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PubMed2004

Disseminated linear calcinosis cutis associated with the Koebner phenomenon in an infant with congenital acute monocytic leukaemia.

We present a unique case of an infant with acute monocytic leukaemia who presented at birth with multiple rubbery, erythematous to violaceous subcutaneous nodules secondary to leukaemia cutis. As these infiltrates regressed with chemotherapy, numerous white to yellow linear confluent papules appeared in a scratch-like pattern. These lesions were widely disseminated but were concentrated across her face, trunk and extremities with relative sparing of the napkin area and back. We propose that these lesions represent a form of dystrophic calcinosis cutis that occurred secondary to koebnerization in an infant with congenital leukaemia cutis.

Calcinosis↗
PubMed1994

Granulocytic sarcoma presenting as a diffuse renal mass before hematological manifestations of acute myelogenous leukemia.

Granulocytic sarcoma is a rare tumor composed of granulocytic precursor cells. The most common sites of involvement include the bones, soft tissue, lymph nodes and skin. Granulocytic sarcoma usually develops as a delayed manifestation of the disease process in patients with known acute myelogenous leukemia. To our knowledge we report the first case of renal granulocytic sarcoma presenting as the first sign of acute myelogenous leukemia. Computerized tomography and magnetic resonance imaging revealed diffuse nonspecific global tumor infiltration of the kidney and ureter. Renal granulocytic sarcoma was diagnosed on open renal biopsy and acute myelogenous leukemia on bone marrow biopsy. There has been partial response to combination chemotherapy. Renal granulocytic sarcoma may diffusely involve the kidney as the only manifestation of acute myelogenous leukemia and it should be included in the differential diagnosis of the many disease entities with global renal involvement since initial treatment is combination chemotherapy.

Diagnosis, Differential↗
PubMed2005

Synovial tissue inflammation in early and late osteoarthritis.

OBJECTIVE: To compare selected immunohistological features of inflammation in synovial tissue from patients with early and late osteoarthritis (OA). METHODS: Synovial tissue samples were obtained from 10 patients with knee pain, normal radiographs, and arthroscopic manifestations of OA (early OA), and from 15 patients with OA undergoing knee joint arthroplasty (late OA). Conventional immunohistochemical techniques were used to measure microscopic manifestations of inflammation. The inflammatory cell infiltrate, blood vessel formation, and angiogenic factors, NF-kappaB activation, expression of tumour necrosis factor alpha (TNFalpha) and interleukin 1beta (IL1beta), and the presence of cyclo-oxygenase (COX)-1 and COX-2 were quantified. Fibroblast-like synoviocytes (FLS) were isolated from early and late OA tissue samples to compare in vitro production of prostaglandin E2 (PGE2) RESULTS: Synovial tissue from patients with early OA demonstrated significantly greater CD4+ (p = 0.017) and CD68+ (p<0.001) cell infiltration, blood vessel formation (p = 0.01), vascular endothelial growth factor (p = 0.001), and intercellular adhesion molecule-1 expression (p<0.001). Numbers of cells producing TNFalpha and IL1beta were also significantly greater in early OA (p<0.001). Manifestations of inflammation in early OA were associated with increased expression of the NF-kappaB1 (p<0.001) and RelA (p = 0.015) subunits, and with increased COX-2 expression (p = 0.04). Cytokine-induced PGE2 production by cultured FLS was similar in both groups. CONCLUSION: Increased mononuclear cell infiltration and overexpression of mediators of inflammation were seen in early OA, compared with late OA. Isolated FLS were functionally similar in both groups, consistent with microenvironmental differences in the synovial tissue during different phases of OA. These observations may have important therapeutic implications for some patients during the early evolution of OA.

Aged↗
PubMed2001

Orbital mass secondary to precursor T-cell acute lymphoblastic leukemia: a rare presentation.

We describe a 40-year-old woman with a history of precursor T-cell acute lymphoblastic leukemia who developed an orbital mass associated with diffuse infiltration of the paranasal sinuses. The clinical and radiologic findings suggested an orbital abscess. Examination of orbital and ethmoid sinus biopsy specimens revealed relapse of precursor T-cell acute lymphoblastic leukemia. Although orbital involvement by granulocytic sarcoma (also known as extramedullary myeloid cell tumor and chloroma) with or without concurrent acute myeloid leukemia is well described in the literature, similar presence of acute lymphoblastic leukemia of either precursor T-cell or B-cell lineage is rare.

