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Nasal chondromesenchymal hamartoma causing sleep-disordered breathing in an infant
Abstracts/Sleep Medicine 16 (2015) S2–S199
discontinuation of pramipexole medication. The sleep study was repeated after the re-continuation of pramipexole. Leg movements were marked manually and PWA dips were detected using a custom script. Temporal relationship between leg movements and transient PWA phenomena was assessed both during wakefulness and sleep. Events with marked disturbance in the plethysmogram signal were excluded from the analysis. Results: A total of 2606 leg movements were included in the analysis. We found that half of the patients had transient dips in PWA following the start of the leg movements. In these patients, the PWA dipped by an average of 24% (range 12–30%) during sleep and 12% (range 6–20%) during wakefulness. PWA phenomena did not herald the leg movements but started 2–10 seconds after the movement. Most commonly, the dip lasted for 10–20 seconds. The other half of the patients did not show similar dips. When present, both leg movements and PWA dips were suppressed by pramipexole. Conclusion: The results suggest that PLM are associated with transient peripheral vasoconstriction in some patients with RLS. The vasoconstriction is in tight temporal relationship with the leg movement. Pramipexole suppresses both PLM and the associated transient vasoconstriction. http://dx.doi.org/10.1016/j.sleep.2015.02.464
Association of co-incidental hypertension with periodic limb movements during sleep in hospital based cross-sectional study K. Yang 1, Y. Cho 2, Y. Hwangbo 3, D. Kim 4, M. Song 5 1 Division of Epilepsy and Sleep Disorders, Department of Neurology, Soonchunhyang University College of Medicine, Cheonan, South Korea 2 Department of Neurology, Dongsan Medical Center, Keimyung University School of Medicine, Daegu, South Korea 3 Department of Preventive Medicine, Soonchunhyang University College of Medicine, Cheonan, South Korea 4 Division of Epilepsy and Sleep Disorders, Department of Neurology, Cheonan Hospital, Soonchunhyang University College of Medicine, Cheonan, South Korea 5 Graduate School of Nursing, Dongsan Medical Center, Keimyung University School of Medicine, Daegu, South Korea
Introduction: There is growing evidence of an increased cardiovascular risk including hypertension in patients with PLMS. In a recent multiethnic cohort study, the association between prevalent hypertension and PLMS varied according to the racial difference. We studied whether PLMS is associated with hypertension in Korean adults. Materials and methods: We investigated 1152 man and woman above 18 years old in two tertiary university hospitals, who had performed in-lab polysomnography (PSG) from 2012 to 2014. The subjects completed a sleep questionnaire prior to PSG study. Individual’s sleep states over the previous 1 month were recorded from their answers to a question. The data were derived from well formed sleep questionnaire-PSG registry. The Epworth Sleepiness Scale – Korean version (KESS), Insomnia Severity Index – Korean version (KISI), Pittsbergh Sleepiness Quality Index – Korean version (KPSQI), and Beck Depression Inventory (BDI), the symptoms of restless legs syndrome (RLS), body mass index (BMI), and the presence of cardiovascular disease were consisted. The presence of hypertension was defined as self-reported, ascertained by a “yes” response to the question, “Have you ever been told by a doctor or health professional that you have hypertension?” We analyzed the association between periodic limb movement index (PLMI), periodic limb movement arousal index (PLMAI) and hypertension. PLMI and PLMAI in three categories were PLMI <5, 5 to <30, and ≥30; and PLMAI <1, 1
S179
to <5, and ≥5. Covariates; age, sex, BMI, RLS, apnea–hypopnea index (AHI), arousal index (AI), and average oxygen saturation were adjusted by stepwise multivariate regression model. Results: The subjects had a mean age of 50.5 ± 14.9 years and 58.3% (n = 671) were men. The PLMI and PLMAI increased with advanced mean age, respectively (p < 0.001). The most of the subjects were in the lowest PLMI and PLMAI categories; 78% with PLMI < 5, 12.6% with 5 to <30, 9.4% with PLMI ≥ 30, 81.1% with PLMAI < 1, 11% with 1 to <5, and 7.9% with PLMAI ≥ 5. Three hundred one (26.1%) subjects had hypertension. Co-incidental hypertension was associated with PLMAI (p = 0.036) but not with PLMI (p = 0.15) in chisquare analysis. Finally, it was not associated with PLMI (p = 0.7) and PLMAI (p = 0.4) after adjusting for all covariates. Conclusion: In a retrospective hospital based study, there was no association between co-incidental hypertension and PLMS in Korean adults. http://dx.doi.org/10.1016/j.sleep.2015.02.465
