Case report: a unique pediatric case of a primary CD8 expressing ALK-1 positive anaplastic large cell lymphoma of skeletal muscle

Diagnostic Pathology

Alexander Marx¹  Andreas Rosenwald⁶  Dietmar Dinter³  Matthias Dürken⁴  Hanns-Peter Scharf²  Torsten Schattenberg²  Eva Geissinger⁶  Timo Gaiser⁵ 

[1] Institute of Pathology, Medical Faculty Mannheim, University of Heidelberg, Theodor-Kutzer-Ufer 1-3, 68167 Mannheim, Germany;Orthopedic Clinic, Medical Faculty Mannheim, University of Heidelberg, Theodor-Kutzer-Ufer 1-3, 68167 Mannheim, Germany;Radiology and Nuclear Medicine, Medical Faculty Mannheim, University of Heidelberg, Theodor-Kutzer-Ufer 1-3, 68167 Mannheim, Germany;Department of Pediatrics, Medical Faculty Mannheim, University of Heidelberg, Theodor-Kutzer-Ufer 1-3, 68167 Mannheim, Germany;Section of Cancer Genomics, Center for Cancer Research, National Cancer Institute, National Institutes of Health, National Insitutes of Health, 50 South Drive, Bldg. 50, Bethesda, MD 20892, USA;Institute of Pathology, University of Würzburg, Josef-Schneider-Straße 2/Bau E2, 97080 Würzburg, Germany

关键词: Pediatric lymphoma;    CD30;    Anaplastic large cell lymphoma;    ALK-1;   

Others  :  808115
DOI  :  10.1186/1746-1596-7-38

 received in 2012-02-23, accepted in 2012-04-12,  发布年份 2012

【 摘 要 】

Primary involvement of skeletal muscle is a very rare event in ALK-1 positive anaplastic large cell lymphoma (ALCL). We describe a case of a 10-year old boy presenting with a three week history of pain and a palpable firm swelling at the dorsal aspect of the left thigh. Histological examination of the lesion revealed a tumoral and diffuse polymorphic infiltration of the muscle by large lymphoid cells. Tumor cells displayed eccentric, lobulated "horse shoe" or "kidney-shape" nuclei. The cells showed immunohistochemical positivity for CD30, ALK-1, CD2, CD3, CD7, CD8, and Perforin. Fluorescence in situ hybridization analysis revealed a characteristic rearrangement of the ALK-1 gene in 2p23 leading to the diagnosis of ALK-1 positive ALCL. Chemotherapy according to the ALCL-99-NHL-BFM protocol was initiated and resulted in a complete remission after two cycles. This case illustrates the unusual presentation of a pediatric ALCL in soft tissue with a good response to chemotherapy.

【 授权许可】

   
2012 Gaiser et al; licensee BioMed Central Ltd.

【 图 表 】

Figure 1.

Figure 2.

