Granulocytic Sarcoma Presenting with Severe Adenopathy (Cervical Lymph Nodes, Tonsils, and Adenoids) in a Child with Juvenile Myelomonocytic Leukemia and Successful Treatment with Allogeneic Bone Marrow Transplantation

Imamura, Toshihiko; Matsuo, Seiichiro; Yoshihara, Takao; Chiyonobu, Tomohiro; Mori, Kumiko; Ishida, Hiroyuki; Nishimura, Yasuhiko; Kasubuchi, Yasuo; Naya, Mayumi; Morimoto, Akira; Hibi, Shigeyoshi; Imashuku, Shinsaku

doi:10.1532/ijh97.04040

Cited by 22 publications

(18 citation statements)

References 23 publications

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“…Notably, the subjects treated with Auto/AlloBMT corresponded to long survivors, whereas those who received conventional therapies most often rapidly died of disease (OS at 48 months: 76 vs 0%; see Figure 3). Although not based on a randomized clinical trial, our study provides robust support to the recent concept -based on anecdotal observations 5,15,41 -that patients with MS should undergo high-dose therapies as front-line approach. The latter seems to represent the only chance to achieve CR and to possibly cure the disease.…”

Section: Discussionsupporting

confidence: 68%

“…[1][2][3][4][5][6][7][8][9][10][11][12][13][14][15] It may develop de novo or concurrently with acute myeloid leukemia (AML), myeloproliferative disorder (MPD) or myelodysplastic syndrome (MDS). 1,2,6,11,[13][14][15][16][17][18][19][20] Interestingly, MS may be the first evidence of AML or precede it by months or years. 1,13 Finally, it can represent the initial manifestation of relapse in a previously treated AML in remission.…”

Section: Introductionmentioning

confidence: 99%

“…1,4,5,12,40,41 However, recent studies suggest that either highly specific drugs 42 or aggressive schedules should also be applied to the latter, if one aims to achieve stable remission and cure. 5,14,15 So far, no definitive conclusion has been drawn concerning MS optimal treatment.…”

Section: Introductionmentioning

confidence: 99%

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Myeloid sarcoma: clinico-pathologic, phenotypic and cytogenetic analysis of 92 adult patients

et al. 2006

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Myeloid sarcoma (MS) is a rare neoplasm whose knowledge is largely based on case reports and/or technically dated contributions. Ninety-two MSs in adulthood with clinical data available were evaluated both morphologically and immunohistochemically. Seventy-four cases were also studied by fluorescent in situ hybridization on tissue sections and/or conventional karyotyping on bone marrow or peripheral blood. Histologically, 50% of the tumors were of the blastic type, 43.5% either monoblastic or myelomonocytic and 6.5% corresponded to different histotypes. CD68/KP1 was the most commonly expressed marker (100%), followed by myeloperoxidase (83.6%), CD117 (80.4%), CD99 (54.3%), CD68/PG-M1 (51%), CD34 (43.4%), terminal-deoxy-nucleotidyl-transferase (31.5%), CD56 (13%), CD61/linker for activation of T cells (2.2%), CD30 (2.2%) and CD4 (1.1%). Foci of plasmacytoid monocyte differentiation were observed in intestinal cases carrying inv16. Chromosomal aberrations were detected in about 54% of cases: monosomy 7(10.8%), trisomy 8(10.4%) and mixed lineage leukemia-splitting (8.5%) were the commonest abnormalities, whereas t(8;21) was rare (2.2%). The behavior was dramatic irrespective of presentation, age, sex, phenotype and cytogenetics. Most if not all, long survivors received bonemarrow transplantation. The present report expands the spectrum of our knowledge showing that MS has frequent monoblastic/myelomonocytic differentiation, displays distinctive phenotypic profile, carries chromosomal aberrations other than t(8;21), and requires supra-maximal therapy.

