Lymph node involvement by Langerhans cell histiocytosis: a clinicopathologic and immunohistochemical study of 20 cases

Edelweiss, Marcia; Medeiros, L. Jeffrey; Suster, Saul; Moran, César A.

doi:10.1016/j.humpath.2007.03.015

Cited by 56 publications

(43 citation statements)

References 19 publications

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“…The most common sites of nodal involvement are cervical, inguinal, supraclavicular and axillary. 1 Limited disease usually has an excellent clinical outcome while systemic disease is typically associated with significant morbidity and mortality. 2 Several findings support the notion that Langerhans cell histiocytosis is a clonal proliferation of phenotypically immature Langerhans cells including occurrence in homozygous twins in very rare cases.…”

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confidence: 99%

“…The broad histologic differential diagnosis for LCH includes Kimura disease, dermatopathic lymphadenitis, classical Hodgkin lymphoma and allergic granulomatosis. 1 In contrast, Rosai-Dorfman disease is characterized by a non-neoplastic proliferation of S100-positive, CD1a-negative histiocytes with unusual features including emperipolesis of lymphocytes and other hematopoietic cells. Nodal involvement is most common, with 80% of cases showing cervical adenopathy and 43% of patients showing involvement in extranodal sites.…”

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Co-occurrence of Langerhans cell histiocytosis and Rosai–Dorfman disease: possible relationship of two histiocytic disorders in rare cases

O’Malley¹,

Duong

Barry³

et al. 2010

Modern Pathology

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Rosai-Dorfman disease and Langerhans cell histiocytosis are both disorders of accessory immune cells. Two cases have been previously reported of concurrent Langerhans cell histiocytosis and Rosai-Dorfman disease. In this report, we characterize the findings and selected molecular studies in nine additional cases. Histology was reviewed. Immunohistochemical stains were performed on all cases in which slides or blocks were available. A combination of CD1a, S-100, CD3, CD20, langerin, CD68, CD163, CD21, CD35 and CD123 immunohistochemical stains were performed. High-resolution array comparative genomic hybridization was performed on six samples from five cases. In these cases, seven were female and two male, with an average age of 25 years (15 monthsÀ59 years). A majority of the cases were identified in lymph node. Areas of Langerhans cell histiocytosis had a typical appearance with the existence of bland 'coffee-bean' nuclei, clear cytoplasm and associated eosinophils. The immunophenotype was typical, including expression of CD1a, S100, CD68 and langerin. In areas of Rosai-Dorfman disease, there was emperipolesis seen in all cases. Cells were intermediate-large in size with large round nuclei and ample clear or pale cytoplasm. The lesional cells were positive for S100, CD68, CD163, without expression of langerin or CD1a. Array comparative genomic hybridization showed gains and/or losses in four of the six samples. One case showed no gains or losses and one additional case showed gains and losses in the Langerhans cell histiocytosis, while no abnormalities were discovered in the Rosai-Dorfman disease component. These findings are comparable to those seen in previous studies of Langerhans cell histiocytosis. We report the clinical and pathologic findings of the combination of Langerhans cell histiocytosis and Rosai-Dorfman disease. Furthermore, we suggest on the basis of evidence from our cases that, when simultaneous, the two entities may be pathophysiologically related.

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Co-occurrence of Langerhans cell histiocytosis and Rosai–Dorfman disease: possible relationship of two histiocytic disorders in rare cases

O’Malley¹,

Duong

Barry³

et al. 2010

Modern Pathology

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“…Sin embargo, en raras ocasiones, la HCL puede afectar de forma primaria a los ganglios linfáticos sin existir enfermedad en otras localizaciones. 7 Desde nuestro mejor conocimiento, solo se ha descrito, en la literatura, un caso más de HCL localizada que afecta a ganglios linfáticos cervicales en lactantes menores de 6 meses y se presentó en el período neonatal. 2 También hay descrito un caso de un niño de 3 meses de edad, pero la afectación ganglionar era axilar.…”

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Histiocitosis de células de Langerhans presentada como adenitis aislada en un lactante. Caso clínico

