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PubMed1995

[Mycosis fungoides bullosa].

We report on a male patient with mycosis fungoides (MF) with blisters mainly occurring on clinically uninvolved skin. This rare association has to be differentiated from other bullous diseases of the skin. The clinical picture has been known since the end of the last century and has been described as mycosis fungoides bullosa. The pathomechanism, however, has not yet been exactly identified. The different clinical and histological features of the disease are discussed.

Aged↗
PubMed2002

Transformation in mycosis fungoides: the role of methotrexate.

BACKGROUND: Large cell transformation in patients with mycosis fungoides (MF) has been well reported in the literature. Although the risk factors have not been clearly elucidated, advanced stage seems to be associated with a higher incidence of transformation. Because MF is a rare disorder, little is known about the influence of other factors such as immunosuppressive therapy in the occurrence of transformation. OBJECTIVE: We questioned the role of methotrexate (MTX) in the transformation of MF to large cell lymphoma (LCL). METHODS: We identified all patients with MF who were registered in our cutaneous lymphoma database. Transformation was defined by the presence of large cells exceeding 25% of the infiltrate in at least one skin biopsy. In one patient, we followed the histologic, immunophenotypic, and genotypic changes taking place as transformation occurred. RESULTS: A total of 134 patients with MF were identified. Of 21 patients who received MTX, 3 transformed, and of the 113 patients in the non-MTX group, only 2 transformed. The incidence of transformation in the patients who received MTX was significantly higher than in those who did not receive the drug (14.3% vs. 1.8%; p = 0.03). This significance was maintained, even after controlling for stage and sex. For one patient who transformed, we demonstrated an identical dominant T-cell clone in all skin specimens, including the large cell lymphoma. CONCLUSION: Our results demonstrate a significant association between MTX exposure and transformation to LCL in patients with MF. In light of the small sample size, short followup of patients, and the inherent tendency of mycosis fungoides to transform, the role for MTX in transformation is unproven and needs to be confirmed in a multicenter study.

Adult↗
PubMed1985

Chromosome changes in mycosis fungoides in an XYY male.

A 32-year-old white male was diagnosed as having mycosis fungoides in 1976; bone marrow biopsy and aspiration in August 1984 revealed infiltration with neoplastic cells. Cytogenetic analysis of the cells from the bone marrow specimen showed that 48 of 50 metaphases contained an extra Y chromosome (i.e., 47,XYY). The remaining two cells were hypotetraploid and hyperpentaploid, respectively, with a common marker derived from chromosome #2. The metaphases obtained by PHA stimulation of peripheral blood cells showed a 47,XYY pattern. An interleukin 2 (IL-2) dependent T-cell line was established from the patient's blood mononuclear cells; all metaphases of this line had an extra Y chromosome. Thus, this case is one of a mycosis fungoides developing in an XYY male.

Adult↗
PubMed1990

Mycosis fungoides in children and adolescents.

The clinical and histologic findings in five young patients with mycosis fungoides are reviewed. The skin specimen from a 16-year-old boy had an infiltrate predominantly Leu-1+, Leu-4+, Leu-3a + 3b+, OKT6+, Leu-2a-, Leu-8-, and Leu-9-, and rearrangements were noted in T cell receptor gene (beta chain [constant region probe] and gamma chain [J region probe]); no rearrangements were found in a histologically normal lymph node or peripheral blood. The skin specimen from an 11-year-old girl contained predominantly Leu-4+ and Leu-9+ lymphoid cells. No T cell receptor gene rearrangements were found in the skin or in an involved lymph node. The variations in clinical, histologic, and immunopathologic features suggest that mycosis fungoides in young patients represents a heterogeneous subgroup of cutaneous T cell lymphoma.

Adolescent↗
PubMed1995

CD8-positive tumor-infiltrating lymphocytes influence the long-term survival of patients with mycosis fungoides.

