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Sleep terrors are a non-rapid eye movement (NREM) sleep–related parasomnia characterized by abrupt terror with a frightening scream. However, it remains unknown whether sleep terrors occur only from NREM sleep. We evaluated 3 cases of a sleep terrors–like parasomnia that occurred exclusively during arousals from rapid eye movement (REM) sleep. These parasomnia events occurred at REM sleep-wake transitions, manifesting with screaming or crying, similar to sleep terrors, without alertness or clear orientation. The patients were all young-adult females without notable medical conditions. REM sleep behavior disorder and nocturnal frontal lobe epilepsy were not detected based on their video-polysomnographic findings. These 3 cases should be provisionally diagnosed as “Parasomnia, Unspecified” according to the International Classification of Sleep Disorders, third edition; however, the phenomenological diagnosis is proposed to be “Disorders of Arousal from REM Sleep.” Our reported cases indicate that sleep terrors may also arise from REM sleep. CITATION: Futenma K, Inoue Y, Saso A, Takaesu Y, Yamashiro Y, Matsuura M. Three cases of parasomnias similar to sleep terrors occurring during sleep-wake transitions from REM sleep. J Clin Sleep Med. 2022;18(2):669–675.

The prevalence of sleep-disordered breathing (SDB) is reportedly very high. Among SDBs, the incidence of obstructive sleep apnea (OSA) is higher than previously believed, with patients having moderate-to-severe OSA accounting for approximately 20% of adult males and 10% of postmenopausal women not only in Western countries but also in Eastern countries, including Japan. Since 1998, when health insurance coverage became available, the number of patients using continuous positive airway pressure (CPAP) therapy for sleep apnea has increased sharply, with the number of patients about to exceed 500,000 in Japan. Although the “Guidelines for Diagnosis and Treatment of Sleep Apnea Syndrome (SAS) in Adults” was published in 2005, a new guideline was prepared to indicate the standard medical care based on the latest trends, as supervised by and in cooperation with the Japanese Respiratory Society and the “Survey and Research on Refractory Respiratory Diseases and Pulmonary Hypertension” Group, of Ministry of Health, Labor and Welfare and other related academic societies, including the Japanese Society of Sleep Research, in addition to referring to the previous guidelines. Since sleep apnea is an interdisciplinary field covering many areas, this guideline was prepared including 36 clinical questions (CQs). In the English version, therapies and managements for SAS, which were written from CQ16 to 36, were shown. The Japanese version was published in July 2020 and permitted as well as published as one of the Medical Information Network Distribution Service (Minds) clinical practice guidelines in Japan in July 2021.

(Pro)renin receptor ((P)RR), a receptor for renin and prorenin, is implicated in the pathophysiology of diabetes mellitus, hypertension and their complications. Soluble (P)RR (s(P)RR) is composed of extracellular domain of (P)RR and thus exists in blood. We have reported that plasma concentrations of s(P)RR were elevated in male patients with obstructive sleep apnea syndrome (OSAS). The aim of the present study was to clarify the difference in plasma s(P)RR concentrations between male and female OSAS patients. Plasma s(P)RR concentrations were studied in 289 subjects (206 males and 83 females) consisting of 259 OSAS patients and 30 non-OSAS control subjects. The 259 OSAS patients were classified into mild (5 ≤ apnea hypopnea index (AHI) < 15 events/h), moderate (15 ≤ AHI < 30), and severe OSAS (AHI ≥ 30). Plasma s(P)RR levels were significantly elevated in all three OSAS groups compared to non-OSAS control subjects (AHI < 5) in the entire cohort and male subjects, whereas in female subjects, the significant elevation was found only in severe OSAS. Plasma s(P)RR levels were significantly correlated with AHI in both sexes, with a higher r value found in male subjects (male r = 0.413, p < 0.0001; female r = 0.263, p < 0.05). Importantly, when OSAS patients (26 males and 15 females) with AHI ≥ 20 underwent continuous positive airway pressure treatment, plasma s(P)RR levels were significantly decreased. In conclusion, plasma s(P)RR levels are elevated in both male and female OSAS patients in parallel with the disease severity.

