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Article ; Online: Unprovoked Arterial Thrombosis: Clinical Presentation of Fibromuscular Dysplasia.

Picca, Andrew / Sankar, Amanda D / Jacobson-Kelly, Amanda E / Rodriguez, Vilmarie

2023 Volume 63, Issue 2, Page(s) 235–238

MeSH term(s)	Humans ; Fibromuscular Dysplasia/diagnosis ; Fibromuscular Dysplasia/diagnostic imaging ; Thrombosis/diagnostic imaging ; Thrombosis/drug therapy ; Thrombosis/etiology
Language	English
Publishing date	2023-04-18
Publishing country	United States
Document type	Journal Article
ZDB-ID	207678-0
ISSN	1938-2707 ; 0009-9228
ISSN (online)	1938-2707
ISSN	0009-9228
DOI	10.1177/00099228231169098
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Zs.A 454: Show issues

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Article ; Online: Pediatric arterial thrombosis: A single-institution cohort study of patient characteristics and thrombosis outcomes.

Abdelghani, Eman / Agarwal, Shreya / Stanek, Joseph / Sankar, Amanda / Kerlin, Bryce A / Rodriguez, Vilmarie

Pediatric blood & cancer

2023 Volume 71, Issue 1, Page(s) e30756

Abstract: Background: Arterial thrombosis (AT) is an increasingly recognized complication in pediatrics. Consensus clinical practice guidelines suggest immediate removal of the indwelling arterial catheter and a short course (5-7 days) of anticoagulation. The ... ...

Abstract	Background: Arterial thrombosis (AT) is an increasingly recognized complication in pediatrics. Consensus clinical practice guidelines suggest immediate removal of the indwelling arterial catheter and a short course (5-7 days) of anticoagulation. The optimal duration and modality of antithrombotic therapy in children are yet to be determined. Aims: Describe treatment patterns and outcomes in pediatric patients with AT and explore predictors for complete thrombus resolution or long-term complications. Methods: Single-institution retrospective study. Patients were identified by ICD-9 and ICD-10 codes for the diagnosis of AT or reports of AT on ultrasound from January 1, 2012, to October 1, 2022. Descriptive and logistic regression analyses were used. Results: 101 patients were included. The median age was 2.2 months. The most common underlying diagnoses were congenital heart disease (39.6%) and infection (22.8%). A majority of patients had symptomatic thrombosis in an extremity, and 78% were catheter-associated. 81% of patients received anticoagulation with a median duration of 35 days. Out of the 70 patients who were treated with anticoagulation alone and had a follow-up imaging, 70% had complete resolution after 90 days of anticoagulation. No clear predictors of complete resolution were identified. Eighteen patients had long-term sequelae secondary to arterial insufficiency. Those with infection-associated AT were more likely to have long-term complications. The major and clinically relevant non-major bleeding rate was 11%. Conclusion: Duration of anticoagulation was widely variable, and 70% of patients achieved complete resolution by 90 days of anticoagulation. A significant proportion of patients developed long-term sequelae secondary to arterial insufficiency. Sepsis/infection at the time of diagnosis with AT was more likely to be associated with long-term complications.
MeSH term(s)	Humans ; Child ; Infant ; Cohort Studies ; Retrospective Studies ; Anticoagulants/therapeutic use ; Thrombosis/etiology ; Thrombosis/drug therapy ; Catheterization/adverse effects ; Arterial Occlusive Diseases/complications ; Treatment Outcome
Chemical Substances	Anticoagulants
Language	English
Publishing date	2023-10-30
Publishing country	United States
Document type	Journal Article
ZDB-ID	2131448-2
ISSN	1545-5017 ; 1545-5009
ISSN (online)	1545-5017
ISSN	1545-5009
DOI	10.1002/pbc.30756
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Zs.A 1131: Show issues

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Article ; Online: Intracardiac thrombi in pediatrics: anticoagulation approach and treatment outcomes.

Agarwal, Shreya / Abdelghani, Eman / Stanek, Joseph R / Sankar, Amanda / Cua, Clifford L / Kerlin, Bryce A / Rodriguez, Vilmarie

Research and practice in thrombosis and haemostasis

2023 Volume 7, Issue 8, Page(s) 102266

Abstract: Background: Intracardiac thrombi (ICT) are associated with significant morbidity and mortality. Anticoagulation is the first line of treatment and may be complemented by thrombectomy or thrombolysis. However, optimal anticoagulant duration remains ill- ... ...

