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PubMed2004

Occupational risk factors for mycosis fungoides: a European multicenter case-control study.

Mycosis fungoides (MF) is a rare disease with an unknown etiology. Its distribution suggests that occupational exposures may play a role. In the present study, we searched for occupational factors associated with MF. A European multicenter case-control study on seven rare cancers, including MF, was conducted from 1995 to 1997. Patients between 35 and 69 years of age diagnosed with MF (n = 134) were identified and their diagnoses were checked by a reference pathologist who classified 83 cases as definitive, 35 cases as possible, and 16 cases as not histologically verified. Of the 118 histologically verified cases, 104 were interviewed, of which 76 were definitive cases. As controls, we selected population controls and colon cancer controls to serve all seven case groups. Altogether, 833 colon cancer controls and 2071 population controls were interviewed. The response rate was 91.5% for cases (76 of the 83 definitive cases), and 66.6% for controls. A high risk of MF for men was observed in the industries of other non-metallic mineral products (Odds Ratio [OR] 5.3, 95% confidence interval [CI] = 1.7-16.2) and of wholesale trade (OR 3.6, 95% CI = 1.3-10.5). A high risk was found for female employees in the sector of pulp paper manufacture (OR 14.4, 95% CI = 2.2-95.1). The male occupations with the highest risks were glass formers, potters, and ceramics workers (OR 17.9, 95% CI = 5.4-59.4) and technical salesmen (OR 8.6, 95% CI = 2.4-30.8). For women, the occupations associated with the highest risks were government executives (OR 4.8, 95% CI = 1.0-22.6) and railway and road vehicles loaders (OR 3.9, 95% CI = 1.0-14.0). The results suggest that some occupational factors are associated with MF. Working as glass formers, pottery, and ceramics workers carried the highest risk, and these findings deserve further attention and replication. Females working in the paper and pulp industries may also be exposed to carcinogens of relevance to MF.

Adult↗
PubMed2000

Unilesional mycosis fungoides: a study of seven cases.

BACKGROUND: Unilesional mycosis fungoides (MF) is a rare variant of cutaneous T-cell lymphoma (CTCL), characterized by a solitary lesion clinically and by histopathological features indistinguishable from those of MF, and typically having a benign course. OBJECTIVE: To describe the clinicopathological features of a series of patients with unilesional MF. METHODS: The records of cases of unilesional MF identified during a 10-year period in two medical departments were reviewed. RESULTS: There were 7 patients: 6 males, 1 female; mean age at the time of diagnosis: 32 years; age range: 12-58 years; 3 were below the age of 18 years. The mean pretherapy follow-up period was 9 years (range: 2-20 years). In 5 patients, the eruption consisted of a characteristic patch or plaque of MF located on the trunk or upper extremity; in 2 it was atypical - in 1, a hypopigmented patch, and in 1, a plaque indistinguishable from MF-associated follicular mucinosis. Histopathologically all the lesions exhibited features characteristic of MF, with CD3+ lymphoid cells. In 6 cases (with available fresh frozen tissue) there was a predominance of CD3+ CD4+ cells; in 1 of 5 there was deletion of CD7, and in 3 of 5 there was an overexpression of IL-2 receptor. T-cell receptor gamma gene rearrangement was found in 1 of 4 cutaneous lesions tested; in 2 cases it was found in the blood but not in the skin. Treatment modalities included localized electron beam, excision, topical nitrogen mustard or topical steroids and sunbathing, resulting in all cases in a sustained complete clinical response. In 1 patient, however, there were 2 local recurrences and in yet another patient there was distant cutaneous spread 3.5 years after therapy. CONCLUSIONS: Unilesional MF is a rare variant of CTCL, has heterogeneous clinical manifestations and can affect any age group, including children. The histopathological and immunophenotypical features are in general indistinguishable from those observed in multilesional MF. Although it is a unifocal event, there may occasionally be cutaneous spread with the appearance of noncontiguous lesions, even a long time after therapy. Whether all cases represent minimal-stage IA MF or whether some are actually T-cell pseudolymphoma remains to be clarified.

Adolescent↗
PubMed1993

Clinical significance of serum adenosine deaminase activity in patients with mycosis fungoides.

