Sleep-Disordered Breathing in the Context of Pulmonary Hypertension in Pediatric Patients with Co-Morbid Conditions: Case Series and Review of the Literature

Abstract Introduction Turner syndrome (TS) is a common genetic disorder that affects phenotypic females with partial or complete absence of one X chromosome. It typically presents with characteristic facial appearance, neck webbing, lymphedema, linear growth failure, and ovarian insufficiency. TS is also associated with other disorders, though sleep related disorders are not commonly reported. We present a case series of pediatric patients diagnosed with TS and assess their risk for sleep disordered breathing. Methods This study utilized retrospective chart review of the electronic medical record at the University of North Carolina at Chapel Hill from April 2014 to January 2019. Only pediatric patients under the age of 18 years who had previously undergone polysomnography and carrying the diagnosis of Turner syndrome were included in this study. Polysomnography results were reviewed. Results Retrospective chart analysis yielded ten (10) patients who qualified for inclusion. The mean age was 8.3 years (age range 1-15 years). Nine (9) patients were found to have sleep disordered breathing ranging from upper airway resistance syndrome to moderate sleep apnea (AHI range 1.2 to 6.2). Six (6) patients were found to have elevated periodic limb movement indices (PLM index range 5.1 to 30). Parasomnias and hypoventilation were not seen. Conclusion Our case series illustrates that sleep disordered breathing may be more common in TS than previously realized. Eklund et al. found that females with TS had more retrognathic mandibles and maxillas, shorter mandibles, and larger cranial base angles. These findings may indicate elevated risk of sleep apnea. Further studies are needed to define the overall risk of sleep disordered breathing in TS. Support None.

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Impact of Tonsillectomy on Obesity in Pediatric Patients With Sleep-Disordered Breathing

OTO Open ◽

10.1177/2473974x211059105 ◽

2021 ◽

Vol 5 (4) ◽

pp. 2473974X2110591

Author(s):

Rohith S. Voora ◽

Daniela Carvalho ◽

Wen Jiang

Keyword(s):

Sleep Disordered Breathing ◽

Weight Status ◽

Pediatric Patients ◽

Case Series ◽

Surgical Indication ◽

Strongly Correlated ◽

Retrospective Case ◽

Academic Center ◽

Disordered Breathing

Objective Both tonsillar hypertrophy and obesity contribute to pediatric sleep-disordered breathing (SDB). Tonsillectomy addresses anatomical obstruction causing SDB; however, it may adversely affect the obesity profile postoperatively. Herein, we investigate posttonsillectomy body mass index (BMI) changes in pediatric patients. Study Design Retrospective case series. Setting Tertiary, pediatric urban academic center. Methods All patients undergoing tonsillectomy from January 1, 2016, to December 31, 2016, were included. Patients’ age, sex, surgical indication, and preoperative BMI were recorded. Postoperative BMI data were collected between March 1, 2016, and December 31, 2017. Statistical analysis was performed using a generalized regression model, using BMI percentile-for-age weight status. Results A total of 1153 patients were included (50% female), with age ranging from 2.0 to 19.5 years (mean [SD], 7.6 [4.0]). The majority (87.8%) had tonsillectomy for SDB. Of the cohort, 560 (48.6%) had available follow-up BMI data. The BMI percentile on the day of the surgery had a median of 65.8, and the BMI percentile on follow-up had a median of 76.4. The median time to follow-up was 197 days with a range of 50 to 605 days. Higher postoperative BMI percentile strongly correlated to higher preoperative BMI percentile ( P < .001), as well as younger age ( P < .001), male sex ( P = .0005), and SDB as a surgical indication ( P = .003). Conclusion We observed a significant increase in BMI percentile following tonsillectomy, which accounted for a significantly higher proportion of the cohort being classified as overweight or obese postoperatively. These findings necessitate greater preoperative counseling, closer follow-up, and adjunctive measures for obesity management in pediatric patients undergoing tonsillectomy.

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Sleep‐disordered breathing and nocturnal hypoventilation in children with the MECP2 duplication syndrome: A case series and review of the literature

American Journal of Medical Genetics Part A ◽

10.1002/ajmg.a.61790 ◽

2020 ◽

Vol 182 (10) ◽

pp. 2437-2441

Author(s):

Amber Baelen ◽

Louise Verhoustraeten ◽

Sandra Kenis ◽

Marije Meuwissen ◽

An Boudewyns ◽

...

