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Article ; Online: 50 Years Ago in TheJournal ofPediatrics: Cystic Fibrosis with Extensive Fat Replacement of the Liver.

Palermo, Joseph J

2015 Volume 168, Page(s) 49

MeSH term(s)	Cystic Fibrosis/history ; Fatty Liver/history ; History, 20th Century ; Humans ; Pediatrics/history
Language	English
Publishing date	2015-10-24
Publishing country	United States
Document type	Classical Article ; Historical Article ; Journal Article
ZDB-ID	3102-1
ISSN	1097-6833 ; 0022-3476
ISSN (online)	1097-6833
ISSN	0022-3476
DOI	10.1016/j.jpeds.2015.08.061
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Article ; Online: 50 years ago in the Journal of Pediatrics: trypsinogen deficiency disease.

Palermo, Joseph J

The Journal of pediatrics

2015 Volume 166, Issue 2, Page(s) 308

MeSH term(s)	Deficiency Diseases/history ; History, 20th Century ; Humans ; Infant ; Pediatrics/history ; Periodicals as Topic/history ; Trypsinogen/deficiency ; Trypsinogen/history
Chemical Substances	Trypsinogen (9002-08-8)
Language	English
Publishing date	2015-02
Publishing country	United States
Document type	Historical Article ; Journal Article
ZDB-ID	3102-1
ISSN	1097-6833 ; 0022-3476
ISSN (online)	1097-6833
ISSN	0022-3476
DOI	10.1016/j.jpeds.2014.08.062
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Article ; Online: Associations of Liver Stiffness Measured by Ultrasound Shear-Wave Elastography With Portal Hypertension and Circulatory Failure in Individuals With Fontan Circulation.

Derinkuyu, Betul E / Dillman, Jonathan R / Lubert, Adam M / Palermo, Joseph J / Opotowsky, Alexander R / Trout, Andrew T

AJR. American journal of roentgenology

2023 Volume 222, Issue 1, Page(s) e2329640

Abstract: BACKGROUND. ...

Abstract	BACKGROUND.
MeSH term(s)	Humans ; Male ; Female ; Young Adult ; Adult ; Child ; Fontan Procedure ; Elasticity Imaging Techniques/methods ; Retrospective Studies ; Hypertension, Portal ; Ascites/pathology ; Liver/diagnostic imaging ; Liver Cirrhosis/pathology
Language	English
Publishing date	2023-08-02
Publishing country	United States
Document type	Journal Article
ZDB-ID	82076-3
ISSN	1546-3141 ; 0361-803X ; 0092-5381
ISSN (online)	1546-3141
ISSN	0361-803X ; 0092-5381
DOI	10.2214/AJR.23.29640
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Article ; Online: The impact of the COVID-19 pandemic on individuals with Fontan circulation: Focus on gaps in care.

Mahendran, Arjun / Fahnhorst, Sarah / Alexander, Craig / Brown, Nicole / Cnota, James F / Divanovic, Allison / Heydarian, Haleh / Hirsch, Russel / Opotowsky, Alexander R / Palermo, Joseph J / Szugye, Cassandra / Lubert, Adam M

International journal of cardiology. Congenital heart disease

2023 Volume 11, Page(s) 100439

Abstract: Background: Gaps in subspecialty cardiology care could potentially delay identification and care for multi-organ complications common in patients with Fontan circulation. This study analyzed the frequency of gaps in care for individuals with Fontan ... ...

