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  1. Article ; Online: Pulmonary Manifestations of Systemic Vasculitis in Children.

    Cuceoglu, Muserref Kasap / Ozen, Seza

    Pediatric clinics of North America

    2020  Volume 68, Issue 1, Page(s) 167–176

    Abstract: Vasculitides are defined according to the vessel size involved, and they tend to affect certain organ systems. Pulmonary involvement is rare in the common childhood vasculitides, such as Kawasaki disease, IgA vasculitis (Henoch Schonlein purpura). On the ...

    Abstract Vasculitides are defined according to the vessel size involved, and they tend to affect certain organ systems. Pulmonary involvement is rare in the common childhood vasculitides, such as Kawasaki disease, IgA vasculitis (Henoch Schonlein purpura). On the other hand, lung involvement is common in a rare pediatric vasculitis, granulomatosis with polyangiitis (GPA) (Wegener granulomatosis), where respiratory system findings are common. A criterion in the Ankara 2008 classification criteria for GPA is the presence of nodules, cavities, or fixed infiltrates. The adult data suggest that rituximab may be an alternative to cyclophosphamide in induction treatment.
    MeSH term(s) Child ; Humans ; Lung Diseases/diagnosis ; Lung Diseases/etiology ; Lung Diseases/therapy ; Pulmonary Circulation ; Systemic Vasculitis/complications ; Systemic Vasculitis/diagnosis ; Systemic Vasculitis/therapy
    Language English
    Publishing date 2020-12-23
    Publishing country United States
    Document type Journal Article ; Review
    ZDB-ID 215711-1
    ISSN 1557-8240 ; 0031-3955
    ISSN (online) 1557-8240
    ISSN 0031-3955
    DOI 10.1016/j.pcl.2020.09.014
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  2. Article: Hypersensitivity to Biological Treatments in Juvenile Idiopathic Arthritis: How Should It Be Managed?

    Kasap Cuceoglu, Muserref / Basaran, Ozge / Soyer, Ozge / Ozen, Seza

    Journal of clinical medicine

    2022  Volume 11, Issue 24

    Abstract: Juvenile idiopathic arthritis (JIA) is one of the most frequent diseases in the practice of pediatric rheumatology. JIA treatments have been modified and improved with the use of biological drugs along with technological innovations. However, different ... ...

    Abstract Juvenile idiopathic arthritis (JIA) is one of the most frequent diseases in the practice of pediatric rheumatology. JIA treatments have been modified and improved with the use of biological drugs along with technological innovations. However, different types of hypersensitivity reactions to biological drugs have also been reported. Anaphylaxis and infusion reactions occurring during the intravenous infusion require a critical approach in the acute period. On the other hand, the detection of drug-related late-type reactions and the development of antibodies to the agent highlight the need for an understanding of the drug-induced etiology to prevent the patient from continuing the treatment with the culprit drug. The chronic disease process, concomitant immune dysregulation, and multiple drug use may result in these hypersensitivity reactions. In this review, the hypersensitivity reactions to the biological treatments used in patients with juvenile idiopathic arthritis and the management of these conditions are discussed.
    Language English
    Publishing date 2022-12-08
    Publishing country Switzerland
    Document type Journal Article ; Review
    ZDB-ID 2662592-1
    ISSN 2077-0383
    ISSN 2077-0383
    DOI 10.3390/jcm11247291
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  3. Article ; Online: Subclinical enthesitis in enthesitis-related arthritis and sacroiliitis associated with familial Mediterranean fever.

    Sener, Seher / Atalay, Erdal / Yildiz, Adalet Elcin / Kasap Cuceoglu, Muserref / Basaran, Ozge / Batu, Ezgi Deniz / Bilginer, Yelda / Ozen, Seza

    Modern rheumatology

    2023  Volume 34, Issue 3, Page(s) 607–613

    Abstract: Objectives: In our study, we investigated the presence of subclinical enthesitis by ultrasonography (US) in asymptomatic patients with enthesitis-related arthritis (ERA) and sacroiliitis associated with familial Mediterranean fever (FMF).: Methods: A ...