Adult↗
PubMed2004

Thoracic CT findings of adult T-cell leukemia or lymphoma.

OBJECTIVE: The aim of this study was to assess pulmonary CT findings in patients with adult T-cell leukemia or lymphoma. MATERIALS AND METHODS: We retrospectively reviewed CT scans of the lung in 87 patients with adult T-cell leukemia or lymphoma who had undergone chest CT between January 1996 and March 2002 at two institutions. The CT scans were interpreted by two chest radiologists working in consensus. Parenchymal abnormalities (ground-glass attenuation, consolidation, nodules, thickening of bronchovascular bundles, interlobular septal thickening, honeycombing, crazy-paving appearance, and bronchiectasis) were evaluated, as were enlarged lymph nodes, pleural effusion, and pleural thickening. In 46 patients who underwent surgical biopsy or autopsy, CT-pathologic correlation was performed with the actual specimens by a pathologist and two chest radiologists. RESULTS: On the CT scans, abnormal findings were seen in 60 patients (69.0%). CT findings consisted of ground-glass attenuation (n = 37), centrilobular nodules (n = 25), thickening of bronchovascular bundles (n = 22), and consolidation (n = 13). These abnormalities were predominantly seen in the peripheral lung parenchyma (n = 26). Pathologically, these findings corresponded with atypical lymphocyte infiltration along the interstitium and the alveolar spaces. Pleural effusion and enlarged lymph nodes were found in 22 and 27 patients, respectively. CONCLUSION: CT findings in patients with adult T-cell leukemia or lymphoma consisted mainly of ground-glass attenuation, centrilobular nodules, and thickening of the bronchovascular bundles in the peripheral lung. These findings, although nonspecific, are considered suggestive of thoracic involvement in patients with adult T-cell leukemia or lymphoma.

Adult↗
PubMedDate not supplied

CD23 negative chronic lymphocytic leukemia of the tonsil: report of a case.

We present the case of atypical chronic lymphocytic leukemia presenting as a tonsillar mass in an elderly man. Histological examination of the tumor revealed diffuse submucosal infiltration by small lymphocytes. On flow cytometry, a monoclonal B lymphocytic population expressing CD5, CD19, lambda light chain, but not expressing CD10, CD23, or FMC7 activities, was observed. A diagnostic conundrum occurred with the demonstration on molecular cytogenetic analysis of the chromosomal translocation abnormality, t(11;14)(q13;q32). The diagnosis of CD23 negative chronic lymphocytic leukemia was made after further molecular studies failed to detect the bcl-1 gene rearrangement.

Aged↗
PubMed1992

Renal biopsy diagnosis of acute lymphocytic leukemia.

Hyperuricemia, due to inborn errors of metabolism, dehydration, or tumor lysis, may cause renal insufficiency. Hyperuricemia from tumor lysis syndrome in malignancy is usually associated with electrolyte disturbances such as hyperkalemia, hyperphosphatemia or hyper or hypocalcemia. Tumor infiltration into the kidneys can occur, yet this accounts for renal insufficiency in only 1% of patients. This infiltration of tumor cells into the kidneys is usually associated with evidence of malignancy elsewhere as identified by physical exam, radiographic studies, and examination of the peripheral smear or bone marrow. We report an unusual presentation of a child with acute lymphocytic leukemia presenting with acute renal failure, nephromegaly and hyperuricemia without electrolyte disturbances or systemic evidence of tumor elsewhere. We stress the importance of kidney biopsy in order to identify the etiology of the renal failure and hyperuricemia.

Acute Kidney Injury↗
PubMed2000

Leukaemia cutis and leukaemic vasculitis.

