Testicular relapse of acute promyelocytic leukemia after allogeneic BMT

Abstract

Summary:analysis of the marrow revealed trisomy 8 and t(15;17) ( Table 1). He was treated with Ara-C and amsacrine and a second remission was achieved after two courses. FollowAfter treatment of acute leukemia (typically ALL and the monocytic variants of AML), relapse may occur at up marrow cytogenetic analysis was normal. This hospitalization was complicated by Aspergillus flavus pneumonia sites other than the marrow. Isolated extramedullary relapse of acute promyelocytic leukemia (APL) howrequiring ICU admission and treatment with 3000 mg of amphotericin B. He was eventually discharged home on cisever, is rare. We describe such an event in a man who underwent allogeneic BMT for APL in second relapse retinoic acid in October 1985.Because of concern about Aspergillus reactivation, an and 4 years later presented with testicular relapse. The marrow was morphologically and cytogenetically norallogeneic BMT was considered inappropriate at this time.He remained well on cis-retinoic acid until December 1987 mal, but RT-PCR analysis revealed the specific PML/RAR chimeric RNA transcript.when he developed hemoptysis. Bronchoscopy was performed and later right upper lobectomy for presumed AsperKeywords: testicular relapse; acute promyelocytic leukemia; allogeneic BMT gillus infection. The pathology of the resected lung, however, revealed necrotizing granulomas consistent with active pulmonary tuberculosis. He was therefore treated with an 8-month course of anti-tuberculous medication. Acute promyelocytic leukemia (APL) is a distinct subtype Treatment with cis-retinoic acid continued. of AML characterized by a consumptive coagulopathy, a Second relapse occurred in September 1990. Cytogenetic specific chromosomal rearrangement, a defined molecular analysis of the marrow revealed tetrasomy 8 and t(15;17) abnormality in a retinoid receptor, and a relatively good (Table 1). He was treated with Ara-C and DNR but a folprognosis. The (15;17) chromosomal translocation fuses the low-up marrow revealed persistent disease. In October PML gene on chromosome 15 with the RAR gene on chro-1990, he underwent an HLA-matched sibling donor BMT mosome 17, resulting in a hybrid gene PML/RAR which using BU, CY conditioning and CsA with methylprednisoencodes a fusion protein, undoubtedly involved in leukelone as GVHD prophylaxis. His hospital course was relamogenesis. 1 Both combination chemotherapy 2 and all-trans retinoic acid (ATRA) 3 are effective treatments.We describe an unusual case in which isolated testicular Table 1 Karyotype of marrow cells at various time points from relapse of APL occurred after allogeneic BMT. 1984-1994 Marrow karyotypeCase report Diagnosis (March 1984) Failed analysisA 34-year-old East Indian man presented in March 1984First relapse (August 1985) 47, XY, +8, t(15;17) [10] with APL. Remission was achieved with high-dose Ara-C Second remission (September 1985) 46, XY [25] and DNR, and consolidated with the same drugs.

High incidence of extramedullary relapse of AML after busulfan/cyclophosphamide conditioning and allogeneic stem cell transplantation

“…31 None of the three AML-M3 patients in our study had exposure to ATRA prior to SCT, although one (UPN 537), as we previously reported, 32 had received extensive isotretinoin (13-cis retinoic acid) maintenance therapy before transplant. Notably, of the three AML-M3 EM relapse patients, one was of East Indian, one was of Asian, and one was of Northern European descent.…”

Bone marrow vs extramedullary relapse of acute leukemia after allogeneic hematopoietic cell transplantation: risk factors and clinical course

“…Of the 78 patients with AML, 26 had normal cytogenetics, nine t(8;21), six t(15;17), and two inv(16) at the time of diagnosis. When we assigned the patients with AML according to the Southwest Oncology Group (SWOG) criteria, 28 17 showed favorable, 28 intermediate, 15 unfavorable, and 10 undetermined risk status. Of the 36 patients with ALL, 26 patients showed pre-B cell type and eight T-cell type on immunophenotyping.…”

“…This remarkable effect is due to the graft-versus-leukemia (GVL) effect that occurs after allogeneic HCT. Although the overall frequency of acute leukemia relapse is less after allogeneic HCT, a high proportion of extramedullary relapses [4][5][6][7] in extremely diverse sites has been reported, including the brain, [8][9][10][11] head and neck, [8][9][10]12 gastrointestinal tract, 13 breast, 14,15 liver, 9 pancreas, 16 urogenital tract, 13,17 spinal canal and paravertebral tissue, 8 bone and periosseous tissue, 13,14,18 pleura, 14 pericardium, 9 peritoneum, 19 and skin. 20 The median time from allogeneic HCT to acute leukemia relapse was longer in cases with extramedullary relapse with or without bone marrow involvement when compared to cases of bone marrow only relapse.…”

Isolated extramedullary relapse after autologous bone marrow transplantation for acute myeloid leukemia: Case report and review of the literature

“…The prognosis is poor, but prolonged survival has been observed in some of these patients, with 13 out of 52 evaluable patients alive having a median survival time of 49 months (range, 11-121). The most effective treatment appeared to be a combined modality of HDCHT and RT, as 7 (54%) out of 13 living patients have been treated with this combined modality [4][5][6][7][8][9][10][11][12][13][14][15][16][17][18][19]. The patient in this report showed two factors associated to IEMR, namely, M4 FAB phenotype and intermediate cytogenetic risk group, including aberration of chromosome 8.…”

“…Our review of the literature disclosed 72 cases of IEMR post alloBMT and 3 cases of IEMR post autoBMT, including our case [2,4,[6][7][8][9][10][11][12][13][14][15][16][17][18][19]. Table I summarizes the clinical and cytogenetic characteristics of the patients.…”