    Abstract Objectives: In our study, we investigated the presence of subclinical enthesitis by ultrasonography (US) in asymptomatic patients with enthesitis-related arthritis (ERA) and sacroiliitis associated with familial Mediterranean fever (FMF).
    Methods: A total of 50 patients, including 35 patients with ERA and 15 with sacroiliitis associated with FMF, were included in the study. All patients were evaluated with US by a paediatric radiologist. Enthesis of seven tendons (common extensor and flexor tendons, quadriceps tendon, proximal and distal patellar tendon, Achilles tendon, and plantar fascia) was examined on both sides.
    Results: Subclinical enthesitis was detected in 10 ERA (28.5%) and three FMF (20%) patients. Enthesitis was radiologically diagnosed in 16 (2.3%) out of 700 evaluated entheseal sites. The most frequent sites of enthesitis were Achilles (37.5%) and quadriceps (31.3%) tendons. All patients were in clinical remission and had no active complaints, and acute phase reactants were within normal limits. Therefore, the patients were followed up without treatment change. However, disease flare-up was observed in three of these patients (23.1%) during the follow-up, and their treatments were intensified.
    Conclusions: Our results showed that the US can be particularly helpful in detecting subclinical enthesitis and predicting disease flare-ups.
    MeSH term(s) Child ; Humans ; Sacroiliitis/complications ; Sacroiliitis/diagnostic imaging ; Familial Mediterranean Fever/complications ; Familial Mediterranean Fever/diagnostic imaging ; Symptom Flare Up ; Enthesopathy/complications ; Enthesopathy/diagnostic imaging ; Arthritis, Juvenile/complications ; Achilles Tendon/diagnostic imaging
    Language English
    Publishing date 2023-05-31
    Publishing country England
    Document type Journal Article
    ZDB-ID 2078157-X
    ISSN 1439-7609 ; 1439-7595
    ISSN (online) 1439-7609
    ISSN 1439-7595
    DOI 10.1093/mr/road053
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  4. Article ; Online: COVID-19 in paediatric rheumatology patients treated with b/tsDMARDs: a cross-sectional patient survey study.

    Kasap Cuceoglu, Muserref / Batu, Ezgi Deniz / Bilginer, Yelda / Özen, Seza

    Annals of the rheumatic diseases

    2020  Volume 80, Issue 6, Page(s) e95

    MeSH term(s) Antirheumatic Agents/therapeutic use ; COVID-19 ; Child ; Cross-Sectional Studies ; Humans ; Rheumatology ; SARS-CoV-2
    Chemical Substances Antirheumatic Agents
    Keywords covid19
    Language English
    Publishing date 2020-07-06
    Publishing country England
    Document type Letter ; Comment
    ZDB-ID 7090-7
    ISSN 1468-2060 ; 0003-4967
    ISSN (online) 1468-2060
    ISSN 0003-4967
    DOI 10.1136/annrheumdis-2020-218341
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  5. Article ; Online: A Child with Refractory and Relapsing Anti-3-Hydroxy-3-Methylglutaryl-Coenzyme A Reductase Myopathy: Case-Based Review.

    Sener, Seher / Batu, Ezgi Deniz / Sari, Seher / Kasap Cuceoglu, Muserref / Yildiz, Adalet Elcin / Talim, Beril / Aydingoz, Ustun / Ozen, Seza / Haliloglu, Goknur

    Journal of neuromuscular diseases

    2023  Volume 10, Issue 2, Page(s) 279–291

    Abstract: Background/objective: Anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase (anti-HMGCR) myopathy is rare in children. Here, we present a boy with relapsing refractory anti-HMGCR myopathy along with a systematic literature review.: Case report: 17- ... ...

