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PubMed1992

Infantile osteopetrosis: report of two cases.

Two cases of infantile osteopetrosis are reported. Both were males aged four and eight months at presentation. They presented with osteosclerotic change of the bone, leukoerythroblastic anemia, optic atrophy, hepatosplenomegaly and frequent infection. The histology of the bone showed thickened bone trabeculae with little osteoclastic activity, although in one patient the number of osteoclasts increased, while in the other they did not. One received a bone marrow transplant (BMT) but died from disseminated cytomegaloviral infection, pulmonary hemorrhage and sepsis. The post-transplant marrow histology showed evidence of engraftment and osteoclastic activity. The other only received a course of prednisolone, which was of little help. His condition has followed a natural course with progressive visual impairment and marrow failure. Our cases suggest that infantile osteopetrosis should be taken into consideration in dealing with infants who present with early marrow failure and that patients of infantile osteopetrosis should receive BMT. BMTs appear to be the only cure. They should be given as early as possible to avoid major consequences and severe infection.

Bone Marrow Transplantation↗

PubMed2003

Reversal of marrow fibrosis in agnogenic myeloid metaplasia by allogeneic peripheral blood stem cell transplantation.

Agnogenic myeloid metaplasia (AMM) is a clonal hematopoietic stem cell disorder characterized by bone marrow fibrosis, extramedullary hemopoiesis, splenomegaly and a leukoerythroblastic blood picture. Current standard therapies using hydroxyurea, interferon, androgens or corticosteroids have not shown to prolong survival of patients with AMM. In this study, we performed a curative approach using an HLA-matched sibling as a donor for allogeneic peripheral blood stem cell transplantation (PBSCT) for a 45-year-old woman with AMM. Busulfan and cyclophosphamide were given as a conditioning regimen from day -7 to day -2 with cyclosporinA and methotrexate as post-transplant immunosuppressive therapy. Donor PBSCs were mobilized by G-CSF at 16 microg/kg/day for five days and transplantation was performed on March 2-3, 2000. The patient rapidly engrafted within 2 weeks after PBSC infusion without evidence of graft versus host disease. Her blood counts and bone marrow 2 years after transplantation were normal with full donor pattern by molecular analysis. In conclusion, marrow fibrosis can be reverted to normal by allogeneic PBSCT. Allogeneic PBSCT should thus be offered to AMM patients if an HLA-matched sibling is available. This report represents the first SCT for AMM in Thailand.

Busulfan↗

PubMed2005

[Myelofibrosis with myeloid metaplasia: diagnosis and treatment].

Myelofibrosis with myeloid metaplasia is the rarest myeloproliferative syndrom. Diagnosis is often easy in classical form, characterised by splenomegaly, leukoerythroblastic blood reaction and tear-drop erythrocytes on blood count and myelofibrosis on bone marrow biopsy. Evolution is highly variable with a median overal survival of 40 to 60 months and numerous prognostic factors especially anemia. No treatment has been demonstrated to improve survival (apart from allogenic bone marrow transplant). The rarity and the complexity of the disease are the most important difficulties for the definition of standardized diagnostic, prognostic and therapeutic criteria.

Bone Marrow Transplantation↗

PubMed2006

Idiopathic myelofibrosis without dacryocytes.

Idiopathic myelofibrosis (IMF) typically presents with marrow fibrosis, splenomegaly, progressive anemia, and a leukoerythroblastic blood smear with dacryocytes. We present a patient with IMF who did not have dacryocytes.

Aged↗

PubMed1990

Primary myelofibrosis in children: report of 4 cases.

From 1982 to 1985, four cases of primary myelofibrosis were diagnosed in our department. Three were boys and one was a girl. Their ages ranged from 7 months to 15 years. The diagnosis was made based on anemia, leukoerythroblastic change and presence of giant platelets in the peripheral blood, and a bone marrow biopsy showing myelofibrosis. Most of them had anemia, fever, and hepatosplenomegaly on admission. The anemia was severe and refractory to repeated transfusions and steroid therapy in 3 out of the 4 cases. Splenectomy was performed in 1 case, but without satisfactory results. The clinical course and blood pictures in one case resembled leukemia of megakaryocyte lineage (M7), but results of marker studies of the blast cells ruled out the possibility of M7. Three of them underwent leukemic transformation within 2 years and died soon after. The other one died of sepsis 2 weeks after diagnosis. Myelofibrosis in childhood occurs rarely, however, when it does, it always runs a rapid and fatal course.

