- Email: [email protected]
Recurrent hypersomnia due to occult hepatic encephalopathy
Sleep Medicine 13 (2012) 321–323
Contents lists available at SciVerse ScienceDirect
Sleep Medicine journal homepage: www.elsevier.com/locate/sleep
Letters to the Editor Beneficial effects of continuous positive airway pressure therapy in a pediatric intestinal transplant recipient with obstructive sleep apnea To the editor, The association of pediatric obstructive sleep apnea (OSA) with mortality and respiratory diseases was demonstrated in a recent study [1]. We encountered a pediatric intestinal posttransplant recipient with OSA in whom continuous positive airway pressure (CPAP) therapy may have a preventive effect on respiratory tract infection. A 3-year-old Japanese boy received an intestinal transplant from a living donor (his mother) because of short-bowel syndrome. During the 5 years following transplantation, he required hospitalization 19 times, including four episodes of acute rejection and three of respiratory tract infection. At the age of four, he developed severe pneumonia and congestive heart failure and needed noninvasive positive pressure ventilation. He was subsequently hospitalized twice for pneumonia. At the age of eight, severe pediatric OSA was diagnosed based on daytime sleepiness, snoring, and 3% oxygen desaturation index (3% ODI) of 10.1 [2]. Nasal auto-set CPAP therapy with the pressure between 4 and 12 cm H2O was started, and the 3% ODI decreased from 10.1 to 1.8. After the beginning of CPAP therapy, the number of hospitalizations markedly decreased to six times during the next seven years. The causes of six hospitalizations were abdominal problems (including suspected rejection) for five of the times and influenza without pneumonia for another time. He experienced no further episodes of severe respiratory tract infection. This course suggests a significant association between OSA and respiratory tract infection. Pulmonary aspiration of gastric contents is common in children with apnea episodes or recurrent pneumonia [3]. CPAP therapy can reduce gastroesophageal reflux [4], which may prevent pneumonitis from gastric acid aspiration. CPAP therapy was also reported to decrease the risk of postoperative pulmonary complications, atelectasis, and pneumonia in patients undergoing abdominal surgery [5]. In patients with OSA, CPAP therapy can also decrease microatelectasis that otherwise would facilitate lower respiratory tract infection. The detection of OSA and application of CPAP therapy can provide an additional benefit in managing immunosuppressed children, especially when experiencing recurrent airway infections. Conflicts of interest The ICMJE Uniform Disclosure Form for Potential Conflicts of Interest associated with this article can be viewed by clicking on the following link: doi:10.1016/j.sleep.2011.10.019. References [1] Tarasiuk A, Greenberg-Dotan S, Simon-Tuval T, et al. Elevated morbidity and health care use in children with obstructive sleep apnea syndrome. Am J Respir Crit Care Med 2007;175:55–61.
[2] Saito H, Araki K, Ozawa H, et al. Pulse-oximetery is useful in determining the indications for adeno-tonsillectomy in pediatric sleep-disordered breathing. Int J Pediatr Otorhinolaryngol 2007;71:1–6. [3] Ravelli AM, Panarotto MB, Verdoni L, Consolati V, Bolognini S. Pulmonary aspiration shown by scintigraphy in gastroesophageal reflux-related respiratory disease. Chest 2006;130:1520–6. [4] Kerr P, Shoenut JP, Millar T, Buckle P, Kryger MH. Nasal CPAP reduces gastroesophageal reflux in obstructive sleep apnea syndrome. Chest 1992;101:1539–44. [5] Ferreyra GP, Baussano I, Squadrone V, et al. Continuous positive airway pressure for treatment of respiratory complications after abdominal surgery: a systematic review and meta-analysis. Ann Surg 2008;247:617–26.
