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Article ; Online: Response to the Comment on "Fetal Risk Stratification and Outcomes in Children With Prenatally Diagnosed Lung Malformations: Results From a Multi-institutional Research Collaborative by Kunisaki et al".

Kunisaki, Shaun M

2021 Volume 274, Issue 6, Page(s) e891

MeSH term(s)	Child ; Female ; Humans ; Lung/diagnostic imaging ; Pregnancy ; Prenatal Diagnosis ; Risk Assessment
Language	English
Publishing date	2021-04-14
Publishing country	United States
Document type	Letter ; Comment
ZDB-ID	340-2
ISSN	1528-1140 ; 0003-4932
ISSN (online)	1528-1140
ISSN	0003-4932
DOI	10.1097/SLA.0000000000004898
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Article: Narrative review of congenital lung lesions.

Kunisaki, Shaun M

Translational pediatrics

2021 Volume 10, Issue 5, Page(s) 1418–1431

Abstract: This article reviews the contemporary diagnosis and management of antenatally diagnosed congenital lung lesions. These anomalies, which include congenital pulmonary airway malformation (CPAM) (formerly congenital cystic adenomatoid malformation), ... ...

Abstract	This article reviews the contemporary diagnosis and management of antenatally diagnosed congenital lung lesions. These anomalies, which include congenital pulmonary airway malformation (CPAM) (formerly congenital cystic adenomatoid malformation), bronchopulmonary sequestration (BPS), bronchogenic cyst, and congenital lobar emphysema (CLE), are relatively rare but are increasingly encountered by clinicians because of the improved resolution and enhanced sensitivity of fetal ultrasound. Serial assessment of these lesions throughout pregnancy remains the norm rather than the exception. Perinatal management strategies may differ based on initial size and growth patterns of these masses until delivery. Fetal magnetic resonance imaging and other diagnostic testing can sometimes be helpful in providing additional prognostic information. Over the last decade, maternal steroids have become standard of care in the management of larger lesions at risk for nonimmune hydrops. As a result, fetal surgical procedures, including open resection, thoracoamniotic shunting, and ex utero intrapartum treatment (EXIT), are less uncommonly performed. Decisions regarding whether delivery of these fetuses should occur in a tertiary care center with pediatric surgery coverage versus delivery at a local community hospital are now highly relevant in most prenatal counseling discussions with families. Large lung malformations may require urgent surgical removal in the early postnatal period because of respiratory distress. Other complications, such as recurrent pneumonia, pneumothorax, and cancer, are indications for postnatal lung resection on an elective basis. Many children are good candidates for minimally invasive (thoracoscopic) surgical approaches as an alternative to resection by thoracotomy. In the vast majority of cases, the overall prognosis remains excellent.
Language	English
Publishing date	2021-06-18
Publishing country	China
Document type	Journal Article ; Review
ZDB-ID	2901309-4
ISSN	2224-4344 ; 2224-4344 ; 2224-4336
ISSN (online)	2224-4344
ISSN	2224-4344 ; 2224-4336
DOI	10.21037/tp-20-133
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Article: Management of long-gap esophageal atresia.

Penikis, Annalise B / Sescleifer, Anne M / Kunisaki, Shaun M

Translational pediatrics

2024 Volume 13, Issue 2, Page(s) 329–342

Abstract: A uniquely challenging subset of infants diagnosed with esophageal atresia (EA) are those born with long-gap EA (LGEA). The common unifying feature in infants with LGEA is that the proximal and distal segments of the esophagus are too far apart to enable ...