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Section: Discussionsupporting

confidence: 68%

Section: Introductionmentioning

confidence: 99%

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Myeloid sarcoma: clinico-pathologic, phenotypic and cytogenetic analysis of 92 adult patients

et al. 2006

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“…From a therapeutic perspective, data from the literature suggest that GSs are extremely sensitive to focal irradiation or chemotherapy; however, their role is not well defined (13,(16)(17)(18)(19)(20)(21)(22)(23)(24)(25)(26)(27)(28). The optimal treatment for the GS-AML association remains uncertain.…”

Section: Discussionmentioning

confidence: 99%

Immunohistochemical patterns in the differential diagnosis of rhinopharyngeal granulocytic sarcoma

et al. 2016

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Abstract. Granulocytic sarcoma (GS) is a rare extramedullary manifestation of acute myeloid leukemia (AML). GS may develop simultaneously to AML or as a relapse of leukemia, particularly following allogeneic hematopoietic stem cell transplant. Subperiosteal bone, lymph nodes and skin are commonly involved, whereas rhinopharyngeal involvement is less common, with only 14 cases reported in the literature. Due to its rarity, rhinopharyngeal GS may lead to diagnostic pitfalls, particularly when it is poorly differentiated or is without concomitant marrow involvement. Thus, immunohistochemical findings play a key role in diagnosis. The current report describes a case of a 53-year-old female suffering from rhinopharyngeal GS and with a history of AML treated with chemotherapy and radiotherapy, focusing on the importance of the immunohistochemical pattern to assess the right diagnosis. Recent studies have demonstrated that the immunophenotype is of utmost importance for the diagnosis of GS. The high expression of myeloperoxidase (MPO) is common in GS; however, ~30% of GSs do not contain MPO. Therefore, the presence of other markers is required to confirm the diagnosis of GS.

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“…In contrast, pulmonary complications after tonsillectomy are uncommon and are easily misdiagnosed or inappropriately managed [1]. The occurrence of adenopathy in patients with myelodysplastic syndrome (MDS)-associated extramedullary myeloid cell tumors has rarely been reported, and only two cases have been reported in which the tonsils were involved [2,3]. We herein report a case of myeloid sarcoma manifesting as a non-healing tonsillar ulcer in a 57-year-old male smoker.…”

Section: Introductionmentioning

confidence: 96%

Post-tonsillectomy pulmonary complication in a patient with tonsillar myeloid sarcoma

Cheng

Ueng

et al. 2011

Int J Hematol

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Myeloid sarcoma in a patient with myelodysplastic syndrome (MDS) manifesting as a non-healing tonsillar ulcer is an extremely rare occurrence. We report the case of a 57-year-old male smoker with a non-healing tonsillar ulcer who underwent tonsillectomy to rule out tonsillar carcinoma after failed antibiotic therapy. On postoperative day 2, he presented with a temperature of 40°C and white blood cell count of 34700/μL. Antibiotic therapy was begun; however, he died 1 day later due to pulmonary infection and septic shock. Though extremely rare, tonsillar involvement of MDS should be considered in the differential diagnosis of a non-healing tonsillar lesion. When definitive diagnosis requires a tissue sample, punch biopsy may be preferable to tonsillectomy in a patient who may be immunocompromised, and appropriate prophylactic antibiotics should be administered.

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Granulocytic Sarcoma Presenting with Severe Adenopathy (Cervical Lymph Nodes, Tonsils, and Adenoids) in a Child with Juvenile Myelomonocytic Leukemia and Successful Treatment with Allogeneic Bone Marrow Transplantation

Cited by 22 publications

References 23 publications

Myeloid sarcoma: clinico-pathologic, phenotypic and cytogenetic analysis of 92 adult patients

Myeloid sarcoma: clinico-pathologic, phenotypic and cytogenetic analysis of 92 adult patients

Immunohistochemical patterns in the differential diagnosis of rhinopharyngeal granulocytic sarcoma

Post-tonsillectomy pulmonary complication in a patient with tonsillar myeloid sarcoma

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