bluuebunny/crossref_metadata_2025_split huggingface dataset:
1027 Title: Hypersomnolence following resection of a craniopharyngioma
Authors: Rehman Sofiya , Khan Safia , Carter Gregory , Luong Shan 
Published: May 2023

Abstract

Introduction
Craniopharyngiomas are rare tumors that arise from Rathke’s pouch remnants along the pituitary-hypothalamic axis. The tumor and its treatment can lead to significant neurological and endocrinological complications including alterations in appetite, endocrine insufficiency, circadian rhythms and sleep. We report the case of a woman who developed hypersomnia following resection of a craniopharyngioma

Report of case(s)
This 38-year-old female with neither significant past medical history of sleep, medical, neurological, or psychiatric disorders nor a family history of narcolepsy presented with galactorrhea and amenorrhea. Imaging showed a pituitary tumor. She underwent resection of the tumor with a tissue diagnosis of an adamantinomatous craniopharyngioma. MRI post procedure showed post-surgical changes without new lesions. Diabetes insipidus and panhypopituitarism developed for which appropriate replacement therapy was initiated. She gained approximately one hundred pounds in the weeks post-surgery. Her family described new onset snoring in supine sleeping position and witnessed apneas. She denied hypnagogic hallucinations, cataplexy, or sleep paralysis. She presented to the sleep medicine clinic a few months later for evaluation of persistent hypersomnolence.

Conclusion
The most common sleep disorders post craniopharyngioma resection are sleep-related breathing disorder, hypersomnia, and circadian rhythm disorder. Sleep and appetite are felt to be altered because of direct surgical compression, injury or vascular compromise of structures involved in the control of sleep, wakefulness, and appetite. This can produce hypothalamic dysfunction induced obesity with secondary sleep-related breathing disorder and worsening hypersomnolence. In this woman the temporal relationship between the surgery and the onset of symptoms is most suggestive of this hypothalamic injury, resulting in abnormal regulation of both appetite and sleep. Improvements in weight control and hypersomnia with use of stimulant therapy in the setting of secondary obstructive sleep apnea have been reported and in addition to standard positive airway pressure therapies give hope for the management of these complications of craniopharyngioma resection.

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1029 Inspire Confidence In CPAP
Authors: Rodriguez Nathaniel 
Published: May 2023

Abstract

Introduction
Obstructive sleep apnea (OSA) is defined as the repetitive episodes of complete or partial occlusion of the airway occurring during sleep. OSA is thought to play a role in many different diseases including anxiety, depression, excessive day time sleepiness, and GERD. Treatment of OSA can often be difficult as many patients cannot tolerate CPAP.

Report of case(s)
The patient is a 76-year-old male who was diagnosed with OSA in 1991. He had undergone Uvulopalatopharyngoplasty in 1997 and maxillomandibular advancement in 1999 but continued to have snoring and witnessed apneas and excessive daytime sleepiness (EDS). A repeat PSG in 2019 showed severe sleep apnea with an AHI of 37.3. The patient had been unable to tolerate his CPAP at home due to discomfort from the pressure. He was then referred for evaluation and subsequent placement of a hypoglossal nerve stimulator known as inspire by ENT. On follow-up at the sleep clinic, the patient complained that he was still having daytime sleepiness and snoring despite compliance with INSPIRE. A home study was done to evaluate the effectiveness of INSPIRE and his AHI was found to be 46. INSPIRE was reconfigured and a repeat sleep study was done which showed similar results. A decision was made to try combination therapy with INSPIRE and CPAP at a lower pressure than previously. His original CPAP settings were 12cmH20 and with combo therapy we were able to change this to autoPAP 7-10, which the patient tolerated well and showed good control of his sleep apnea with an AHI of 4.6.

Conclusion
For those with moderate to severe OSA, the gold standard therapy is CPAP, but not every patient can tolerate CPAP therapy. One of the reasons is the CPAP pressure being too high. In 2014 the FDA approved a new treatment option for those with moderate to severe sleep apnea with the hypoglossal nerve stimulator. In our patient, multiple different INSPIRE configurations were attempted but he still had uncontrolled OSA and EDS. We were able to use combination therapy with INSPIRE and CPAP to adequately control his OSA without needing the high pressure that he could not tolerate previously.