Abstract	A uniquely challenging subset of infants diagnosed with esophageal atresia (EA) are those born with long-gap EA (LGEA). The common unifying feature in infants with LGEA is that the proximal and distal segments of the esophagus are too far apart to enable primary anastomosis via a single operation in the newborn period. Although any type of EA can technically result in a long gap, the Gross type A variant occurs in 8% of all EA cases and is most commonly associated with LGEA. In this review, we provide an evidence-based approach to the current challenges and management strategies employed in LGEA. There are fortunately a range of available surgical techniques for LGEA repair, including delayed primary repair, staged repair based on longitudinal traction strategies to lengthen both ends (e.g., Foker procedure, internal traction), and esophageal replacement using other portions of the gastrointestinal tract. The literature on the management of LGEA has long been dominated by single-center retrospective reviews, but the field has recently witnessed increased multi-center collaboration that has helped to increase our understanding on how to best manage this challenging patient population. Delayed primary repair is strongly preferred as the initial approach in management of LGEA in the United States as well as several European countries and is supported by the American Pediatric Surgery Association recommendations. Should esophageal replacement be required in cases where salvaging the native thoracic esophagus is not possible, gastric conduits are the preferred approach, based on the relative simplicity of the operation, low postoperative morbidity, and long-term durability. Long-term followup for monitoring of swallowing function, nutritional status, aspiration/respiratory illnesses, gastroesophageal reflux, and associated comorbidities is essential in the comprehensive care of these complex patients.
Language	English
Publishing date	2024-02-27
Publishing country	China
Document type	Journal Article ; Review
ZDB-ID	2901309-4
ISSN	2224-4344 ; 2224-4344 ; 2224-4336
ISSN (online)	2224-4344
ISSN	2224-4344 ; 2224-4336
DOI	10.21037/tp-23-453
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Article: Patient Characteristics, Treatment, and Survival in Pleuropulmonary Blastoma: An Analysis from the National Cancer Database.

Chidiac, Charbel / Sferra, Shelby R / Kunisaki, Shaun M / Rhee, Daniel S

Children (Basel, Switzerland)

2024 Volume 11, Issue 4

Abstract: Pleuropulmonary blastoma (PPB) is a rare childhood tumor originating from the lung or pleura, typically treated with surgery, chemotherapy (CTx), and/or radiation therapy (RTx). This study aimed to assess patient and tumor features, treatment methods, ... ...

Abstract	Pleuropulmonary blastoma (PPB) is a rare childhood tumor originating from the lung or pleura, typically treated with surgery, chemotherapy (CTx), and/or radiation therapy (RTx). This study aimed to assess patient and tumor features, treatment methods, and survival rates in PPB. We retrospectively analyzed PPB patients under 18 from 2004 to 2019, using the National Cancer Database (NCDB). We assessed 5-year overall survival (OS) rates based on disease presentation and treatment regimens, using Kaplan-Meier curves and Cox proportional regression. Among 144 cases identified, 45.9% were female, with a median age of 2 years (interquartile range 1-3). In all, 62.5% of tumors originated from the lung, and 10.4% from the pleura. Moreover, 6.9% were bilateral, and the median tumor size was 8.9 cm, with 4.2% presenting with metastases. The 5-year OS rate was 79.6%, with no significant change over time (2004-2009, 77.6%; 2010-2014, 90.8%; 2015-2019, OS 75.3%;
Language	English
Publishing date	2024-04-02
Publishing country	Switzerland
Document type	Journal Article
ZDB-ID	2732685-8
ISSN	2227-9067
ISSN	2227-9067
DOI	10.3390/children11040424
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Article ; Online: Reply to Letter to the Editor regarding: Association of operative approach with outcomes in neonates with esophageal atresia and tracheoesophageal fistula.

Etchill, Eric W / Rhee, Daniel S / Kunisaki, Shaun M

Journal of pediatric surgery

2022 Volume 57, Issue 10, Page(s) 482

MeSH term(s)	Abnormalities, Multiple/surgery ; Esophageal Atresia/surgery ; Humans ; Infant, Newborn ; Tracheoesophageal Fistula/surgery
Language	English
Publishing date	2022-06-18
Publishing country	United States
Document type	Letter ; Comment
ZDB-ID	80165-3
ISSN	1531-5037 ; 0022-3468
ISSN (online)	1531-5037
ISSN	0022-3468
DOI	10.1016/j.jpedsurg.2022.06.004
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Article: Amniotic Fluid Stem Cells for the Treatment of Surgical Disorders in the Fetus and Neonate.

Kunisaki, Shaun M

Stem cells translational medicine

2018 Volume 7, Issue 11, Page(s) 767–773

Abstract: Over the past decade, amniotic fluid-derived stem cells have emerged as a novel experimental approach aimed at improving outcomes in children with congenital anomalies, including spina bifida, heart defects, and diaphragmatic hernia. Interest in these ... ...