BACKGROUND: Nonneoplastic mononuclear cells commonly infiltrate lesions of mycosis fungoides. OBJECTIVE: We sought to determine the immunophenotypic characteristics of these cells and to determine whether the presence of CD8+ tumor-infiltrating lymphocytes has an impact on prognosis. METHODS: Skin biopsy specimens from 78 patients were stained with immunopleroxidase techniques to determine their phenotypic characteristics. The proportion of CD8+ tumor-infiltrating lymphocytes was quantified and compared with stage of disease and survival rate. RESULTS: Patients with more limited T-stage disease tended to have a higher proportion of CD8+ cells in their skin biopsy specimens, compared with patients with more advanced T-stage disease. Within each T-stage patients with a larger proportion of CD8+ cells had a better survival rate than those with fewer CD8+ cells (p < 0.05 for T1 and T3). A multivariate analysis confirmed the importance of T stage (p = 0.0006), overall stage (p = 0.0112), and CD8 positivity (p = 0.0335) in this cohort of patients. CONCLUSION: CD8+ tumor-infiltrating lymphocytes in mycosis fungoides correlate with improved survival rate and may exert an antitumor effect rather than being mere bystander cells.

Actuarial Analysis↗
PubMed1995

[Association of mycosis fungoides and Jaccoud syndrome].

We report here a case of a 49 year-old woman who developed a unilateral Jaccoud's arthropathy 9 years after a mycosis fungoides of the left hand. This rheumatic affection, described in 1866, is a deformation of the hands or of the feet linked to a periarticular disease without any bone lesion. Many causes have been registered but the most frequent etiology is the systemic lupus erythematosus. The mycosis fungoides had never been reported as yet to be responsible to the Jaccoud's arthropathy.

Female↗
PubMed1983

Palliative radiation treatment of cutaneous mycosis fungoides--a dose response.

Between 1966 and 1981, 20 patients (191 lesions) underwent palliative radiation therapy for control of biopsy-proven cutaneous mycosis fungoides. Six patients (47 lesions) and an additional 34 lesions from the remaining 14 patients with complete response to treatment were excluded from the study because of follow-up of less than one year. Included in the remaining 110 lesions were all recurrences and all partial responses. The modalities for treatment included superficial X rays, Cobalt-60 or electron beam irradiation. The total tumor doses employed ranged from 600-4000 cGy. The 110 lesions (14 patients) were retrospectively analyzed to determine the dose required for local control of the lesions. Fifty-three percent of the lesions were classified as plaques, 20% as tumors less than or equal to 3 cm in diameter, and 27% as tumors greater than 3 cm in diameter. Complete response to treatment was observed in 95% of the plaque lesions, 95% of the tumors less than or equal to 3 cm in diameter and 93% of tumor greater than 3 cm in diameter. A complete response to treatment was noted in all lesions receiving greater than 2000 cGy. In the total population of lesions having a complete response, a local infield recurrence rate of 42% was noted in the group receiving less than or equal to 1000 cGy, 32% in those receiving 1001-2000 cGy, 21% in those receiving 2001-3000 cGy, and 0% in the group receiving greater than 3000 cGy. No infield recurrence was seen when the treated lesion received a total tumor dose greater than or equal to TDF of 49. Of those lesions which recurred, the mean time to recurrence for the first three dose ranges above were 5 months, 10 months and 16 months respectively. Eighty-three percent of the 30 recurrences were seen within one year of treatment; 100% of the recurrences occurred within two years of treatment. The data from this study indicate that tumor doses equivalent to at least 3000 cGy at 200 cGy per fraction, five fractions per week (TDF greater than or equal to 49) are needed for adequate local control of cutaneous mycosis fungoides lesions.

Cobalt Radioisotopes↗
PubMed1987

The indications for total cutaneous electron beam radiation therapy of mycosis fungoides.

From 1977 to 1984, we treated 34 patients with mycosis fungoides and 9 patients with B cutaneous lymphomas. Eighteen patients with mycosis fungoides were treated with total skin electron irradiation (TSEI) and had a minimum follow-up of 15 months (range 15 months to 7 years). The lowest electron energy of the linear accelerator was 8 MeV therefore we placed a plexiglas screen between the patient and the machine; the resulting electron energy was 4 MeV. The total dose was 30 Gy delivered in 12 fractions over 40 days. There were 8 males and 10 females. The median age was 48 years (ranging from 13 to 78 years). All patients were staged as follows: Stage A = superficial lesions covering less than 50% of the body surface; Stage B = superficial lesions covering more than 50% of the body surface; Stage C = tumors involving the skin, lymph nodes and/or visceral organs. Five patients with Stage A (5/5) and 5 patients with Stage B (5/5) had a complete remission, 1 stage A patient relapsed 6 months after completion of treatment. All the Stage B patients recurred between 3 and 15 months. The recurrences were localized to the skin and were well controlled with topical nitrogen mustard or puvatherapy. Among the Stage C patients, 3 did not respond to treatment and died of their disease; the remaining 5 patients achieved complete remission but they all relapsed from 2 to 9 months following completion of treatment. The median follow-up was 32 months and the average time for relapse was 6.5 months. All relapses except one (15 months) occurred within the first year. We feel that total skin electron irradiation is indicated in Stage A and B patients. However, we feel Stage C patients should receive TSEI for palliative purposes only.

