Clinicopathological features of primary thyroid Burkitt’s lymphoma: a systematic review and meta-analysis

Abstract Background Primary thyroid Burkitt’s lymphoma (BL) is an extremely rare and highly aggressive form of non-Hodgkin’s lymphoma; only isolated case reports are available for patients with this disease. Methods We analyzed the clinicopathological features of thyroid BL by conducting a meta-anal...

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Main Authors: Toshitetsu Hayashi, Mitsuyoshi Hirokawa, Seiji Kuma, Miyoko Higuchi, Ayana Suzuki, Risa Kanematsu, Takumi Kudo, Naomi Katsuki, Reiji Haba, Akira Miyauchi
Format: Article
Language:English
Published: BMC 2020-02-01
Series:Diagnostic Pathology
Subjects:
Online Access:https://doi.org/10.1186/s13000-020-00933-z
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author Toshitetsu Hayashi
Mitsuyoshi Hirokawa
Seiji Kuma
Miyoko Higuchi
Ayana Suzuki
Risa Kanematsu
Takumi Kudo
Naomi Katsuki
Reiji Haba
Akira Miyauchi
author_facet Toshitetsu Hayashi
Mitsuyoshi Hirokawa
Seiji Kuma
Miyoko Higuchi
Ayana Suzuki
Risa Kanematsu
Takumi Kudo
Naomi Katsuki
Reiji Haba
Akira Miyauchi
author_sort Toshitetsu Hayashi
collection DOAJ
description Abstract Background Primary thyroid Burkitt’s lymphoma (BL) is an extremely rare and highly aggressive form of non-Hodgkin’s lymphoma; only isolated case reports are available for patients with this disease. Methods We analyzed the clinicopathological features of thyroid BL by conducting a meta-analysis of 21 known patients (including ours) and compared them to those of extrathyroidal BL. Results There were 13 men and 8 women with a median age of 39.3 years (range, 6–75 years). The median follow-up was 46.5 months (range, 0.5–361 months). Six patients (28.6%) had stage I disease, 2 (9.5%) had stage II, 2 (9.5%) had stage III, and 11 (52.4%) had stage IV. Five of 7 tested patients with thyroid BL (71.4%) had histological evidence of underlying Hashimoto’s thyroiditis. Ki-67 labeling indices exceeding 90% in all 19 patients tested (100%). Fluorescence in situ hybridization performed on 12 patient samples revealed that all (100%) had MYC rearrangement. Among the 16 patients for whom follow-up data were available, 4 died of disease-related causes. Kaplan-Meier analysis revealed that the 12- and 60-month overall survival rates for patients with thyroid BL were 87.5 and 70.7%, respectively. Conclusions Ours was the largest study of thyroid BL and its detailed clinicopathological features to date. Thyroid BL is not associated with underlying Epstein-Barr virus infection but is closely linked to Hashimoto’s thyroiditis; patients generally have good overall survival and respond well to intensive chemotherapy. The correct pathological diagnosis is essential for treatment selection and outcome improvement.
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spelling doaj.art-9f4229f89c3b42c4b642c1266106bdb02022-12-21T22:41:31ZengBMCDiagnostic Pathology1746-15962020-02-011511810.1186/s13000-020-00933-zClinicopathological features of primary thyroid Burkitt’s lymphoma: a systematic review and meta-analysisToshitetsu Hayashi0Mitsuyoshi Hirokawa1Seiji Kuma2Miyoko Higuchi3Ayana Suzuki4Risa Kanematsu5Takumi Kudo6Naomi Katsuki7Reiji Haba8Akira Miyauchi9Department of Diagnostic Pathology and Cytology, Kuma HospitalDepartment of Diagnostic Pathology and Cytology, Kuma HospitalDepartment of Diagnostic Pathology and Cytology, Kuma HospitalDepartment of Diagnostic Pathology and Cytology, Kuma HospitalDepartment of Diagnostic Pathology and Cytology, Kuma HospitalDepartment of Diagnostic Pathology and Cytology, Kuma HospitalDepartment of Internal Medicine, Kuma HospitalDepartment of Diagnostic Pathology, Takamatsu Red Cross HospitalDepartment of Diagnostic Pathology, Faculty of Medicine, Kagawa UniversityDepartment of Surgery, Kuma HospitalAbstract Background Primary thyroid Burkitt’s lymphoma (BL) is an extremely rare and highly aggressive form of non-Hodgkin’s lymphoma; only isolated case reports are available for patients with this disease. Methods We analyzed the clinicopathological features of thyroid BL by conducting a meta-analysis of 21 known patients (including ours) and compared them to those of extrathyroidal BL. Results There were 13 men and 8 women with a median age of 39.3 years (range, 6–75 years). The median follow-up was 46.5 months (range, 0.5–361 months). Six patients (28.6%) had stage I disease, 2 (9.5%) had stage II, 2 (9.5%) had stage III, and 11 (52.4%) had stage IV. Five of 7 tested patients with thyroid BL (71.4%) had histological evidence of underlying Hashimoto’s thyroiditis. Ki-67 labeling indices exceeding 90% in all 19 patients tested (100%). Fluorescence in situ hybridization performed on 12 patient samples revealed that all (100%) had MYC rearrangement. Among the 16 patients for whom follow-up data were available, 4 died of disease-related causes. Kaplan-Meier analysis revealed that the 12- and 60-month overall survival rates for patients with thyroid BL were 87.5 and 70.7%, respectively. Conclusions Ours was the largest study of thyroid BL and its detailed clinicopathological features to date. Thyroid BL is not associated with underlying Epstein-Barr virus infection but is closely linked to Hashimoto’s thyroiditis; patients generally have good overall survival and respond well to intensive chemotherapy. The correct pathological diagnosis is essential for treatment selection and outcome improvement.https://doi.org/10.1186/s13000-020-00933-zThyroid Burkitt’s lymphomaHashimoto’s thyroiditisMeta-analysisClinicopathological features
spellingShingle Toshitetsu Hayashi
Mitsuyoshi Hirokawa
Seiji Kuma
Miyoko Higuchi
Ayana Suzuki
Risa Kanematsu
Takumi Kudo
Naomi Katsuki
Reiji Haba
Akira Miyauchi
Clinicopathological features of primary thyroid Burkitt’s lymphoma: a systematic review and meta-analysis
Diagnostic Pathology
Thyroid Burkitt’s lymphoma
Hashimoto’s thyroiditis
Meta-analysis
Clinicopathological features
title Clinicopathological features of primary thyroid Burkitt’s lymphoma: a systematic review and meta-analysis
title_full Clinicopathological features of primary thyroid Burkitt’s lymphoma: a systematic review and meta-analysis
title_fullStr Clinicopathological features of primary thyroid Burkitt’s lymphoma: a systematic review and meta-analysis
title_full_unstemmed Clinicopathological features of primary thyroid Burkitt’s lymphoma: a systematic review and meta-analysis
title_short Clinicopathological features of primary thyroid Burkitt’s lymphoma: a systematic review and meta-analysis
title_sort clinicopathological features of primary thyroid burkitt s lymphoma a systematic review and meta analysis
topic Thyroid Burkitt’s lymphoma
Hashimoto’s thyroiditis
Meta-analysis
Clinicopathological features
url https://doi.org/10.1186/s13000-020-00933-z
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