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1032 Mixed sleep apnea management in a pediatric patient with Arnold Chiari type 1 and septo-optic dysplasia leading to hypopituitarism
Authors: Ghildiyal Naomi , Pant Alok , Rosales William , Chernyshev Oleg , Fahmy Omar 
Published: May 2023

Abstract

Introduction
Mixed Sleep apnea and Central sleep apnea are commonly noted in a patient with Arnold Chiari Malformation and definitive treatment of Arnold Chiari Malformation is surgical decompression.

Report of case(s)
The patient at the time of diagnosis of Mixed sleep apnea was 7 years old. She had already undergone tonsillectomy and adenoidectomy at the age of 4 years. Due to continued Sleep-disordered breathing, PSG was done in 2016 which showed predominantly mixed and central events. MRI was done which revealed Chiari 1 malformation with cerebellar tonsils extending 6 mm below the foramen magnum. The patient also due to septo-optic dysplasia and hypopituitarism followed with pediatric endocrinology and was on supplementation of Desmopressin, Hydrocortisone, Levothyroxine, and Somatropin. The mother at the time of pregnancy was a substance abuser, was incarcerated and the primary care was assumed by the patient’s legal guardian. The legal guardian was not interested in surgery for Arnold Chiari Malformation as she feared long-term outcomes and requested alternate treatment options. The patient underwent another split study and started on CPAP. The patient did not tolerate the CPAP and was transitioned to BiPAP. During her last clinic visit, the patient’s compliance had improved and she had achieved therapeutic AHI. The patient was also counseled on the effects of GH supplementation and untreated sleep apnea and is regularly followed by Sleep Medicine and Pediatric Endocrinology. The patient after transitioning to BiPAP has responded well to treatment for Mixed sleep apnea. Despite being on GH supplementation, with careful monitoring of hormone levels by Pediatric Endocrinology and symptom control by Sleep Medicine, the patient has shown remarkable improvement.

Conclusion
Mixed and central sleep apnea secondary to Arnold Chiari Malformation responds well to surgical decompression with improvement in sleep-disordered breathing. In a pediatric patient with complicated medical history with needing stress dose steroids, not a candidate for surgery, and intolerant to CPAP, BiPAP as a treatment modality showed good control of Sleep Apnea.

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1034 A Rare Case of ROHHAD Syndrome
Authors: Saha Anand , Bashir Ahsan , Francis Natalie , Stanton Crystal , Coughlin Kevin 
Published: May 2023

Abstract

Introduction
Early diagnosis of pediatric syndromes that are rare and involve multiple systems can be challenging. Rapid onset obesity, hypoventilation, hypothalamic dysfunction, and autonomic dysregulation characterizes ROHHAD syndrome which, if undiagnosed, can lead to potentially harmful outcomes including deferring radiological workup for a neural crest tumor (which has an over 50% association) and cardiopulmonary arrest.

Report of case(s)
A 10-year-old female with a history of obesity, precocious puberty, recurrent hypothermia, recurrent sodium derangements, bilateral ptosis, seizure disorder, and GERD initially presented to sleep clinic as a 4-year-old in 2017 when she recently began snoring with excessive daytime sleepiness as a result of rapid onset of weight gain. BMI at that time was 99th percentile and Epworth was 11. An in-lab polysomnography revealed severe OSA with AHI 26.4 events/hour; nocturnal hypoxemia with TS90 27% and O2 nadir 70%; and sleep related hypoventilation based on elevated pCO2 on end-tidal capnometry. Patient underwent adenotonsillectomy, revision adenoidectomy, and was started on CPAP before transitioning to BiPAP over the span of the next several years with serial polysomnographies ultimately showing significant improvement of sleep apnea with AHI 5.4 events/hour. Sleep-related hypoventilation, however, remained persistent with blood gases showing pCO2 as high as 89. PHOX2B gene testing was negative and secondary causes of hypoventilation were ruled out. During this time, she was also followed by Endocrinology for suspected hypothalamic dysfunction resulting in precocious puberty, deceleration of growth rate, sodium derangements. Autonomic dysfunction was also suspected due to recurrent hypothermia including a recent admission for Temperature &lt; 95F and ophthalmologic abnormalities including bilateral ptosis. At a multidisciplinary care conference, a working diagnosis of ROHHAD syndrome was made based on the rapid onset of obesity, hypoventilation on sleep studies and CBGs, hypothalamic dysfunction, and autonomic degeneration. Workup for neural crest tumor with MRI chest and abdomen unremarkable. Outside academic consultation was sought for management which recommended shared decision-making regarding steroids, immunosuppressive therapy (eg rituximab, cyclophosphamide), and IVIG.