Adenosine deaminase is one of the key enzymes in purine nucleotide degradation. This enzyme exists in most of the human tissues and the activity is high in lymphatic tissues, especially in T lymphocytes. Elevated adenosine deaminase activity in T cell leukemia has been reported, and its inhibitor, deoxycoformycin, has been developed as an antitumor agent. In some types of leukemia, serum adenosine deaminase activity increases in accordance with the severity of the disease. Although mycosis fungoides rarely involves peripheral blood, tumor cells do invade the skin. In order to evaluate the clinical significance of adenosine deaminase in mycosis fungoides, adenosine deaminase activity was measured in sera of 15 patients with mycosis fungoides at various stages. The mean enzyme activity was 23.2 IU/l, which was high with statistical significance compared with healthy controls (P < 0.001). Nine of twelve patients in the plaque stage (T2N0M0, IB) showed higher adenosine deaminase activity than did the normal population. The mean adenosine deaminase activity in sera in the patients in the plaque stage (T2N0M0, IB) was as high as 19.0 IU/l (range 13.7-21.4) with statistical significance compared with healthy control (P < 0.001). Three tumor stage patients without visceral involvement (T3N0M0, IIB) showed higher levels of adenosine deaminase activity (19.7, 21.5, 24.4 IU/l). An erythrodermic patient (T4N0M0, III) also had a high adenosine deaminase activity 28.4 IU/l. Two tumor stage patients with organ involvement (T3N0M1, IVB) exhibited extremely high adenosine deaminase activity (60.9, 32.2 IU/l). The adenosine deaminase activity in sera showed a tendency to become higher with the extension of the stages.(ABSTRACT TRUNCATED AT 250 WORDS)

Adenosine Deaminase↗
PubMed1985

Topical nitrogen mustard in early mycosis fungoides. A 12-year experience.

A 12-year experience in thirty-three patients suffering from early mycosis fungoides in plaque stage confirms the effectiveness of topical nitrogen mustard therapy. Fourteen patients were in complete remission at the latest time of observation and 7 in partial remission. The probability of freedom from relapse was approximately 50% after 6 and 12 years. Three deaths attributable to mycosis fungoides were recorded. Three patients had to discontinue treatment due to contact dermatitis to nitrogen mustard. Specific precautions were undertaken in order to protect personnel handling the drug. No damaging or toxic effects were observed among staff personnel and no hematological side-effects were observed among the patients. The treatment as a whole was well tolerated.

Administration, Topical↗
PubMed1986

Hypopigmented mycosis fungoides. Speculations about the mechanism of hypopigmentation.

Hypopigmented, nonatrophic macules are an unusual manifestation of lesions of early mycosis fungoides. Presented herein is a patient with such lesions and an unusual mask-like hypopigmentation of the face. We review both the clinical and microscopic findings in this patient and in previously reported patients with hypopigmented lesions of mycosis fungoides. Finally, speculation is made as to the mechanisms of the hypopigmentation.

Adult↗
PubMed1990

Preliminary observations on the phenotypes of lymphoid cells in mycosis fungoides using anti-human-thymocyte monoclonal antibodies.

The phenotypes of lymphoid cells from twelve patients with mycosis fungoides (including 5 patients in the tumor stage and 7 in the pretumor stage) were determined immunohistochemically using anti-thymocyte monoclonal antibodies (HIT1, HIT2, and Leu 6). The results showed significant differences between the numbers of HIT1 and HIT2 positive cells in tumor and pretumor stage lesions, indicating that these two monoclonal antibodies may be of help in the staging of mycosis fungoides and in making a prognosis. Also, we found that some Leu 6 (+) cells in tumor stage lesions were round or oval in shape without dendritic processes, suggesting that they may be immature lymphocytes.

ADP-ribosyl Cyclase↗
PubMedDate not supplied

Mycosis fungoides cells in sputum. A case report.

Atypical lymphoid cells were identified in the sputum of a patient known to have mycosis fungoides. Although the lungs are the second most common organ to be affected when there is extracutaneous dissemination of mycosis fungoides, this is the first report of pulmonary involvement diagnosed by the cytologic identification of "mycosis cells" in the sputum.

Adult↗
PubMed1981

[Alternations of dermal lymphocytes in mycosis fungoides, a study with the scanning electron microscope (author's transl)].

Isolated dermal lymphocytes from the infiltrate of mycosis fungoides show on scanning electron microscopic examination following characteristics, which differ from normal lymphocyte surface: loss of the round cell shape, increase of absolute length of microvilli, increase of lymphocytes with smooth surface and mitoses. These alterations concern all immunologic subpopulations and may indicate a stimulation of the whole dermal infiltrate in mycosis fungoides.

Cell Membrane↗
PubMed1988

Treatment of pulmonary mycosis fungoides with whole-lung radiation therapy.

Visceral involvement is a relatively common but poorly appreciated occurrence in patients with mycosis fungoides. The lungs are the most common site of visceral involvement. Here we present two cases of mycosis fungoides with pulmonary parenchymal involvement, one of them proven by biopsy. Both patients were treated with whole-lung radiation therapy, with complete disappearance of the lesions and improvement of respiratory symptoms. There was no significant radiation-induced pulmonary toxicity. One patient continues to be alive with no recurrence in the lungs 17 months after radiation therapy.

