Castleman Disease: A Multicenter Case Series from Turkey

Yıl: 2022 Cilt: 39 Sayı: 2 Sayfa Aralığı: 130 - 135 Metin Dili: İngilizce DOI: 10.4274/tjh.galenos.2022.2021.0670 İndeks Tarihi: 07-06-2022

Castleman Disease: A Multicenter Case Series from Turkey

Öz:
Objective: Castleman disease (CD) is a rare disease also known as angiofollicular lymph node hyperplasia. The two main histological subtypes are the hyaline vascular and plasma cell variants. It is further classified as unicentric CD (UCD) or multicentric CD (MCD) according to the anatomical distribution of the disease and the number of lymph nodes involved. The aim of this multicenter study was to evaluate all cases of CD identified to date in Turkey to set up a national registry to improve the early recognition, treatment, and follow-up of CD. Materials and Methods: Both adult (n=130) and pediatric (n=10) patients with lymph node or involved field biopsy results reported as CD were included in the study. Patients’ demographic information, clinical and laboratory characteristics, imaging study results, treatment strategies, and clinical outcomes were evaluated retrospectively. Results: A total of 140 patients (69 male and 71 female) with a diagnosis of UCD (n=73) or MCD (n=67) were included. The mean age was 39 years in the UCD group and 47 years in the MCD group. Female patients were more common in the UCD group. The most common histological subtype was hyaline vascular for both UCD and MCD patients. Asymptomatic patients were more common in the UCD group. Anemia, elevations of acute phase reactants, and hypoalbuminemia were more common in the MCD group. The most commonly used treatment strategies for UCD were surgical excision, rituximab, and radiotherapy, respectively. All UCD patients were alive at a median of 19.5 months of follow-up. The most commonly used treatment strategies for MCD were methyl prednisolone, R-CHOP, R-CVP, and rituximab. Thirteen MCD patients had died at a median of 34 months of follow-up. Conclusion: This study is important in presenting the patient characteristics and treatment strategies for CD from Turkey, with the potential of increasing awareness about CD. Treatment data may help in making decisions, particularly in countries that do not have access to siltuximab. However, larger prospective studies are needed to make definitive conclusions.
Anahtar Kelime:

Castleman Hastalığı: Türkiye’den Çok Merkezli Bir Olgu Serisi

Öz:
Amaç: Anjiyofolliküler lenf nodu hiperplazisi olarak da bilinen Castleman hastalığı (CH), nadir bir hastalık olup başlıca hiyalin vasküler ve plazma hücreli olmak üzere 2 histolojik alt tipi vardır. Hastalığın anatomik yayılımı ve tutulan lenf nodu bölgelerinin sayısına göre unisentrik (UCH) ya da multisentrik (MCH) olarak sınıflandırılır. Bu çok merkezli çalışmanın amacı bugüne kadar Türkiye’de tanımlanan tüm CH olgularını tanımlamak, ulusal bir veri tabanı oluşturarak CH’de erken tanı, tedavi ve takip sürecine katkı sağlamaktır. Gereç ve Yöntemler: Çalışmaya lenf nodu ya da tutulan alandan yapılmış biyopsi sonucu CH olarak rapor edilen hem erişkin (n=130) hem de pediyatrik (n=10) hastalar dahil edildi. Hastaların demografik bilgileri, klinik ve laboratuvar özellikleri, görüntüleme bulguları, aldıkları tedaviler ve tedavi sonuçları geriye dönük olarak değerlendirildi. Bulgular: Dahil edilen 140 hastanın 69’u kadın, 71’i erkekti. Yetmiş üç hasta UCH, 67 hasta MCH olarak sınıflandırılmıştı. Yaş ortalaması UCH’de 39, MCH’de 47 yıl idi. Kadın hastalar UCH’de daha fazlaydı. Hem UCH hem de MCH için en sık histolojik alt tip hiyalin vaskülerdi. Asemptomatik hastalar UCH’de daha fazlaydı. Anemi, akut faz reaktanı yüksekliği ve hipoalbuminemi MCH’de daha sıktı. UCH’de en sık uygulanan tedaviler sırasıyla cerrahi eksizyon, rituksimab ve radyoterapiydi. Median 19,5 aylık takipte tüm UCH’li hastalar hayattaydı. MCH’de 1. basamak tedaviler metil prednizolon, R-CHOP, R-CVP ve rituksimab idi. Median 34 aylık takipte 13 MCH’li hasta kaybedilmişti. Sonuç: Çalışmamız Türkiye’deki CH hastalarının özellikleri ve tedavi yaklaşımlarını yansıtması açısından önemli olup hastalıkla ilgili farkındalığın arttırılması potansiyeline sahiptir. Tedavi verileri özellikle ülkemizde olduğu gibi siltuksimaba ulaşımı zor olan ülkelerde tedavi seçimi konusunda fikir verebilir. Kesin sonuçlar çıkarmak için büyük ölçekli prospektif çalışmalara ihtiyaç vardır.