Correlation between upper airway obstruction sites during sleep video fluoroscopy and natural overnight sleep J. Ahn, W. Lee, H. Kim, J. Kim Bundang Hospital, Seoul National University, South Korea
Introduction: Sleep video fluoroscopy (SVF) is useful to assess dynamic pharyngeal obstruction in obstructive sleep apnea syndrome but it is conducted within several minutes of drug-induced sleep. This study aimed to evaluate the correlation between upper airway obstruction sites within short duration of drug-induced sleep and full overnight natural sleep. Materials and methods: This study included patients who underwent both SVF during midazolam induced short (about 15 minutes) sleep and pharyngeal manometry (PM) during overnight polysomnography (PSG) from 2004 to 2007. PM measures percents of retropalatal and retroglossal obstruction events during respiratory distress events. On the other hand, SVF measures the obstruction level during desaturation events. In SVF, obstruction levels are soft palate, tongue base and both levels. Results: Among a total of 102 patients, there were 94 male and 15 female patients. The mean age was 44.2 ± 10.4 years old. The mean percent of retroglossal obstruction events in PM was 30.5 ± 30.0%. In SVF, there were 57 patients with soft palatal level obstruction, 7 with tongue base level and 38 with both levels. SVF results had a weak correlation (R2 = 0.05, p = 0.035) with PM results while sex, age, body mass index and PSG parameters had no correlation. Among 45 cases of tongue base associated obstruction in SVF, 44 showed retroglossal obstruction events in PM. However, among 57 cases of soft palatal level obstruction, only nine showed absolute retropalatal obstruction events. SVF had a low sensitivity 47.8% and high specificity 90.0%. Conclusion: Although SVF provides information of the obstruction sites, it weakly represents obstruction events during the overnight sleep. In particular, soft palatal level obstruction in SVF could not exclude retroglossal obstruction events in natural sleep. http://dx.doi.org/10.1016/j.sleep.2015.02.466
Nasal chondromesenchymal hamartoma causing sleepdisordered breathing in an infant J. Bae, S. Kim, Y. Park Department of Otorhinolaryngology, School of Medicine, Ewha Womans University, South Korea
S180
Abstracts/Sleep Medicine 16 (2015) S2–S199
Introduction: Infants are vulnerable to sleep-related airway obstruction due to both anatomical and physiological predispositions. In this report, we present a rare case of huge nasal chondromesenchymal hamartoma causing sleep-disordered breathing that was successfully treated with image-guided endoscopic surgery. Materials and methods: A 10-month-old boy presented with a 6-month history of nasal congestion and mouth breathing. Before 1 month, he showed loud snoring, episodes of breathing cessation, irritability, and sternal recession during sleep witnessed by parents,. Computed tomography scan revealed a large, minimally enhancing homogeneous mass in the right nasal cavity. Surgical excision via endoscopic approach was done. Intraoperatively, the mass was solid and appeared to originate from the roof of the right nasal cavity. But it was easily freed from the septum, the nasal floor, the middle turbinate. Resection of the mass was carried out in a piecemeal manner with microdebrider and cutting forceps. No significant bleeding occurred during the operation, and complete resection of the tumor mass was achieved. Results: The recovery of the patient was uneventful, and he was discharged a day later. The patient’s sleep disturbance was completely resolved. Histopathologic examination showed multiple, irregular islands of chondroid tissue within spindle cell pattern with occasional binucleated mesenchymal cell proliferation. The spindle cells possessed reactivity for vimentin, S-100 protein, smooth muscle actin, epithelial membrane antigen, and myelin basic protein were negative in immunohistochemical studies. At a 12-month follow-up, there was no evidence of recurrence. Conclusion: Huge nasal chondromesenchymal hamartoma is an extremely rare condition that can account for sleep-disordered breathing in infants. http://dx.doi.org/10.1016/j.sleep.2015.02.467
Acoustic analysis of whole night snoring events in habitual snoring children J. Bae 1, H. Lee 2, S. Kim 1, Y. Park 1 1 Department of Otorhinolaryngology, School of Medicine, Ewha Womans University, South Korea 2 Department of Neurology, School of Medicine, Ewha Womans University, South Korea