Abstract	Background: Gaps in subspecialty cardiology care could potentially delay identification and care for multi-organ complications common in patients with Fontan circulation. This study analyzed the frequency of gaps in care for individuals with Fontan circulation during the COVID-19 pandemic and associated demographic and clinical factors. Methods: This retrospective study evaluated individuals with Fontan circulation followed at our center since 2010. A gap in care was defined as an absence of any formal cardiology provider-patient contact (clinic visit or telehealth) for >15 months. Results: Over a third of 308 patients with Fontan circulation experienced at least one gap in care between 2010 and 2022, and 77 experienced a gap in care during the COVID-19 pandemic. Of this latter group, 27 (35%) had never experienced a prior gap in cardiology care until the pandemic. Those who experienced gaps in care during the pandemic were on average older (18.0 [IQR 9.6-25.6] vs. 14.2 [7.2-21.2] years, p = 0.01), more likely to be of Black/African American race (23.4% vs 7.4%, p = 0.001), and less likely to have a diagnosis of protein-losing enteropathy or plastic bronchitis (0% vs. 8.6%, p = 0.005). Those without a gap in care during the pandemic were more likely to have utilized telehealth visits (13% vs 3%, p = 0.02). Conclusion: Gaps in care are common and appear to have been exacerbated by the COVID-19 pandemic in those with a Fontan circulation. Such gaps are particularly common among African American and adult patients, and may potentially be mitigated by expanding telehealth access.
Language	English
Publishing date	2023-01-11
Publishing country	Netherlands
Document type	Journal Article
ISSN	2666-6685
ISSN (online)	2666-6685
DOI	10.1016/j.ijcchd.2023.100439
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Article ; Online: Isosorbide DiNitrate Effect on Hemodynamic Profile, Liver Stiffness, and Exercise Tolerance in Fontan Circulation (The NEET Clinical Trial).

Bigelow, Amee M / Riggs, Kyle W / Morales, David L S / Opotowsky, Alexander R / Lubert, Adam M / Dillman, Jonathan R / Veldtman, Gruschen R / Heydarian, Haleh C / Trout, Andrew T / Cooper, David S / Goldstein, Stuart L / Chin, Clifford / Palermo, Joseph J / Ollberding, Nicholas J / Mays, Wayne A / Alsaied, Tarek

Pediatric cardiology

2023

Abstract: After Fontan operation, decreased venous capacitance and venoconstriction are adaptive mechanisms to maintain venous return and cardiac output. The consequent higher venous pressure may adversely impact end-organ function, exercise capacity and result in ...

Abstract	After Fontan operation, decreased venous capacitance and venoconstriction are adaptive mechanisms to maintain venous return and cardiac output. The consequent higher venous pressure may adversely impact end-organ function, exercise capacity and result in worse clinical outcomes. This pilot study evaluated the safety and effect of isosorbide dinitrate (ISDN), a venodilator, on exercise capacity, peripheral venous pressure (PVP), and liver stiffness in patients with Fontan circulation. In this prospective single-arm trial, 15 individuals with Fontan circulation were evaluated at baseline and after 4 weeks of therapeutic treatment with ISDN. Primary aims were to assess the safety of ISDN and the effect on maximal exercise. We also aimed to evaluate the effect of ISDN on ultrasound-assessed liver stiffness, markers of submaximal exercise, and PVP at rest and peak exercise. Repeated measures t-tests were used to assess change in variables of interest in response to ISDN. Mean age was 23.5 ± 9.2 years (range 11.2-39.0 years), and 10/15 (67%) were male. There was no statistically significant change in peak VO
Language	English
Publishing date	2023-04-21
Publishing country	United States
Document type	Journal Article
ZDB-ID	800857-7
ISSN	1432-1971 ; 0172-0643
ISSN (online)	1432-1971
ISSN	0172-0643
DOI	10.1007/s00246-023-03156-3
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Article ; Online: Abdominal CT and MRI Findings of Portal Hypertension in Children and Adults with Fontan Circulation.

Zafar, Faizeen / Lubert, Adam M / Trout, Andrew T / Katz, David A / Palermo, Joseph J / Opotowsky, Alexander R / Lorts, Angela / Chin, Clifford / Powell, Adam W / Villa, Chet R / Anwar, Nadeem / Morales, David L / Dillman, Jonathan R / Alsaied, Tarek

Radiology

2022 Volume 303, Issue 3, Page(s) 557–565

Abstract: Background Portal hypertension in the Fontan circulation is a function of elevated systemic venous pressure and liver fibrosis. Purpose To quantify the prevalence of radiologic evidence of portal hypertension and elevated VAST score (one point each for ... ...