Adolescent↗

PubMed1989

[The use of MCNU to a patient of primary myelofibrosis complicated with pericardial effusion and proteinuria].

A case of primary myelofibrosis complicated with pericardial effusion and proteinuria is described. A 66-year-old female was admitted to our hospital because of abdominal fullness and shortness of breath. On admission, hepatosplenomegaly and pericardial effusion were observed. Blood examination revealed leukoerythroblastic anemia and thrombocytosis with tear drop cells and giant platelets. Bone marrow aspiration was dry tap and its biopsy showed remarkable myelofibrosis. Urinalysis indicated severe proteinuria. Although neutrophilic alkaline phosphatase score was low, no signs of acute blastic crisis of chronic myelogenous leukemia was found. The diagnosis of an atypical type of primary myelofibrosis was obtained. Administration of MCNU was started in August 1987. Hepatosplenomegaly, pericardial effusion and proteinuria were gradually improved after the administration. The etiology of the pericardial effusion and proteinuria were not obvious, however, these facts suggest that these abnormal findings might be related to PMF itself and MCNU was effective to PNF.

Aged↗

PubMed1988

Nonimmunologic hydrops fetalis resulting from intrauterine human parvovirus B-19 infection: report of two cases.

Two cases of nonimmunologic hydrops fetalis associated with intrauterine human parvovirus B-19 infection are reported. In these cases, hydrops fetalis was diagnosed with ultrasound at 21 and 22 weeks' gestation after 10 or more days of maternal flu-like symptoms. The outcome was stillbirth in one case and neonatal death in the other. In both cases, intrauterine infection by human parvovirus B-19 was confirmed based on two findings: maternal serum positive for human parvovirus B-19 immunoglobulin M (IgM) antibody, and human parvovirus B-19 DNA detected in the fetal organs using Southern blotting and hybridization with a 32P-labeled probe. Laboratory tests on cord blood demonstrated a red blood cell count of 163 x 10(4)/microL, and nucleated red cells numbering 1267 per 50 white cells in the live-birth case. Histologic examinations of fetal tissues demonstrated leukoerythroblastic reaction in the liver and spleen, granular hemosiderin deposition in hepatocytes and Kupffer cells, and bilirubin deposition in the intercellular space in the liver. This evidence indicates that, in some fetuses with intrauterine human parvovirus B-19 infection, hydropic changes may be induced by the sudden decrease in oxygen-carrying capacity of the blood due to severe anemia caused by the infection.

Adult↗

PubMed1986

Tumors of unknown origin in the bone marrow.

Tumors of unknown origin represent a common presentation of malignancy. However, tumors of unknown origin presenting as bone marrow metastases are infrequently reported. The records of 11,820 bone marrow biopsy procedures over a 15-year period at a university hospital and a veteran's hospital were reviewed, disclosing 25 cases of tumor of unknown primary site. Most of the patients were elderly and presented with bone pain or abdominal pain. Anemia, thrombocytopenia, and a leukoerythroblastic blood picture were common hematologic findings. Examination for detection of the primary site was usually unrewarding. The median survival of patients was very short (18 days) and therapy seldom altered survival.

Adenocarcinoma↗

PubMed1985

Clinicopathologic aspects of acute leukemias in the dog.

Nineteen cases of canine acute leukemia were diagnosed during a 4-year period. Two main categories were identified on the basis of cytologic, hematologic, and clinical features: acute lymphoid leukemia and acute myelogenous leukemia. Clinical features included history of weight loss, anorexia, shifting limb lameness, and incoordination. Physical findings were characterized by hepatomegaly, splenomegaly, mild generalized lymphadenopathy, and pallor. Ocular lesions were found in 29% of dogs with acute myelogenous leukemia. Hematologic abnormalities included anemia, thrombocytopenia, pancytopenia, leukemia, and leukoerythroblastic reactions. Results of therapy were discouraging.