Introduction: Habitual snoring, a symptom that may indicate the presence of the OSA, is also common in children. The exact mechanism and etiology remains still unclear. We aimed to record and analyze whole night snoring events in these patients. The correlation between clinical parameters and snoring pattern also was investigated. Materials and methods: Twenty-one pediatric patients with a chief complaint of habitual snoring participated in this study. Demographic data, clinical examination including oral and nasal endoscopy, and skull lateral view were obtained. Overnighit PSG in the sleep laboratory or portable home PSG data were obtained for diagnosis of potential sleep-disordered breathing. The recording of the snoring sounds were performed by two independent digital recording systems. The intensities of the recorded snoring sounds, the frequency power spectrum was created by fast Fourier transformation. Average intensity and power spectral density parameters were calculated, the sound segments are classified into normal breath, snore, and apnea segments. A correlation analysis was done between full night breathing sound, PSG data, and clinical data. Results: The mean age of participated was 6.4 (from 3.5 to 8 years old), body mass index was 13.4, Brodsky tonsil size was 3.4, pulse oximetry saturation level was 94%, and AHI was 17.2. The average
intensity of snoring sound and proportion of apnea segments were associated with AHI, tonsil and adenoid size, BMI. In particular, tonsil size was strongly correlated with snoring sound intensity. Conclusion: In habitual snoring children, including tonsil and adenoid hypertrophy patients, acoustic analysis of snoring sounds seems to be a promising additional diagnostic tool by itself. http://dx.doi.org/10.1016/j.sleep.2015.02.468
Effects of endocannabinoids system on glucose metabolism of chronic intermittent hypoxia rats W. Bei The Second Hospital of Shanxi Medical University, China
Introduction: This experiment by CIH of OSAHS model simulation of pathophysiological changes in rats, which ECS disorder in CIH-induced glucose metabolic disorders play a role in the mechanism are discussed. Materials and methods: Forty-eight rats were divided into groups by random permutation table method: normal control group (which were filled with compressed 21% air oxygen concentration), intermittent hypoxia group (which were given intermittent hypoxic exposure every day 8 h), intermittent hypoxia + rimonabant intervention group [which were given intraperitoneal injection of rimonabant (1 mg/kg/d) before the exposure]. This experiment ended after 4 and 6 weeks, then the level of fasting plasma glucose, insulin, C-peptide and the expression of ECS receptor CB1 in rats of each group were tested, and then evaluated CIH on the indexes of glucose metabolic abnormalities. Results: The results showed that the level of fasting plasma glucose, insulin, C-peptide and the expression of ECS receptor CB1 in rats of each intermittent hypoxia groups were significantly increased, as compared with the level of those quotas of the normal controls (p < 0.05). These measured indexes increased more (p < 0.05) when the time of intermittent hypoxia was extended and after giving rimonabant intervention; the above indexes were reduced significantly (p < 0.05). The level of CB1 was significantly positively correlated with the level of fasting plasma glucose, insulin, C-peptide (r = 0.856, 0.758, 0.827, p < 0.05). Conclusion: Endocannabinoids system (ECS) may play a role in the abnormal glucose metabolism caused by CIH. http://dx.doi.org/10.1016/j.sleep.2015.02.469
Study on assessing subjective sleep quality in patients with obstructive sleep apnea syndrome and its effective factors Q. Cai 1, J. Xu 2 1 The Tumor Hospital of Xinjiang, China 2 Urumqi General Hospital of Lanzhou Command of PLA, China
Introduction: To assess the subjective sleep quality in patients with obstructive sleep apnea syndrome, and investigate the effect of the influencing factors on subjective sleep quality of the patients. Materials and methods: This study used a descriptive survey; 190 of the patients were inquired questionnaire type survey, which including: general condition of the patient, the Pittsburgh sleep quality index scale (PSQI) assessment of subjective sleep quality and self rating Depression Scale (SDS) assessment of depression symptoms, each patient after all night polysomnography and sleep status data acquisition.
