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Artikel ; Online: Short-term Outcomes of Corticosteroid Monotherapy in COVID-19-Associated Multisystem Inflammatory Syndrome in Children-Handle With Caution-Reply.

Villacis-Nunez, D Sofia / Prahalad, Sampath / Jaggi, Preeti

2022 Band 176, Heft 9, Seite(n) 946–947

Mesh-Begriff(e)	Adrenal Cortex Hormones/therapeutic use ; COVID-19/complications ; COVID-19/drug therapy ; Child ; Humans ; SARS-CoV-2 ; Systemic Inflammatory Response Syndrome
Chemische Substanzen	Adrenal Cortex Hormones
Sprache	Englisch
Erscheinungsdatum	2022-07-11
Erscheinungsland	United States
Dokumenttyp	Journal Article ; Comment
ZDB-ID	2701223-2
ISSN	2168-6211 ; 2168-6203
ISSN (online)	2168-6211
ISSN	2168-6203
DOI	10.1001/jamapediatrics.2022.2322
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Impact of follow-up adherence on disease activity in childhood-onset systemic lupus erythematosus (cSLE).

Nelson, Meghan Corrigan / Mosley, Colleen / Villacis-Nunez, D Sofia / Rouster-Stevens, Kelly / Thakral, Amit

Lupus

2023 Band 32, Heft 6, Seite(n) 799–803

Abstract: Background/purpose: Systemic lupus erythematosus (SLE) is a multisystem autoimmune disease, with a potential for significant disease damage, morbidity, and mortality. In comparison to the adult population, childhood-onset SLE (cSLE) tends to be more ... ...

Abstract	Background/purpose: Systemic lupus erythematosus (SLE) is a multisystem autoimmune disease, with a potential for significant disease damage, morbidity, and mortality. In comparison to the adult population, childhood-onset SLE (cSLE) tends to be more aggressive given the higher preponderance of renal and neuropsychiatric disease and increased disease activity. There is a paucity of literature examining relationship between disease activity, rheumatology follow-up visits, and health care utilization. The objective of this study is to determine whether adherence with outpatient clinic visits would affect disease activity in patients with childhood-onset systemic lupus erythematosus (cSLE). Methods: 41 children <18 years of age at time of diagnosis with SLE who met Systemic Lupus International Collaborative Clinics (SLICC) criteria and not evaluated in clinic within the previous 120-day period were identified as eligible for inclusion. Patients were continuously searched between December 2021 and July 2022 for eligibility evaluation. Through retrospective chart review, we assessed disease activity (SLE Disease Activity Index) at the last clinic visit. The patients were stratified into two cohorts of lower and higher disease activity, with SLE disease activity index (SLEDAI) ≤ 3 and SLEDAI ≥ 4, respectively. Descriptive statistics and Willcox Rank Sum (numerical variables) and Fisher's test (categorical variables) were used to compare these two groups. Results: Clinical, epidemiological, and serological data were compared between the two groups, with observed statistically significant differences to include current use of high dose prednisone associated with higher SLEDAI scores ( Conclusion: Our findings suggest that cSLE patients with higher disease activity are at risk for increased health care utilization with respect to ED visits as well as hospitalizations in the setting of follow-up nonadherence. While further studies are required to enhance our understanding of this association, this links the importance of disease-related outcome and routine outpatient visits in this particularly vulnerable patient population.
Mesh-Begriff(e)	Child ; Adult ; Humans ; Lupus Erythematosus, Systemic/drug therapy ; Lupus Erythematosus, Systemic/epidemiology ; Lupus Erythematosus, Systemic/diagnosis ; Retrospective Studies ; Follow-Up Studies ; Age of Onset ; Prednisone ; Severity of Illness Index
Chemische Substanzen	Prednisone (VB0R961HZT)
Sprache	Englisch
Erscheinungsdatum	2023-04-26
Erscheinungsland	England
Dokumenttyp	Journal Article
ZDB-ID	1154407-7
ISSN	1477-0962 ; 0961-2033
ISSN (online)	1477-0962
ISSN	0961-2033
DOI	10.1177/09612033231173530
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Pituitary Sarcoidosis in a Pediatric Patient Successfully Treated With Adalimumab and Methotrexate.