Soriano-Ramos

et al. 2016

Arch Argent Pediat

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RESUMENLa histiocitosis de células de Langerhans es una enfermedad infrecuente en el lactante y su presentación como una adenitis cervical aislada sin otra sintomatología es excepcional en estos pacientes. Se describe el caso de una lactante de 3 meses de edad que presentaba una tumoración cervical en el ángulo mandibular derecho, con mala respuesta al tratamiento antibiótico. Se realizó una punción-aspiración con aguja fina, que confirmó el diagnóstico de histiocitosis de células de Langerhans. El estudio de extensión no mostró afectación sistémica. Debe considerarse la histiocitosis de células de Langerhans en el diagnóstico diferencial de una masa cervical subaguda de evolución tórpida en los lactantes de corta edad y se debe plantear la realización de una punción-aspiración con aguja fina de manera precoz para establecer el diagnóstico. Palabras clave: histiocitosis de células de Langerhans, lactante, linfadenitis. ABSTRACTLangerhans cell histiocytosis in infants is a rare condition, and presentation as an isolated cervical adenitis is exceptional at this age. We describe the case of a 3-month-old female infant presenting with a neck mass in the right mandibular angle with poor response to antibiotic treatment. Fine needle aspiration was performed and confirmed the diagnosis of Langerhans cell histiocytosis with complementary tests showing no features of systemic involvement. Langerhans cell histiocytosis should be considered in the differential diagnosis of subacute neck masses with poor outcome in infants and physicians should consider performing a fine needle aspiration to establish the diagnosis.

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“…One case of cystic lymph nodes is not described. Although the Birbeck granule is a diagnostic ultrastructural hallmark, Edelweiss and colleagues 8 have shown a broad spectrum of histologic changes in lymph nodes involved by LCH, from very subtle and focal changes to massive and diffuse changes. The cytologic features can also vary from the absence of eosinophils to a prominent component of multinucleated giant cells.…”

Section: Discussionmentioning

confidence: 99%

Sonography of Biopsy-Proven Langerhans Cell Histiocytosis in Lymph Nodes of the Neck

Park

Suh

Seol

et al. 2009

Journal of Ultrasound in Medicine

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Objective. The purpose of this series is to describe the sonographic findings of lymph nodes of the neck involved by Langerhans cell histiocytosis (LCH). Methods. We reviewed and interpreted available images of lymph nodes in patients with cervical lymphadenopathies involved by LCH for the past decade. In all patients, each abnormal lymph node was pathologically confirmed by the use of a sonographically guided core needle biopsy or an incisional biopsy. Results. We characterized 3 different kinds of imaging findings for cervical lymph nodes involved by LCH: (1) a lymphomalike lesion, (2) a lesion similar to reactive lymphadenopathy, and (3) a cystic lymph node. Conclusions. There was no specific imaging finding to diagnose lymphadenopathy involved by LCH. In our study, we characterized 3 different imaging patterns of lymph node involvement by LCH. This study represents an initial step to organize specific findings for LCH. Key words: cervical lymph node; Langerhans cell histiocytosis; sonography. The clinical manifestations of LCH vary from localized to generalized and from adolescent to progressive. 3 Patients with generalized LCH have a poorer prognosis than patients with localized LCH. Cervical lymph node involvement of LCH usually occurs in patients with a known generalized form of LCH. 4,5 Fortunately, cervical lymph nodes are easy to approach for an invasive study because of their superficial location. Therefore, before an invasive study, familiarity with the imaging findings of LCH involving lymph nodes is important.

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Lymph node involvement by Langerhans cell histiocytosis: a clinicopathologic and immunohistochemical study of 20 cases

Cited by 56 publications

References 19 publications

Co-occurrence of Langerhans cell histiocytosis and Rosai–Dorfman disease: possible relationship of two histiocytic disorders in rare cases

Co-occurrence of Langerhans cell histiocytosis and Rosai–Dorfman disease: possible relationship of two histiocytic disorders in rare cases

Histiocitosis de células de Langerhans presentada como adenitis aislada en un lactante. Caso clínico

Sonography of Biopsy-Proven Langerhans Cell Histiocytosis in Lymph Nodes of the Neck

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