Adolescent↗
PubMed1993

Total scalp treatment of mycosis fungoides: the 4 x 4 technique.

PURPOSE: A technique for treating mycosis fungoides confined to the scalp using a method known as the 4 x 4 technique is presented. METHODS AND MATERIALS: Uniform dose distribution on the scalp and acceptable "hot spots" along five match lines is accomplished by using four sets of four fields (i.e., 4 x 4) on the patient. Precise and reproducible patient and field alignment was accomplished with a solid thermoplastic mask, which is the surface on which match lines are drawn. In-vivo dosimetry (thermoluminescent dosimeters and film) are easily attached to the mask which also provides a portion of the 7 mm bolus used to shift the characteristic 6 MeV electron depth dose toward the skin surface. RESULTS: In-vivo dosimetry demonstrated that single fraction match line doses are within 25% to 30% of central axis dose. Shifting these match lines to four locations reduces these "hot spots" to satisfactory levels (less than 120%). Three patients have been treated with this technique and each patient continues to have a complete clinical response at 14 to 21 months post treatment. In addition, each patient has excellent cosmetic results with no evidence of acute or chronic side effects at the match lines. CONCLUSION: The 4 x 4 technique has proven to be useful in the treatment of mycosis fungoides confined to the scalp.

Adult↗
PubMed1995

Multiple courses of high-dose total skin electron beam therapy in the management of mycosis fungoides.

PURPOSE: A retrospective analysis was undertaken to determine the indications for, the efficacy of, and the long-term complications of two courses of total skin electron beam therapy for mycosis fungoides. METHODS AND MATERIALS: A retrospective analysis of 15 patients with the pathologic diagnosis of mycosis fungoides treated in the Department of Radiation Oncology at Stanford University Medical Center between 1968 and 1990 was performed. All patients received two courses of high-dose electron beam therapy to the skin. The mean dose for the total skin treatment for the first course was 32.6 Gy and 23.4 Gy for the second course of treatment. RESULTS: Following the first course of total skin electron beam therapy, 11 of 15 had a complete response, with a mean duration of 11.6 months. All patients received adjuvant therapies between the first and second courses of high-dose total skin electron beam therapy. The mean interval between the first and the second courses of therapy was 41.3 months. Patients were restaged prior to commencement of their second course of high-dose total skin electron beam therapy, resulting in upstaging in six. The second course of therapy resulted in six complete responses and nine partial responses. Twelve of these patients have since died, 1 is lost to follow-up, and 2 are living with disease. The long-term side effects in the two living patients include pigmentation changes, alopecia, and diffuse xerosis. CONCLUSION: Delivery of two courses of total skin electron beam therapy is technically feasible, tolerable, and efficacious. The dose to the total skin was reduced for the second course of therapy in all cases. The criteria used to screen patients included initial good response to total skin electron treatment, long disease-free interval, exhaustion of other therapeutic modalities, and generalized skin involvement at relapse. Long-term toxicities were mild in severity and generally consisted of generalized xerosis, scattered telangiectasias, pigmentation changes, and partial alopecia.

Adult↗
PubMed2006

Minimal residual disease in hypopigmented mycosis fungoides.

We describe the case of a 13-year-old boy with stage I hypopigmented mycosis fungoides in whom minimal residual disease was detected with T-cell receptor gamma-polymerase chain reaction after the disease was in complete clinical remission. We further cloned and sequenced the T-cell receptor gamma-polymerase chain reaction product of the lesion in remission and found that the original T-cell clone still existed in decreased amounts. The patient was followed up for 3 1/2 years without any new lesions developing. The clinical significance of this residual malignant T-cell clone in mycosis fungoides remains to be elucidated.

Adolescent↗
PubMed2006

Clinicopathologic features and T-cell receptor gene rearrangement findings of mycosis fungoides palmaris et plantaris.