Villacis-Nunez, D Sofia / Thakral, Amit

Journal of investigative medicine high impact case reports

2021 Band 9, Seite(n) 23247096211012191

Abstract: Neurosarcoidosis is a rare phenomenon in the pediatric population, with only a few cases reported in the literature worldwide. While hypothalamo-pituitary involvement is known to occur, direct infiltration of the pituitary gland and isolated anterior ... ...

Abstract	Neurosarcoidosis is a rare phenomenon in the pediatric population, with only a few cases reported in the literature worldwide. While hypothalamo-pituitary involvement is known to occur, direct infiltration of the pituitary gland and isolated anterior pituitary dysfunction without diabetes insipidus is seldom observed. A high index of suspicion is required for diagnosis of neurosarcoidosis, and treatment can be challenging due to lack of standardized guidelines. We present the case of a 17-year-old female with known sarcoidosis of the lacrimal glands, who developed severe headache and neurologic symptoms secondary to granulomatous infiltration of the pituitary gland and infundibulum due to neurosarcoidosis. She was successfully treated with corticosteroids, methotrexate, and adalimumab, with complete radiologic resolution. This is the first documented pediatric case of neurosarcoidosis with radiologic granulomatous infiltration of the pituitary gland, manifesting as partial anterior hypopituitarism, in the form of central hypothyroidism, without diabetes insipidus.
Mesh-Begriff(e)	Adalimumab/therapeutic use ; Adolescent ; Central Nervous System Diseases/drug therapy ; Child ; Female ; Humans ; Methotrexate/therapeutic use ; Pituitary Gland ; Sarcoidosis/drug therapy
Chemische Substanzen	Adalimumab (FYS6T7F842) ; Methotrexate (YL5FZ2Y5U1)
Sprache	Englisch
Erscheinungsdatum	2021-04-30
Erscheinungsland	United States
Dokumenttyp	Case Reports ; Journal Article
ZDB-ID	2710326-2
ISSN	2324-7096 ; 2324-7096
ISSN (online)	2324-7096
ISSN	2324-7096
DOI	10.1177/23247096211012191
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Severe Immediate and Delayed Hypersensitivity Reactions to Biologics in a Toddler With Systemic Juvenile Idiopathic Arthritis.

Villacis-Nunez, D Sofia / Bilcha, Kassahun / Spraker, Mary / Rouster-Stevens, Kelly / Cooley, Anthony

Journal of investigative medicine high impact case reports

2022 Band 10, Seite(n) 23247096221077836

Abstract: Many pediatric rheumatic diseases can be safely managed with biologic therapy. Severe allergic reactions to these medications are uncommon. We report the case of a 2-year-old male with systemic-onset juvenile idiopathic arthritis and secondary macrophage ...