This double-blind study evaluated the efficacy and safety of modafinil for treating excessive daytime sleepiness in Japanese patients with obstructive sleep apnea syndrome (OSAS).Patients with residual excessive sleepiness (Epworth Sleepiness Scale [ESS] ≥ 11) on optimal nasal continuous positive airway pressure (nCPAP) therapy (apnea-hypopnea index ≤ 10) were randomized to either 200 mg modafinil (n = 52) or placebo (n = 62) once daily for 4 weeks. Outcomes included baseline-week 4 changes in ESS total score, sleep latency on maintenance of wakefulness test (SL-MWT), nocturnal polysomnography, Pittsburgh Sleep Quality Index (PSQI), and safety.All 114 randomized patients completed the study. Mean change in ESS total score (-6.6 vs -2.4, p0.001) and SL-MWT (+2.8 vs -0.4 minutes, p = 0.009) were significantly greater with modafinil than with placebo. ESS total score decreased from11 to11 at the final assessment in 69.2% of modafinil-treated patients and 30.6% of placebo-treated patients (p0.001). Corresponding rates at week 1 were 57.7% and 33.9% (p = 0.014). Changes in nocturnal polysomnography, PSQI, and apnea-hypopnea index from baseline to the final assessment were similar in both groups. Adverse drug reactions occurred in 36.5% and 22.6% of patients in the modafinil and placebo groups, respectively (p = 0.146).Once-daily modafinil was effective and well tolerated for managing residual daytime sleepiness in Japanese OSAS patients with residual excessive daytime sleepiness on optimal nCPAP therapy.

Craniofacial morphological anomalies can be divided into two principal categories: skeletal anomalies and soft tissue anomalies. This study examined the hypothesis that the assessment of indices representing both skeletal and soft tissue can be used to appropriately identify the risk factor of obstructive sleep apnea–hypopnea syndrome (OSAHS). 232 suspected OSAHS male patients were examined with polysomnography and divided into two groups (202 males with OSAHS and 30 male controls without OSAHS). Cephalometric analysis was performed on all patients to evaluate craniofacial morphological anomalies. The measurement sites were as follows: skeletal morphology; soft tissue morphology; mixed morphology including mandibular plane to hyoid bone (MP-H); and jaw soft tissue (JS) ratio; a novel ratio we defined, between the area of jaw and area of tongue with soft palate. JS ratio increased with AHI as well as MP-H. MP-H and JS ratio showed significant but weak correlation with apnea–hypopnea index. JS ratio was significantly associated with an increased risk for severe OSAHS, even after adjusting age and BMI, its odds ratio was the greatest among these variables. These results showed that mixed craniofacial, skeletal and soft tissue morphology are correlated with AHI, and JS ratio may be a useful parameters to explain the characteristics of OSAHS in male patients.

Sleep-disordered breathing is commonly associated with periodic limb movements (PLMS). Nasal continuous positive airway pressure (nCPAP) is the most widely used treatment for sleep-disordered breathing. However, it is not clear whether nCPAP treatment of apnea also has a systematic effect on PLMS. We studied 15 patients with sleep-disordered breathing and PLMS in a split-night protocol in order to confirm the acute effects of nCPAP on PLMS. Although the PLMS index (PLMSI) did not change statistically (baseline, 38.7 +/- 20.5; CPAP, 31.3 +/- 17.0; p = ns), the PLMS-related index (PLMS-ArI) decreased significantly on nCPAP (baseline, 17.8 +/- 10.1; CPAP, 9.2 +/- 5.7; p < 0.05). Whether the reduced PLMS-ArI is sustained with chronic nCPAP is unknown and a matter of future investigation.

Craniofacial morphological anomalies can be divided into two principal categories: skeletal anomalies and soft tissue anomalies. This study examined the hypothesis that the assessment of indices representing both skeletal and soft tissue can be used to appropriately identify the risk factor of obstructive sleep apnea–hypopnea syndrome (OSAHS). 232 suspected OSAHS male patients were examined with polysomnography and divided into two groups (202 males with OSAHS and 30 male controls without OSAHS). Cephalometric analysis was performed on all patients to evaluate craniofacial morphological anomalies. The measurement sites were as follows: skeletal morphology; soft tissue morphology; mixed morphology including mandibular plane to hyoid bone (MP-H); and jaw soft tissue (JS) ratio; a novel ratio we defined, between the area of jaw and area of tongue with soft palate. JS ratio increased with AHI as well as MP-H. MP-H and JS ratio showed significant but weak correlation with apnea–hypopnea index. JS ratio was significantly associated with an increased risk for severe OSAHS, even after adjusting age and BMI, its odds ratio was the greatest among these variables. These results showed that mixed craniofacial, skeletal and soft tissue morphology are correlated with AHI, and JS ratio may be a useful parameters to explain the characteristics of OSAHS in male patients.