Abstract	Background Portal hypertension in the Fontan circulation is a function of elevated systemic venous pressure and liver fibrosis. Purpose To quantify the prevalence of radiologic evidence of portal hypertension and elevated VAST score (one point each for varices, ascites, splenomegaly, and thrombocytopenia) of 2 or greater in children and adults with Fontan circulation and to determine the association with hemodynamics and adverse outcomes. Materials and Methods This was a retrospective study of individuals with Fontan circulation who underwent abdominal MRI or CT for focal liver lesion surveillance between January 2012 and December 2019. Portal hypertension was defined as the presence of at least two of the following: varices, ascites, or splenomegaly. Fontan deterioration was defined as a composite of heart failure signs or symptoms requiring diuretic escalation, placement of a ventricular assist device, heart transplant, or death. Relationships between variables and the composite end point were assessed using univariable and multivariable logistic regression. Results A total of 123 patients (age range, 9-55 years; 32 children) were evaluated (median age, 23 years; IQR, 17-30 years; 63 male patients). Median time since diagnosis of Fontan circulation was 16 years (IQR, 12-23 years). Twenty-five of the 123 patients (20%) had radiologic evidence of portal hypertension, and 34 (28%) had a VAST score of 2 or greater. Fontan deterioration occurred in 25 of the 123 patients (20%); median follow-up duration was 0.4 year (IQR, 0.1-3.1 years). Compared with patients who had Fontan circulation without deterioration, patients with Fontan deterioration were more likely to have moderate or severe ventricular systolic dysfunction (
MeSH term(s)	Adolescent ; Adult ; Ascites/etiology ; Child ; Fontan Procedure/adverse effects ; Heart Defects, Congenital/diagnostic imaging ; Heart Defects, Congenital/surgery ; Humans ; Hypertension, Portal/diagnostic imaging ; Hypertension, Portal/etiology ; Magnetic Resonance Imaging ; Male ; Middle Aged ; Retrospective Studies ; Splenomegaly/etiology ; Tomography, X-Ray Computed ; Varicose Veins ; Young Adult
Language	English
Publishing date	2022-03-15
Publishing country	United States
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	80324-8
ISSN	1527-1315 ; 0033-8419
ISSN (online)	1527-1315
ISSN	0033-8419
DOI	10.1148/radiol.211037
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Article ; Online: Hepatic Steatosis in Patients With Single Ventricle and a Fontan Circulation.

Katz, David A / Peck, Daniel / Lubert, Adam M / Possner, Mathias / Zafar, Faizeen / Trout, Andrew T / Palermo, Joseph J / Anwar, Nadeem / Dillman, Jonathan R / Powell, Adam W / Xanthakos, Stavra A / Opotowsky, Alexander R / Veldtman, Gruschen / Alsaied, Tarek

Journal of the American Heart Association

2021 Volume 10, Issue 9, Page(s) e019942

Abstract: Background Hepatic steatosis, caused by nonalcoholic fatty liver disease, is a leading cause of chronic liver disease. The interplay between hepatic steatosis and the development of liver disease following the Fontan procedure is not well understood. ... ...

Abstract	Background Hepatic steatosis, caused by nonalcoholic fatty liver disease, is a leading cause of chronic liver disease. The interplay between hepatic steatosis and the development of liver disease following the Fontan procedure is not well understood. This study examined the prevalence and associations of hepatic steatosis in patients with a Fontan circulation. Methods and Results This was a single-center retrospective study of 95 patients with a Fontan circulation with liver magnetic resonance imaging performed between 2012 and 2019. The average age at magnetic resonance imaging was 21.5±8.5 years. The percent liver fat signal was determined using magnetic resonance chemical shift-encoded proton density fat fraction imaging. Hepatic steatosis was defined as liver fat ≥5% and was present in 10.5% of the cohort. The presence of hepatic steatosis was associated with higher body mass index (29±4 versus 24±6 kg/m
MeSH term(s)	Adolescent ; Adult ; Child ; Echocardiography ; Fatty Liver/diagnosis ; Fatty Liver/epidemiology ; Fatty Liver/etiology ; Female ; Follow-Up Studies ; Fontan Procedure/adverse effects ; Heart Defects, Congenital/diagnosis ; Heart Defects, Congenital/surgery ; Heart Ventricles/abnormalities ; Heart Ventricles/diagnostic imaging ; Heart Ventricles/surgery ; Humans ; Liver/pathology ; Magnetic Resonance Imaging ; Male ; Middle Aged ; Ohio/epidemiology ; Postoperative Complications/diagnosis ; Postoperative Complications/epidemiology ; Postoperative Complications/etiology ; Prevalence ; Prognosis ; Retrospective Studies ; Young Adult
Language	English
Publishing date	2021-04-21
Publishing country	England
Document type	Journal Article
ZDB-ID	2653953-6
ISSN	2047-9980 ; 2047-9980
ISSN (online)	2047-9980
ISSN	2047-9980
DOI	10.1161/JAHA.120.019942
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Article ; Online: Fontan-Associated Dyslipidemia.