BACKGROUND: Leukaemia cutis (LC) and leukaemic vasculitis (LV) are uncommon but important features of haematological malignancy. OBJECTIVES: We aimed to evaluate clinicopathological features of a series of such cases. METHODS: Thirty-four patients with LC were studied. Nineteen had acute myeloblastic leukaemia, six had acute lymphoblastic leukaemia, three had myelodysplastic syndrome-refractory anaemia with excess blast in transformation, three had non-Hodgkin's lymphoma in leukaemic phase, two had chronic lymphocytic leukaemia and one had multiple myeloma. RESULTS: Cutaneous lesions were widely dispersed in 15 patients, but were restricted to the extremities in eight patients. In other patients lesions were localized on the face, hands and feet. Cutaneous lesions were generally erythematous, maculopapular, squamous, nodular, haemorrhagic or necrotic. Two localizations were very interesting: in one case, lesions were confined to the site of previous varicella zoster virus infection; in the other case, leukaemic infiltration occurred within a lesion of cutaneous leishmaniasis. All of the patients had high white blood cell count and/or other findings of high tumour burden. Vasculitis with leukaemic cell infiltration (LV) was detected in 24 patients, and was high-grade LV in 16 patients. Patients with high-grade LV tended to have a higher white blood cell count and other findings associated with an aggressive clinical course compared with patients not having LV. Thirteen patients with LV died within 1 month but five patients lived more than 1 year. CONCLUSIONS: LC, and especially LV, is generally an indicator of poor prognosis. Its pathogenesis must be evaluated further with additional studies, including adhesion molecules, angiogenic factors and other biological parameters.

Adolescent↗
PubMed1995

Satisfactory remission achieved by PUVA therapy in a case of crisis-type adult T-cell leukaemia/lymphoma with generalized cutaneous leukaemic cell infiltration.

We used PUVA therapy in a patient with crisis-type adult T-cell leukaemia/lymphoma and generalized cutaneous leukaemic cell infiltration. PUVA proved very effective in reducing leukaemic cells and in clearing the eruption. To understand the way in which PUVA produced a reduction in the number of leukaemic cells, we examined peripheral blood cells by light and electron microscopy. Light microscopy was of little help, but electron microscopy revealed that PUVA induced apoptosis-like changes in circulating leukaemic cells. This suggests that apoptosis-like changes in leukaemic cells might be the reason for the success of this treatment.

Apoptosis↗
PubMed2005

[The clinical and immunological manifestations of infiltrative pulmonary tuberculosis under natural zinc deficiency].

Patients with infiltrative pulmonary tuberculosis who lived in zinc-deficient areas (the level of mobile zinc in the soil is < 0.1 mg/kg, and its dietary level is 9.1 mg/day) were found to have a significant depression of the T-cell link of the immunity system along with enhanced activation apoptosis and decreased phagocytic activity of neutrophils. The detected immunological disorders determine a sluggish and asymptomatic onset and an unfavorable course of the disease with a predominance of destructive and bacillary forms.

Adult↗
PubMed2004

Alemtuzumab-induced remission of both severe paraneoplastic pemphigus and leukaemic bone marrow infiltration in a case of treatment-resistant B-cell chronic lymphocytic leukaemia.

BACKGROUND: Alemtuzumab (MabCampath; ILEX Pharmaceuticals, Geneva, Switzerland) is a humanised monoclonal antibody directed against CD52. It belongs to a new group of monoclonal antibodies with anti-neoplastic effects used in chronic lymphocytic leukaemia (CLL) either as first-line treatment or in those cases resistant to alkylating drugs. Paraneoplastic pemphigus (PNP) is a severe mucocutaneus disease mostly associated with B-cell lymphoproliferative disorders. Independent of the course of the underlying malignancy, this disease is often resistant to conventional immunosuppressive treatment and may lead to death as a result of infectious complications. CASE PRESENTATION: We report a case where an ongoing long-term remission of PNP has been induced by alemtuzumab in a patient with an underlying B-CLL. A 68-yr-old male with a 4-yr history of B-CLL presented with a widespread blistering eruption on the extremities and trunk and a severe stomatitis. The diagnosis of PNP relied on the clinical, histological and direct immunofluorescence findings. Despite intensive treatment strategies with various immunosuppressive drugs and antibiotics, blisters continued to develop and the patient was deteriorating. When treated with alemtuzumab the mucocutaneous lesions healed almost completely within a few weeks and the patients' general condition improved significantly. After 12 wk of treatment with alemtuzumab, the CLL infiltration of the bone marrow previously quantified at 75-80% remitted completely. Twelve months later, the patient was still in remission with only a small residual ulceration on the lip and one on the penis. CONCLUSIONS: Based on this case report we recommend treatment with alemtuzumab to severe cases of PNP in CLL. However, further follow-up of this case is needed in order to assess the long-term effect of alemtuzumab treatment in PNP.