Kiminobu Tanizawa Department of Respiratory Medicine, Kyoto University, Graduate School of Medicine, 54 Shogoin-kawaracho, Sakyo-ku, Kyoto 606-8507, Japan E-mail address: [email protected] Shinya Okamoto Department of Transplantation Surgery, Kyoto University, Graduate School of Medicine, 54 Shogoin-kawaracho, Sakyo-ku, Kyoto 606-8507, Japan E-mail address: [email protected] Shinji Uemoto Department of Transplantation Surgery, Kyoto University, Graduate School of Medicine, 54 Shogoin-kawaracho, Sakyo-ku, Kyoto 606-8507, Japan E-mail address: [email protected]
⇑
Kazuo Chin Department of Respiratory Care and Sleep Control Medicine, Kyoto University, Graduate School of Medicine, 54 Shogoin-kawaracho, Sakyo-ku, Kyoto 606-8507, Japan ⇑ Tel.: +81 75 751 3852; fax: +81 75 751 3854. E-mail address: [email protected] Available online 9 December 2011
1389-9457/$ - see front matter Ó 2011 Elsevier B.V. All rights reserved. doi:10.1016/j.sleep.2011.10.019
Recurrent hypersomnia due to occult hepatic encephalopathy
To the Editor Hepatic encephalopathy can give rise to stupor. However, the pathology is typically structural damage and is usually chronic rather than intermittent. We report a case of hepatic vascular dysfunction giving rise to recurrent hypersomnia.
322
Letters to the Editor / Sleep Medicine 13 (2012) 321–323
Fig. 1. Thirty second epoch tracing of polysomnography recorded during sleep demonstrates triphasic waveforms most prominent in the anterior leads. Some leads have been omitted, and others re-referenced for clarity. Abbreviations: Fp1-C3, Frontopolar-central electroencephalogram (left); Fp2-C4, Frontopolar-central electroencephalogram (right); O1-A2, left occipital-right ear reference; O2-A1, right occipital-left ear reference; LOC-A2, left electrooculogram; ROC-A1, right electrooculogram; EMG1EMG2 – chin electromyogram; L leg, left anterior tibialis electromyography (EMG); R leg, right anterior tibialis EMG; snore – microphone; nasal pressure – transducer; thor effort – thoracic inductance plethysmography; abdo effort – abdominal inductance plethysmography; SpO2 – oxygen saturation.
A 67-year-old presented with recurrent stupor for years. At the beginning of an episode, the patient became lethargic and had difficulty ambulating. Episodes lasted for 12–36 h and occurred weekly. Between episodes the patient was normal. He did not have other signs of hepatic disease. EEGs demonstrated mild generalized slowing. Treatment with anti-epileptics for suspected epilepsy failed to help. A metabolic screening was negative. He was referred to us for assessment of potential recurrent hypersomnia. Polysomnography during an episode demonstrated a sleep latency of 6 min with a sleep efficiency of 91.4, and no REM sleep. There was no significant apnea/limb movement. Prominent triphasic waveforms and generalized slowing were seen (see Fig. 1). Consequently, the patient was transferred for medical evaluation. Liver function tests were abnormal (bilirubin 24 [<20], AST 37 [<37], ALP 139 [40–120]), as was an ammonia (75 mmol/L [<50]). CT of abdomen demonstrated thrombosis of portal veins. He was diagnosed with hepatic encephalopathy on the basis of vascular dysfunction. The patient was treated with lactulose and, after four years, he remains episode-free. Other considerations included Kleine–Levin syndrome (KLS) and idiopathic recurrent stupor (IRS). KLS was unlikely as he did not have compulsive eating or hypersexuality [1]. IRS was ruled out by EEG recordings not showing generalized fast activity [2,3]. Our case was instructive as the patient did not exhibit typical signs of hepatic dysfunction; he did not have continuous stupor. The patient’s syndrome was a hypersomnia due to a medical condition [4]. This case demonstrates the value of polysomnography in directing appropriate investigations and therapy for a treatable condition that might have been otherwise confused with recurrent hypersomnia [5]. Conflicts of interest The ICMJE Uniform Disclosure Form for Potential Conflicts of Interest associated with this article can be viewed by clicking on the following link: doi:10.1016/j.sleep.2011.10.020.
