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Article ; Online: Long-term effects of systemic gene therapy On gait in a canine model of myotubular myopathy.

Pierson, Christopher R

2017 Volume 56, Issue 5, Page(s) 839–840

MeSH term(s)	Animals ; Dogs ; Gait ; Genetic Therapy ; Myopathies, Structural, Congenital ; Protein Tyrosine Phosphatases, Non-Receptor/genetics
Chemical Substances	Protein Tyrosine Phosphatases, Non-Receptor (EC 3.1.3.48)
Language	English
Publishing date	2017-05-23
Publishing country	United States
Document type	Editorial ; Comment
ZDB-ID	438353-9
ISSN	1097-4598 ; 0148-639X
ISSN (online)	1097-4598
ISSN	0148-639X
DOI	10.1002/mus.25681
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Zs.A 1449: Show issues			Location: Je nach Verfügbarkeit (siehe Angabe bei Bestand) bis Jg. 1994: Bestellungen von Artikeln über das Online-Bestellformular Jg. 1995 - 2021: Lesesall (1.OG) ab Jg. 2022: Lesesaal (EG)
Zs.MO 551: Show issues

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Article ; Online: Histopathologic and Molecular Features of Central Nervous System Embryonal Tumors for Integrated Diagnosis Reporting.

Cole, Bonnie L / Pierson, Christopher R

Surgical pathology clinics

2020 Volume 13, Issue 4, Page(s) 783–800

Abstract: Embryonal tumors of the pediatric central nervous system are challenging clinically and diagnostically. These tumors are aggressive, and patients often have poor outcomes even with intense therapy. Proper tumor classification is essential to patient care, ...

Abstract	Embryonal tumors of the pediatric central nervous system are challenging clinically and diagnostically. These tumors are aggressive, and patients often have poor outcomes even with intense therapy. Proper tumor classification is essential to patient care, and this process has undergone significant changes with the World Health Organization recommending histopathologic and molecular features be integrated in diagnostic reporting. This has especially impacted the workup of embryonal tumors because molecular testing has resulted in the identification of clinically relevant tumor subgroups and new entities. This review summarizes recent developments and provides a framework to workup embryonal tumors in diagnostic practice.
MeSH term(s)	Biomarkers, Tumor ; Central Nervous System Neoplasms/classification ; Central Nervous System Neoplasms/diagnosis ; Central Nervous System Neoplasms/pathology ; Child ; Humans ; Medulloblastoma/classification ; Medulloblastoma/diagnosis ; Medulloblastoma/pathology ; Neoplasms, Germ Cell and Embryonal/classification ; Neoplasms, Germ Cell and Embryonal/diagnosis ; Neoplasms, Germ Cell and Embryonal/pathology ; Rhabdoid Tumor/classification ; Rhabdoid Tumor/diagnosis ; Rhabdoid Tumor/pathology
Chemical Substances	Biomarkers, Tumor
Language	English
Publishing date	2020-10-09
Publishing country	United States
Document type	Journal Article ; Review
ISSN	1875-9157
ISSN (online)	1875-9157
DOI	10.1016/j.path.2020.08.005
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Article ; Online: Neuropathology of Surgically Managed Epilepsy Specimens.

Thomas, Diana L / Pierson, Christopher R

Neurosurgery

2020 Volume 88, Issue 1, Page(s) 1–14

Abstract: Epilepsy is characterized as recurrent seizures, and it is one of the most prevalent disorders of the human nervous system. A large and diverse profile of different syndromes and conditions can cause perturbations in neural networks that are associated ... ...