Keyword(s):

Sleep Disordered Breathing ◽

Case Series ◽

Review Of The Literature ◽

Nocturnal Hypoventilation ◽

Mecp2 Duplication Syndrome ◽

Duplication Syndrome ◽

Disordered Breathing

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Masses of the Sternoclavicular Area: Case Series and Review of the Literature

Clinical Medicine Insights Pediatrics ◽

10.1177/11795565211021600 ◽

2021 ◽

Vol 15 ◽

pp. 117955652110216

Author(s):

Parisa Oviedo ◽

Morgan Bliss

Keyword(s):

English Language ◽

Pediatric Patients ◽

Tertiary Care ◽

Case Series ◽

Review Of The Literature ◽

Incision And Drainage ◽

Surgical Interventions ◽

Ganglion Cysts ◽

Icd 10 ◽

Bronchogenic Cysts

Objective: Masses of the sternoclavicular area are rare, and are not well described in the literature. We aim to present a series of patients with masses in this location and to review all reported English language cases of sternoclavicular masses in pediatric patients. Methods: This is a case series of pediatric patients with masses of the sternoclavicular area presenting to a tertiary care pediatric hospital from 2010 through 2017. Data was collected by using ICD-9 and ICD-10 codes to query the electronic medical record. Chart review included age at presentation, mass characteristics, medical and surgical interventions, and pathology results. A review of the literature was then performed. Results: Ten patients with masses overlying the sternoclavicular area were identified. Four patients presented with abscess and were treated with incision and drainage. Three of these patients were then treated with staged excision once infection cleared. Two additional patients were treated with primary excision. Four patients were treated with observation. The most common histopathologic finding was epidermoid. One patient was found to have a dermoid cyst, and 1 had a congenital cartilaginous rest. Conclusion: Epidermoids and dermoids are the most common masses overlying the sternoclavicular area. Controversy remains regarding the embryologic origin of sternoclavicular masses. The differential for masses in this area also includes branchial remnants, bronchogenic cysts, ganglion cysts, or septic arthritis.

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558 A Surface Electrode Adjacent to Vagal Nerve Stimulator Lead Can Aid in Characterizing VNS Mediated Sleep Disordered Breathing

SLEEP ◽

10.1093/sleep/zsab072.556 ◽

2021 ◽

Vol 44 (Supplement_2) ◽

pp. A220-A221

Author(s):

Jeremy Chan ◽

Joanna Wrede

Keyword(s):

Sleep Disordered Breathing ◽

Pediatric Patients ◽

Refractory Epilepsy ◽

Vagal Nerve ◽

Apnea Hypopnea Index ◽

Surface Electrode ◽

Patients Âgés ◽

Vagal Nerve Stimulator ◽

Obstructive Sleep ◽

Disordered Breathing

Abstract Introduction Vagal nerve stimulators (VNS) are a nonpharmacological treatment for patients with refractory epilepsy. The VNS can decrease seizure frequency by over 75% in 40% of pediatric patients with refractory epilepsy. An underrecognized side effect is sleep disordered breathing (SDB). The purpose of this study was to demonstrate how a sensor placed adjacent to the VNS lead can distinguish whether SDB is due to VNS discharge. Methods Five pediatric patients (ages: 5–8) with refractory epilepsy with VNS were referred to our sleep center for concern for SDB. Each patient underwent a polysomnogram (PSG) that included a standard PSG montage with a surface electrode placed adjacent to their left lateral neck to detect VNS discharge. VNS associated apnea hypopnea index (vAHI) was calculated by determining the number of hypopneas and obstructive apneas occurring during VNS discharge. Results Of the 5 patients, three met pediatric criteria for obstructive sleep apnea (OSA). Patient 1 had an obstructive AHI (oAHI) of 21.3 events/hr with a vAHI accounting for 79% of the total (16.8 events/hr), patient 2 had an oAHI of 16.6 events/hr with a vAHI accounting for 57% of the total (9.5 events/hr), and patient 3 had an oAHI of 1.9 events/hr with vAHI accounting for 68% of the total (1.3 events/hr). Because of these findings, the VNS settings of all 3 patients were changed with the goal of reducing SDB due to VNS discharge. Upon repeat PSG, patient 2 had reduced OSA with an oAHI of 3 events/hr, with no events associated with VNS discharge. The remaining 2 patients did not exhibit VNS associated SDB, however, both experienced increased respiratory rate during VNS discharge. Conclusion We demonstrated that a surface electrode adjacent to the VNS is able to temporally co-register VNS discharges and enabled us to directly correlate SDB to VNS stimulation in 3 patients with refractory epilepsy. Because of our findings, we titrated the VNS parameters in all 3 patients, with one showing resolution of VNS associated SDB on repeat PSG. We propose that an added surface electrode to detect VNS discharge be considered as standard practice in PSG studies of patients with VNS. Support (if any):

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Fenestrated Transcatheter ASD Closure in Adults with Diastolic Dysfunction and/or Pulmonary Hypertension: Case Series and Review of the Literature

Congenital Heart Disease ◽

10.1111/chd.12367 ◽

2016 ◽

Vol 11 (6) ◽

pp. 663-671 ◽

Cited By ~ 13

Author(s):

Ayman Abdelkarim ◽

Daniel S. Levi ◽

Bao Tran ◽

Joanna Ghobrial ◽

Jamil Aboulhosn

Keyword(s):

Pulmonary Hypertension ◽

Diastolic Dysfunction ◽

Case Series ◽

Review Of The Literature ◽

Asd Closure

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Parent–child co-sleeping in children with co-morbid conditions and sleep-disordered breathing

Sleep And Breathing ◽

10.1007/s11325-018-1710-y ◽

2018 ◽

Vol 23 (1) ◽

pp. 327-332 ◽

Cited By ~ 1

Author(s):

Lynda Sidhoum ◽

Alessandro Amaddeo ◽

Jorge Olmo Arroyo ◽

Livio De Sanctis ◽

Sonia Khirani ◽

...

