinoma with clinical and electroencephalographic features resembling complex partial seizures. J Zhejiang Univ Sci B 9(6):496–499CrossRefPubMed Wang S, Hu HT, Wen SQ, Wang ZJ, Zhang BR, Ding MP (2008) An insulinoma with clinical and electroencephalographic features resembling complex partial seizures. J Zhejiang Univ Sci B 9(6):496–499CrossRefPubMed
Metadata
Title
Insulinoma presenting as idiopathic hypersomnia
Authors
Michelangelo Maestri
Fabio Monzani
Enrica Bonanni
Elisa Di Coscio
Fabio Cignoni
Angela Dardano
Alfonso Iudice
Luigi Murri
Publication date
01-06-2010
Publisher
Springer Milan
Published in
Neurological Sciences / Issue 3/2010
Print ISSN: 1590-1874
Electronic ISSN: 1590-3478
DOI
https://doi.org/10.1007/s10072-009-0207-7

Other articles of this Issue 3/2010

Neurological Sciences 3/2010 Go to the issue