Abstract	Epilepsy is characterized as recurrent seizures, and it is one of the most prevalent disorders of the human nervous system. A large and diverse profile of different syndromes and conditions can cause perturbations in neural networks that are associated with epilepsy. Advances in neuroimaging and electrophysiological monitoring have enhanced our ability to localize the neuropathological lesions that alter the neural networks giving rise to epilepsy, whereas advances in surgical management have resulted in excellent seizure control in many patients following resections. Histopathologic study using a variety of special stains, molecular analysis, and functional studies of these resected tissues has facilitated the neuropathological characterization of these lesions. Here, we review the neuropathology of common structural lesions that cause epilepsy and are amenable to neurosurgical resection, such as hippocampal sclerosis, focal cortical dysplasia, and its associated principal lesions, including long-term epilepsy-associated tumors, as well as other malformations of cortical development and Rasmussen encephalitis.
MeSH term(s)	Adult ; Brain Diseases/complications ; Brain Diseases/pathology ; Epilepsy/etiology ; Epilepsy/pathology ; Epilepsy/surgery ; Female ; Humans ; Male ; Neuropathology
Language	English
Publishing date	2020-11-24
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	135446-2
ISSN	1524-4040 ; 0148-396X
ISSN (online)	1524-4040
ISSN	0148-396X
DOI	10.1093/neuros/nyaa366
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Zs.A 1424: Show issues			Location: Je nach Verfügbarkeit (siehe Angabe bei Bestand) bis Jg. 1994: Bestellungen von Artikeln über das Online-Bestellformular Jg. 1995 - 2021: Lesesall (1.OG) ab Jg. 2022: Lesesaal (EG)
Zs.MO 539: Show issues

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Article: Gene therapy in myotubular myopathy: promising progress and future directions.

Pierson, Christopher R

Annals of translational medicine

2015 Volume 3, Issue 5, Page(s) 61

Language	English
Publishing date	2015-05-20
Publishing country	China
Document type	Journal Article
ZDB-ID	2893931-1
ISSN	2305-5847 ; 2305-5839
ISSN (online)	2305-5847
ISSN	2305-5839
DOI	10.3978/j.issn.2305-5839.2015.01.11
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Article ; Online: Neuropathology of Mowat-Wilson Syndrome.

Conces, Miriam R / Hughes, Anna / Pierson, Christopher R

Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society

2020 Volume 23, Issue 4, Page(s) 322–325

Abstract: Mowat-Wilson syndrome (MWS) is a syndromic form of Hirschsprung disease that is characterized by variable degrees of intellectual disability, characteristic facial dysmorphism, and a diverse set of other congenital malformations due to haploinsufficiency ...

Abstract	Mowat-Wilson syndrome (MWS) is a syndromic form of Hirschsprung disease that is characterized by variable degrees of intellectual disability, characteristic facial dysmorphism, and a diverse set of other congenital malformations due to haploinsufficiency of
MeSH term(s)	Autopsy ; Biomarkers/metabolism ; Brain/metabolism ; Brain/pathology ; Facies ; Fatal Outcome ; Hirschsprung Disease/diagnosis ; Hirschsprung Disease/genetics ; Hirschsprung Disease/metabolism ; Hirschsprung Disease/pathology ; Humans ; Intellectual Disability/diagnosis ; Intellectual Disability/genetics ; Intellectual Disability/metabolism ; Intellectual Disability/pathology ; Male ; Microcephaly/diagnosis ; Microcephaly/genetics ; Microcephaly/metabolism ; Microcephaly/pathology ; Young Adult ; Zinc Finger E-box Binding Homeobox 2/genetics ; Zinc Finger E-box Binding Homeobox 2/metabolism
Chemical Substances	Biomarkers ; ZEB2 protein, human ; Zinc Finger E-box Binding Homeobox 2
Language	English
Publishing date	2020-04-06
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	1463498-3
ISSN	1615-5742 ; 1093-5266
ISSN (online)	1615-5742
ISSN	1093-5266
DOI	10.1177/1093526620903956
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Article ; Online: Submucosal Nerve Diameter in the Rectum Increases With Age: An Important Consideration for the Diagnosis of Hirschsprung Disease.

Conces, Miriam R / Beach, Sarah / Pierson, Christopher R / Prasad, Vinay

Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society

2021 Volume 25, Issue 3, Page(s) 263–269

Abstract: Introduction: Hypertrophic submucosal nerves, defined as ≥40 µm in diameter, are considered supportive of a diagnosis of HSCR, but the effect of age on nerve diameter has not been well-studied. We sought to determine the distribution of the largest ... ...