Abstract	Many pediatric rheumatic diseases can be safely managed with biologic therapy. Severe allergic reactions to these medications are uncommon. We report the case of a 2-year-old male with systemic-onset juvenile idiopathic arthritis and secondary macrophage activation syndrome (MAS), whose treatment was complicated by severe allergic reactions to biologics, including drug reaction with eosinophilia and systemic symptoms (DRESS)/drug-induced hypersensitivity reaction (DIHR) likely due to anakinra, and anaphylactoid reaction to intravenous tocilizumab. These required transition to canakinumab, cyclosporine, and corticosteroids, with later development of interstitial lung disease and MAS flare needing transition from canakinumab to tofacitinib, which led to disease control. Whether lung disease is a manifestation of DRESS/DIHR to canakinumab remains unclear. High index of suspicion of hypersensitivity reactions for timely diagnosis and drug discontinuation is critical, especially in patients with active disease who might be at increased risk of these adverse events.
Mesh-Begriff(e)	Antirheumatic Agents/adverse effects ; Arthritis, Juvenile/complications ; Arthritis, Juvenile/drug therapy ; Biological Products/adverse effects ; Child, Preschool ; Humans ; Hypersensitivity/complications ; Hypersensitivity/drug therapy ; Hypersensitivity, Delayed/chemically induced ; Hypersensitivity, Delayed/complications ; Hypersensitivity, Delayed/drug therapy ; Macrophage Activation Syndrome/chemically induced ; Macrophage Activation Syndrome/complications ; Macrophage Activation Syndrome/drug therapy ; Male
Chemische Substanzen	Antirheumatic Agents ; Biological Products
Sprache	Englisch
Erscheinungsdatum	2022-02-28
Erscheinungsland	United States
Dokumenttyp	Case Reports ; Journal Article
ZDB-ID	2710326-2
ISSN	2324-7096 ; 2324-7096
ISSN (online)	2324-7096
ISSN	2324-7096
DOI	10.1177/23247096221077836
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel: Improving Hepatitis B Screening Prior to Rituximab: A Quality Improvement Project.

Villacis-Nunez, D Sofia / Orenstein, Evan / Selvaggio, Phyllis / Rouster-Stevens, Kelly / Wang, Chia-Shi / Thakral, Amit

Children (Basel, Switzerland)

2023 Band 10, Heft 7

Abstract: Rituximab, used in the treatment of some rheumatic and kidney diseases, can lead to hepatitis B virus (HBV) reactivation; HBV screening is recommended for those starting this medication. We aimed to improve by 50% the proportion of patients undergoing ... ...

Abstract	Rituximab, used in the treatment of some rheumatic and kidney diseases, can lead to hepatitis B virus (HBV) reactivation; HBV screening is recommended for those starting this medication. We aimed to improve by 50% the proportion of patients undergoing HBV screening by implementing multimodal interventions to support clinicians in this evidence-based practice. We conducted a quality improvement project from November 2020 to June 2022 at a tertiary care pediatric hospital system, including patients with rheumatic and/or kidney diseases starting rituximab. Multimodal interventions targeting clinicians included electronic health tools (dot phrase, display of screening recommendations and screening results in rituximab order sets/therapy plans), educational meetings, and e-mail/paper reminders. The primary outcome was the proportion of patients with complete HBV screening, while the secondary outcome was utilization of each laboratory component, tracked using statistical process control charts. Pre- and post-intervention data were compared using Fisher's test. One hundred eighty-two patients who had been prescribed rituximab were included, of which 98 (54%) were post-intervention. The proportions of patients undergoing complete HBV screening (6% vs. 44%;
Sprache	Englisch
Erscheinungsdatum	2023-06-30
Erscheinungsland	Switzerland
Dokumenttyp	Journal Article
ZDB-ID	2732685-8
ISSN	2227-9067
ISSN	2227-9067
DOI	10.3390/children10071142
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Quality of life measures and physical activity in childhood systemic lupus erythematosus.

Nelson, Meghan Corrigan / Gibson, Sinclair / Villacis-Nunez, D Sofia / Kimi Chan, Lai Hin / Ponder, Lori / Prahalad, Sampath / Moorthy, L Nandini

Lupus

2022 Band 31, Heft 9, Seite(n) 1114–1120

Abstract: Childhood systemic lupus erythematosus (cSLE) is a life-long disease with significant morbidity and mortality, and with associated significant impact on health-related quality of life (HRQOL). Previous literature supports that physical activity has ... ...

