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Letters to the Editor

Indian Pediatrics 2005; 42:1166-1167

Testicular Involvement in Blast Crisis of Chronic Myeloid Leukemia


A 17-year-old boy presented with complaints of weakness, low-grade fever and abdominal distension of one-month duration. He had pallor and massive splenomegaly. Complete blood count showed hemoglobin 7.9 g/dL, WBC count 245900/mm3 with 30% neutrophils, 10% lymphocytes, 3% myeloblasts and 57% myelocytes and metamyelocytes. The platelet count was 93000/mm3. Bone marrow aspiration revealed a hypercellular marrow consistent with a diagnosis of chronic myeloid leukemia. Cytogenetic analysis confirmed the presence of Philadelphia chromosome.

Patient was started on Imatinib mesylate in the dose of 400 mg/day. He tolerated Imatinib well for 2 months. Splenomegaly regressed and hematological remission was achieved. In the third month, patient developed myelosuppression with prolonged pancyto-penia for which Imatinib was withheld. Bone marrow aspiration and biopsy performed during this period showed a regenerating marrow. Imatinib was finally resumed after 11 weeks in the dose of 300 mg/day but was withheld within 10 days as the patient again developed thrombocytopenia.

Four weeks later, patient presented with fever, weakness and decreased hearing. He was pale, had generalized lymphadenopathy, mild hepatosplenomegaly and bilateral painless testicular enlargement. Complete blood count revealed hemoglobin 3.5 g/dL WBC count 4400/mm3 with 58% neutrophils, 28% lymphocytes 4% monocytes and 6% blasts. The platelet count was 18000/mm3. The bone marrow was hypercellular with 85% blasts consistent with blast crisis of CML. Immunophenotyping revealed CD10 and CD20 positive B-lineage lymphoblastic leukemia. Scrotal sonography showed bilateral enlarged testis with decreased echogenicity and coarse echotextue. The cerebrospinal fluid cytology was positive for blasts. The patient was started on induction chemotherapy as per the MCP 841 Protocol for acute lymphoblastic leukemia. He responded well initially with resolution of hepato-splenomegaly, testiculomegaly and CNS symptoms but died during induction chemotherapy due to septicemia.

Our patient achieved hematological remission with Imatinib. In view of prolonged myelosupression, Imatinib was withheld and patient rapidly progressed to lymphoid blast crisis with testicular and CNS involvement. Testicular involvement is extremely rare in blast crisis of chronic myeloid leukemia. In acute lymphoblastic leukemia, it is uncommon at presentation but being a sanctuary site, testicular relapses are common.

Literature reviews revealed only four reports of testicular involvement in blast crises of CML(1-4). Two of these four patients were children who had simultaneous involvement of testes and CNS. The prognosis of blast crisis in CML is poor. Remissions can be achieved using high dose chemotherapy and stem cell transplant but are usually short lived.

Anupama Borker,
S.H. Advani,

Department of Medical and
Pediatric Oncology,
Asian Institute of Oncology,
S.L. Raheja Hospital,
Mahim, Mumbai 400 016, India.
E-mail: [email protected]

 

References

1. Beedassy A, Topolsky D, Styler M, Crilley P. Extramedullary blast crisis in a patient with chronic myeloid leukemia in complete cyto-genetic and molecular remission on interferon alfa therapy. Leukemia 2000; 24: 733-735.

2. Carlson NL, Erichson G, Lissel L. Simultaneous meningeal and testicular lymphoblastic transformation of Philadelphia positive CML in a three year old. Anticancer Res 1990; 10: 1739-1741.

3. Ohyashiki JH, Ohyashiki K, Shimizu H, Miki M, Kimura N, Mori S, Fujisawa K, Akatsuka J, Toyama K. Testicular tumor as the first manifestation of B-lymphoid blastic crisis in a case of Ph-positive chronic myelogenous leukemia. Am J Hematol 1988; 29: 164- 167.

4. Kusumakumari P, Kumar SR, Pillai GR. Unusual course of chronic myeloid leukemia. A report. Am J Clin Oncol 1994; 17: 19-21.

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