discontinuation of pramipexole medication. The sleep study was repeated after the re-continuation of pramipexole. Leg movements were marked manually and PWA dips were detected using a custom script. Temporal relationship between leg movements and transient PWA phenomena was assessed both during wakefulness and sleep. Events with marked disturbance in the plethysmogram signal were excluded from the analysis. Results: A total of 2606 leg movements were included in the analysis. We found that half of the patients had transient dips in PWA following the start of the leg movements. In these patients, the PWA dipped by an average of 24% (range 12–30%) during sleep and 12% (range 6–20%) during wakefulness. PWA phenomena did not herald the leg movements but started 2–10 seconds after the movement. Most commonly, the dip lasted for 10–20 seconds. The other half of the patients did not show similar dips. When present, both leg movements and PWA dips were suppressed by pramipexole. Conclusion: The results suggest that PLM are associated with transient peripheral vasoconstriction in some patients with RLS. The vasoconstriction is in tight temporal relationship with the leg movement. Pramipexole suppresses both PLM and the associated transient vasoconstriction. http://dx.doi.org/10.1016/j.sleep.2015.02.464
Association of co-incidental hypertension with periodic limb movements during sleep in hospital based cross-sectional study K. Yang 1, Y. Cho 2, Y. Hwangbo 3, D. Kim 4, M. Song 5 1 Division of Epilepsy and Sleep Disorders, Department of Neurology, Soonchunhyang University College of Medicine, Cheonan, South Korea 2 Department of Neurology, Dongsan Medical Center, Keimyung University School of Medicine, Daegu, South Korea 3 Department of Preventive Medicine, Soonchunhyang University College of Medicine, Cheonan, South Korea 4 Division of Epilepsy and Sleep Disorders, Department of Neurology, Cheonan Hospital, Soonchunhyang University College of Medicine, Cheonan, South Korea 5 Graduate School of Nursing, Dongsan Medical Center, Keimyung University School of Medicine, Daegu, South Korea
Introduction: There is growing evidence of an increased cardiovascular risk including hypertension in patients with PLMS. In a recent multiethnic cohort study, the association between prevalent hypertension and PLMS varied according to the racial difference. We studied whether PLMS is associated with hypertension in Korean adults. Materials and methods: We investigated 1152 man and woman above 18 years old in two tertiary university hospitals, who had performed in-lab polysomnography (PSG) from 2012 to 2014. The subjects completed a sleep questionnaire prior to PSG study. Individual’s sleep states over the previous 1 month were recorded from their answers to a question. The data were derived from well formed sleep questionnaire-PSG registry. The Epworth Sleepiness Scale – Korean version (KESS), Insomnia Severity Index – Korean version (KISI), Pittsbergh Sleepiness Quality Index – Korean version (KPSQI), and Beck Depression Inventory (BDI), the symptoms of restless legs syndrome (RLS), body mass index (BMI), and the presence of cardiovascular disease were consisted. The presence of hypertension was defined as self-reported, ascertained by a “yes” response to the question, “Have you ever been told by a doctor or health professional that you have hypertension?” We analyzed the association between periodic limb movement index (PLMI), periodic limb movement arousal index (PLMAI) and hypertension. PLMI and PLMAI in three categories were PLMI <5, 5 to <30, and ≥30; and PLMAI <1, 1
S179
to <5, and ≥5. Covariates; age, sex, BMI, RLS, apnea–hypopnea index (AHI), arousal index (AI), and average oxygen saturation were adjusted by stepwise multivariate regression model. Results: The subjects had a mean age of 50.5 ± 14.9 years and 58.3% (n = 671) were men. The PLMI and PLMAI increased with advanced mean age, respectively (p < 0.001). The most of the subjects were in the lowest PLMI and PLMAI categories; 78% with PLMI < 5, 12.6% with 5 to <30, 9.4% with PLMI ≥ 30, 81.1% with PLMAI < 1, 11% with 1 to <5, and 7.9% with PLMAI ≥ 5. Three hundred one (26.1%) subjects had hypertension. Co-incidental hypertension was associated with PLMAI (p = 0.036) but not with PLMI (p = 0.15) in chisquare analysis. Finally, it was not associated with PLMI (p = 0.7) and PLMAI (p = 0.4) after adjusting for all covariates. Conclusion: In a retrospective hospital based study, there was no association between co-incidental hypertension and PLMS in Korean adults. http://dx.doi.org/10.1016/j.sleep.2015.02.465