References [1] Arnulf I, Zeitzer JM, File J, Farber N, Mignot E. Kleine–Levin syndrome: a systematic review of 186 cases in the literature. Brain 2005;128: 2763–76. [2] Lugaresi E, Montagna P, Tinuper P, et al. Endozepine stupor. Recurring stupor linked to endozepine-4 accumulation. Brain 1998;121:127–33. [3] Cortelli P, Avallone R, Baraldi M, Zeneroli ML, Mandrioli J, Corsi L. Endozepines in recurrent stupor. Sleep Med Rev 1995;9:477–87. [4] Billiard M, Jaussent I, Dauvilliers Y, Besset A. Recurrent hypersomnia: a review of 339 cases. Sleep Med Rev 2011;15:247–57. [5] American Academy of Sleep Medicine. International classification of sleep disorders. Diagnostic and coding manual, 2nd ed. Westchester, Illinois: American Academy of Sleep Medicine; 2005.
Mark I. Boulos Department of Medicine (Neurology), Sunnybrook Health Sciences Centre, Toronto, Ontario, Canada Jennifer Singerman Department of Medicine (Neurology), Sunnybrook Health Sciences Centre, Toronto, Ontario, Canada
⇑
Brian J. Murray Department of Medicine (Neurology), Sunnybrook Health Sciences Centre, Toronto, Ontario, Canada ⇑ Address: Sunnybrook Health Sciences Centre, M1600-2075 Bayview Avenue, Toronto, Ontario, Canada M4N 3M5. Tel.: +1 416 480 6100x2461; fax: +1 416 480 4674. E-mail address: [email protected] Available online 9 December 2011
1389-9457/$ - see front matter Ó 2011 Elsevier B.V. All rights reserved. doi:10.1016/j.sleep.2011.10.020
Contents lists available at SciVerse ScienceDirect
Sleep Medicine journal homepage: www.elsevier.com/locate/sleep
Letters to the Editor Beneficial effects of continuous positive airway pressure therapy in a pediatric intestinal transplant recipient with obstructive sleep apnea To the editor, The association of pediatric obstructive sleep apnea (OSA) with mortality and respiratory diseases was demonstrated in a recent study [1]. We encountered a pediatric intestinal posttransplant recipient with OSA in whom continuous positive airway pressure (CPAP) therapy may have a preventive effect on respiratory tract infection. A 3-year-old Japanese boy received an intestinal transplant from a living donor (his mother) because of short-bowel syndrome. During the 5 years following transplantation, he required hospitalization 19 times, including four episodes of acute rejection and three of respiratory tract infection. At the age of four, he developed severe pneumonia and congestive heart failure and needed noninvasive positive pressure ventilation. He was subsequently hospitalized twice for pneumonia. At the age of eight, severe pediatric OSA was diagnosed based on daytime sleepiness, snoring, and 3% oxygen desaturation index (3% ODI) of 10.1 [2]. Nasal auto-set CPAP therapy with the pressure between 4 and 12 cm H2O was started, and the 3% ODI decreased from 10.1 to 1.8. After the beginning of CPAP therapy, the number of hospitalizations markedly decreased to six times during the next seven years. The causes of six hospitalizations were abdominal problems (including suspected rejection) for five of the times and influenza without pneumonia for another time. He experienced no further episodes of severe respiratory tract infection. This course suggests a significant association between OSA and respiratory tract infection. Pulmonary aspiration of gastric contents is common in children with apnea episodes or recurrent pneumonia [3]. CPAP therapy can reduce gastroesophageal reflux [4], which may prevent pneumonitis from gastric acid aspiration. CPAP therapy was also reported to decrease the risk of postoperative pulmonary complications, atelectasis, and pneumonia in patients undergoing abdominal surgery [5]. In patients with OSA, CPAP therapy can also decrease microatelectasis that otherwise would facilitate lower respiratory tract infection. The detection of OSA and application of CPAP therapy can provide an additional benefit in managing immunosuppressed children, especially when experiencing recurrent airway infections. Conflicts of interest The ICMJE Uniform Disclosure Form for Potential Conflicts of Interest associated with this article can be viewed by clicking on the following link: doi:10.1016/j.sleep.2011.10.019. References [1] Tarasiuk A, Greenberg-Dotan S, Simon-Tuval T, et al. Elevated morbidity and health care use in children with obstructive sleep apnea syndrome. Am J Respir Crit Care Med 2007;175:55–61.
