Spontaneous Remission in Patients with Acute Myeloid Leukemia with t(8;21) or Cutaneous Myeloid Sarcoma: Two Case Reports and a Review of the Literature
Abstract:Spontaneous remission (SR) in patients with acute myeloid leukemia (AML) is rare. We herein present two such cases. The first case was of AML-M2 accompanied by a bone marrow cytogenetic analysis revealing 46, XY, t(8;21)(q22,q22). The second case was of isolated cutaneous myeloid sarcoma (MS) that progressed to AML within seven months. Both of the patients had symptoms of infection and anemia and were therefore treated with antibiotics and transfusions. The SR lasted for two months and one month, respectively.… Show more
“…8,11,13 A statistically significant correlation between fusion transcripts involving the core-binding factor (CBF) complex (RUNX1-RUNX1T1 and CBFB-MYH11) and de novo MS has been reported in other studies. 12,[17][18][19] In our patient cohort, we also found an association between these fusion genes and de novo MS. According to the literature, orbital presentations of MS are often associated with t(8;21) (q22;q22) or KMT2A rearrangements, while the presence of an inv (16) in MS cases seems to be associated with intestinal involvement.…”
De novo myeloid sarcoma mostly presented isolated. Lesions were often localized at skin and lymph nodes. Genetic aberrations frequently involved core-binding factor rearrangements in de novo cases and a complex karyotype in secondary cases.
“…8,11,13 A statistically significant correlation between fusion transcripts involving the core-binding factor (CBF) complex (RUNX1-RUNX1T1 and CBFB-MYH11) and de novo MS has been reported in other studies. 12,[17][18][19] In our patient cohort, we also found an association between these fusion genes and de novo MS. According to the literature, orbital presentations of MS are often associated with t(8;21) (q22;q22) or KMT2A rearrangements, while the presence of an inv (16) in MS cases seems to be associated with intestinal involvement.…”
De novo myeloid sarcoma mostly presented isolated. Lesions were often localized at skin and lymph nodes. Genetic aberrations frequently involved core-binding factor rearrangements in de novo cases and a complex karyotype in secondary cases.
“…Potential other contributing factors including granulocyte-colony stimulating factor (GCSF) administration and hormonal changes (both of which could also impact on innate immune function) have been reported [43]. There is also data suggesting that certain biological subsets of AML may be more likely to undergo SR, specifically FAB subtypes M4/M5 which constitute about half of all reported cases of SR in AML, and certain cytogenetics [3, 4]. Of note, patients with NPM1 mutant AML similar to our patient may also be predisposed towards SR [3, 44].…”
Section: Discussionmentioning
confidence: 99%
“…There is also data suggesting that certain biological subsets of AML may be more likely to undergo SR, specifically FAB subtypes M4/M5 which constitute about half of all reported cases of SR in AML, and certain cytogenetics [3, 4]. Of note, patients with NPM1 mutant AML similar to our patient may also be predisposed towards SR [3, 44]. The presence of NPM1 mutation, found in approximately 35% of AML cases, is known to confer favorable prognosis [45, 46].…”
Section: Discussionmentioning
confidence: 99%
“…It denotes either partial or complete morphologic disappearance of AML without administration of antileukemic therapy [2, 3]. About 100 cases were reported up to 1955 while less than 10 reports were published between 1955 and 1985 [4–7].…”
Spontaneous remission (SR) of acute myeloid leukemia (AML) is a very rare phenomenon. AML characterized by FLT3 internal tandem duplication (FLT3 ITD) is typically associated with an aggressive clinical course with rapid progression, relapse, and short overall survival in the absence of transplantation. We report here the first case of SR of FLT3 ITD mutant AML in the literature. Our patient was an elderly woman with relapsed NPM1 and FLT3 ITD mutant AML whose disease underwent SR for a brief duration without precipitating cause. We review the potential immune mechanisms underlying SR in AML and discuss the implications for novel immunotherapeutic approaches for FLT3 mutant AML.
“…For certain patients, where myeloid sarcomas are accompanied by other hematological diseases, systemic chemotherapy or combined treatment with surgical removal, local radiotherapy and systemic chemotherapy may be performed, depending on the particular situation of each individual patient (8). Although previous studies have reported the spontaneous remission of myeloid sarcomas (25), the majority of these were accompanied with other hematological diseases (18). Therefore, treatment is required to improve the survival rate of patients with myeloid sarcoma.…”
Abstract.The current study presents a case of cluster of differentiation (CD)56 + myeloid sarcoma in a patient that initially presented with skin lesions, and provides evidence for the clinical and differential diagnosis of myeloid sarcoma. The patient of the present case report was a 65-year-old man who was admitted to hospital with a six-month history of bilateral purple-red papules and nodules, which were present on the upper limbs of the patient and had spread over his whole body one month prior to admission to the hospital. Pathological examination demonstrated a diffuse infusion of primitive round cells at the papillary dermis and subcutaneous tissues. The infiltrated cells were 40-60 µm in diameter and morphologically identical. Immunohistochemical examination revealed that the cells expressed myeloperoxidase, CD56, CD43 and T-cell intracytoplasmic antigen. In addition, several cells expressed CD34, and 90% of the cells expressed Ki67. While the majority of cells in myeloid sarcoma do not express CD56, the present case was a myeloid sarcoma that expressed CD56, which is extremely rare. The sarcoma in the present patient progressed rapidly, and the patient died eight months following the onset of disease. Clinicians should be aware of CD56 + myeloid sarcoma, which is easily misdiagnosed and inappropriately treated. Consequently, myeloid sarcoma may have a high malignancy and poor outcome for patients.
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