Anahtar Kelime:

Belge Türü: Makale Makale Türü: Araştırma Makalesi Erişim Türü: Erişime Açık
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APA Gündüz E, Kirkizlar H, ÜMİT E, karaman gülsaran s, Ozkocaman V, Özkalemkaş F, candar ö, ELVERDİ T, Kucukyurt S, Paydas S, Ceneli O, KARAKUŞ S, MARAL S, Ekinci O, İpek Y, Kis C, Guven Z, AKDENİZ A, CELKAN T, Eroğlu küçükdiler A, Akgun Cagliyan G, ÖZÇELİK ŞENGÖZ C, Karatas A, BULDUK T, ÖZCAN A, belen f, CANBOLAT A, Kartal I, Ören H, Toret E, Özdemir N, Bakanay S (2022). Castleman Disease: A Multicenter Case Series from Turkey. , 130 - 135. 10.4274/tjh.galenos.2022.2021.0670
Chicago Gündüz Eren,Kirkizlar Hakki Onur,ÜMİT Elif Gülsüm,karaman gülsaran sedanur,Ozkocaman Vildan,Özkalemkaş Fahir,candar ömer,ELVERDİ TUĞRUL,Kucukyurt Selin,Paydas Semra,Ceneli Ozcan,KARAKUŞ Sema,MARAL SENEM,Ekinci Omer,İpek Yıldız,Kis Cem,Guven Zeynep Tugba,AKDENİZ Aydan,CELKAN TÜLIN TIRAJE,Eroğlu küçükdiler Ayşe hilal,Akgun Cagliyan Gulsum,ÖZÇELİK ŞENGÖZ CEYDA,Karatas Ayse,BULDUK TUBA,ÖZCAN Alper,belen fatma burcu,CANBOLAT Aylin,Kartal Ibrahim,Ören Hale,Toret Ersin,Özdemir Nihal,Bakanay Sule Mine Castleman Disease: A Multicenter Case Series from Turkey. (2022): 130 - 135. 10.4274/tjh.galenos.2022.2021.0670
MLA Gündüz Eren,Kirkizlar Hakki Onur,ÜMİT Elif Gülsüm,karaman gülsaran sedanur,Ozkocaman Vildan,Özkalemkaş Fahir,candar ömer,ELVERDİ TUĞRUL,Kucukyurt Selin,Paydas Semra,Ceneli Ozcan,KARAKUŞ Sema,MARAL SENEM,Ekinci Omer,İpek Yıldız,Kis Cem,Guven Zeynep Tugba,AKDENİZ Aydan,CELKAN TÜLIN TIRAJE,Eroğlu küçükdiler Ayşe hilal,Akgun Cagliyan Gulsum,ÖZÇELİK ŞENGÖZ CEYDA,Karatas Ayse,BULDUK TUBA,ÖZCAN Alper,belen fatma burcu,CANBOLAT Aylin,Kartal Ibrahim,Ören Hale,Toret Ersin,Özdemir Nihal,Bakanay Sule Mine Castleman Disease: A Multicenter Case Series from Turkey. , 2022, ss.130 - 135. 10.4274/tjh.galenos.2022.2021.0670
AMA Gündüz E,Kirkizlar H,ÜMİT E,karaman gülsaran s,Ozkocaman V,Özkalemkaş F,candar ö,ELVERDİ T,Kucukyurt S,Paydas S,Ceneli O,KARAKUŞ S,MARAL S,Ekinci O,İpek Y,Kis C,Guven Z,AKDENİZ A,CELKAN T,Eroğlu küçükdiler A,Akgun Cagliyan G,ÖZÇELİK ŞENGÖZ C,Karatas A,BULDUK T,ÖZCAN A,belen f,CANBOLAT A,Kartal I,Ören H,Toret E,Özdemir N,Bakanay S Castleman Disease: A Multicenter Case Series from Turkey. . 2022; 130 - 135. 10.4274/tjh.galenos.2022.2021.0670