[2] Saito H, Araki K, Ozawa H, et al. Pulse-oximetery is useful in determining the indications for adeno-tonsillectomy in pediatric sleep-disordered breathing. Int J Pediatr Otorhinolaryngol 2007;71:1–6. [3] Ravelli AM, Panarotto MB, Verdoni L, Consolati V, Bolognini S. Pulmonary aspiration shown by scintigraphy in gastroesophageal reflux-related respiratory disease. Chest 2006;130:1520–6. [4] Kerr P, Shoenut JP, Millar T, Buckle P, Kryger MH. Nasal CPAP reduces gastroesophageal reflux in obstructive sleep apnea syndrome. Chest 1992;101:1539–44. [5] Ferreyra GP, Baussano I, Squadrone V, et al. Continuous positive airway pressure for treatment of respiratory complications after abdominal surgery: a systematic review and meta-analysis. Ann Surg 2008;247:617–26.
Kiminobu Tanizawa Department of Respiratory Medicine, Kyoto University, Graduate School of Medicine, 54 Shogoin-kawaracho, Sakyo-ku, Kyoto 606-8507, Japan E-mail address: [email protected] Shinya Okamoto Department of Transplantation Surgery, Kyoto University, Graduate School of Medicine, 54 Shogoin-kawaracho, Sakyo-ku, Kyoto 606-8507, Japan E-mail address: [email protected] Shinji Uemoto Department of Transplantation Surgery, Kyoto University, Graduate School of Medicine, 54 Shogoin-kawaracho, Sakyo-ku, Kyoto 606-8507, Japan E-mail address: [email protected]
⇑
Kazuo Chin Department of Respiratory Care and Sleep Control Medicine, Kyoto University, Graduate School of Medicine, 54 Shogoin-kawaracho, Sakyo-ku, Kyoto 606-8507, Japan ⇑ Tel.: +81 75 751 3852; fax: +81 75 751 3854. E-mail address: [email protected] Available online 9 December 2011
1389-9457/$ - see front matter Ó 2011 Elsevier B.V. All rights reserved. doi:10.1016/j.sleep.2011.10.019
Recurrent hypersomnia due to occult hepatic encephalopathy
To the Editor Hepatic encephalopathy can give rise to stupor. However, the pathology is typically structural damage and is usually chronic rather than intermittent. We report a case of hepatic vascular dysfunction giving rise to recurrent hypersomnia.
322
Letters to the Editor / Sleep Medicine 13 (2012) 321–323
Fig. 1. Thirty second epoch tracing of polysomnography recorded during sleep demonstrates triphasic waveforms most prominent in the anterior leads. Some leads have been omitted, and others re-referenced for clarity. Abbreviations: Fp1-C3, Frontopolar-central electroencephalogram (left); Fp2-C4, Frontopolar-central electroencephalogram (right); O1-A2, left occipital-right ear reference; O2-A1, right occipital-left ear reference; LOC-A2, left electrooculogram; ROC-A1, right electrooculogram; EMG1EMG2 – chin electromyogram; L leg, left anterior tibialis electromyography (EMG); R leg, right anterior tibialis EMG; snore – microphone; nasal pressure – transducer; thor effort – thoracic inductance plethysmography; abdo effort – abdominal inductance plethysmography; SpO2 – oxygen saturation.