Lubert, Adam M / Alsaied, Tarek / Palermo, Joseph J / Anwar, Nadeem / Urbina, Elaine M / Brown, Nicole M / Alexander, Craig / Almeneisi, Hassan / Wu, Fred / Leventhal, Andrew R / Aldweib, Nael / Mendelson, Michael / Opotowsky, Alexander R

Journal of the American Heart Association

2021 Volume 10, Issue 7, Page(s) e019578

Abstract: Background Hypocholesterolemia is a marker of liver disease, and patients with a Fontan circulation may have hypocholesterolemia secondary to Fontan-associated liver disease or inflammation. We investigated circulating lipids in adults with a Fontan ... ...

Abstract	Background Hypocholesterolemia is a marker of liver disease, and patients with a Fontan circulation may have hypocholesterolemia secondary to Fontan-associated liver disease or inflammation. We investigated circulating lipids in adults with a Fontan circulation and assessed the associations with clinical characteristics and adverse events. Methods and Results We enrolled 164 outpatients with a Fontan circulation, aged ≥18 years, in the Boston Adult Congenital Heart Disease Biobank and compared them with 81 healthy controls. The outcome was a combined outcome of nonelective cardiovascular hospitalization or death. Participants with a Fontan (median age, 30.3 [interquartile range, 22.8-34.3 years], 42% women) had lower total cholesterol (149.0±30.1 mg/dL versus 190.8±41.4 mg/dL,
MeSH term(s)	Adult ; Biomarkers/blood ; Cholesterol/blood ; Dyslipidemias/blood ; Dyslipidemias/epidemiology ; Dyslipidemias/etiology ; Female ; Follow-Up Studies ; Fontan Procedure/adverse effects ; Heart Defects, Congenital/blood ; Heart Defects, Congenital/surgery ; Humans ; Incidence ; Male ; Middle Aged ; Postoperative Complications/blood ; Postoperative Complications/epidemiology ; Postoperative Complications/etiology ; Prognosis ; Prospective Studies ; United States/epidemiology ; Young Adult
Chemical Substances	Biomarkers ; Cholesterol (97C5T2UQ7J)
Language	English
Publishing date	2021-03-31
Publishing country	England
Document type	Journal Article ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't
ZDB-ID	2653953-6
ISSN	2047-9980 ; 2047-9980
ISSN (online)	2047-9980
ISSN	2047-9980
DOI	10.1161/JAHA.120.019578
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Article ; Online: Relation of Liver Volume to Adverse Cardiovascular Events in Adolescents and Adults With Fontan Circulation.

Lubert, Adam M / Opotowsky, Alexander R / Palermo, Joseph J / Alsaied, Tarek / Szugye, Cassandra / Anwar, Nadeem / Tiao, Gregory M / Lorts, Angela / Dillman, Jonathan R / Trout, Andrew T

The American journal of cardiology

2021 Volume 165, Page(s) 88–94

Abstract: Elevated central venous pressure in those with Fontan circulation causes liver congestion and hepatomegaly. We assessed if liver volume by magnetic resonance imaging (MRI) is associated with adverse cardiovascular outcomes. Retrospective study of 122 ... ...