Abstract	Childhood systemic lupus erythematosus (cSLE) is a life-long disease with significant morbidity and mortality, and with associated significant impact on health-related quality of life (HRQOL). Previous literature supports that physical activity has positive impact on HRQOL in patients with chronic diseases, including cSLE. We sought to describe the physical activity of our patients with cSLE and determine the relationship between physical activity, SLE activity, treatment modalities and HRQOL in cSLE. Children ≤18 years of age with cSLE and their parents were enrolled and completed corresponding child and parent Simple Measure of Impact of Lupus Erythematosus in Youngsters
Mesh-Begriff(e)	Adolescent ; Age of Onset ; Child ; Exercise ; Humans ; Lupus Erythematosus, Systemic/drug therapy ; Quality of Life ; Retrospective Studies ; Severity of Illness Index
Sprache	Englisch
Erscheinungsdatum	2022-06-06
Erscheinungsland	England
Dokumenttyp	Journal Article
ZDB-ID	1154407-7
ISSN	1477-0962 ; 0961-2033
ISSN (online)	1477-0962
ISSN	0961-2033
DOI	10.1177/09612033221106154
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Successful Medical-Surgical Management of Intracardiac Thrombosis and Pulmonary Pseudoaneurysms in an Adolescent With Hughes-Stovin Syndrome.

Villacis-Nunez, D Sofia / West, Zachary / Khan, Adil Y / Rodriguez, Fred / Shane, Andi L / Rytting, Heather / Shashidharan, Subhadra / Clifton, Matthew S / Woods, Gary / Clabby, Martha / Prahalad, Sampath

Journal of investigative medicine high impact case reports

2023 Band 11, Seite(n) 23247096231166672

Abstract: We present an adolescent male with a single intracardiac mass and pulmonary emboli, complicated by peripheral venous thrombosis and subsequent development of pulmonary pseudoaneurysms, leading to diagnosis of Hughes-Stovin syndrome. Remission was ... ...

Abstract	We present an adolescent male with a single intracardiac mass and pulmonary emboli, complicated by peripheral venous thrombosis and subsequent development of pulmonary pseudoaneurysms, leading to diagnosis of Hughes-Stovin syndrome. Remission was achieved with cyclophosphamide, corticosteroids, and pseudoaneurysm resection and maintained with infliximab and methotrexate.
Mesh-Begriff(e)	Male ; Humans ; Adolescent ; Aneurysm, False/complications ; Aneurysm, False/therapy ; Syndrome ; Pulmonary Artery ; Aneurysm/complications ; Aneurysm/diagnosis ; Vasculitis/complications ; Thrombosis/drug therapy ; Thrombosis/etiology
Sprache	Englisch
Erscheinungsdatum	2023-04-07
Erscheinungsland	United States
Dokumenttyp	Journal Article ; Case Reports
ZDB-ID	2710326-2
ISSN	2324-7096 ; 2324-7096
ISSN (online)	2324-7096
ISSN	2324-7096
DOI	10.1177/23247096231166672
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Outcomes of COVID-19 in a cohort of pediatric patients with rheumatic diseases.

Villacis-Nunez, D Sofia / Rostad, Christina A / Rouster-Stevens, Kelly / Khosroshahi, Arezou / Chandrakasan, Shanmuganathan / Prahalad, Sampath

Pediatric rheumatology online journal

2021 Band 19, Heft 1, Seite(n) 94

Abstract: Background: There are few reports of COVID-19 in pediatric patients with rheumatic diseases. This study describes the clinical presentation and outcomes of COVID-19 in this population.: Methods: We analyzed a single-center case series of pediatric ... ...

