Indolent T‐lymphoblastic proliferation concomitant with acinic cell carcinoma mimicking T‐lymphoblastic lymphoma: case report and literature review

Yasuda, Hajime; Tsutsui, Miyuki; Ota, Yasunori; Tanaka, Masaru; Komatsu, Norio

doi:10.1111/his.13433

Cited by 6 publications

(6 citation statements)

References 11 publications

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“…Because of the rarity of the disease and the lack of diagnostic guidelines, the diagnosis was based on pathological and clinical similarities to previous documented cases, a er having ruled out T-cell lymphoma [6]. ese findings include: e presence of T-cells expressing TdT and CD3, the presence of a heterogeneous population of immature T cells following the maturation pattern of normal thymocytes, small T-cells without significant morphological atypia, the lack of clonality and a clinically nonprogressive and indolent course.…”

Section: Discussionmentioning

confidence: 99%

Sirolimus for the Treatment of Airway Obstruction due to Indolent T-Lymphoblastic Proliferation

Moughames

Kiess

Akst

et al. 2019

Case Reports in Immunology

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Introduction. Indolent T-lymphoblastic proliferation (iT-LBP) is a rare nonmalignant entity that presents as a proliferation of T-lymphoblasts. We report a first such case with a recurrent laryngeal obstruction presentation that was successfully controlled with Sirolimus. Case presentation. This is the case of a 29-year-old female who presented with a recurrent significant lymphoid hyperplasia in the adenoid and tongue base region as well as a right cervical lymph node. After repeated adenoidectomies and tonsillectomies, and based on pathological and clinical findings she was diagnosed with iT-LBP. Trials of radiotherapy and immunotherapy with cyclosporine and rituximab all failed to control the progression of the disease. Sirolimus was finally able to restrict the growth and improve her symptoms. Conclusion. While It-LBP does not usually require treatment, it is important to report cases in which treatment was crucial for the survival of the patient, and the effective role of Sirolimus in doing so, without any major adverse effects.

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Section: Discussionmentioning

confidence: 99%

Sirolimus for the Treatment of Airway Obstruction due to Indolent T-Lymphoblastic Proliferation

Moughames

Kiess

Akst

et al. 2019

Case Reports in Immunology

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Section: Methodsmentioning

confidence: 99%

“…As of January 2022, 25 papers involving iT‐LBP were identified in the literature 1–4,6,9–28 in addition to one older review 5 . All papers were included in the study 1–4,6,9–28 . All identified cases were reviewed by two of the authors (AS and RSO) for the diagnostic criteria initially put forth by Ohgami et al in 2013 (Table 1).…”

Section: Methodsmentioning

confidence: 99%

“…5 All papers were included in the study. [1][2][3][4]6,[9][10][11][12][13][14][15][16][17][18][19][20][21][22][23][24][25][26][27][28] All identified cases were reviewed by two of the authors (AS and RSO) for the diagnostic criteria initially put forth by Ohgami et al in 2013 (Table 1). Since re-review of pathologic slides of the cases identified was not possible the described cases have been described to be present in various conditions and tissues, especially in relation to Castleman disease.…”

Section: Selection Of Casesmentioning

confidence: 99%

“…The terminology “indolent T‐lymphoblastic proliferation” (iT‐LBP) was first coined by Velankar et al in 1999, in retrospect of a male patient with a T‐lymphoblastic proliferation in the upper aerodigestive tract which remained stable over 16 years 1 . Since then, several studies analyzing reactive T‐lymphoblasts and T‐lymphoblastic proliferations in tumors and secondary lymphoid organs have been reported, and many more cases of iT‐LBP have been described 1–29 . In particular, iT‐LBP has been found in association with entities including Castleman disease (CD), follicular dendritic cell tumor/sarcoma (FDCT/S), and peripheral T‐cell lymphoma (PTCL).…”

Section: Introductionmentioning

confidence: 99%

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Indolent T‐lymphoblastic proliferation: A systematic review of the literature analyzing the epidemiologic, clinical, and pathologic features of 45 cases

Saglam

Singh

Gollapudi

et al. 2022

Int J Lab Hematology

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An indolent T‐lymphoblastic proliferation (iT‐LBP) is a rare benign disorder characterized by an abnormal expansion of immature T‐cells, which morphologically can mimic malignancy. Since the first case was described in 1999, dozens more have been reported in the literature. However, the epidemiologic, clinical, pathologic, and biologic features of this disease have not been well described. Here, we retrospectively reviewed all known cases reported in the literature to better understand this entity. A PubMed search up to January 2022 highlighted 25 papers describing cases/case series of iT‐LBP, one of which was a case presentation in a slide workshop. Except for 9 of the cases in one of the papers, where it was evident that the number of CD3+/TdT+ cells were too few to conform with a diagnosis of iT‐LBP, all papers and all the cases reported were included in the study amounting to a total of 45 cases. Clinicopathologic characteristics were analyzed using descriptive statistics and frequencies. Our analysis highlighted the previously known association with Castleman disease and Castleman‐like features and underlined its association with dendritic cell proliferations in general, as well as uncovering high frequency of concurrence with hepatocellular carcinoma and autoimmune diseases, most notably myasthenia gravis, paraneoplastic pemphigus and paraneoplastic autoimmune multiorgan syndrome. Furthermore, the co‐expression of CD4 and CD8 and high prevalence of extranodal disease and recurrences were other less well described features that were revealed.

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2021

Flow Cytometry of Hematological Malignancies

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Indolent T‐lymphoblastic proliferation concomitant with acinic cell carcinoma mimicking T‐lymphoblastic lymphoma: case report and literature review

Abstract: iT-LBP should always be considered as a differential diagnosis of T-lymphoblastic lymphoma, as the two disorders show extremely similar traits.

Cited by 6 publications

References 11 publications

Sirolimus for the Treatment of Airway Obstruction due to Indolent T-Lymphoblastic Proliferation

Sirolimus for the Treatment of Airway Obstruction due to Indolent T-Lymphoblastic Proliferation

Indolent T‐lymphoblastic proliferation: A systematic review of the literature analyzing the epidemiologic, clinical, and pathologic features of 45 cases

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