Keyword(s):

Sleep Disordered Breathing ◽

Morbid Conditions ◽

Disordered Breathing ◽

Parent Child

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574 Sleep-disordered breathing symptoms in children who co-sleep: Are caregivers better reporters?

SLEEP ◽

10.1093/sleep/zsab072.572 ◽

2021 ◽

Vol 44 (Supplement_2) ◽

pp. A226-A227

Author(s):

Nimra Alvi ◽

Allison Clarke ◽

Pallavi Patwari

Keyword(s):

Sleep Disordered Breathing ◽

Chart Review ◽

Retrospective Chart Review ◽

Caregiver Distress ◽

Inclusion Criteria ◽

Demographic Information ◽

Older Children ◽

Obstructive Sleep ◽

Morbid Conditions ◽

Disordered Breathing

Abstract Introduction Medical consensus advises against co-sleeping for infants to protect against SIDS, but co-sleeping in older children is often dismissed if not associated with caregiver distress. While some families may choose to co-sleep due to cultural, circumstantial, or psychosocial factors, this choice can also be due to medical concerns warranting greater caregiver attention. We aimed to explore characteristics of co-sleeping children referred for sleep disordered breathing and hypothesized that children with polysomnogram confirmed obstructive sleep apnea (OSA) would have higher caregiver-reported sleep disordered breathing symptoms as compared to children without confirmed OSA. Methods Caregivers who accompanied their child for polysomnogram were asked to complete a questionnaire that included sleep-related symptoms of sleep-disordered breathing (snoring, apnea, gasping/choking), restlessness, and parasomnias. Inclusion criteria are age >1.0 years and <18 years, baseline study for sleep disordered breathing, and completed questionnaire. Retrospective chart review included demographic information, BMI, co-morbid conditions, and polysomnogram results. The cohort was divided into 2 groups based on polysomnogram confirmed diagnosis of “snoring” or “OSA”. Results Of 75 co-sleeping children, 27 (36%) had a diagnosis of snoring and 48 (64%) of OSA. The cohort was similar in age, gender, and insurance type for snoring and OSA groups (Average 5.7 +/- 2.6 yrs and 5.4 +/- 2.9 yrs, respectively; 41% and 35% female, respectively; 44% and 50% Medicaid, respectively). Notable differences in the snoring and OSA groups were found with BMI z-score (1.6 +/- 4.6 and 1.0 +/- 1.5, respectively) and absence of co-morbid conditions (44% and 63%, respectively). Regarding reported symptoms, the snoring-group compared to OSA-group had lower report of gasping/choking (19% vs 29%), bedwetting (7% vs 13%), and nightmares (7% vs 15%); and had higher report of movement (74% vs 60%), kicking (48% vs 31%), and startle/jump (30% vs 19%). Conclusion Although we predicted that co-sleeping would be associated with increased caregiver vigilance, witnessed sleep-disordered-breathing symptoms was only higher for report of gasping/choking and did not differ significantly for report of snoring and apnea in children with and without OSA. Interestingly, co-sleeping in children without OSA appeared to be more strongly related to report of sleep disruption in the form of restless sleep. Support (if any):

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The effect of lateral head position on sleep disordered breathing: A case series

Sleep Medicine ◽

10.1016/j.sleep.2012.10.008 ◽

2013 ◽

Vol 14 (2) ◽

pp. 220-221 ◽

Cited By ~ 2

Author(s):

Sandeep Riar ◽

Sushanth Bhat ◽

Besher Kabak ◽

Divya Gupta ◽

Irving Smith ◽

...

Keyword(s):

Sleep Disordered Breathing ◽

Case Series ◽

Head Position ◽

Lateral Head ◽

Disordered Breathing

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Sleep-Disordered Breathing in 2 Pediatric Patients on Peritoneal Dialysis

Peritoneal Dialysis International ◽

10.3747/pdi.2014.00205 ◽

2016 ◽

Vol 36 (1) ◽

pp. 109-112 ◽

Cited By ~ 1

Author(s):

Neha Sharma ◽

Elizabeth Harvey ◽

Reshma Amin

Keyword(s):

Quality Of Life ◽

Chronic Kidney Disease ◽

Sleep Disordered Breathing ◽

Pediatric Patients ◽

Central Sleep Apnea ◽

Cognitive Health ◽

Nocturnal Hypoventilation ◽

Underlying Mechanisms ◽

Disordered Breathing

Sleep-disordered breathing (SDB) is prevalent in children with chronic kidney disease (CKD), and has the potential to worsen vascular and neuro-cognitive health and quality of life. We present 2 children with CKD who experience central sleep apnea and nocturnal hypoventilation and discuss the possible underlying mechanisms in relation to CKD and dialysis.

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