BACKGROUND: Mycosis fungoides palmaris et plantaris (MFPP), characterized by hyperkeratotic patches or plaques confined to the palms and soles, is rare and easy to misdiagnose because of the clinical similarity to psoriasis, cutaneous inflammatory dermatoses, and dermatophytic infections. The literature about MFPP mostly consists of case reports with short-term follow-up. OBJECTIVE: Our purpose was to evaluate the clinicopathologic features, T-cell receptor (TCR) gene rearrangement findings, and prognosis of MFPP. PATIENTS AND METHODS: This retrospective study has been reviewed in the clinicopathologic, TCR gamma gene rearrangement findings and follow-up study of 12 patients with MFPP. RESULTS: The duration of diseases ranged from 9 months to 25 years with a mean duration of 5.3 years. Clinically, hyperkeratotic patches and plaques were observed in all cases, with 6 cases having developed on the palms and soles and 6 cases on the palms only. In TNM classifications, all cases were confined to T1N0M0 (stage IA) showing an early stage of mycosis fungoides (MF). Histopathologic findings revealed marked hyperkeratosis, parakeratosis with plasma, epidermotropism, convoluted lymphocytes, haloed lymphocytes, dense infiltrate of lymphocytes in all 12 cases (100%), Pautrier's microabscess in 9 cases (75%), a wiry bundle of collagen in 11 cases (91.7%) and basilar epidermotropism in 3 cases (25%). TCR gamma gene rearrangement was performed except for one case and monoclonality was detected in 10 of 11 cases. In the comparison group with cutaneous inflammatory dermatoses, all cases showed polyclonality. Treatment was done with Re-PUVA (acitretin and PUVA), ultraviolet A1, as well as systemic acitretin and methotrexate. Most patients showed a good response. In the follow-up study of 9 cases for a mean period of 47.6 months, only one patient's skin lesions were extended to the trunk and face, but the other patients had no sign of extracutaneous involvement. LIMITATIONS: These results were obtained from patients with MFPP in Korea. A cooperative study with other ethnic groups will be helpful. CONCLUSIONS: If a patient has recalcitrant palmoplantar dermatosis, MFPP should be suspected and histopathologic studies with TCR gene rearrangement should be done for early diagnosis of MFPP.

Adolescent↗
PubMed2006

A phase II open-label study of recombinant human interleukin-12 in patients with stage IA, IB, or IIA mycosis fungoides.

BACKGROUND: Interleukin-12 (IL-12) increases Th(1) cytokines, natural killer (NK) cells, and cytotoxic T-cell activities. Progression of mycosis fungoides is associated with Th(2) cytokines produced by a clonal proliferation of epidermotropic T-helper cells. OBJECTIVE: To determine the safety and efficacy of subcutaneous recombinant human IL-12 (rhIL-12) in early mycosis fungoides (MF; stage IA-IIA) in a multi-center, open label clinical trial. METHODS: rhIL-12 was administered biweekly (100 ng/kg for 2 weeks; 300 ng/kg thereafter). A modified severity-weighted assessment tool (SWAT) and the longest diameter of 5 index lesions measured efficacy. RESULTS: Twenty-three MF patients (stage IA, 12 patients; IB, 9; and IIA, 2) had previously received >3 therapies. Ten of 23 patients (43%) achieved partial responses (PR); 7 (30%) achieved minor responses; and 5 (22%) had stable disease. The duration of PRs ranged from 3 to more than 45 weeks. Twelve (52%) ultimately progressed with mean time to progressive disease of 57 days (range, 28-805). Ten completed 6 months of therapy; 1 completed 24 months. Of patients not completing 6 months of therapy, 6 progressed and 6 others discontinued because of adverse events or withdrew consent. Seventeen patients had treatment-related adverse events that were generally mild or moderate in severity, including asthenia, headache, chills, fever, injection site reaction, pain, myalgia, arthralgia, elevated aspartate and alanine aminotransferase levels, anorexia, and sweating. One patient in PR died of hemolytic anemia, possibly exacerbated by rhIL-12 treatment. LIMITATIONS: The original company was purchased during the conduct of the trial and rhIL-12 is currently unavailable. The quality of life data were not available for inclusion. CONCLUSION: Twice-weekly subcutaneously administered rhIL-12 (100 ng/kg escalated to 300 ng/kg) showed antitumor activity with a response rate of 43% in refractory patients. It was relatively well-tolerated in early-stage MF.

Adjuvants, Immunologic↗
PubMed1980

Photochemotherapy in mycosis fungoides. A study of 73 patients.