Correlation between upper airway obstruction sites during sleep video fluoroscopy and natural overnight sleep J. Ahn, W. Lee, H. Kim, J. Kim Bundang Hospital, Seoul National University, South Korea
Introduction: Sleep video fluoroscopy (SVF) is useful to assess dynamic pharyngeal obstruction in obstructive sleep apnea syndrome but it is conducted within several minutes of drug-induced sleep. This study aimed to evaluate the correlation between upper airway obstruction sites within short duration of drug-induced sleep and full overnight natural sleep. Materials and methods: This study included patients who underwent both SVF during midazolam induced short (about 15 minutes) sleep and pharyngeal manometry (PM) during overnight polysomnography (PSG) from 2004 to 2007. PM measures percents of retropalatal and retroglossal obstruction events during respiratory distress events. On the other hand, SVF measures the obstruction level during desaturation events. In SVF, obstruction levels are soft palate, tongue base and both levels. Results: Among a total of 102 patients, there were 94 male and 15 female patients. The mean age was 44.2 ± 10.4 years old. The mean percent of retroglossal obstruction events in PM was 30.5 ± 30.0%. In SVF, there were 57 patients with soft palatal level obstruction, 7 with tongue base level and 38 with both levels. SVF results had a weak correlation (R2 = 0.05, p = 0.035) with PM results while sex, age, body mass index and PSG parameters had no correlation. Among 45 cases of tongue base associated obstruction in SVF, 44 showed retroglossal obstruction events in PM. However, among 57 cases of soft palatal level obstruction, only nine showed absolute retropalatal obstruction events. SVF had a low sensitivity 47.8% and high specificity 90.0%. Conclusion: Although SVF provides information of the obstruction sites, it weakly represents obstruction events during the overnight sleep. In particular, soft palatal level obstruction in SVF could not exclude retroglossal obstruction events in natural sleep. http://dx.doi.org/10.1016/j.sleep.2015.02.466
Nasal chondromesenchymal hamartoma causing sleepdisordered breathing in an infant J. Bae, S. Kim, Y. Park Department of Otorhinolaryngology, School of Medicine, Ewha Womans University, South Korea
S180
Abstracts/Sleep Medicine 16 (2015) S2–S199
Introduction: Infants are vulnerable to sleep-related airway obstruction due to both anatomical and physiological predispositions. In this report, we present a rare case of huge nasal chondromesenchymal hamartoma causing sleep-disordered breathing that was successfully treated with image-guided endoscopic surgery. Materials and methods: A 10-month-old boy presented with a 6-month history of nasal congestion and mouth breathing. Before 1 month, he showed loud snoring, episodes of breathing cessation, irritability, and sternal recession during sleep witnessed by parents,. Computed tomography scan revealed a large, minimally enhancing homogeneous mass in the right nasal cavity. Surgical excision via endoscopic approach was done. Intraoperatively, the mass was solid and appeared to originate from the roof of the right nasal cavity. But it was easily freed from the septum, the nasal floor, the middle turbinate. Resection of the mass was carried out in a piecemeal manner with microdebrider and cutting forceps. No significant bleeding occurred during the operation, and complete resection of the tumor mass was achieved. Results: The recovery of the patient was uneventful, and he was discharged a day later. The patient’s sleep disturbance was completely resolved. Histopathologic examination showed multiple, irregular islands of chondroid tissue within spindle cell pattern with occasional binucleated mesenchymal cell proliferation. The spindle cells possessed reactivity for vimentin, S-100 protein, smooth muscle actin, epithelial membrane antigen, and myelin basic protein were negative in immunohistochemical studies. At a 12-month follow-up, there was no evidence of recurrence. Conclusion: Huge nasal chondromesenchymal hamartoma is an extremely rare condition that can account for sleep-disordered breathing in infants. http://dx.doi.org/10.1016/j.sleep.2015.02.467
Acoustic analysis of whole night snoring events in habitual snoring children J. Bae 1, H. Lee 2, S. Kim 1, Y. Park 1 1 Department of Otorhinolaryngology, School of Medicine, Ewha Womans University, South Korea 2 Department of Neurology, School of Medicine, Ewha Womans University, South Korea