Abstract	Elevated central venous pressure in those with Fontan circulation causes liver congestion and hepatomegaly. We assessed if liver volume by magnetic resonance imaging (MRI) is associated with adverse cardiovascular outcomes. Retrospective study of 122 patients with Fontan circulation who were >10 years old and had a liver MRI with magnetic resonance elastography. Liver volume (ml) was measured by manual segmentation from axial T2-weighted images and was indexed to body surface area. The composite outcome included death, heart transplant, ventricular assist device placement, or nonelective cardiovascular hospitalization. The median age at the time of MRI was 18.9 (interquartile range 15.8 to 25.9) years, and 47% of the patients were women. The mean indexed liver volume was 1,133 ± 180 ml/m
MeSH term(s)	Adolescent ; Adult ; Central Venous Pressure ; Elasticity Imaging Techniques ; Female ; Fontan Procedure ; Heart Defects, Congenital/surgery ; Heart Failure/epidemiology ; Heart Failure/therapy ; Heart Transplantation/statistics & numerical data ; Heart-Assist Devices/statistics & numerical data ; Hepatomegaly/diagnostic imaging ; Hepatomegaly/epidemiology ; Hospitalization ; Humans ; Liver/diagnostic imaging ; Liver/pathology ; Magnetic Resonance Imaging ; Male ; Mortality ; Organ Size ; Proportional Hazards Models ; Young Adult
Language	English
Publishing date	2021-12-07
Publishing country	United States
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	80014-4
ISSN	1879-1913 ; 0002-9149
ISSN (online)	1879-1913
ISSN	0002-9149
DOI	10.1016/j.amjcard.2021.10.045
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Article ; Online: Relation of Magnetic Resonance Elastography to Fontan Circulatory Failure in a Cohort of Pediatric and Adult Patients.

Brayer, Samuel W / Zafar, Faizeen / Lubert, Adam M / Trout, Andrew T / Palermo, Joseph J / Opotowsky, Alexander R / Anwar, Nadeem / Dillman, Jonathan R / Alsaied, Tarek

Pediatric cardiology

2021 Volume 42, Issue 8, Page(s) 1871–1878

Abstract: Elevated magnetic resonance elastography (MRE)-derived liver stiffness may be associated with worse outcomes in people with Fontan circulation. We sought to evaluate the association between liver stiffness and Fontan failure or portal hypertension. ... ...

Abstract	Elevated magnetic resonance elastography (MRE)-derived liver stiffness may be associated with worse outcomes in people with Fontan circulation. We sought to evaluate the association between liver stiffness and Fontan failure or portal hypertension. Single center cross-sectional retrospective study of people with Fontan circulation who underwent MRE between 2011 and 2020. The cohort was divided into adult (age ≥ 21 years) and pediatric (< 21 years) groups. Fontan circulatory failure (FF) was defined as any of the following: death, transplantation, ventricular assist device, heart failure symptoms requiring escalation of diuretics. Radiologic portal hypertension was defined as the presence of one or more of the following: splenomegaly, ascites, or gastrointestinal varices. 128 patients were included (average age = 22.6 ± 8.7 years) and 58 (45%) were children. Median liver stiffness was 4.3 kPa (interquartile range (IQR) 3.8-5.8) for the entire cohort. Thirty patients (23%) developed FF (16 adults, 14 children). Liver stiffness was higher in adults with FF compared to those without FF (4.9 (IQR 4.0-6.0) vs. 4.2 (IQR 3.8-4.7) kPa, p = 0.04). There was no difference in liver stiffness between pediatric patients with and without FF (4.4 (IQR 4.1-5.4) vs. 4.4 (IQR 3.8-5.0), p = 0.5). Adults with radiologic portal hypertension and adults with moderate or severe atrioventricular valve regurgitation had higher liver stiffness than adults without. MRE-derived liver stiffness is associated with atrioventricular valve regurgitation, portal hypertension, and poor clinical outcomes in adults with Fontan circulation. There was no association between liver stiffness and FF in pediatric patients. This difference may be due to the progressive nature of Fontan-associated liver disease.
MeSH term(s)	Adolescent ; Adult ; Child ; Cross-Sectional Studies ; Elasticity Imaging Techniques ; Fontan Procedure/adverse effects ; Humans ; Liver/diagnostic imaging ; Liver/pathology ; Liver Cirrhosis/pathology ; Retrospective Studies ; Shock/pathology ; Young Adult
Language	English
Publishing date	2021-08-26
Publishing country	United States
Document type	Journal Article
ZDB-ID	800857-7
ISSN	1432-1971 ; 0172-0643
ISSN (online)	1432-1971
ISSN	0172-0643
DOI	10.1007/s00246-021-02707-w
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