Paraneoplastic syndrome and intrathoracic Castleman disease

Hung, Iou‐Jih; Lin, Jainn-Jim; Yang, Chao-Ping

doi:10.1002/pbc.20499

Cited by 10 publications

(14 citation statements)

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“…Yet, cases of unexplained irondeficiency anemia (no stainable iron store in bone-marrow samples and/or low serum ferritin levels) have been reported of children or young patients with CD6 -10 whose conditions were refractory to long-term oral iron supplementation and failed ironabsorption testing. 6,7 It is interesting to note that, as in our case, all of these patients had localized CD with mixedcellularity type, the rarest form, and tumor resection completely corrected the anemia and inflammation. [6][7][8]10 The mechanism of iron deficiency in these cases of CD, without an alternative cause of iron deficiency, had not been elucidated to date.…”

Section: Discussionmentioning

confidence: 80%

“…In addition to occasional autoimmune hemolytic anemia, mild inflammatory anemia is common with CD, but unexplained irondeficiency anemia may also be observed, particularly in children. [6][7][8][9][10] With overproduction of IL-6 as a hallmark, 11 CD could be an interesting human model for studying the contribution of the IL-6/hepcidin pathway in the pathogenesis of ACD.…”

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confidence: 99%

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Iron-Deficiency Anemia in Castleman Disease: Implication of the Interleukin 6/Hepcidin Pathway

et al. 2010

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In addition to occasional autoimmune hemolytic anemia, unexplained iron-deficiency anemia has been reported in childhood Castleman disease (CD). The recent discovery of hepcidin has regenerated the research on iron metabolism. This hormone is a key regulator of iron homeostasis, mainly by inhibiting intestinal iron absorption. Liver expression of hepcidin increases in response to interleukin 6 (IL-6). With chronic overproduction of IL-6 as a hallmark, CD could be an interesting human model for studying the contribution of the IL-6/hepcidin pathway in the pathogenesis of anemia of chronic disease. We report here the case of a 16-year-old boy with chronic iron-deficiency anemia (plasma ferritin: 19 μg/L; plasma iron: 2.2 μmol/L; negative bone marrow Perls' Prussian blue stain), inflammatory syndrome (C-reactive protein: 108 mg/L), and growth retardation for the previous 2 years. Diagnostic workup revealed a large mesenteric mass corresponding to localized CD of mixed histologic type. Resection of the tumor resulted in complete resolution of iron-deficiency anemia and inflammatory syndrome. Parallel variations of plasma IL-6, C-reactive protein, and hepcidin concentrations, together with tumor immunohistochemistry, strongly suggested that IL-6 synthesized by the tumor caused both the inflammation and iron deficiency through enhancement of hepcidin production by the liver. The results of this unique case study (1) explain the mechanism of iron deficiency observed in some children with CD, (2) confirm in vivo the regulatory effect of IL-6 in human hepcidin production, and (3) suggest that iron deficiency is a causal link between IL-6 and anemia of chronic disease.

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Section: Discussionmentioning

confidence: 80%

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confidence: 99%

Iron-Deficiency Anemia in Castleman Disease: Implication of the Interleukin 6/Hepcidin Pathway

et al. 2010

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“…15,16 La coexistencia con enfermedades autoinmunes ha sido poco reportada en la literatura médica. En la publicación de Ginebra y cols en 2004, se detalló la asociación con ciertas enfermedades autoinmunes (tabla), siendo su variedad de células plasmática y mixta como la más frecuente en esta serie, y el lupus eritematoso sistémico, enfermedad mixta de tejido conectivo, miastenia gravis y síndrome de Sjögren como las más frecuentes.…”

Section: Discussionunclassified

“…28 Los mecanismos etiopatogénicos asociados en su desarrollo no están del todo claro; se ha postulado una relación entre la sobreproducción de interleuquina 6, niveles altos de factor de crecimiento endotelial vascular y estimuladores de linfocitos B. 8,11,16,28 La mortalidad asociada a la enfermedad de Castleman en su variante multicéntrica se asocia a procesos infecciosos y malignidad. 10,23,27 Con relación a la polimiositis, y principalmente la dermatomiositis, se observó hasta el 81 % de casos con malignidad hematológica, principalmente linfomas.…”

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Coexistencia entre polimiositis y enfermedad de Castleman; presentación de un caso.