Vancouver Gündüz E,Kirkizlar H,ÜMİT E,karaman gülsaran s,Ozkocaman V,Özkalemkaş F,candar ö,ELVERDİ T,Kucukyurt S,Paydas S,Ceneli O,KARAKUŞ S,MARAL S,Ekinci O,İpek Y,Kis C,Guven Z,AKDENİZ A,CELKAN T,Eroğlu küçükdiler A,Akgun Cagliyan G,ÖZÇELİK ŞENGÖZ C,Karatas A,BULDUK T,ÖZCAN A,belen f,CANBOLAT A,Kartal I,Ören H,Toret E,Özdemir N,Bakanay S Castleman Disease: A Multicenter Case Series from Turkey. . 2022; 130 - 135. 10.4274/tjh.galenos.2022.2021.0670
IEEE Gündüz E,Kirkizlar H,ÜMİT E,karaman gülsaran s,Ozkocaman V,Özkalemkaş F,candar ö,ELVERDİ T,Kucukyurt S,Paydas S,Ceneli O,KARAKUŞ S,MARAL S,Ekinci O,İpek Y,Kis C,Guven Z,AKDENİZ A,CELKAN T,Eroğlu küçükdiler A,Akgun Cagliyan G,ÖZÇELİK ŞENGÖZ C,Karatas A,BULDUK T,ÖZCAN A,belen f,CANBOLAT A,Kartal I,Ören H,Toret E,Özdemir N,Bakanay S "Castleman Disease: A Multicenter Case Series from Turkey." , ss.130 - 135, 2022. 10.4274/tjh.galenos.2022.2021.0670
ISNAD Gündüz, Eren vd. "Castleman Disease: A Multicenter Case Series from Turkey". (2022), 130-135. https://doi.org/10.4274/tjh.galenos.2022.2021.0670
APA Gündüz E, Kirkizlar H, ÜMİT E, karaman gülsaran s, Ozkocaman V, Özkalemkaş F, candar ö, ELVERDİ T, Kucukyurt S, Paydas S, Ceneli O, KARAKUŞ S, MARAL S, Ekinci O, İpek Y, Kis C, Guven Z, AKDENİZ A, CELKAN T, Eroğlu küçükdiler A, Akgun Cagliyan G, ÖZÇELİK ŞENGÖZ C, Karatas A, BULDUK T, ÖZCAN A, belen f, CANBOLAT A, Kartal I, Ören H, Toret E, Özdemir N, Bakanay S (2022). Castleman Disease: A Multicenter Case Series from Turkey. Turkish Journal of Hematology, 39(2), 130 - 135. 10.4274/tjh.galenos.2022.2021.0670
Chicago Gündüz Eren,Kirkizlar Hakki Onur,ÜMİT Elif Gülsüm,karaman gülsaran sedanur,Ozkocaman Vildan,Özkalemkaş Fahir,candar ömer,ELVERDİ TUĞRUL,Kucukyurt Selin,Paydas Semra,Ceneli Ozcan,KARAKUŞ Sema,MARAL SENEM,Ekinci Omer,İpek Yıldız,Kis Cem,Guven Zeynep Tugba,AKDENİZ Aydan,CELKAN TÜLIN TIRAJE,Eroğlu küçükdiler Ayşe hilal,Akgun Cagliyan Gulsum,ÖZÇELİK ŞENGÖZ CEYDA,Karatas Ayse,BULDUK TUBA,ÖZCAN Alper,belen fatma burcu,CANBOLAT Aylin,Kartal Ibrahim,Ören Hale,Toret Ersin,Özdemir Nihal,Bakanay Sule Mine Castleman Disease: A Multicenter Case Series from Turkey. Turkish Journal of Hematology 39, no.2 (2022): 130 - 135. 10.4274/tjh.galenos.2022.2021.0670