Abstract	Background: Intracardiac thrombi (ICT) are associated with significant morbidity and mortality. Anticoagulation is the first line of treatment and may be complemented by thrombectomy or thrombolysis. However, optimal anticoagulant duration remains ill-defined. High-risk features of ICT that may warrant long-term anticoagulation therapy have not been established. Objectives: To describe anticoagulation duration and patterns of ICT resolution. To identify potential risk factors for persistent ICT despite anticoagulation. Methods: A single-institution retrospective chart review identified patients diagnosed with ICT by echocardiogram between January 2014 and March 2022. Descriptive statistics and logistic regression were used. Results: Fifty-one patients with ICT were identified. Median age at diagnosis was 9.2 years (IQR, 0.4-15.2). The most common underlying diagnoses were congenital heart disease (41%), infection (25%), and malignancy (24%). The majority of ICT were in the right atrium ( Conclusion: Our study demonstrates the wide variability in duration of anticoagulation for children with ICT. Majority of the individuals benefit from 3-to-6 month treatment; however, individuals with a central venous line may benefit from a longer course of anticoagulation. Further large-scale studies are recommended to validate our findings.
Language	English
Publishing date	2023-11-24
Publishing country	United States
Document type	Journal Article
ISSN	2475-0379
ISSN (online)	2475-0379
DOI	10.1016/j.rpth.2023.102266
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Article ; Online: A regional anticoagulation program improves safety and outcomes for both children and adults.

Rodriguez, Vilmarie / Stanek, Joseph / Cua, Clifford L / Sankar, Amanda / Giver, Jean / Monda, Kay / Canini, Joan / Dunn, Amy L / Kerlin, Bryce A

Journal of thrombosis and thrombolysis

2023 Volume 56, Issue 1, Page(s) 27–36

Abstract: Background: Evidence-based anticoagulation programs usually serve a local, adult patient population. Here we report outcomes for a regional combined pediatric-adult program.: Aims: The aims of this study were: (1) Compare the pre- vs. post- ... ...

Abstract	Background: Evidence-based anticoagulation programs usually serve a local, adult patient population. Here we report outcomes for a regional combined pediatric-adult program. Aims: The aims of this study were: (1) Compare the pre- vs. post-implementation quality of therapy (% time in therapeutic range (%TTR) and compliance). (2) Assess anticoagulant-relevant outcomes (bleeding and thrombotic complications). Methods: Data were collected for the years 2014-2019. Rosendaal linear interpolation was used to calculate %TTR. Bleeding complications were categorized using ISTH-SSC standard nomenclature and new thrombotic events were reviewed. Results: The patients were divided into a long-term warfarin group (N = 308), 80.2% of whom had cardiac-related therapeutic indications (median age 24y), and a second group (N = 114) comprised of short-term and non-warfarin long-term anticoagulation (median age 16y). Median %TTR for those on long-term warfarin was 78.9%. The incidence of major and clinically relevant non-major bleeding events was 1.65 and 2.43 /100 person-years of warfarin use, respectively. Thromboembolism (TE) incidence was 0.78/100 patient-years of warfarin use. Neither bleeding nor thrombosis was associated with %TTR (p = 0.48). Anticoagulant indication was the only variable associated with bleeding risk (p = 0.005). The second group had no on-therapy TE events but 7.9% experienced bleeding. Complete data were available for a randomly sampled pre-program warfarin group (N = 26). Median %TTR improved from 17.5 to 87% pre- vs. post-implementation. Similarly, compliance (defined as ≥ 1 INR/month) improved by 34.3%. Conclusions: In conclusion, this program significantly improved and sustained %TTR and compliance. The lack of association between bleeding and thrombosis events and %TTR may be related to the high median %TTR (> 70%) achieved by this approach.
MeSH term(s)	Humans ; Child ; Adult ; Young Adult ; Adolescent ; International Normalized Ratio ; Warfarin/adverse effects ; Anticoagulants/adverse effects ; Blood Coagulation ; Hemorrhage/chemically induced ; Hemorrhage/drug therapy ; Thromboembolism/drug therapy ; Thrombosis/drug therapy ; Treatment Outcome
Chemical Substances	Warfarin (5Q7ZVV76EI) ; Anticoagulants
Language	English
Publishing date	2023-04-24
Publishing country	Netherlands
Document type	Journal Article
ZDB-ID	1230645-9
ISSN	1573-742X ; 0929-5305
ISSN (online)	1573-742X
ISSN	0929-5305
DOI	10.1007/s11239-023-02806-w
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Zs.A 4352: Show issues

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Article: The evolution of recombinant factor replacement for hemophilia.