【 参考文献 】

• [1]Stein H, Mason DY, Gerdes J, O'Connor N, Wainscoat J, Pallesen G, Gatter K, Falini B, Delsol G, Lemke H, et al.: The expression of the Hodgkin's disease associated antigen Ki-1 in reactive and neoplastic lymphoid tissue: evidence that Reed-Sternberg cells and histiocytic malignancies are derived from activated lymphoid cells. Blood 1985, 66:848-858.
• [2]Stansfeld AG, Diebold J, Noel H, Kapanci Y, Rilke F, Kelenyi G, Sundstrom C, Lennert K, van Unnik JA, Mioduszewska O, et al.: Updated Kiel classification for lymphomas. Lancet 1988, 1:292-293.
• [3]Swerdlow SH, Campo E, Harris NL, Jaffe ES, Pileri SA, Stein H, Thiele J, Vardiman JW: WHO Classification of Tumours of Haematopoietic and Lymphoid Tissues. IARC Fourth edition. 2008.
• [4]Fischer P, Nacheva E, Mason DY, Sherrington PD, Hoyle C, Hayhoe FG, Karpas A: A Ki-1 (CD30)-positive human cell line (Karpas 299) established from a high-grade non-Hodgkin's lymphoma, showing a 2;5 translocation and rearrangement of the T-cell receptor beta-chain gene. Blood 1988, 72:234-240.
• [5]Morris SW, Kirstein MN, Valentine MB, Dittmer K, Shapiro DN, Look AT, Saltman DL: Fusion of a kinase gene, ALK, to a nucleolar protein gene, NPM, in non-Hodgkin's lymphoma. Science 1995, 267:316-317.
• [6]Kuefer MU, Look AT, Pulford K, Behm FG, Pattengale PK, Mason DY, Morris SW: Retrovirus-mediated gene transfer of NPM-ALK causes lymphoid malignancy in mice. Blood 1997, 90:2901-2910.
• [7]Laver JH, Kraveka JM, Hutchison RE, Chang M, Kepner J, Schwenn M, Tarbell N, Desai S, Weitzman S, Weinstein HJ, Murphy SB: Advanced-stage large-cell lymphoma in children and adolescents: results of a randomized trial incorporating intermediate-dose methotrexate and high-dose cytarabine in the maintenance phase of the APO regimen: a Pediatric Oncology Group phase III trial. J Clin Oncol 2005, 23:541-547.
• [8]Driss M, Abbes I, Mrad K, Sassi S, Oubich F, Barsaoui S, Romdhane KB: Primary CD30/ALK-1 positive anaplastic large cell lymphoma of the skeletal muscle in a child. Pathologica 2009, 101:97-100.
• [9]Ishii E, Honda K, Nakagawa A, Urago K, Oshima K: Primary CD30/Ki-1 positive anaplastic large cell lymphoma of skeletal muscle with der(17)t(1;17)(q11;p11). Cancer Genet Cytogenet 2000, 122:116-120.
• [10]Wu L, Wang Y, Fu SL, Huang L, Tongji FC, Qi JY: Anaplastic large cell lymphoma with primary involvement of skeletal muscle: a rare case report and review of the literature. Pediatr Hematol Oncol 2009, 26:142-149.
• [11]Kounami S, Shibuta K, Yoshiyama M, Mitani Y, Watanabe T, Takifuji K, Yoshikawa N: Primary Anaplastic Large Cell Lymphoma of the Psoas Muscle: A Case Report and Literature Review. Acta Haematol 2012, 127:186-188.
• [12]Murphy SB: Classification, staging and end results of treatment of childhood non-Hodgkin's lymphomas: dissimilarities from lymphomas in adults. Semin Oncol 1980, 7:332-339.
• [13]Bishara MR, Ross C, Sur M: Primary anaplastic large cell lymphoma of the breast arising in reconstruction mammoplasty capsule of saline filled breast implant after radical mastectomy for breast cancer: an unusual case presentation. Diagn Pathol 2009, 4:11. BioMed Central Full Text
• [14]Savage KJ, Harris NL, Vose JM, Ullrich F, Jaffe ES, Connors JM, Rimsza L, Pileri SA, Chhanabhai M, Gascoyne RD, et al.: ALK- anaplastic large-cell lymphoma is clinically and immunophenotypically different from both ALK + ALCL and peripheral T-cell lymphoma, not otherwise specified: report from the International Peripheral T-Cell Lymphoma Project. Blood 2008, 111:5496-5504.
• [15]Stein H, Foss HD, Durkop H, Marafioti T, Delsol G, Pulford K, Pileri S, Falini B: CD30(+) anaplastic large cell lymphoma: a review of its histopathologic, genetic, and clinical features. Blood 2000, 96:3681-3695.
• [16]Recavarren RA, Yang J: Cytomorphologic features of primary anaplastic large cell lymphoma of the psoas muscle: a case report and literature review. Diagn Cytopathol 2010, 38:208-212.
• [17]Chim CS, Loong F, Ooi GC, Srivastava G, Liang R: Primary skeletal muscle lymphoma. Am J Med 2002, 112:79-80.
• [18]Wiendl H, Mitsdoerffer M, Weller M: Express and protect yourself: the potential role of HLA-G on muscle cells and in inflammatory myopathies. Hum Immunol 2003, 64:1050-1056.
• [19]Delsol G, Lamant L, Mariame B, Pulford K, Dastugue N, Brousset P, Rigal-Huguet F, al Saati T, Cerretti DP, Morris SW, Mason DY: A new subtype of large B-cell lymphoma expressing the ALK kinase and lacking the 2; 5 translocation. Blood 1997, 89:1483-1490.