A Case of Acute Myelogenous Leukemia (FAB; M5a) Presenting Isolated Testicular Relapse

We encountered a 3-year-old boy with isolated left testicular relapse of acute myelogenous leukemia (AML : FAB classification M5a) that occurred 11 months after the completion of chemotherapy. The bone marrow specimen revealed a massive infiltration of monoblasts with chromosomes involving t (9; 11)...

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Published inThe Japanese Journal of Pediatric Hematology Vol. 19; no. 4; pp. 220 - 223
Main Authors NAKAGAWA, Atsuko, KOBAYASHI, Ryoji, SATO, Tomonobu, IGUCHI, Akihiro, NAKAJIMA, Masahide
Format Journal Article
LanguageJapanese
Published THE JAPANESE SOCIETY OF PEDIATRIC HEMATOLOGY/ONCOLOGY 2005
特定非営利活動法人 日本小児血液・がん学会
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ISSN0913-8706
1884-4723
DOI10.11412/jjph1987.19.220

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Abstract We encountered a 3-year-old boy with isolated left testicular relapse of acute myelogenous leukemia (AML : FAB classification M5a) that occurred 11 months after the completion of chemotherapy. The bone marrow specimen revealed a massive infiltration of monoblasts with chromosomes involving t (9; 11) (p22; q23) at the initial diagnosis. At the time of recurrence, histopathological findings on biopsied testis mimicked those of non-Hodgkin's lymphoma (NHL); however, MLL/AF9 chimera mRNA was detected from blasts of testis by reverse transcription-polymerase chain reaction (RT-PCR). On the basis of this finding, he was diagnosed as having testicular relapse of acute myelogenous leukemia. Although a bone marrow specimen revealed infiltration of leukemic cells and monoblasts were detected in cerebrospinal fluid, he has again been in remission with AML-99 protocol. As testicular relapse in AML is extremely rare, it was difficult for us to diagnose this case. It is suggested that RT-PCR is useful to diagnose overt testicular relapse of AML, even though histopathological findings mimic those of NHL.
AbstractList We encountered a 3-year-old boy with isolated left testicular relapse of acute myelogenous leukemia (AML : FAB classification M5a) that occurred 11 months after the completion of chemotherapy. The bone marrow specimen revealed a massive infiltration of monoblasts with chromosomes involving t (9; 11) (p22; q23) at the initial diagnosis. At the time of recurrence, histopathological findings on biopsied testis mimicked those of non-Hodgkin's lymphoma (NHL); however, MLL/AF9 chimera mRNA was detected from blasts of testis by reverse transcription-polymerase chain reaction (RT-PCR). On the basis of this finding, he was diagnosed as having testicular relapse of acute myelogenous leukemia. Although a bone marrow specimen revealed infiltration of leukemic cells and monoblasts were detected in cerebrospinal fluid, he has again been in remission with AML-99 protocol. As testicular relapse in AML is extremely rare, it was difficult for us to diagnose this case. It is suggested that RT-PCR is useful to diagnose overt testicular relapse of AML, even though histopathological findings mimic those of NHL. 初発時の化学療法終了から11ヵ月後に左睾丸の無痛性腫大で再発した急性骨髄性白血病 (FAB;M5a) の3歳男児例を経験した.初発時に認められたmonoblastは, t (9 : 11) (P22 ; q23) の染色体異常を有していた.再発時に摘出した精巣の病理所見からは, 当初2次性非ポジキンリンパ腫 (NHL) の発生が疑われたが, reverse transcription-polymerase chain reaction (RT-PCR) 法により精巣組織からMLL-AF9キメラmRNAが検出されたことから原疾患の再発と診断した.その後の検査で骨髄・中枢神経系への芽球の浸潤も認め, 化学療法を開始し, 寛解に至った.急性骨髄性白血病 (AML) の精巣再発はきわめてまれであり, かっ同一クローンでもあり, 再発時にphenotypeが異なっていたため本症例ではその診断に苦慮したが, RT-PCR法が確定診断に有用であった.
We encountered a 3-year-old boy with isolated left testicular relapse of acute myelogenous leukemia (AML : FAB classification M5a) that occurred 11 months after the completion of chemotherapy. The bone marrow specimen revealed a massive infiltration of monoblasts with chromosomes involving t (9; 11) (p22; q23) at the initial diagnosis. At the time of recurrence, histopathological findings on biopsied testis mimicked those of non-Hodgkin's lymphoma (NHL); however, MLL/AF9 chimera mRNA was detected from blasts of testis by reverse transcription-polymerase chain reaction (RT-PCR). On the basis of this finding, he was diagnosed as having testicular relapse of acute myelogenous leukemia. Although a bone marrow specimen revealed infiltration of leukemic cells and monoblasts were detected in cerebrospinal fluid, he has again been in remission with AML-99 protocol. As testicular relapse in AML is extremely rare, it was difficult for us to diagnose this case. It is suggested that RT-PCR is useful to diagnose overt testicular relapse of AML, even though histopathological findings mimic those of NHL.