A 67-year-old presented with recurrent stupor for years. At the beginning of an episode, the patient became lethargic and had difficulty ambulating. Episodes lasted for 12–36 h and occurred weekly. Between episodes the patient was normal. He did not have other signs of hepatic disease. EEGs demonstrated mild generalized slowing. Treatment with anti-epileptics for suspected epilepsy failed to help. A metabolic screening was negative. He was referred to us for assessment of potential recurrent hypersomnia. Polysomnography during an episode demonstrated a sleep latency of 6 min with a sleep efficiency of 91.4, and no REM sleep. There was no significant apnea/limb movement. Prominent triphasic waveforms and generalized slowing were seen (see Fig. 1). Consequently, the patient was transferred for medical evaluation. Liver function tests were abnormal (bilirubin 24 [<20], AST 37 [<37], ALP 139 [40–120]), as was an ammonia (75 mmol/L [<50]). CT of abdomen demonstrated thrombosis of portal veins. He was diagnosed with hepatic encephalopathy on the basis of vascular dysfunction. The patient was treated with lactulose and, after four years, he remains episode-free. Other considerations included Kleine–Levin syndrome (KLS) and idiopathic recurrent stupor (IRS). KLS was unlikely as he did not have compulsive eating or hypersexuality [1]. IRS was ruled out by EEG recordings not showing generalized fast activity [2,3]. Our case was instructive as the patient did not exhibit typical signs of hepatic dysfunction; he did not have continuous stupor. The patient’s syndrome was a hypersomnia due to a medical condition [4]. This case demonstrates the value of polysomnography in directing appropriate investigations and therapy for a treatable condition that might have been otherwise confused with recurrent hypersomnia [5]. Conflicts of interest The ICMJE Uniform Disclosure Form for Potential Conflicts of Interest associated with this article can be viewed by clicking on the following link: doi:10.1016/j.sleep.2011.10.020.
References [1] Arnulf I, Zeitzer JM, File J, Farber N, Mignot E. Kleine–Levin syndrome: a systematic review of 186 cases in the literature. Brain 2005;128: 2763–76. [2] Lugaresi E, Montagna P, Tinuper P, et al. Endozepine stupor. Recurring stupor linked to endozepine-4 accumulation. Brain 1998;121:127–33. [3] Cortelli P, Avallone R, Baraldi M, Zeneroli ML, Mandrioli J, Corsi L. Endozepines in recurrent stupor. Sleep Med Rev 1995;9:477–87. [4] Billiard M, Jaussent I, Dauvilliers Y, Besset A. Recurrent hypersomnia: a review of 339 cases. Sleep Med Rev 2011;15:247–57. [5] American Academy of Sleep Medicine. International classification of sleep disorders. Diagnostic and coding manual, 2nd ed. Westchester, Illinois: American Academy of Sleep Medicine; 2005.
Mark I. Boulos Department of Medicine (Neurology), Sunnybrook Health Sciences Centre, Toronto, Ontario, Canada Jennifer Singerman Department of Medicine (Neurology), Sunnybrook Health Sciences Centre, Toronto, Ontario, Canada
⇑
Brian J. Murray Department of Medicine (Neurology), Sunnybrook Health Sciences Centre, Toronto, Ontario, Canada ⇑ Address: Sunnybrook Health Sciences Centre, M1600-2075 Bayview Avenue, Toronto, Ontario, Canada M4N 3M5. Tel.: +1 416 480 6100x2461; fax: +1 416 480 4674. E-mail address: [email protected] Available online 9 December 2011
1389-9457/$ - see front matter Ó 2011 Elsevier B.V. All rights reserved. doi:10.1016/j.sleep.2011.10.020