Introduction: Habitual snoring, a symptom that may indicate the presence of the OSA, is also common in children. The exact mechanism and etiology remains still unclear. We aimed to record and analyze whole night snoring events in these patients. The correlation between clinical parameters and snoring pattern also was investigated. Materials and methods: Twenty-one pediatric patients with a chief complaint of habitual snoring participated in this study. Demographic data, clinical examination including oral and nasal endoscopy, and skull lateral view were obtained. Overnighit PSG in the sleep laboratory or portable home PSG data were obtained for diagnosis of potential sleep-disordered breathing. The recording of the snoring sounds were performed by two independent digital recording systems. The intensities of the recorded snoring sounds, the frequency power spectrum was created by fast Fourier transformation. Average intensity and power spectral density parameters were calculated, the sound segments are classified into normal breath, snore, and apnea segments. A correlation analysis was done between full night breathing sound, PSG data, and clinical data. Results: The mean age of participated was 6.4 (from 3.5 to 8 years old), body mass index was 13.4, Brodsky tonsil size was 3.4, pulse oximetry saturation level was 94%, and AHI was 17.2. The average
intensity of snoring sound and proportion of apnea segments were associated with AHI, tonsil and adenoid size, BMI. In particular, tonsil size was strongly correlated with snoring sound intensity. Conclusion: In habitual snoring children, including tonsil and adenoid hypertrophy patients, acoustic analysis of snoring sounds seems to be a promising additional diagnostic tool by itself. http://dx.doi.org/10.1016/j.sleep.2015.02.468
Effects of endocannabinoids system on glucose metabolism of chronic intermittent hypoxia rats W. Bei The Second Hospital of Shanxi Medical University, China
Introduction: This experiment by CIH of OSAHS model simulation of pathophysiological changes in rats, which ECS disorder in CIH-induced glucose metabolic disorders play a role in the mechanism are discussed. Materials and methods: Forty-eight rats were divided into groups by random permutation table method: normal control group (which were filled with compressed 21% air oxygen concentration), intermittent hypoxia group (which were given intermittent hypoxic exposure every day 8 h), intermittent hypoxia + rimonabant intervention group [which were given intraperitoneal injection of rimonabant (1 mg/kg/d) before the exposure]. This experiment ended after 4 and 6 weeks, then the level of fasting plasma glucose, insulin, C-peptide and the expression of ECS receptor CB1 in rats of each group were tested, and then evaluated CIH on the indexes of glucose metabolic abnormalities. Results: The results showed that the level of fasting plasma glucose, insulin, C-peptide and the expression of ECS receptor CB1 in rats of each intermittent hypoxia groups were significantly increased, as compared with the level of those quotas of the normal controls (p < 0.05). These measured indexes increased more (p < 0.05) when the time of intermittent hypoxia was extended and after giving rimonabant intervention; the above indexes were reduced significantly (p < 0.05). The level of CB1 was significantly positively correlated with the level of fasting plasma glucose, insulin, C-peptide (r = 0.856, 0.758, 0.827, p < 0.05). Conclusion: Endocannabinoids system (ECS) may play a role in the abnormal glucose metabolism caused by CIH. http://dx.doi.org/10.1016/j.sleep.2015.02.469
Study on assessing subjective sleep quality in patients with obstructive sleep apnea syndrome and its effective factors Q. Cai 1, J. Xu 2 1 The Tumor Hospital of Xinjiang, China 2 Urumqi General Hospital of Lanzhou Command of PLA, China
Introduction: To assess the subjective sleep quality in patients with obstructive sleep apnea syndrome, and investigate the effect of the influencing factors on subjective sleep quality of the patients. Materials and methods: This study used a descriptive survey; 190 of the patients were inquired questionnaire type survey, which including: general condition of the patient, the Pittsburgh sleep quality index scale (PSQI) assessment of subjective sleep quality and self rating Depression Scale (SDS) assessment of depression symptoms, each patient after all night polysomnography and sleep status data acquisition.