Abstract	Background: There are few reports of COVID-19 in pediatric patients with rheumatic diseases. This study describes the clinical presentation and outcomes of COVID-19 in this population. Methods: We analyzed a single-center case series of pediatric patients with rheumatic diseases and laboratory-confirmed COVID-19. Demographic, baseline and COVID-19 associated clinical features were compared between ambulatory and hospitalized patients using univariate analysis. Results: Fifty-five cases were identified: 45 (81.8%) in the ambulatory group and 10 (18.2%) hospitalized. African American race (OR 7.78; 95% CI [1.46-55.38]; p = 0.006) and cardiovascular disease (OR 19.40; 95% CI 2.45-254.14; p = 0.001) predominated in hospitalized patients. Active rheumatic disease (OR 11.83; 95% CI 1.43-558.37; p = 0.01), medium/high-dose corticosteroid use (OR 14.12; 95% CI [2.31-106.04]; p = 0.001), mycophenolate use (OR 8.84; 95% CI [1.64-63.88]; p = 0.004), rituximab use (OR 19.40; 95% CI [2.45-254.14]; p = 0.001) and severe immunosuppression (OR 34.80; 95% CI [3.94-1704.26]; p = < 0.001) were associated with increased odds of hospitalization. Fever (OR 7.78; 95% CI [1.46-55.38]; p = 0.006), dyspnea (OR 26.28; 95% CI [2.17-1459.25]; p = 0.003), chest pain (OR 13.20; 95% CI [1.53-175.79]; p = 0.007), and rash (OR 26.28; 95% CI [2.17-1459.25]; p = 0.003) were more commonly observed in hospitalized patients. Rheumatic disease flares were almost exclusive to hospitalized patients (OR 55.95; 95% CI [5.16-3023.74]; p < 0.001).. One patient did not survive. Conclusions: Medium/high-dose corticosteroid, mycophenolate and rituximab use, and severe immunosuppression were risk factors for hospitalization. Fever, dyspnea, chest pain, and rash were high-risk symptoms for hospitalization. Rheumatic disease activity and flare could contribute to the need for hospitalization.
Mesh-Begriff(e)	Adolescent ; COVID-19/complications ; COVID-19/diagnosis ; COVID-19/pathology ; COVID-19/therapy ; Child ; Child, Preschool ; Female ; Hospitalization ; Humans ; Infant ; Infant, Newborn ; Male ; Rheumatic Diseases/complications ; Rheumatic Diseases/pathology ; Rheumatic Diseases/therapy ; Rheumatic Diseases/virology ; Treatment Outcome ; Young Adult
Sprache	Englisch
Erscheinungsdatum	2021-06-21
Erscheinungsland	England
Dokumenttyp	Journal Article
ZDB-ID	2279468-2
ISSN	1546-0096 ; 1546-0096
ISSN (online)	1546-0096
ISSN	1546-0096
DOI	10.1186/s12969-021-00568-4
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Short-term Outcomes of Corticosteroid Monotherapy in Multisystem Inflammatory Syndrome in Children.

Villacis-Nunez, D Sofia / Jones, Kaitlin / Jabbar, Aysha / Fan, Lucie / Moore, Whitney / Peter, Andrew S / Henderson, Michaela / Xiang, Yijin / Kelleman, Michael S / Sherry, Whitney / Chandrakasan, Shanmuganathan / Oster, Matthew E / Jaggi, Preeti / Prahalad, Sampath

JAMA pediatrics

2022 Band 176, Heft 6, Seite(n) 576–584

Abstract: Importance: Optimal agents and duration of primary treatment for multisystem inflammatory syndrome in children (MIS-C) remain unclear.: Objective: To compare short-term patient outcomes based on initial treatment with corticosteroids, intravenous ... ...