Adult↗
PubMed1994

Prognosis with newly diagnosed mycosis fungoides after total skin electron radiation of 30 or 35 GY.

PURPOSE: To determine the prognosis of new patients with T1-4N0-1B0M0 mycosis fungoides treated with total skin electron beam radiation. METHODS AND MATERIALS: 25 consecutive patients received 30 Gy with 3 or 4 MeV electrons in 1977-1980; 121 received 35 Gy with 4 MeV in 1980-1992. Response rates, relapse-free survival, and overall and cause-specific survivals were assessed by explicit criteria. The relationships of T, N, gender, age, and radiation technique to prognosis were investigated by regression statistics. RESULTS: The average age was 55 years and the male:female ratio was 1:4. Forty-four percent were T1N0 and 34% were T2N0. The overall complete response rate was 82%, and lower T status, more radiation, and female gender were independently and positively associated with response. Median follow-up was 5.2 years. T1 patients who entered remission had a higher relapse-free survival compared to T2 through T4 patients. Thirty-four percent of T1 patients remained relapse-free at 6 years, compared to fewer than 20% of T2-4 patients. For all 146 patients the median overall survival was not reached at 15 years. Only 8 of 29 deaths were related to mycosis fungoides and these were significantly associated with higher T. The 54 T1N0 patients who had 35 Gy had a 10-year mycosis fungoides-specific survival of 100%. CONCLUSION: Total skin electron beam radiation gives good results with T1N0B0M0 disease. T3-4 disease is less likely to respond, it relapses more quickly, and it implies a poorer survival, but radiation offers palliation. T2 responds like T1, but relapses like T3-4. T2 also implies an intermediate survival. These results have implications for staging, informed consent, optimizing radiation treatment, and clinical trials.

Female↗
PubMed1977

Cytophotometric studies on mycosis fungoides and other cutaneous reticuloses.

Feulgen-DNA cytophotometry was carried out in skin imprint preparations of patients with mycosis fungoides, reticulum-cell sarcoma, lymphomatoid papulosis and Sézary's syndrome. These patients proved to have aneuploid and polyploid DNA histograms. In tumour stage of mycosis fungoides a more bimodal distribution of the DNA values was found. Of 79 cases of suspected malignant reticulosis of the skin, 32 showed an abnormal DNA histogram with 5 percent or more tetraploid and hypertetraploid cells. Thirty of these patients developed a malignant skin reticulosis during the 4-year follow-up period. It is concluded that DNA cytophotometry has value as a supplement to routine morphological histopathology in malignant cutaneous reticulosis.

DNA, Neoplasm↗
PubMed1978

Selective immunological evaluation of mycosis fungoides.

Tests of specific aspects of immune function were conducted on 15 patients with clinically and histologically evident mycosis fungoides of various stages. These tests included peripheral blood lymphocyte counts, T and B cell counts, mitogen stimulation tests to phytohemagglutinin (PHA), concanavalin A (con A), and pokeweed mitogen, as well as determinations of IgE levels and antinuclear factor (ANF). We found that mycosis fungoides patients, as a group, had normal peripheral blood lymphocyte, T cell, and B cell counts, but had decreased stimulation to PHA and con A. When evaluated by stage of disease, the peripheral blood lymphocytes, T and B cell counts did not differ significantly from normal, at least until stage V. Concanavalin A stimulation was decreased by stage II disease, and PHA was decreased by stage III, indicating at least a selective immunological deficit in the plaque stage or in later disease. Values of IgE and ANF were rarely abnormal.

Adult↗
PubMed1981

Acquired factor VIII inhibitor in a patient with mycosis fungoides.

Acquired factor VIII inhibitors have been noted in patients with hemophilia A (factor VIII deficiency), in nonhemophilic individuals with various collagen-vascular diseases, in certain normal women following parturition, and occasionally in elderly individuals with no underlying diseases. This study describes the first reported instance of a factor VIII inhibitor in a patient with mycosis fungoides who had bleeding manifested by gross hematuria. Treatment with corticosteroids and cryoprecipitate was followed by cessation of hematuria within two weeks. The patient had one episode of shoulder pain presumably related to hemarthrosis. Immunosuppressive therapy with cyclophosphamide was instituted in an attempt to decrease antibody production and control skin involvement of mycosis fungoides. Factor VIII inhibitor level rose to 100 Bethesda units without further serious bleeding. There was no peripheral blood evidence of Sézary syndrome. It is possible that some patients with cutaneous T-cell lymphomas, such as mycosis fungoides and Sézary syndrome, may have an increase in helper T-cells which may lead to excessive B-cell activity and overproduction of antibodies.