    Abstract Background/objective: Anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase (anti-HMGCR) myopathy is rare in children. Here, we present a boy with relapsing refractory anti-HMGCR myopathy along with a systematic literature review.
    Case report: 17-year-old boy with five years of muscle weakness, rash, high creatinine kinase (CK) levels, and muscle biopsy compatible with inflammatory myopathy was diagnosed with juvenile dermatomyositis. He was treated with corticosteroids, intravenous immunoglobulin (IVIG), and methotrexate. His muscle weakness improved with this treatment although never completely resolved. CK levels decreased from ∼15000 U/L to ∼3000 U/L. At the age of 15, muscle weakness relapsed after an upper respiratory tract infection; pulse corticosteroid treatment was administered. The re-evaluated muscle biopsy showed a necrotizing pattern and the HMGCR antibody was positive confirming anti-HMGCR myopathy when he was 16. The diagnostic delay was 50 months. Disease activity was monitored by Medical Research Council score, MRI and functional tests. Despite corticosteroids, methotrexate, IVIG, cyclosporine A, and rituximab therapies, muscle weakness improved only slightly during the first three months and remained stable afterwards.Results of the Literature Search:We identified 16 articles describing 50 children (76% female) with anti-HMGCR myopathy by reviewing the English literature up to March 1st, 2022. Proximal muscle weakness was the most common clinical symptom (70.8%). Corticosteroids (84.8%), IVIG (58.7%), and methotrexate (56.5%) were preferred in most cases. Complete remission was achieved in nine patients (28.1%).
    Conclusion: Diagnosis and management of children with anti-HMGCR myopathy are challenging. Complete remission is achieved in only one third of these patients. Imaging biomarkers may aid treatment.
    MeSH term(s) Male ; Humans ; Child ; Female ; Adolescent ; Oxidoreductases/therapeutic use ; Coenzyme A/therapeutic use ; Immunoglobulins, Intravenous/therapeutic use ; Methotrexate/therapeutic use ; Delayed Diagnosis ; Autoantibodies ; Muscular Diseases/pathology ; Muscle Weakness
    Chemical Substances Oxidoreductases (EC 1.-) ; Coenzyme A (SAA04E81UX) ; Immunoglobulins, Intravenous ; Methotrexate (YL5FZ2Y5U1) ; Autoantibodies
    Language English
    Publishing date 2023-01-22
    Publishing country Netherlands
    Document type Systematic Review ; Case Reports ; Journal Article
    ISSN 2214-3602
    ISSN (online) 2214-3602
    DOI 10.3233/JND-221557
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  6. Article ; Online: Two siblings with Majeed syndrome and neutropenia.

    Kasap Cuceoglu, Muserref / Batu, Ezgi Deniz / Yildiz, Adalet Elcin / Kaya Akca, Ummusen / Atalay, Erdal / Sener, Seher / Balik, Zeynep / Basaran, Ozge / Bilginer, Yelda / Ozen, Seza

    Archives of rheumatology

    2022  Volume 37, Issue 4, Page(s) 638–640

    Language English
    Publishing date 2022-09-20
    Publishing country Turkey
    Document type Journal Article
    ZDB-ID 3012972-2
    ISSN 2618-6500 ; 2148-5046
    ISSN (online) 2618-6500
    ISSN 2148-5046
    DOI 10.46497/ArchRheumatol.2022.9437
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  7. Article: COVID-19 in paediatric rheumatology patients treated with b/tsDMARDs: a cross-sectional patient survey study

    Kasap Cuceoglu, Muserref / Batu, Ezgi Deniz / Bilginer, Yelda / Özen, Seza

    Ann. rheum. dis

    Keywords covid19
    Publisher WHO
    Document type Article
    Note WHO #Covidence: #635207
    Database COVID19

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  8. Article ; Online: The role of vascular inflammation markers in deficiency of adenosine deaminase 2.

    Kaya Akca, Ummusen / Sag, Erdal / Unal, Sule / Kasap Cuceoglu, Muserref / Bilginer, Yelda / Ozen, Seza

    Seminars in arthritis and rheumatism

    2021  Volume 51, Issue 4, Page(s) 839–844

    Abstract: Objectives: The first objective was to assess the role of vascular inflammatory factors in the pathogenesis of deficiency of adenosine deaminase 2 (DADA2) and to compare these markers among DADA2 patients with different phenotypes. We also aimed to ... ...