Abstract	Introduction: Hypertrophic submucosal nerves, defined as ≥40 µm in diameter, are considered supportive of a diagnosis of HSCR, but the effect of age on nerve diameter has not been well-studied. We sought to determine the distribution of the largest nerve diameter in ganglionic rectal biopsies and the significance of hypertrophic submucosal nerves in the diagnosis of Hirschsprung disease (HSCR) based on age. Methods: Rectal biopsies performed in the evaluation of HSCR were retrospectively reviewed from 179 patients (151 ganglionic biopsies, 28 aganglionic biopsies), and the diameter of the largest submucosal nerve was measured. Results: In non-Hirschsprung disease (non-HSCR) biopsies, submucosal nerve diameter increased with age. In patients <1 year, the average diameter was 34.1 ± 11.6 µm but increased to 50.8 ± 17.3 µm after 1 year of age. Submucosal nerves ≥40 µm in diameter were significantly associated with HSCR across all ages [HSCR = 25/28 (89.3%) vs non-HSCR = 59/151 (39.1%), p < 0.0001] and remained significant in patients <1 year of age [HSCR = 22/24 (91.7%) vs non-HSCR = 19/91 (20.9%), p < 0.0001]. Conclusions: The diameter of submucosal nerves increases with age, and ≥40 µm nerves are common after 1 year of age.
MeSH term(s)	Biopsy ; Hirschsprung Disease/diagnosis ; Hirschsprung Disease/pathology ; Humans ; Hypertrophy/pathology ; Rectum/pathology ; Retrospective Studies
Language	English
Publishing date	2021-11-18
Publishing country	United States
Document type	Journal Article
ZDB-ID	1463498-3
ISSN	1615-5742 ; 1093-5266
ISSN (online)	1615-5742
ISSN	1093-5266
DOI	10.1177/10935266211049689
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Article ; Online: Neuropathology of Septo-optic Dysplasia: A Report of 4 Autopsy Cases.

Blackburn, Jessica / Thomas, Diana L / Hughes, Anna / Pierson, Christopher R

Journal of child neurology

2020 Volume 36, Issue 2, Page(s) 105–115

Abstract: Septo-optic dysplasia (SOD) is defined by the presence of 2 or more features in a diagnostic triad: (1) optic nerve hypoplasia, (2) pituitary dysfunction, and (3) midline forebrain anomalies. SOD arises due to diverse pathogenetic mechanisms including ... ...

Abstract	Septo-optic dysplasia (SOD) is defined by the presence of 2 or more features in a diagnostic triad: (1) optic nerve hypoplasia, (2) pituitary dysfunction, and (3) midline forebrain anomalies. SOD arises due to diverse pathogenetic mechanisms including acquired and genetic factors, and it shows considerable clinical and phenotypic variability. Our knowledge of SOD is incomplete in part because of a paucity of published neuropathology data, so we reviewed the autopsy neuropathology of 4 SOD patients. All patients met SOD criteria according to the triad. Additional neuropathologic findings included malformations involving non-forebrain structures and possible secondary phenomena. Autopsies demonstrate that SOD patients often have additional neuropathologic findings beyond the triad and we feel that use of the term SOD-complex appropriately underscores this diversity and its likely clinical impact. This study suggests that autopsies enhance our understanding of SOD and may be an asset in performing needed clinical and phenotypic correlation studies.
MeSH term(s)	Adult ; Autopsy ; Brain/pathology ; Child ; Child, Preschool ; Humans ; Infant ; Male ; Retrospective Studies ; Septo-Optic Dysplasia/diagnosis ; Septo-Optic Dysplasia/pathology
Language	English
Publishing date	2020-09-14
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	639288-x
ISSN	1708-8283 ; 0883-0738
ISSN (online)	1708-8283
ISSN	0883-0738
DOI	10.1177/0883073820954071
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Zs.A 2301: Show issues

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Article ; Online: Reoperation following strabismus surgery among Medicare beneficiaries: Associations with geographic region, academic affiliation, surgeon volume, and adjustable suture technique.

Christensen, Michael R / Pierson, Kasey / Leffler, Christopher Theodore

Indian journal of ophthalmology

2018 Volume 66, Issue 8, Page(s) 1149–1153

Abstract: Purpose: The objective of this study was to determine the associations of strabismus surgery reoperation rates in a large national database of provider payments with geographic region, practice type and volume, and the availability of adjustable suture ... ...