Conclusion
For ROHHAD syndrome, there are very few documented cases in the United States and there is no known laboratory marker; diagnosis remains clinical and takes multidisciplinary collaboration, education, and a high degree of suspicion.

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1038 Treating Obstructive Sleep Apnea Without Batting an Eye: A Case of Lacrimal Duct Air Regurgitation Complicating CPAP Therapy
Authors: Taich Zachary , Rodríguez Astrid Carrión , Hsieh Caleb 
Published: May 2023

Abstract

Introduction
Dacryocystorhinostomy (DCR) is a surgical procedure that re-opens the nasolacrimal duct in patients with nasolacrimal duct obstruction and relieves symptoms of epiphora (watery eyes) by allowing tear drainage back to nasal mucosa. This procedure, however, can present a unique challenge for patients with obstructive sleep apnea (OSA) on continuous positive airway pressure (CPAP).

Report of case(s)
We present a 65 year-old man with a history of nasolacrimal duct obstruction status-post DCR who initially presented with daytime hypersomnolence and multiple nighttime awakenings. He underwent polysomnography (PSG) demonstrating moderate OSA with Apnea-hypopnea index (AHI) 18.5 events per hour. After initiation of CPAP, he reported eye pain, dryness and a sensation of air blowing into his eye. Both mandibular advancement device and BPAP (bilevel positive airway pressure) were attempted, unfortunately neither was found to be sufficiently effective.

Conclusion
This patient experienced a potential complication of DCR called lacrimal duct air regurgitation (LADR), which occurs due to the potential for reflux of air through the relatively dilated anastomosis from nasopharynx to puncta of the eye. By reducing end expiratory pressure, BPAP may in theory relieve expiratory LADR. A total-face mask was then employed as, covering the eyes allows for pressure equalization across the duct with resultant LADR improvement. This improved eye discomfort, but adherence was limited due to bulkiness of the mask. Lacrimal duct plugging may also be explored to reduce air reflux. This is an example of an uncommon complication of PAP therapy and highlights the unique challenges in maintaining adherence in this setting.

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Water- and feed-based arginine impacts on gut integrity in weanling pigs
Authors: Greiner Laura , Humphrey Dalton , Kerr Brian , Becker Spenser , Breuer Sophie , Hagen Chloe , Elefson Sarah , Haydon Keith 
Published: May 2023

Abstract
Two hundred and forty newly weaned pigs (PIC, Hendersonville, TN) were used to determine if supplementing additional arginine (Arg) either in the water or in the feed, and the combinations thereof, improved intestinal integrity and growth performance in nursery pigs. Each of the 80 pens contained three pigs (21 ± 2 d of age) which were randomly allotted to treatments in 4 × 3 factorial arrangement consisting of four water treatments (0%, 4%, 8%, and 12% Arg stock delivered through a 1:128 medication delivery system) in combination with three dietary Arg treatments (1.35%, 1.55%, and 1.75% standardized ileal digestible Arg; SID). Pigs and feeders were weighed at the d0, d6 (water and diet change), d20 (diet change), and d41 for the calculation of average daily gain (ADG), average daily feed intake (ADFI), and feed efficiency (G:F). Eighty pigs, 1 pig/pen, were euthanized at d6 for ileum evaluation of villus height and crypt depth. The remaining pigs were taken off the Arg-water treatment and fed phase-2 diets formulated to contain 1.35%, 1.55%, and 1.75% SID Arg. All pigs received a common diet from d20 to d41. Data were analyzed by pen as repeated measures (SAS 9.4). No interaction between water- and dietary-Arg was detected on nursery pig growth performance. There was a significant quadratic effect of SID Arg in the feed on pig final body weight (BW), ADG, ADFI, and G:F (P ≤ 0.037), where feeding 1.55% dietary Arg tended to improve growth performance compared to the 1.35% level for the 41 d of the trial (P ≤ 0.088). The use of the stock 8% Arg in the water resulted in a reduction in crypt depth (0:132.5, 4:140.7, 8:117.3, 12:132.0; P ≤ 0.01) and an improvement in intestinal permeability. The 4% oral Arg significantly reduced villous height:crypt depth ratio (0:2.50, 4:2.09, 8:2.56, 12:2.43; P ≤ 0.02). In conclusion, the feeding of 1.55% Arg resulted in an improvement in nursery pig ADG, ADFI, G:F, and final BW but did not alter intestinal villi morphology; however, the use of Arg in the water resulted in an improvement in intestinal villi, but no phenotypical change in piglet growth in the nursery.
Bacteroides fragilis expresses three proteins similar to Porphyromonas gingivalisHmuY: Hemophore‐like proteins differentially evolved to participate in heme acquisition in oral and gut microbiomes
Authors: Antonyuk Svetlana V. , Siemińska Klaudia , Śmiga Michał , Strange Richard W. , Wagner Mateusz , Barnett Katie J. , Olczak Teresa 
Published: May 2023