Aged↗
PubMed2007

Extramedullary infiltration at diagnosis and prognosis in children with acute myelogenous leukemia.

BACKGROUND: Extramedullary infiltration (EMI) is an occasional clinical symptom in childhood acute myelogenous leukemia (AML), but there is considerable controversy regarding the prognostic significance of EMI in AML. PROCEDURE: We evaluated the frequency and prognostic significance of EMI at diagnosis of AML in children. RESULTS: Of 240 cases of de novo AML excluding children with Down syndrome and acute promyelocytic leukemia, 56 (23.3%) showed EMI at diagnosis. Patients with EMI had a higher initial WBC count and a higher proportion of M4/M5 morphological variants. The complete remission rate following induction chemotherapy was lower in patients with EMI. However, the overall survival and event-free survival did not differ between patients with and without EMI. A detailed analysis showed that patients with EMI with a WBC count at diagnosis of over 100 x 10(9)/L or infiltration into the central nervous system are likely to have a poor prognosis. CONCLUSIONS: CNS leukemia and EMI together with a WBC count of >100 x 10(9)/L at diagnosis of AML are high risk factors for relapse, and alternative treatment approaches for patients with these characteristics should be explored.

Acute Disease↗
PubMed1994

Spread of malignant lymphoid cells into rat central nervous system with intact and disrupted blood-brain barrier.

The pathways of spread of malignant lymphoid cells into the central nervous system (CNS) were studied using a T lymphoblastic leukaemia/lymphoma model of inbred PVG rats. The effects of intraperitoneal, intracarotid, intravenous, intrathecal and intracerebral routes of transplantation were analysed, and the significance of the blood-brain barrier (BBB) in preventing neoplastic cell invasion was studied by disrupting the BBB with focal cold injury. Extraneurally transplanted cells appeared first in the dura and subarachnoid space. From the latter they spread further into the perivascular space of penetrating cortical vessels. Parenchymal tumour cell foci were only seen in terminally ill rats, usually associated with damage to the vessel wall. Intrathecal transplantation did not accelerate the progression of the disease. Intracerebrally transplanted cells readily produced parenchymal infiltrates with diffuse invasion into the white matter, perivascular spreading into the cortex, and contralateral extension along the corpus callosum. Parenchymal invasion did not occur immediately after disruption of the BBB, but in the chronic phase neoplastic cells infiltrated the injured area. In conclusion, the model closely resembles human CNS leukaemia. Malignant cells appeared to enter the CNS through the deficient BBB of the subarachnoid vessels, whereas the BBB of the intracerebral vessels and perivascular glia limitans were very resistant to leukaemic cell invasion. This underlines the difference between the subarachnoid and perivascular v. intraparenchymal compartments. Preceding BBB damage may predispose to brain metastases. The parenchymal dissemination of malignant cells was similar to that in primary CNS lymphoma and it followed the same spreading pathways as the extracellular fluid.

Animals↗
PubMedDate not supplied

Ocular involvement in leukemia--a study of 288 cases.

Two hundred and eighty-eight newly diagnosed cases of leukemia (164 males and 124 females, 167 adults and 121 children, 245 acute and 43 chronic, 151 myeloid and 137 lymphoid), aged between 6 weeks and 78 years, were examined for eye changes in the oncology wards within 2 days of diagnosis before starting chemotherapy. Ocular lesions were present in 102 patients (35.4%)--retinal vascular changes in 91 (31.6%); infiltration of ocular tissues in 5 (1.7%), and neuro-ophthalmic signs in 6 (2.1%) cases. Some of the patients had more than one ocular lesion in one or both eyes. The eye changes were seen more often in adults (49.1%) than in children (16.5%), and in myeloid leukemia (41.0%) than in lymphoid leukemia (29.2%). Eye symptoms were present in 29 patients (10%) at initial diagnosis. Since ocular lesions were detected in many asymptomatic leukemia patients, eye examination should be included as a part of routine evaluation at initial diagnosis in these patients.