Lam

Cobos

Mejias³

et al. 2015

Rev. Med. UCSG

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ResumenLa coexistencia entre la enfermedad de Castleman y polimiopatia inflamatoria idiopática es poco conocida por cuanto existe pocos casos referentes a estas dos patologías juntas. Las características histopatológicas de los nódulos linfáticos asociados en estas dos entidades son similares, haciendo su diferenciación más difícil. La resolución quirúrgica del tumor conlleva, en la mayoría de casos a la mejoría de las manifestaciones autoinmunes, aunque en ciertos casos, el uso de glucocorticoides, radioterapia, quimioterapia y tratamiento biológicos pueden ser usados cuando la opción quirúr-gica no es suficiente. Se propone como parte de la fisiopatología de la enfermedad de Castleman, compartida en ciertas enfermedades autoinmunes, la inmunoactivación celular asociada con niveles altos de interleukina 6 y otros reactantes de fase aguda, así como ciertos procesos infecciosos como el virus de inmunodeficiencia adquirida y el virus del Epstein-Barr. Se presenta el caso de una mujer de 17 años con diagnóstico de enfermedad de Castleman variante hialino vascular y polimiositis.PALABRAS CLAVE: hiperplasia de ganglio linfático gigante, ganglios linfáticos, miositis. Abstract Coexistence between Castleman disease and idiopathic inflammatory polymyopathy is little known because there are few cases concerning these two diseases together. The histopathologic features of lymph nodes associated in these two entities are similar, making their differentiation more difficult. Surgical tumor resolution involves, in most cases, the improvement of autoimmune manifestations, although in certain cases, the use of glucocorticoids, radiotherapy, chemotherapy, and biological treatment may be possible when the surgical option is not enough. It is proposed as part of Castleman disease pathophysiology, shared in certain autoimmune diseases, cell immune activation associated with high levels of interleukin-6 and other acute phase reactants, as well as certain infectious diseases such as human immunodeficiency virus and Epstein-Barr virus. The case of a 17 year old woman diagnosed with Castleman disease hyaline vascular variant and polymyositis is presented.KEYWORDS: giant lymph node hyperplasia, lymph nodes, myositis. ResumoA coexistência entre a doença de Castlemans e a polimiopatia inflamatória idiopática é pouco conhecida como há poucos casos relativos a essas duas patologias juntas. As características histopatológicas dos gânglios linfáticos associados nestas duas entidades são semelhantes, tornando difícil sua diferenciação. A resolução cirúrgica do tumor envolve, na maioria dos casos a melhoria das manifestações autoimunes, no entanto em alguns casos, o uso de glicocorticoides, radioterapia, quimioterapia e tratamento biológico podem ser utilizados quando a opção cirúrgica não é suficiente. É proposto como parte da fisiopatologia da doença Castlemans, partilhada em certas doenças autoimunes, inmunoativação celular associada a altos níveis de interleucina 6 e de outros reactantes de fase aguda, bem como certos processos infecciosos...

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“…In children, the most common cause is Castleman's disease (also known as angiofollicular hyperplasia, giant lymph node hyperplasia, localized nodal hyperplasia, benign giant lymphoma, or lymphoid hamartoma), which is a rare lymphoproliferative disorder that is disproportionately associated with PNP in children. 75,76,77 Some have proposed that the disease is a response to a viral infection such as Epstein-Barr virus or human herpes virus-8. 77 In adults, the most common causes are nonHodgkins lymphoma, chronic lymphocytic leukemia, Castleman's disease, sarcoma and thymoma.…”

Section: What Are the Differences Between Pemphigus Vulgaris In Childmentioning

confidence: 99%

Bullous diseases: Kids are not just little people

Marathe

Morel

2015

Clinics in Dermatology

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Paraneoplastic syndrome and intrathoracic Castleman disease

Cited by 10 publications

References 23 publications

Iron-Deficiency Anemia in Castleman Disease: Implication of the Interleukin 6/Hepcidin Pathway

Iron-Deficiency Anemia in Castleman Disease: Implication of the Interleukin 6/Hepcidin Pathway

Coexistencia entre polimiositis y enfermedad de Castleman; presentación de un caso.

Bullous diseases: Kids are not just little people

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