AbstractOral and gut microbiomes are important for the maintenance of homeostasis in the human body. Altered or disturbed mutualism between their members results in dysbiosis with local injury and subsequent systemic diseases. The high bacterial density causes intense competition among microbiome residents to acquire nutrients, including iron and heme, the latter of high importance for heme auxotrophic members of the Bacteroidetes phylum. Our main hypothesis is that the heme acquisition mechanism, with the leading role played by a novel HmuY family of hemophore‐like proteins, can be used to fulfill nutritional requirements and increase virulence. We characterized HmuY homologs expressed by Bacteroides fragilis and compared their properties with the first representative of this family, the HmuY protein of Porphyromonas gingivalis. In contrast to other Bacteroidetes members, B. fragilis produces three HmuY homologs (Bfr proteins). All bfr transcripts were produced at higher levels in bacteria starved of iron and heme (fold change increase ~60, ~90, and ~70 for bfrA, bfrB, and bfrC, respectively). X‐ray protein crystallography showed that B. fragilis Bfr proteins are structurally similar to P. gingivalis HmuY and to other homologs, except for differences in the potential heme‐binding pockets. BfrA binds heme, mesoheme, and deuteroheme, but preferentially under reducing conditions, using Met175 and Met146 to coordinate heme iron. BfrB binds iron‐free protoporphyrin IX and coproporphyrin III, whereas BfrC does not bind porphyrins. HmuY is capable of heme sequestration from BfrA, which might increase the ability of P. gingivalis to cause dysbiosis also in the gut microbiome.
“Uncrossed Central Facial Paralysis” Caused by Pontine Infarction
Authors: Zhao Liwen , Ren Bingcheng 
Published: May 2023

Introduction:
We report a patient with extraordinary pontine infarction-induced contralateral central facial palsy and weakened limb strength.

Case Report:
This is a 66-year-old man with left arm movement difficulty for 10 days and worsening over the last 1 day. His left nasolabial fold flattening and left arm strength and sensory were decreased. He could not complete the finger-nose test well with his right hand. Magnetic resonance and magnetic resonance angiography tests confirmed his right pontine acute infarction but without large vessel stenosis or occlusion.

Conclusion:
“Uncrossed paralysis” patients may present with contralateral face and body weakness with pontine infarcts, if the infarct occurs above the level of the facial nucleus head, and may be simmilar with the higher level pontine lesions or cerebrum semisphere infarction, which need particular attention during clinical practice.
Use of Subcutaneous Injection of Epinephrine and Triamcinolone with Tranexamic Acid Reduces Blood Loss, Transfusion Rates, and Length of Stay in Open Sagittal Craniosynostosis Repair
Authors: Ackerman Laurie L. , Snider Anna A. , Ye Jian 
Published: May 2023

Objective:
In 2017, we adopted the use of triamcinolone/epinephrine (TAC/Epi) scalp injection and later added tranexamic acid (TXA) in open sagittal synostosis surgery. We believe that this reduced blood loss and transfusion rates.

Methods:
A total of 107 consecutive patients operated for sagittal synostosis aged <4 months from 2007 to 2019 were retrospectively reviewed. We collected demographics [age, sex, weight at surgery, and length of stay (LOS)], intraoperative information [estimated blood loss (EBL)], administration of packed red blood cell, transfusion of plasmalyte/albumen, operating time, baseline hemoglobin (Hb) and hematocrit (Hct), type of local anesthetic (1/4% bupivacaine vs. TAC/Epi), and use/volume of TXA. Hb, Hct, coagulation studies, and platelets at 2 hours postoperatively and postoperative day (POD) 1 were recorded.