Abstract	Importance: Optimal agents and duration of primary treatment for multisystem inflammatory syndrome in children (MIS-C) remain unclear. Objective: To compare short-term patient outcomes based on initial treatment with corticosteroids, intravenous immunoglobulin (IVIG), or both. Design, setting, and participants: This retrospective cohort study included patients in a tertiary-care pediatric hospital system who had MIS-C per the Centers for Disease Control and Prevention case definition during the period March 2020 to February 2021. Exposures: Immunomodulatory therapy within the first 24 hours (patients in the intensive care unit [ICU]) or 48 hours (non-ICU patients): corticosteroids alone, IVIG alone, and IVIG plus corticosteroids. Main outcomes and measures: Primary outcome was failure of initial therapy, defined as therapy escalation due to fever or worsening or lack of improvement of laboratory, cardiac, or noncardiac clinical factors after 24 hours (ICU patients) or 48 hours (non-ICU patients) from time of therapy initiation, per clinician assessment. Secondary outcomes included presence of complications, cardiovascular outcomes, fever duration, length of hospital and ICU stays, corticosteroid use duration, and need for readmission. Results: Among 228 eligible patients, 215 patients were included in the univariate analysis; median age was 8 years, and 135 (62.8%) were boys. There were 69 patients in the corticosteroids group, 31 patients in the IVIG group, and 115 patients in the IVIG plus corticosteroids group. Patients in the corticosteroids group had milder disease at presentation. After propensity score weighting including 179 patients (68 in the corticosteroids group and 111 in the IVIG plus corticosteroids group), rates of initial treatment failure were similar between groups. Among patients whose initial treatment failed, treatment failure in the IVIG plus corticosteroids group was more likely to be based on laboratory parameters (odds ratio [OR], 1.96; 95% CI, 1.07-3.60) and less likely to be based on cardiovascular markers (OR, 0.39; 95% CI, 0.2-0.76), per clinician assessment. Patients in the IVIG plus corticosteroids group had a longer median inpatient stay (6 vs 5 days; P = .001) and longer median corticosteroid course duration (10 vs 5 days; P = .04) compared with the corticosteroids group. Forty-nine patients (71% of 69 in the corticosteroids group) recovered after receiving corticosteroid monotherapy for 10 days or less. Conclusions and relevance: Corticosteroid monotherapy is a reasonable management option for a subset of patients with MIS-C, particularly those with mild disease.
Mesh-Begriff(e)	Adrenal Cortex Hormones/therapeutic use ; COVID-19/complications ; Child ; Female ; Humans ; Immunoglobulins, Intravenous/therapeutic use ; Male ; Retrospective Studies ; Systemic Inflammatory Response Syndrome ; Treatment Outcome
Chemische Substanzen	Adrenal Cortex Hormones ; Immunoglobulins, Intravenous
Sprache	Englisch
Erscheinungsdatum	2022-03-28
Erscheinungsland	United States
Dokumenttyp	Journal Article
ZDB-ID	2701223-2
ISSN	2168-6211 ; 2168-6203
ISSN (online)	2168-6211
ISSN	2168-6203
DOI	10.1001/jamapediatrics.2022.0292
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Giant Coronary Aneurysms in Multisystem Inflammatory Syndrome in Children Associated With SARS-CoV-2 Infection.

Villacis-Nunez, D Sofia / Hashemi, Sassan / Nelson, Meghan C / Flanagan, Elaine / Thakral, Amit / Rodriguez, Fred / Jaggi, Preeti / Oster, Matthew E / Prahalad, Sampath / Rouster-Stevens, Kelly A

JACC. Case reports

2021 Band 3, Heft 13, Seite(n) 1499–1508

Abstract: Multisystem inflammatory syndrome in children (MIS-C) can cause a myriad of cardiac manifestations, including coronary dilation and aneurysms; giant aneurysms are infrequent. We describe 3patients with giant coronary aneurysms associated with MIS-C, ... ...

Abstract	Multisystem inflammatory syndrome in children (MIS-C) can cause a myriad of cardiac manifestations, including coronary dilation and aneurysms; giant aneurysms are infrequent. We describe 3patients with giant coronary aneurysms associated with MIS-C, including the youngest case reported to date, treated with intravenous immunoglobulin, corticosteroids, and biologic agents. (
Sprache	Englisch
Erscheinungsdatum	2021-10-06
Erscheinungsland	Netherlands
Dokumenttyp	Case Reports
ISSN	2666-0849
ISSN (online)	2666-0849
DOI	10.1016/j.jaccas.2021.06.043
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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