Antibody Formation↗
PubMed1982

Oral retinoid in combination with bleomycin, cyclophosphamide, prednisone and transfer factor in mycosis fungoides.

Oral retinoids seem to have been of great benefit in a non-randomized study on advanced mycosis fungoides using two different chemotherapy regimens, one with retinoid, the other without. Both groups also received a 3-drug chemotherapy with bleomycin, cyclophosphamide and prednisone. Complete remission including all signs of lymph-node involvement was found in 8 of 10 patients of the retinoid treated group, while none went into complete remission in the control group. All in the control group died between 3 and 12 months after therapy, whereas all but one in the retinoid treated group are alive. Other treatment differences between the groups were related to the use of transfer factor, topical treatment, and steroid administration. These differences make a final evaluation of the use of retinoids in mycosis fungoides difficult at the present stage. Further studies are needed.

Administration, Oral↗
PubMed1990

Skin exudate levels of interleukin 6, interleukin 1 and other cytokines in mycosis fungoides.

The role of locally released cytokines in inducing lymphocyte activation and infiltration in the skin lesions of mycosis fungoides has been investigated. The levels of selected cytokines were measured in chamber fluid samples from lesional and control skin. Biologically active interleukin 6 was significantly elevated in lesional samples and a recombinant form of this cytokine was shown to induce lymphocyte migration in an in vitro assay. Biologically active interleukin 1 was detected in all control chamber fluid samples. Significantly reduced levels of this cytokine were present in lesional samples, which may be the result of the release of preformed material. Interleukin 2 and tumour necrosis factor activity, and gamma interferon and granulocyte macrophage colony-stimulating factor immunoreactivity, were not detectable in any of the samples. Interleukins 1 and 6 may play a role in the pathogenesis of the lesional lymphocyte infiltrates in mycosis fungoides.

Adult↗
PubMed1984

Mycosis fungoides with monoclonal gammopathy, hypereosinophilia, and hyper-IgE.

A 61-year-old female with a well-established Mycosis fungoides also had a marked hypereosinophilia, a monoclonal gammopathy IgGk type, and an elevated IgE level. Such unusual association of abnormalities of the immune system in a given Mycosis fungoides patient has not been described before in the absence of any other underlying cause.

Eosinophils↗
PubMed2000

Interleukin 4 and interferon-gamma expression of the dermal infiltrate in patients with erythroderma and mycosis fungoides. An immuno-histochemical study.

BACKGROUND: Erythroderma, or generalized erythema of the skin, may result from different causes. At present it is unclear whether the underlying patho-mechanisms that lead to erythroderma are identical or different depending on the original disease. The aim of this study was to investigate the dermal cytokine profile in different types of erythroderma and mycosis fungoides. METHODS: Snap-frozen skin biopsy specimens from 33 patients with erythroderma were studied. Thirteen had idiopathic erythroderma, 7 erythrodermic atopic dermatitis, 5 Sézary syndrome and 8 had erythroderma from miscellaneous causes. We also studied 6 patients with mycosis fungoides (5 plaques and 1 tumor) and 5 healthy non-atopic volunteers. The biopsies were immunohistochemically stained for interleukin 4 (IL-4) and interferon gamma (IFN-gamma). All positive cells for IL-4 and IFN-gamma in the dermis were counted and the number of positive cells was calculated per mm2. IL-4/IFN-gamma ratio was calculated for each biopsy. RESULTS: The patients with idiopathic erythroderma, atopic dermatitis and miscellaneous erythroderma, all showed more IFN-gamma-positive cells than IL-4-positive cells in the dermis. The median IL-4/ IFN-gamma ratio for these three groups was 0.6, 0.9 and 0.45, respectively. These differences were not statistically significant. All patients with Sézary syndrome however, showed more IL-4-positive cells than IFN-gamma-positive cells. The median IL-4/IFN-gamma ratio was 1.8, which is significantly higher than in the other groups p<0.05). In mycosis fungoides roughly the same number of cells expressed IL-4 and IFN-gamma. The median IL-4/IFN-gamma ratio was 1.0, which is significantly lower than in Sézary syndrome (p<0.05). CONCLUSIONS: The dermal infiltrate in patients with Sezary syndrome mainly shows a T-helper 2 (Th2) cytokine profile, this in contrast to T-helper 1 (Th1) cytokine profile in benign reactive erythroderma. This indicates that although a relative uniform clinical picture of erythroderma is obvious, a different patho-mechanisms may be underlying.

Antibodies, Monoclonal↗