Results:
There were 3 groups: 1/4% bupivacaine/epinephrine (N=64), TAC/Epi (N=13), and TAC/Epi with TXA bolus/infusion intraoperatively (N=30). Groups receiving TAC/Epi or TAC/Epi with TXA had lower mean EBL (P<0.0001), lower rate/amount of packed red blood cell transfusion (P<0.0001), lower prothrombin time/international normalized ratio on POD 1 (P<0.0001), higher platelets (P<0.001), and shorter operative time (P<0.0001). LOS was shortest for TAC/Epi with TXA (P<0.0001). No significant differences between groups were noted on POD 1 Hb, Hct, or partial prothrombin time. Post hoc testing revealed an advantage of TAC/Epi with TXA over TAC/Epi alone for 2-hour postoperative international normalized ratio (P=0.0249), Operating Room time (P=0.0179), and LOS (P=0.0049).

Conclusions:
Use of TAC/Epi alone reduced EBL, LOS, Operating Room time, and improved laboratory values postoperatively in open sagittal synostosis surgery. Addition of TXA further improved operative time and LOS. It is likely that lower rates of transfusion could be tolerated.
Quantifying and estimating ecological network diversity based on incomplete sampling data
Authors: Chiu Chun-Huo , Chao Anne , Vogel Sebastian , Kriegel Peter , Thorn Simon 
Published: May 2023

An ecological network refers to the ecological interactions among sets of species. Quantification of ecological network diversity and related sampling/estimation challenges have explicit analogues in species diversity research. A unified framework based on Hill numbers and their generalizations was developed to quantify taxonomic, phylogenetic and functional diversity. Drawing on this unified framework, we propose three dimensions of network diversity that incorporate the frequency (or strength) of interactions, species phylogenies and traits. As with surveys in species inventories, nearly all network studies are based on sampling data and thus also suffer from under-sampling effects. Adapting the sampling/estimation theory and the iNEXT (interpolation/extrapolation) standardization developed for species diversity research, we propose the iNEXT.link method to analyse network sampling data. The proposed method integrates the following four inference procedures: (i) assessment of sample completeness of networks; (ii) asymptotic analysis via estimating the true network diversity; (iii) non-asymptotic analysis based on standardizing sample completeness via rarefaction and extrapolation with network diversity; and (iv) estimation of the degree of unevenness or specialization in networks based on standardized diversity. Interaction data between European trees and saproxylic beetles are used to illustrate the proposed procedures. The software iNEXT.link has been developed to facilitate all computations and graphics.
This article is part of the theme issue ‘Detecting and attributing the causes of biodiversity change: needs, gaps and solutions’.
Biodiversity–ecosystem function relationships change in sign and magnitude across the Hill diversity spectrum
Authors: Roswell Michael , Harrison Tina , Genung Mark A. 
Published: May 2023

Motivated by accelerating anthropogenic extinctions, decades of biodiversity–ecosystem function (BEF) experiments show that ecosystem function declines with species loss from local communities. Yet, at the local scale, changes in species' total and relative abundances are more common than species loss. The consensus best biodiversity measures are Hill numbers, which use a scaling parameter,ℓ, to emphasize rarer versus more common species. Shifting that emphasis captures distinct, function-relevant biodiversity gradients beyond species richness. Here, we hypothesized that Hill numbers that emphasize rare species more than richness does may distinguish large, complex and presumably higher-functioning assemblages from smaller and simpler ones. In this study, we tested which values ofℓproduce the strongest BEF relationships in community datasets of ecosystem functions provided by wild, free-living organisms. We found thatℓvalues that emphasized rare species more than richness does most often correlated most strongly with ecosystem functions. As emphasis shifted to more common species, BEF correlations were often weak and/or negative. We argue that unconventional Hill diversities that shift emphasis towards rarer species may be useful for describing biodiversity change, and that employing a wide spectrum of Hill numbers can clarify mechanisms underlying BEF relationships.This article is part of the theme issue ‘Detecting and attributing the causes of biodiversity change: needs, gaps and solutions’.