MLA Gündüz Eren,Kirkizlar Hakki Onur,ÜMİT Elif Gülsüm,karaman gülsaran sedanur,Ozkocaman Vildan,Özkalemkaş Fahir,candar ömer,ELVERDİ TUĞRUL,Kucukyurt Selin,Paydas Semra,Ceneli Ozcan,KARAKUŞ Sema,MARAL SENEM,Ekinci Omer,İpek Yıldız,Kis Cem,Guven Zeynep Tugba,AKDENİZ Aydan,CELKAN TÜLIN TIRAJE,Eroğlu küçükdiler Ayşe hilal,Akgun Cagliyan Gulsum,ÖZÇELİK ŞENGÖZ CEYDA,Karatas Ayse,BULDUK TUBA,ÖZCAN Alper,belen fatma burcu,CANBOLAT Aylin,Kartal Ibrahim,Ören Hale,Toret Ersin,Özdemir Nihal,Bakanay Sule Mine Castleman Disease: A Multicenter Case Series from Turkey. Turkish Journal of Hematology, vol.39, no.2, 2022, ss.130 - 135. 10.4274/tjh.galenos.2022.2021.0670
AMA Gündüz E,Kirkizlar H,ÜMİT E,karaman gülsaran s,Ozkocaman V,Özkalemkaş F,candar ö,ELVERDİ T,Kucukyurt S,Paydas S,Ceneli O,KARAKUŞ S,MARAL S,Ekinci O,İpek Y,Kis C,Guven Z,AKDENİZ A,CELKAN T,Eroğlu küçükdiler A,Akgun Cagliyan G,ÖZÇELİK ŞENGÖZ C,Karatas A,BULDUK T,ÖZCAN A,belen f,CANBOLAT A,Kartal I,Ören H,Toret E,Özdemir N,Bakanay S Castleman Disease: A Multicenter Case Series from Turkey. Turkish Journal of Hematology. 2022; 39(2): 130 - 135. 10.4274/tjh.galenos.2022.2021.0670
Vancouver Gündüz E,Kirkizlar H,ÜMİT E,karaman gülsaran s,Ozkocaman V,Özkalemkaş F,candar ö,ELVERDİ T,Kucukyurt S,Paydas S,Ceneli O,KARAKUŞ S,MARAL S,Ekinci O,İpek Y,Kis C,Guven Z,AKDENİZ A,CELKAN T,Eroğlu küçükdiler A,Akgun Cagliyan G,ÖZÇELİK ŞENGÖZ C,Karatas A,BULDUK T,ÖZCAN A,belen f,CANBOLAT A,Kartal I,Ören H,Toret E,Özdemir N,Bakanay S Castleman Disease: A Multicenter Case Series from Turkey. Turkish Journal of Hematology. 2022; 39(2): 130 - 135. 10.4274/tjh.galenos.2022.2021.0670
IEEE Gündüz E,Kirkizlar H,ÜMİT E,karaman gülsaran s,Ozkocaman V,Özkalemkaş F,candar ö,ELVERDİ T,Kucukyurt S,Paydas S,Ceneli O,KARAKUŞ S,MARAL S,Ekinci O,İpek Y,Kis C,Guven Z,AKDENİZ A,CELKAN T,Eroğlu küçükdiler A,Akgun Cagliyan G,ÖZÇELİK ŞENGÖZ C,Karatas A,BULDUK T,ÖZCAN A,belen f,CANBOLAT A,Kartal I,Ören H,Toret E,Özdemir N,Bakanay S "Castleman Disease: A Multicenter Case Series from Turkey." Turkish Journal of Hematology, 39, ss.130 - 135, 2022. 10.4274/tjh.galenos.2022.2021.0670
ISNAD Gündüz, Eren vd. "Castleman Disease: A Multicenter Case Series from Turkey". Turkish Journal of Hematology 39/2 (2022), 130-135. https://doi.org/10.4274/tjh.galenos.2022.2021.0670