Abstract	Over the past decade, amniotic fluid-derived stem cells have emerged as a novel experimental approach aimed at improving outcomes in children with congenital anomalies, including spina bifida, heart defects, and diaphragmatic hernia. Interest in these cells for the treatment of prenatally diagnosed diseases has arisen based on numerous studies demonstrating the relative ease of harvesting an abundant quantity of amniocytes from a small aliquot of fluid, the unique properties of amniocytes themselves, and the beneficial effects of amniotic fluid-derived stem cells in experimental animal models. This report gives a brief overview of the rationale and current status of amniotic fluid stem cell-based therapies, focusing on its relevance to birth defects affecting the fetus and neonate. The author proposes a roadmap for further study that would be required prior to clinical application of amniotic fluid stem cell technologies. Stem Cells Translational Medicine 2018;7:767-773.
MeSH term(s)	Amniotic Fluid/cytology ; Fetal Diseases/therapy ; Heart Defects, Congenital/therapy ; Humans ; Induced Pluripotent Stem Cells/cytology ; Induced Pluripotent Stem Cells/metabolism ; Infant, Newborn ; Myocytes, Cardiac/cytology ; Myocytes, Cardiac/metabolism ; Spinal Dysraphism/therapy ; Stem Cell Transplantation ; Stem Cells/cytology ; Stem Cells/metabolism ; Tissue Engineering
Language	English
Publishing date	2018-08-07
Publishing country	United States
Document type	Journal Article ; Research Support, N.I.H., Extramural
ZDB-ID	2642270-0
ISSN	2157-6580 ; 2157-6564
ISSN (online)	2157-6580
ISSN	2157-6564
DOI	10.1002/sctm.18-0018
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Article ; Online: Surgical Pulmonary and Pleural Diseases in Children: Lung Malformations, Empyema, and Spontaneous Pneumothorax.

Kunisaki, Shaun M / Leys, Charles M

Advances in pediatrics

2020 Volume 67, Page(s) 145–169

MeSH term(s)	Child ; Disease Management ; Empyema, Pleural/surgery ; Humans ; Lung/abnormalities ; Lung/surgery ; Lung Diseases/congenital ; Lung Diseases/surgery ; Pneumonectomy/methods ; Pneumothorax/surgery ; Thoracentesis/methods
Language	English
Publishing date	2020-05-14
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	210524-x
ISSN	1878-1926 ; 0065-3101
ISSN (online)	1878-1926
ISSN	0065-3101
DOI	10.1016/j.yapd.2020.03.006
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Article ; Online: Increased survival following fetoscopic endoluminal tracheal occlusion for diaphragmatic hernia.

Baschat, Ahmet A / Miller, Jena L / Kunisaki, Shaun M

The Journal of pediatrics

2021 Volume 238, Page(s) 338–342

MeSH term(s)	Female ; Fetoscopy ; Hernias, Diaphragmatic, Congenital/surgery ; Humans ; Pregnancy
Language	English
Publishing date	2021-10-27
Publishing country	United States
Document type	Journal Article ; Comment
ZDB-ID	3102-1
ISSN	1097-6833 ; 0022-3476
ISSN (online)	1097-6833
ISSN	0022-3476
DOI	10.1016/j.jpeds.2021.08.061
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Article ; Online: Neurodevelopmental Outcomes in Children After Fetoscopic Endoluminal Tracheal Occlusion for Severe Congenital Diaphragmatic Hernia: Results From a Multidisciplinary Clinic.

Sferra, Shelby R / Penikis, Annalise B / Guo, Matthew / Baschat, Ahmet A / Mogayzel, Peter J / Burton, Vera Joanna / Kunisaki, Shaun M

Journal of pediatric surgery

2024

Abstract: Background: We compared early neurodevelopmental morbidity in young children with severe CDH who underwent FETO to those without fetal therapy.: Methods: We conducted a prospective study of severe CDH patients undergoing FETO (n = 18) at a single ... ...