Background: Because increased sympathetic nervous activity (SNA) in patients with dilated cardiomyopathy (DCM) associated with sleep breathing disorder (SBD) is known to deteriorate the prognosis of cardiac failure, 123I-metaiodobenzylguanidine (MIBG) myocardial scintigraphy was used as the investigative tool in the present study. Methods and Results: The study group comprised 53 patients (47 men, 6 women; mean age 56 ±3 years) with chronic stable DCM. Patients were divided into SBD(+) or SBD(-) group according to 24-h pulse oximetry results. SBD(+) was defined when the 3% oxygen desaturation index was more than 15/h during sleep. In total, 32 patients were SBD(-) and 21 were SBD(+). In both groups, pulse oximetry were performed during sleep and awakening pulse rate, and measurement of the blood levels of catecholamines and B-type natriuretic peptide was performed. MIBG myocardial scintigraphy and echocardiography were performed at the same time. No significant difference was found between the 2 groups in catecholamine levels or left ventricular ejection fraction. However, MIBG had a significantly increased washout rate and a significantly decreased delayed heart to mediastinum ratio in the SBD(+) group compared with the SBD(-) group. Conclusions: SNA is increased in DCM patients when associated with SBD. MIBG myocardial scintigraphy may be a sensitive method of detecting increased SNA. (Circ J 2009; 73: 686 - 690)

Background Obesity and craniofacial abnormalities such as small maxilla and mandible are common features of patients with obstructive sleep apnea (OSA). The authors hypothesized that anatomical imbalance between the upper airway soft-tissue volume and the craniofacial size (rather than each alone) may result in pharyngeal airway obstruction during sleep, and therefore development of OSA. Methods Blind measurements of tongue cross-sectional area and craniofacial dimensions were performed through lateral cephalograms in 50 adult male patients with OSA and 55 adult male non-OSA subjects with various craniofacial dimensions. Results Maxillomandibular dimensions were matched between OSA and non-OSA groups. While the tongue was significantly larger in subjects with larger maxillomandible dimensions, OSA patients had a significantly larger tongue for a given maxillomandible size than non-OSA subjects. The hypothesis was also supported in subgroups matched for both body mass index and maxillomandible dimensions. Conclusions Upper airway anatomical imbalance is involved in the pathogenesis of OSA.

We report a case of sepsis caused by Escherichia coli (E. coli) of serotype O-143. A 78-year-old man developed symptoms of gastroenteritis after ingesting raw meat on noodles. He rapidly developed respiratory failure. Blood culture grew E. coli. The sepsis seemed to have directly spread from a food-borne infection. The development of primary sepsis after ingesting E. coli is very rare. We suspect that bacterial translocation played a major role. Serotype O-143 is recognized in enteroinvasive E. coli (EIEC) as well as in Shigella dysenteriae. The pathogen in the present case is suspected of being EIEC although the isolated E. coli strain was negative for the inv and ipa genes.

The objective of this study was to examine the usefulness of home oximetry for the screening of sleep disorders presenting with excessive daytime sleepiness (EDS). This was accomplished by blinded comparison of diagnosis by oximetry alone versus polysomnographic diagnosis carried out at a sleep disorders center at a tertiary referral hospital. This study included three hundred patients who had been referred because of EDS and suspected sleep apnea. A number of measurements were made. The arterial oxygen saturation (SaO2) data were sampled at 2 Hz and stored digitally during polysomnography (PSG). From the SaO2 data recorded onto paper six scorers calculated the number of desaturations3% per hour (desaturation index: DI) and then made a diagnosis [normal, DI5; mild obstructive sleep apnea (OSA), 5DI20; moderate OSA, 20DI40; severe OSA, DI40]. Upper airway resistance syndrome (UARS) was diagnosed when DI was5 but associated with small fluctuations in SaO2. The diagnosis made by each of six scorers was compared to the clinical diagnosis made independently using PSG. Thirty-one (10.3%) of all the records were rejected by scorers because of inadequate SaO2 signals requiring technologist intervention. Sensitivity of screening for sleep-breathing disorders was 90.0% and specificity was 75.0%. All moderate and severe OSA patients were detected by oximetry. However, among the 66 patients who were classified as normal by oximetry, 1 had mild OSA, 20 had UARS, 9 had periodic limb movements in sleep, 4 had narcolepsy and 2 had a parasomnia. In conclusion, home oximetry may not have sufficient sensitivity and specificity to detect breathing disorders reliably during sleep and is useless for other disorders of sleep.

We hypothesize that breathing disorder related arousal index (B-ArI) can differentiate sleep breathing disorder from simple snorer when apnea hypopnea index (AHI) is low. We studied 54 patients using polysomnography. Breathing disorder related arousal (B-Ar) was defined as arousal accompanied by apnea, hypopnea, desaturation or snort. Mean AHI was 44.2+/-34.0/h, and B-ArI correlated significantly with AHI, desaturation index, percentage total sleep time with SpO2 below 90%. Breathing disorder related arousal index was greater than AHI when AHI was below 20. In 11/54 patients, AHI was below 10, and B-ArI more than 10. Nasal continuous positive airway pressure was applied to six patients and showed reduced B-ArI. Breathing disorder related arousal index may be one of the useful indices for the diagnosis of sleep breathing disorder when AHI is less than 10.