Seventy-three patients with mycosis fungoides were treated with photochemotherapy and followed up for 2 to 39 months. Photochemotherapy produced clinical and histological clearing in a very high proportion of patients with stage 0 to II disease. The response in patients with tumours was less satisfactory, and a high proportion of these patients required local radiotherapy to resistant lesions. Maintenance treatment with photochemotherapy may keep many patients with stage 0 to II disease in remission for up to 3 years, but patients with advanced disease may relapse despite maintenance treatment. Patients with all stages of mycosis fungoides may relapse when maintenance treatment is withdrawn.

Adolescent↗
PubMed1985

Conjunctival involvement with mycosis fungoides in a patient receiving PUVA therapy.

A 40-year-old woman had documented mycosis fungoides of the skin. Over a period of five years, she had undergone intermittent treatment with psoralen long-wave ultraviolet light (PUVA) therapy. Despite regression of the skin lesions, tumor plaques developed in her eyelid and conjunctiva within the area shielded by dark glasses. Light and electron microscopic examination of a biopsy from the conjunctival plaque confirmed that the lesion was a deposit of mycosis fungoides. Local radiotherapy resulted in complete regression of these ocular lesions. The conjunctiva may well be an iatrogenic "sanctuary site" when this disease is treated with PUVA therapy.

Adult↗
PubMed2000

Radiotherapy of advanced mycosis fungoides: indications and results of total skin electron beam and photon beam irradiation.

BACKGROUND: The goals of this retrospective study of advanced mycosis fungoides are (1) to describe the indications of a combination of total skin electron beam and photon beam irradiation and (2) to analyze the results of total body or segmental photon irradiation for patients with extension beyond the skin. METHODS: From January 1975 to December 1995, 45 patients with pathologically-confirmed mycosis fungoides or Sézary syndrome received a combination of TSEB and photon beam irradiation for advanced disease: 34 males and 11 females, mean age 61 years (range 27-87 years). The mean follow-up was 111 months (range 18-244 months, median 85 months). Whole-skin irradiation treatment to a depth of 3-5 mm with a 6-MeV electron beam was produced by a linear accelerator to a total dose of 24-30 Gy in 8-15 fractions, 3-4 times a week. In cases of thick plaques or tumors that were beyond the scope of low energy electron beams or for treating nodal areas (especially in the head and neck area or axilla involvement), regional irradiation (RRT) with Co-60 photon beams was followed by whole-skin electron beam irradiation (15 patients). In cases of diffuse erythrodermia, Sézary syndrome, nodal or visceral involvement, total body irradiation was delivered with a 25-MV photon beam using a split-course regimen to prevent hematological toxicity (22 patients). The first course consisted of 1.25 Gy delivered in ten fractions and 10 days. Subsequently, patients received TSEB. Four to 6 weeks after TSEB, they received a second course of 1.25 Gy. The cumulative TBI dose ranged from 2.5 to 3 Gy in about 3 months. Hemi-body irradiation (HB) with Co-60 (and a bolus) was given in cases of multiple regional tumors with large and thick infiltration of the skin to a dose of 9-12 Gy (using fractions of 1-1.5 Gy/day) which, once flattened, were boosted with whole-skin electron beam therapy (8 patients). RESULTS: At 3 months, the overall response rate was 75% with 23/45 (51%) patients in complete response and 24% in partial response; one patient had stable lesions and 1 patient presented progressive disease. The overall response rate was 81% for T3 patients, 61% for T4, 79% for N1 and 70% for N3. The complete response rate was 67% for T3 and 28% for T4. Sixty-four percent of N1 patients and 41% of N3 had a complete response. The 5-year actuarial overall survival was 37% for T3 and 44% for T4 (P = 0.84). Patients with clinically abnormal lymph nodes that were pathologically negative (N1) presented a 5-year survival of 63%. Patients with pathologically positive lymph nodes (N3) experienced a 5-year survival rate of 32% (P = 0.040). CONCLUSIONS: TSEB provides an excellent quality of life by reducing itching and discharge from the skin. Patients with more advanced disease may be treated and cured by the addition of photon beams in combination with TSEB. A selection of patients with advanced skin disease and regional extension may be cured by a combination of TSEB and photon beam irradiation. The regional treatment allows the use of electrons after the reduction of the plaques or thick tumors and a prophylactic irradiation of the adjacent nodal area.

Adult↗