    Abstract Objectives: The first objective was to assess the role of vascular inflammatory factors in the pathogenesis of deficiency of adenosine deaminase 2 (DADA2) and to compare these markers among DADA2 patients with different phenotypes. We also aimed to investigate differences between DADA2 patients with vasculitic features and classic polyarteritis nodosa (PAN) for the aforementioned markers.
    Methods: The study included eighteen DADA2 patients, ten PAN patients, and eight healthy controls. Plasma levels of sST2, sRAGE, Tie-2, sCD40L, Tie-1, sFlt-1, LIGHT, TNF-α, PlGF, IL-6, IL-18, IL-10, MCP-1 were studied by cytometric bead-based multiplex assay panel.
    Results: Among the DADA2 patients, five had hematological manifestations, 13 had vasculitic findings, and accompanying immunological findings were present in seven patients. Nine patients had neurological findings, five of whom had neuropathy. Plasma levels of Tie-1 and sFlt-1 were higher in the overall DADA2 patients compared to healthy controls and PAN patients (p<0.001 and p = 0.004, respectively). DADA2 patients with PAN-like features had higher sRAGE, Tie-2, and TNF-α levels compared to PAN patients (p = 0.013, p = 0.003, and p = 0.001, respectively). In DADA2 patients with hematological findings, plasma IL-18 levels were higher than those with PAN-like phenotype (p = 0.001). Finally, DADA2 patients with neuropathy had higher sRAGE concentrations than patients without neuropathy and healthy controls (p = 0.03 and p = 0.008, respectively).
    Conclusions: We suggest that the high plasma IL-18 levels observed in DADA2 patients with hematologic manifestations may be associated with an activated IFNγ pathway, and lack of response to anti-TNF treatment. We identified sRAGE as a potential biomarker of neuropathy in DADA2 patients. Higher concentrations of Tie-1, Tie-2, sFlt-1, sRAGE, and TNF-α distinguished DADA2 patients with PAN-like features from PAN patients.
    MeSH term(s) Adenosine Deaminase ; Humans ; Inflammation ; Intercellular Signaling Peptides and Proteins ; Polyarteritis Nodosa ; Tumor Necrosis Factor Inhibitors
    Chemical Substances Intercellular Signaling Peptides and Proteins ; Tumor Necrosis Factor Inhibitors ; ADA2 protein, human (EC 3.5.4.4) ; Adenosine Deaminase (EC 3.5.4.4)
    Language English
    Publishing date 2021-04-28
    Publishing country United States
    Document type Journal Article
    ZDB-ID 120247-9
    ISSN 1532-866X ; 0049-0172
    ISSN (online) 1532-866X
    ISSN 0049-0172
    DOI 10.1016/j.semarthrit.2021.04.013
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  9. Article: The Effect of Biologics in the Treatment of Multisystem Inflammatory Syndrome in Children (Mis-C): A Single-Center Propensity-Score-Matched Study.

    Basaran, Ozge / Batu, Ezgi Deniz / Kaya Akca, Ummusen / Atalay, Erdal / Kasap Cuceoglu, Muserref / Sener, Seher / Balık, Zeynep / Karabulut, Erdem / Kesici, Selman / Karagoz, Tevfik / Ozsurekci, Yasemin / Bilginer, Yelda / Cengiz, Ali Bulent / Ozen, Seza

    Children (Basel, Switzerland)

    2023  Volume 10, Issue 6

    Abstract: Multisystem inflammatory syndrome in children (MIS-C) is a serious condition characterized by excessive inflammation that can arise as a complication of SARS-CoV-2 infection in children. While our understanding of COVID-19 and MIS-C has been advancing, ... ...

    Abstract Multisystem inflammatory syndrome in children (MIS-C) is a serious condition characterized by excessive inflammation that can arise as a complication of SARS-CoV-2 infection in children. While our understanding of COVID-19 and MIS-C has been advancing, there is still uncertainty regarding the optimal treatment for MIS-C. In this study, we aimed to compare the clinical and laboratory outcomes of MIS-C patients treated with IVIG plus corticosteroids (CS) to those treated with IVIG plus CS and an additional biologic drug. We used the propensity score (PS)-matching method to assess the relationships between initial treatment and outcomes. The primary outcome was a left ventricular ejection fraction of less than 55% on day 2 or beyond and/or the requirement of inotrope support on day 2 or beyond. We included 79 MIS-C patients (median age 8.51 years, 33 boys) followed in our center. Among them, 50 children (25 in each group) were allocated to the PS-matched cohort sample. The primary outcome was observed in none of the patients in the IVIG and CS group, while it occurred in eight patients in the IVIG plus CS and biologic group (
    Language English
    Publishing date 2023-06-11
    Publishing country Switzerland
    Document type Journal Article
    ZDB-ID 2732685-8
    ISSN 2227-9067
    ISSN 2227-9067
    DOI 10.3390/children10061045
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  10. Article ; Online: COVID-19 in paediatric rheumatology patients treated with b/tsDMARDs

    Kasap Cuceoglu, Muserref / Batu, Ezgi Deniz / Bilginer, Yelda / Özen, Seza

    Annals of the Rheumatic Diseases

    a cross-sectional patient survey study

    2020  , Page(s) annrheumdis–2020–218341

    Keywords Immunology ; General Biochemistry, Genetics and Molecular Biology ; Immunology and Allergy ; Rheumatology ; covid19
    Language English
    Publisher BMJ
    Publishing country uk
    Document type Article ; Online
    ZDB-ID 7090-7
    ISSN 1468-2060 ; 0003-4967
    ISSN (online) 1468-2060
    ISSN 0003-4967
    DOI 10.1136/annrheumdis-2020-218341
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