Abstract	Purpose: The objective of this study was to determine the associations of strabismus surgery reoperation rates in a large national database of provider payments with geographic region, practice type and volume, and the availability of adjustable suture technique. Methods: Fee-for-service payments to providers for medicare beneficiaries having strabismus surgery between 2012 and 2015 were retrospectively analyzed to identify reoperations in the same calendar year. The adjustable-suture technique was considered to be available to the patient if the patient's surgeon billed for adjustable sutures. Predictors of reoperation in the same calendar year were determined by multivariable logistic regression. Results: Availability of the adjustable suture technique was not associated with reoperation rate in multivariable analysis among 5971 patients having horizontal muscle surgery (odds ratio, [OR] 0.86, P = 0.29), 2840 patients having vertical muscle surgery (OR 0.98, P = 0.93), or 1199 patients having surgery with scarring or restriction (OR 0.86, P = 0.61). For horizontal surgery, the reoperation rate was higher in academic practices (OR 1.67), as compared with community practices, and in the South (OR 2.85) and West (OR 1.92, all P < 0.001). The reoperation rate was unchanged with surgeons in the lowest-quartile of surgical volume. Among surgeons paid for horizontal surgery, 45% of surgeons in the Northeast, the West, or Florida coded for adjustable sutures, compared with 8% of surgeons elsewhere (P < 0.001). Conclusion: The availability of the adjustable-suture technique was not associated with reoperation rate after strabismus surgery in this large national database. Having surgery by a lower-volume surgeon was not associated with a higher reoperation rate. The reoperation rate was higher when surgery was conducted in an academic practice, or in certain regions of the country. Adjustable sutures are largely a bicoastal practice.
MeSH term(s)	Aged ; Fee-for-Service Plans ; Female ; Health Expenditures ; Humans ; Male ; Medicare/economics ; Middle Aged ; Oculomotor Muscles/surgery ; Ophthalmologic Surgical Procedures/economics ; Reoperation ; Retrospective Studies ; Strabismus/economics ; Strabismus/surgery ; Surgeons/supply & distribution ; Suture Techniques/economics ; Suture Techniques/instrumentation ; Sutures/economics ; United States
Language	English
Publishing date	2018-07-23
Publishing country	India
Document type	Journal Article ; Multicenter Study
ZDB-ID	187392-1
ISSN	1998-3689 ; 0301-4738
ISSN (online)	1998-3689
ISSN	0301-4738
DOI	10.4103/ijo.IJO_18_18
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Zs.MO 598: Show issues

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Article ; Online: Remotely operated robotic microscopy for rapid diagnosis of bronchoscopic cytology specimens.

Naso, Julia R / Chan, Jackie / Reisenauer, Janani / Edell, Eric S / Stackhouse, Katherine / Bungum, Aaron O / Vierkant, Robert A / Pierson, Karlyn / Seidl, Andrew / Sturgis, Charles D / Meroueh, Chady / Huang, Yajue / Hartley, Christopher P

Diagnostic cytopathology

2023 Volume 51, Issue 9, Page(s) 554–562

Abstract: Background: Obtaining a diagnosis and treating pulmonary malignancies during the same anesthesia requires either an on-site pathologist or a system for remotely evaluating microscopic images. Cytology specimens are challenging to remotely assess given ... ...