Sankar, Amanda D / Weyand, Angela C / Pipe, Steven W

Transfusion and apheresis science : official journal of the World Apheresis Association : official journal of the European Society for Haemapheresis

2019 Volume 58, Issue 5, Page(s) 596–600

Abstract: Hemophilia A and Hemophilia B are the most common of the severe bleeding disorders and are caused by a deficiency in blood clotting factor VIII or factor IX respectively. Factor replacement therapy has been the cornerstone of treatment to treat life ... ...

Abstract	Hemophilia A and Hemophilia B are the most common of the severe bleeding disorders and are caused by a deficiency in blood clotting factor VIII or factor IX respectively. Factor replacement therapy has been the cornerstone of treatment to treat life threatening bleeds and prevent joint disease. The treatment of hemophilia has evolved tremendously over the past five decades from fresh frozen plasma as the only available therapy to more specific plasma-derived and recombinant-derived factor replacement. Now due to innovations in bioengineering, there are even more efficacious factor replacement options available to patients. Here we review these recent advancements and their impact on the treatment and management of hemophilia.
MeSH term(s)	Factor IX/genetics ; Factor IX/metabolism ; Factor IX/therapeutic use ; Factor VIII/genetics ; Factor VIII/metabolism ; Factor VIII/therapeutic use ; Hemophilia A/blood ; Hemophilia A/drug therapy ; Hemophilia A/genetics ; Hemophilia B/blood ; Hemophilia B/drug therapy ; Hemophilia B/genetics ; Humans ; Recombinant Proteins/therapeutic use
Chemical Substances	Recombinant Proteins ; F8 protein, human (839MOZ74GK) ; Factor VIII (9001-27-8) ; Factor IX (9001-28-9)
Language	English
Publishing date	2019-08-09
Publishing country	England
Document type	Journal Article ; Review
ZDB-ID	2046795-3
ISSN	1878-1683 ; 1473-0502
ISSN (online)	1878-1683
ISSN	1473-0502
DOI	10.1016/j.transci.2019.08.010
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Zs.A 1739: Show issues

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Article ; Online: Hemostatic and Thrombotic Considerations in the Diagnosis and Management of Childhood Arterial Ischemic Stroke: A Narrative Review.

Kumar, Riten / Sun, Lisa R / Rodriguez, Vilmarie / Sankar, Amanda / Sharma, Mukta / Meoded, Avner / Brandão, Leonardo R / Goldenberg, Neil A

Seminars in pediatric neurology

2022 Volume 43, Page(s) 101003

Abstract: Although rare in children, arterial ischemic stroke (AIS) is associated with increased mortality and neurological morbidity. The incidence of AIS after the neonatal period is approximately 1-2/100,000/year, with an estimated mortality of 3-7%. A ... ...

Abstract	Although rare in children, arterial ischemic stroke (AIS) is associated with increased mortality and neurological morbidity. The incidence of AIS after the neonatal period is approximately 1-2/100,000/year, with an estimated mortality of 3-7%. A significant proportion of children surviving AIS experience life-long neurological deficits including hemiparesis, epilepsy, and cognitive delays. The low incidence of childhood AIS coupled with atypical clinical-presentation and lack of awareness contribute to delay in diagnosis and consequently, the early initiation of treatment. While randomized-clinical trials have demonstrated the efficacy and safety of reperfusion therapies including thrombolysis and endovascular thrombectomy in appropriately-selected adult patients, similar data for children are unavailable. Consequently, clinical decisions surrounding reperfusion therapy in childhood AIS are either extrapolated from adult data or based on local experience. The etiology of childhood AIS is multifactorial, often occurring in the setting of both acquired and congenital risk-factors including thrombophilia. While multiple studies have investigated the association of thrombophilia with incident childhood AIS, its impact on stroke recurrence and therefore duration and intensity of antithrombotic therapy is less clear. Despite these limitations, a significant progress has been made over the last decade in the management of childhood AIS. This progress can be attributed to international consortiums, and in selected cohorts to federally-funded clinical trials. In this narrative review, the authors have systematically appraised the literature and summarize the hemostatic and thrombotic considerations in the diagnosis and management of childhood AIS focusing on the evidence supporting reperfusion therapies, relevance of thrombophilia testing, and duration and drug choices for secondary-prophylaxis.
MeSH term(s)	Child ; Infant, Newborn ; Humans ; Brain Ischemia/complications ; Brain Ischemia/diagnosis ; Brain Ischemia/therapy ; Ischemic Stroke ; Hemostatics ; Stroke/diagnosis ; Stroke/etiology ; Stroke/therapy ; Thrombophilia/complications ; Randomized Controlled Trials as Topic
Chemical Substances	Hemostatics
Language	English
Publishing date	2022-10-04
Publishing country	United States
Document type	Review ; Journal Article
ZDB-ID	1290000-x
ISSN	1558-0776 ; 1071-9091
ISSN (online)	1558-0776
ISSN	1071-9091
DOI	10.1016/j.spen.2022.101003
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Zs.A 4772: Show issues