Author SATO, Tomonobu
NAKAGAWA, Atsuko
NAKAJIMA, Masahide
KOBAYASHI, Ryoji
IGUCHI, Akihiro
Author_FL 小林 良二
佐藤 智信
中嶋 雅秀
中川 温子
井口 晶裕
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  fullname: 中川 温子
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  fullname: NAKAGAWA, Atsuko
  organization: Department of Pediatrics, School of Medicine, Hokkaido University
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  organization: Department of Pathology, Aichi Medical University
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  fullname: SATO, Tomonobu
  organization: Department of Pediatrics, School of Medicine, Hokkaido University
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  organization: Department of Pediatrics, School of Medicine, Hokkaido University
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  fullname: NAKAJIMA, Masahide
  organization: Department of Pediatrics, School of Medicine, Hokkaido University
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DocumentTitleAlternate 急性骨髄性白血病(FAB;M5a)における精巣再発の1例
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References 5) Liesveld JL, Dipersio JF, Abboud CN : Integrins and adhesive receptors in normal and leukemic CD34+ progenitor cells : Potential regulatory checkpoints for cellular traffic. Leuk Lymphoma 14 : 19-28, 1994
6) Lal A, Kwan E, Mahr MA, et al : Molecular detection of acute lymphoblastic leukemia in boys with testicular relapse. Mol Pathol 51 : 277-281, 1998
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4) Byrd JC, Edenfield WJ, Shields DJ, et al : Extramedullary myeloid cell tumors in acute nonlymphocytic leukemia : A clinical review. J Clin Oncol 13 : 1800-1816, 1995
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1) Shaffer DW, Burris HA, O'Rourke TJ : Testicular relapse in adult acute myelogenous leukemia. Cancer 70 : 1541-1544, 1992
References_xml – reference: 7) Reinhardt D, Pekrun A, Lakomek M, et al : Primary myelosarcomas are associated with a high rate of relapse : Report on 34 children from the acute myeloid leukemia-Berlin-Frankfurt-Munster studies. Br J Haematol 110 : 863-866, 2000
– reference: 6) Lal A, Kwan E, Mahr MA, et al : Molecular detection of acute lymphoblastic leukemia in boys with testicular relapse. Mol Pathol 51 : 277-281, 1998
– reference: 3) Furman WL, Fontanesi J, Hustu O, et al : Testicular relapse in children with acute nonlymphoblastic leukemia. Cancer 66 : 2095-2098, 1990
– reference: 4) Byrd JC, Edenfield WJ, Shields DJ, et al : Extramedullary myeloid cell tumors in acute nonlymphocytic leukemia : A clinical review. J Clin Oncol 13 : 1800-1816, 1995
– reference: 5) Liesveld JL, Dipersio JF, Abboud CN : Integrins and adhesive receptors in normal and leukemic CD34+ progenitor cells : Potential regulatory checkpoints for cellular traffic. Leuk Lymphoma 14 : 19-28, 1994
– reference: 1) Shaffer DW, Burris HA, O'Rourke TJ : Testicular relapse in adult acute myelogenous leukemia. Cancer 70 : 1541-1544, 1992
– reference: 2) Tobelem G, Jacquillat C, Chastang C, et al : Acute monoblastic leukemia : A clinical and biologic study of 74 cases. Blood 55 : 71-76, 1980
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Snippet We encountered a 3-year-old boy with isolated left testicular relapse of acute myelogenous leukemia (AML : FAB classification M5a) that occurred 11 months...
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StartPage 220
SubjectTerms acute myelogenous leukemia (AML)
reverse transcription-polymerase chain reaction (RT-PCR)
testicular relapse
Title A Case of Acute Myelogenous Leukemia (FAB; M5a) Presenting Isolated Testicular Relapse
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