Acute Disease↗

PubMed1985

[Clinicopathological examination of 12 autopsy cases of carcinomatosis of the bone marrow].

Twelve autopsy cases of carcinomatosis of the bone marrow were examined clinicopathologically. Among them, 7 were gastric adenocarcinoma, and the other 5 were a rectal carcinoid and carcinomas of the lung, prostate, maxilla and kidney, respectively. The gastric cancers were almost all poorly differentiated adenocarcinoma with mucin production and presented poorer prognoses than the other cancers. Leukoerythroblastic anemia, microangiopathic hemolytic anemia and DIC were found more frequently in the gastric cancers than in the others. It is concluded that the evolution of these critical hematologic disorders may be dependent on differences of histologic type, original focus and cancer-host interactions as well as wide-spread skeletal metastases of cancer cells.

Adult↗

PubMed1977

[Changes in the processes of bone marrow cell proliferation and differentiation in white mice under the influence of heparin].

In experiments carried out on albino mice daily heparin injections (250 units/kg) for two weeks resulted in 30% increase of leukocyte count in the venous blood. The femur bone marrow kariocyte count remained unchanged, while the leukoerythroblastic ratio rose from 4.1 to 7.6. Autoradiography demonstrated that the duration of the mitotic cycle and its phases in myeloid cells underwent no change, constituting: T--13 hrs, ts--8 hrs, tG2--1 hour, t(G1+M)--4 hrs. In heparinized animals the curve of labeled mitoses was nearly ideal. It is suggested that there occurred a change in the stem cells differentiation towards the myeloid type in the bone marrow under the effect of heparin.

Animals↗

PubMed1996

Posttransplant T-cell lymphoproliferative disorders--an aggressive, late complication of solid-organ transplantation.

T-cell non-Hodgkin's lymphomas are an uncommon occurrence after solid-organ transplantation. We describe a morphologically and immunophenotypically distinct group of T-cell lymphoproliferative disorders that occurred late in the course of six patients with solid-organ transplants. The patients ranged in age from 31 to 56 years (median, 43). Three were male; all were splenectomized. The interval from transplant to the diagnosis of lymphoma ranged from 4 to 26 years (median, 15). Symptoms at presentation were related to sites of involvement. Pulmonary, marrow, and CNS involvement were present in five, four, and one case, respectively. No patient had lymphadenopathy. Five patients had an elevated lactate dehydrogenase level (range, 226 to 4,880 IU/L; median, 1,220 IU/L). Five of six patients had a leukoerythroblastic reaction. All cases had large-cell histology and frequently contained cytoplasmic granules. Those cases tested expressed CD2, CD3, and CD8 and were negative for B-cell antigens. T-cell receptor beta- and gamma-chain genes were clonally rearranged in three of three and one of three cases, respectively. All T-cell posttransplant lymphoproliferative disorders (T-PTLDs) studied were negative for Epstein-Barr virus (EBV), human T-cell leukemia/lymphoma virus type 1 (HTLV-1), human T-cell leukemia/lymphoma virus type 2 (HTLV-2), and human herpes virus type 8 (HHV-8) genomes. Treatment with acyclovir (three patients) or chemotherapy (three patients) resulted in two responses. All patients had an aggressive course, with a median survival duration of 5 weeks. In conclusion, a clinically aggressive T-PTLD may be a late complication of solid-organ transplantation and does not appear to be related to EBV, HTLV-1, HTLV-2, or HHV-8 infection.

Adult↗

PubMed1998

[Microangiopathic hemolytic anemia. A form of presentation of stomach neoplasm].

The authors report the case of a 53-year-old female with symptomatic anemia and hemorrhagic diathesis. The patient presented thrombocytopenia, microangiopathic hemolytic anemia and leukoerythroblastic reaction in peripheral blood film. The diagnosis of metastatic gastric carcinoma was made by subsequent studies. A review is made of the literature concerning microangiopathic hemolytic anemia, namely its etiology, mechanisms of pathogenesis and association with metastatic carcinoma.

Adenocarcinoma↗

PubMed1957