Abstract	Background: Obtaining a diagnosis and treating pulmonary malignancies during the same anesthesia requires either an on-site pathologist or a system for remotely evaluating microscopic images. Cytology specimens are challenging to remotely assess given the need to navigate through dispersed and three-dimensional cell clusters. Remote navigation is possible using robotic telepathology, but data are limited on the ease of use of current systems, particularly for pulmonary cytology. Methods: Air dried modified Wright-Giemsa stained slides from 26 touch preparations of transbronchial biopsies and 27 smears of endobronchial ultrasound guided fine needle aspirations were scored for ease of adequacy assessment and ease of diagnosis on robotic (rmtConnect Microscope) and non-robotic telecytology platforms. Diagnostic classifications were compared between glass slides and the robotic and non-robotic telecytology assessments. Results: Compared to non-robotic telecytology, robotic telecytology had a greater ease of adequacy assessment and non-inferior ease of diagnosis. The median time to diagnosis using robotic telecytology was 85 s (range 28-190 s). Diagnostic categories were concordant for 76% of cases in robotic versus non-robotic telecytology and 78% of cases in robotic telecytology versus glass slide diagnosis. Weighted Cohen's kappa scores for agreement in these comparisons were 0.84 and 0.72, respectively. Conclusions: Use of a remote-controlled robotic microscope improved the ease of adequacy assessment compared to non-robotic telecytology and enabled strongly concordant diagnoses to be expediently rendered. This study provides evidence that modern robotic telecytology is a feasible and user-friendly method of remotely and potentially intraoperatively rendering adequacy assessments and diagnoses on bronchoscopic cytology specimens.
MeSH term(s)	Humans ; Microscopy ; Cytodiagnosis/methods ; Cytological Techniques/methods ; Biopsy, Fine-Needle/methods ; Telepathology/methods
Language	English
Publishing date	2023-06-08
Publishing country	United States
Document type	Journal Article
ZDB-ID	632710-2
ISSN	1097-0339 ; 8755-1039
ISSN (online)	1097-0339
ISSN	8755-1039
DOI	10.1002/dc.25177
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Zs.A 2117: Show issues

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Article ; Online: STAT3 inhibitor in combination with irradiation significantly inhibits cell viability, cell migration, invasion and tumorsphere growth of human medulloblastoma cells.

Pan, Li / Zhang, Ruijie / Ma, Ling / Pierson, Christopher R / Finlay, Jonathan L / Li, Chenglong / Lin, Jiayuh

Cancer biology & therapy

2021 Volume 22, Issue 7-9, Page(s) 430–439

Abstract: Persistent activation of signal transducer and activator of transcription 3 (STAT3) is frequently reported in cancers and plays important roles in tumor progression. Therefore, directly targeting persistent STAT3 signaling is an attractive cancer ... ...

Abstract	Persistent activation of signal transducer and activator of transcription 3 (STAT3) is frequently reported in cancers and plays important roles in tumor progression. Therefore, directly targeting persistent STAT3 signaling is an attractive cancer therapeutic strategy. The aim of this study is to test the inhibitory efficacy of novel STAT3 small molecule inhibitors, LLY17 and LLL12B, in combination with irradiation in human medulloblastoma cells. Both LLY17 and LLL12B inhibit the IL-6-induced and persistent STAT3 phosphorylation in human medulloblastoma cells. Irradiation using 4 Gy alone exhibits some inhibitory effects on medulloblastoma cell viability, and these effects are further enhanced by combining with either STAT3 inhibitor. Irradiation alone also shows certain inhibitory effects on medulloblastoma cell migration and invasion and the combination of LLY17 or LLL12B with irradiation further demonstrates greater inhibitory effects than monotherapy. STAT3 inhibitor alone or irradiation alone exhibits some suppression of medulloblastoma tumorsphere growth, and the combination of LLY17 or LLL12B and irradiation exhibits greater suppression of tumorsphere growth than monotherapy. Combining either STAT3 inhibitor with irradiation reduces the expression of STAT3 downstream targets, Cyclin D1 and Survivin, and induces apoptosis in medulloblastoma cells. These results support that combination of a potent STAT3 inhibitor such as LLY17 or LLL12B with irradiation is an effective and novel therapeutic approach for medulloblastoma.
MeSH term(s)	Apoptosis ; Cell Line, Tumor ; Cell Movement ; Cell Survival ; Cerebellar Neoplasms/drug therapy ; Cerebellar Neoplasms/radiotherapy ; Humans ; Medulloblastoma/drug therapy ; Medulloblastoma/radiotherapy ; STAT3 Transcription Factor/metabolism
Chemical Substances	STAT3 Transcription Factor ; STAT3 protein, human
Language	English
Publishing date	2021-07-13
Publishing country	United States
Document type	Journal Article ; Research Support, N.I.H., Extramural
ZDB-ID	2146305-0
ISSN	1555-8576 ; 1538-4047
ISSN (online)	1555-8576
ISSN	1538-4047
DOI	10.1080/15384047.2021.1951573
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