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Article ; Online: Multisystem Inflammatory Syndrome in Children and Cardiac Involvement: A Quaternary Center Experience.

Cruz Vidal, Diego / Lee, Simon / Ardoin, Stacy P / Dalmacy, Djhenne / Chaparro, Juan / Blaney, Cristin / Rodriguez, Vilmarie / Sankar, Amanda / Akoghlanian, Shoghik / Lisciandro, Richard / Washam, Matthew / Abraham, Roshini S / Leber, Amy / Eby, Meika / Bennett, Berkeley / Erdem, Guliz

The Pediatric infectious disease journal

2024 Volume 43, Issue 5, Page(s) e160–e163

Abstract: We prospectively analyzed clinical and laboratory characteristics associated with cardiac involvement and severe presentation in multisystem inflammatory syndrome in children. Of 146 patients, 66 (45.2%) had cardiac dysfunction and 26 (17.8%) had ... ...

Abstract	We prospectively analyzed clinical and laboratory characteristics associated with cardiac involvement and severe presentation in multisystem inflammatory syndrome in children. Of 146 patients, 66 (45.2%) had cardiac dysfunction and 26 (17.8%) had coronary artery abnormalities. Lower serum albumin levels, absolute lymphocyte and platelet counts, and elevated ferritin, fibrinogen, d-dimer and interleukin-6 levels were associated with cardiac dysfunction. Possible treatment complications were identified.
MeSH term(s)	Child ; Humans ; COVID-19/complications ; Interleukin-6 ; Laboratories ; Heart Diseases ; Systemic Inflammatory Response Syndrome/diagnosis
Chemical Substances	Interleukin-6
Language	English
Publishing date	2024-02-06
Publishing country	United States
Document type	Journal Article
ZDB-ID	392481-6
ISSN	1532-0987 ; 0891-3668
ISSN (online)	1532-0987
ISSN	0891-3668
DOI	10.1097/INF.0000000000004266
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Article ; Online: Line Associated Thrombosis in Pediatric Patients With NF-κB Pathway Variants.

Michniacki, Thomas F / Atchison, Christie / Walkovich, Kelly / Sankar, Amanda / McGrath, Mary / Weyand, Angela C

Journal of pediatric hematology/oncology

2020 Volume 43, Issue 3, Page(s) e436–e437

Abstract: Our report explores the complex role that nuclear factor-kappa B (NF-κB) plays in thrombosis formation, inflammation, and immunity; while additionally demonstrating that patients with NF-κB pathway pathogenic variants appear to carry a substantial ... ...

Abstract	Our report explores the complex role that nuclear factor-kappa B (NF-κB) plays in thrombosis formation, inflammation, and immunity; while additionally demonstrating that patients with NF-κB pathway pathogenic variants appear to carry a substantial thrombotic risk, particularly when secondary thrombotic risk factors are present. We propose that prophylactic anticoagulation should be strongly considered in such patients during high-risk situations and provide additional hematologic management strategies for those with NF-κB pathway alterations. We hope our work also calls to attention the potential need for a broader assessment of venous thromboembolism risk in patients with immune dysregulation to better delineate which patient populations may benefit from anticoagulation prophylaxis.
MeSH term(s)	Anticoagulants/therapeutic use ; Child, Preschool ; Heparin, Low-Molecular-Weight/therapeutic use ; Humans ; Infant, Newborn ; Male ; NF-kappa B/genetics ; Point Mutation ; Polymorphism, Single Nucleotide ; Thrombosis/drug therapy ; Thrombosis/genetics ; Whole Exome Sequencing
Chemical Substances	Anticoagulants ; Heparin, Low-Molecular-Weight ; NF-kappa B
Language	English
Publishing date	2020-02-03
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	1231152-2
ISSN	1536-3678 ; 1077-4114 ; 0192-8562
ISSN (online)	1536-3678
ISSN	1077-4114 ; 0192-8562
DOI	10.1097/MPH.0000000000001739
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Zs.A 1537: Show issues

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Article: Drive-Through Anticoagulation Clinic During the COVID-19 Pandemic.