Abstract	Background: We compared early neurodevelopmental morbidity in young children with severe CDH who underwent FETO to those without fetal therapy. Methods: We conducted a prospective study of severe CDH patients undergoing FETO (n = 18) at a single North American center from 2015 to 2021 (NCT02710968). Outpatient survivors (n = 12) were evaluated by a multidisciplinary team and compared to expectantly managed CDH patients. Neurodevelopmental outcomes were assessed using the Capute Scales [Clinical Linguistic and Auditory Milestone Scales (CLAMS) and Cognitive Adaptive Test (CAT)], with a developmental quotient (DQ) < 85 indicative of at-risk for delay. Results: At one year, 58% (n = 7) of FETO patients underwent evaluation, with notable concern for language delay (CLAMS median DQ, 80.1 [interquartile range, 67.6-86.7]). FETO scores improved by 24-months, whereas high severity/non-FETO scores declined [CLAMS median DQ (Difference in DQ), 92.3 (+12.2) vs. 77.1 (-13.4), respectively; p = 0.049]. On the initial CAT, FETO patients had concern for visual motor and problem-solving delays, with a median DQ of 81.3 (62.1-89.4). At 24-months, FETO patients had improving scores [Median CAT DQ, 90.8 (+9.5)], whereas high severity/non-FETO [87.5 (-3.0), p = 0.28] had declining scores. Conclusion: These initial data suggest that FETO is associated with favorable neurodevelopmental outcomes at 24-months compared to severe CDH under expectant management. Level of evidence: III.
Language	English
Publishing date	2024-03-20
Publishing country	United States
Document type	Journal Article
ZDB-ID	80165-3
ISSN	1531-5037 ; 0022-3468
ISSN (online)	1531-5037
ISSN	0022-3468
DOI	10.1016/j.jpedsurg.2024.03.041
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Article ; Online: Finite element analysis of esophageal atresia repair with biodegradable polymer sleeves.

Ramaraju, Harsha / Sferra, Shelby R / Kunisaki, Shaun M / Hollister, Scott J

Journal of the mechanical behavior of biomedical materials

2022 Volume 133, Page(s) 105349

Abstract: Esophageal atresia, which occurs in 1 in every 4100 live births, is a potentially lethal congenital malformation resulting in discontinuity of the esophagus. Treatment requires approximating the disconnected esophageal segments and suturing the ends to ... ...

Abstract	Esophageal atresia, which occurs in 1 in every 4100 live births, is a potentially lethal congenital malformation resulting in discontinuity of the esophagus. Treatment requires approximating the disconnected esophageal segments and suturing the ends to restore continuity. Due to excessive anastomotic tension, leaks and strictures are prevalent in primary surgical repair of the esophagus especially in the subset of neonates presenting with long gap atresia (>3 cm between esophageal segments). Extracellular matrix derived scaffolds and biodegradable polymer scaffolds have been investigated in preclinical models for use in alleviating esophageal anastomotic tension with varying degrees of success. We have previously described the suitability of biodegradable shape memory materials for use in a number of soft tissue repair applications. Developing repair strategies addressing esophageal atresia requires a framework for approximating tension at the anastomosis. In this study, we describe a computational framework for approximating esophageal anastomotic tension to study the impact of primary and device supported repair. The esophagus was modeled as an idealized concentric cylinder comprised of mucosal and muscle layers described by nonlinear strain energy functions and a mixed fiber model with a Neo-Hookean base material (FEBIO studio). Sutures were modeled as nonlinear elastic springs carrying only tension, and shape memory polymers were modeled as nonlinear elastic materials using one term Ogden parameters. The impact of suture bite (length of suture from anastomosis), sleeve material properties, sleeve suture strategy, and gap length were evaluated with respect to anastomotic LaGrangian strain, displacement magnitude, and strain energy density. With increasing gap length, there was an increase in anastomotic strain, displacement magnitude and strain energy density. Increasing the suture bite length decreased strain at the anastomosis. Application of the sleeve reduced strain, displacement and strain energy to a greater extent in longer gap atresia. Increasing the number of sutures to apply the sleeve did not decrease the esophageal strain compared to sleeves with lesser number of sutures. Sleeve material testing revealed an interplay between the nonlinear mechanical properties of the selected materials and their contribution to reducing anastomotic tension. Taken together this study provides a unique framework for computational verification of design hypothesis broadly addressing clinical procedure optimization, material design, and device design for surgical repair of esophageal atresia.
MeSH term(s)	Anastomosis, Surgical/methods ; Esophageal Atresia/surgery ; Finite Element Analysis ; Humans ; Infant, Newborn ; Polymers
Chemical Substances	Polymers
Language	English
Publishing date	2022-07-03
Publishing country	Netherlands
Document type	Journal Article
ZDB-ID	2378381-3
ISSN	1878-0180 ; 1751-6161
ISSN (online)	1878-0180
ISSN	1751-6161
DOI	10.1016/j.jmbbm.2022.105349
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