Adolescent↗
PubMed2003

The effects of the levonorgestrel intrauterine system (Mirena coil) on endometrial morphology.

AIMS: The Mirena coil is a levonorgestrel releasing intrauterine device that is in widespread use. This study aims to document the endometrial morphology associated with this device. METHODS: Endometrial specimens from 75 women with the Mirena coil were reviewed and the histological features detailed. RESULTS: Morphological features found in most of the endometria were decidualisation of stroma (72 of 75 cases), atrophy of endometrial glands (65 of 75 cases), a surface papillary pattern (38 of 75 cases), and a stromal inflammatory cell infiltrate (59 of 75 cases). Additional common histological features were the presence of foci of stromal myxoid change (29 of 75 cases) and stromal haemosiderin pigment (24 of 75 cases). Reactive atypia of surface glands, glandular metaplastic changes, stromal necrosis, and stromal calcifications were found in small numbers of cases. CONCLUSION: The endometrial features are characteristic and relatively constant and are in keeping with the effects of both a progestogenic compound and a mechanical device. Pathologists should be aware of these histological features because the Mirena coil is in widespread use.

Adult↗
PubMed2002

CD7+ and CD56+ myeloid/natural killer cell precursor acute leukemia treated with idarubicin and cytosine arabinoside.

We describe a 69-year-old Japanese male with acute leukemia with a CD7+ and CD56+ immunophenotype presenting with multiple lymphadenopathy. He was treated with idarubicin and cytosine arabinoside. Although the leukemia showed partial response, the patient did not achieve complete remission. He died of sepsis due to severe neutropenia after the third course of chemotherapy. His autopsy revealed blast infiltration in the lymph nodes, liver, spleen and vertebral bone marrow. Recently, CD7+ and CD56+ myeloid/natural killer precursor acute leukemia has been associated with a poor prognosis. Our case illustrates that myeloid/natural killer cell precursor acute leukemia shows some response to intensive chemotherapy for acute myeloid leukemia, but such therapy is insufficient to effect a cure. To overcome the resistance of this disease to chemotherapy, further studies should explore other treatment strategies.

Acute Disease↗
PubMed1998

Cutaneous involvement as the first manifestation in a case of T-cell prolymphocytic leukaemia.

Mature T-cell malignancies of extracutaneous origin are rare disorders. T-cell prolymphocytic leukaemia (T-PLL) is the most common form of all mature T-cell leukaemias in adults. Secondary skill involvement by T-PLL has been reported in 25% of patients. A case of T-PLL which presented with cutaneous infiltration mimicking a cellulitis-like lesion resistant to antibiotic therapy is described. The diagnosis of T-PLL was subsequently fully supported by the clinical, laboratory and cytological findings, as well as by the immunophenotypic study of the skin biopsy. The present case stresses the importance of accurate evaluation of skin lesions in the diagnosis of some haematological conditions and gives additional information about T-PLL such as a previously non-reported cytogenetic abnormality [t(6;6)] and lack of cutaneous lymphocytic-associated antigen expression.

Aged↗
PubMed2006

Bridging necrosis and reticulin bridging fibrosis induced by intrahepatic involvement of acute biphenotypic leukemia.

A 47-year-old Japanese woman was diagnosed as having acute biphenotypic leukemia with association of t(9;22)(q34;q11). Cholestatic liver dysfunction arose, and she died of cachexia and intracranial hemorrhage. Autopsy showed unusual hepatic fibrosis. In the liver, bridging infiltration, bridging necrosis and bridging fibrosis by leukemic cells were seen. It seemed that the degree of fibrosis was associated with the number of aggregates of infiltrating leukemic cells. The fibrotic foci were predominantly composed of reticulin and collagen fibers, and distortion of the lobules was observed. Immunohistochemically, dense bundles of alpha-smooth muscle actin (ASMA)-positive stromal cells, namely activated hepatic stellate cells (HSCs), were observed in the immature fibrotic foci as well as along the sinusoids densely infiltrated by leukemic cells. No cells positive for TGF-beta1 or PDGF-BB were identified. In conclusion, extensive intrahepatic involvement by neoplastic cells in adult acute biphenotypic leukemia may cause the unusual "disorganized" hepatic fibrosis.

Becaplermin↗