Giver, Jean / Dunn, Amy L / Sankar, Amanda / Stanek, Joseph / Monda, Kay / Canini, Joan / Kerlin, Bryce A / Rodriguez, Vilmarie

The journal for nurse practitioners : JNP

2021 Volume 18, Issue 1, Page(s) 92–96

Abstract: An innovative approach to anticoagulation management during the COVID-19 pandemic was used at our center that allowed patients to stay in their vehicle while our anticoagulation advanced practice registered nurse obtained blood for point-of-care ... ...

Abstract	An innovative approach to anticoagulation management during the COVID-19 pandemic was used at our center that allowed patients to stay in their vehicle while our anticoagulation advanced practice registered nurse obtained blood for point-of-care international normalized ratio (INR) testing while education and counseling were completed. A significant improvement in the median percentage of INR within the therapeutic range was observed among the patients who used the drive-through clinic. A small group of patients improved compliance to anticoagulation monitoring. Clinical care models, such as this clinic approach may improve patient compliance and adherence to anticoagulation beyond the pandemic needs.
Language	English
Publishing date	2021-09-08
Publishing country	United States
Document type	Journal Article
ZDB-ID	2202063-9
ISSN	1878-058X ; 1555-4155
ISSN (online)	1878-058X
ISSN	1555-4155
DOI	10.1016/j.nurpra.2021.08.026
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Article ; Online: Modulation of the HIF2α-NCOA4 axis in enterocytes attenuates iron loading in a mouse model of hemochromatosis.

Das, Nupur K / Jain, Chesta / Sankar, Amanda / Schwartz, Andrew J / Santana-Codina, Naiara / Solanki, Sumeet / Zhang, Zhiguo / Ma, Xiaoya / Parimi, Sanjana / Rui, Liangyou / Mancias, Joseph D / Shah, Yatrik M

Blood

2021 Volume 139, Issue 16, Page(s) 2547–2552

Abstract: Intestinal iron absorption is activated during increased systemic demand for iron. The best-studied example is iron deficiency anemia, which increases intestinal iron absorption. Interestingly, the intestinal response to anemia is very similar to that of ...

Abstract	Intestinal iron absorption is activated during increased systemic demand for iron. The best-studied example is iron deficiency anemia, which increases intestinal iron absorption. Interestingly, the intestinal response to anemia is very similar to that of iron overload disorders, as both the conditions activate a transcriptional program that leads to a hyperabsorption of iron via the transcription factor hypoxia-inducible factor 2α (HIF2α). However, pathways for selective targeting of intestine-mediated iron overload remain unknown. Nuclear receptor coactivator 4 (NCOA4) is a critical cargo receptor for autophagic breakdown of ferritin and the subsequent release of iron, in a process termed ferritinophagy. Our work demonstrates that NCOA4-mediated intestinal ferritinophagy is integrated into systemic iron demand via HIF2α. To demonstrate the importance of the intestinal HIF2α/ferritinophagy axis in systemic iron homeostasis, whole-body and intestine-specific NCOA4-/- mouse lines were generated and assessed. The analyses revealed that the intestinal and systemic response to iron deficiency was not altered after disruption of intestinal NCOA4. However, in a mouse model of hemochromatosis, ablation of intestinal NCOA4 was protective against iron overload. Therefore, NCOA4 can be selectively targeted for the management of iron overload disorders without disrupting the physiological processes involved in the response to systemic iron deficiency.
MeSH term(s)	Anemia ; Animals ; Basic Helix-Loop-Helix Transcription Factors/metabolism ; Enterocytes/metabolism ; Hemochromatosis/genetics ; Iron/metabolism ; Iron Overload ; Mice ; Nuclear Receptor Coactivators/genetics ; Transcription Factors/metabolism
Chemical Substances	Basic Helix-Loop-Helix Transcription Factors ; NcoA4 protein, mouse ; Nuclear Receptor Coactivators ; Transcription Factors ; endothelial PAS domain-containing protein 1 (1B37H0967P) ; Iron (E1UOL152H7)
Language	English
Publishing date	2021-10-09
Publishing country	United States
Document type	Journal Article ; Research Support, N.I.H., Extramural
ZDB-ID	80069-7
ISSN	1528-0020 ; 0006-4971
ISSN (online)	1528-0020
ISSN	0006-4971
DOI	10.1182/blood.2021013452
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