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PubMed2002

Mycosis fungoides presenting with extensive pyoderma gangrenosum-like ulcers.

Mycosis fungoides (MF) may present with atypical clinical manifestations. Usually it mimics various chronic dermatoses, with the appearance of ulcers during the tumour stage. Infrequently, cutaneous ulcers are the main or initial sign of lymphoma. We report the case of a man who presented multiple skin lesions that clinically appeared to be pyoderma gangrenosum (PG). However, histological and immunohistochemical examination revealed MF. This case illustrates that PG-like ulcers maybe atypical cutaneous manifestations of MF and exceptionally the presenting sign of this disease.

Biopsy, Needle↗

PubMed1982

Home ultraviolet phototherapy of early mycosis fungoides: preliminary observations.

Thirty-one patients with early mycosis fungoides (MF) and three patients with parapsoriasis en plaques were treated with ultraviolet phototherapy (280 to 350 nm) at home using a commercially available light source containing four Westinghouse FS40 lamps. A complete clinical and histologic remission of disease, lasting for a median duration in excess of 18 months, was achieved in nineteen patients (61%) with MF. Although higher complete response rates generally are achieved with other therapeutic modalities, ultraviolet phototherapy with its minimal adverse effects may be indicated for selected patients. Controlled studies are encouraged to evaluate the full potential of conventional phototherapy in the management of MF.

Adult↗

PubMed1975

The treatment of mycosis fungoides and related conditions with particular emphasis on electron therapy.

The difficulties created by the lack of generally accepted views on classification, diagnostic criteria and natural history of the cutaneous lymphomata are discussed. Only the lymphomata which remain confined to the skin throughout the major part of their development are considered. It is emphasized that these are extremely radiosensitive and that there must be some factors as yet unknown which play a part in their unique natural history and response to treatment. The cutaneous lymphomata are considered under three headings: (1) lymphocytoma, (2) lymphosarcoma and reticulum cell sarcoma, (3) mycosis fungoides. The characteristics of the first two are discussed briefly and illustrative examples are given. The relatively benign nature of the lymphocytoma is stressed. It is suggested that the solitary lymphosarcoma of the skin should be treated by radical radiotherapy since long-term survival or cure may be achieved. The treatment of mycosis fungoides is considered in greater detail. The physical advantages of electron therapy are described and details given of whole body surface treatment with electrons produced by a 6 MeV linear accelerator and slowed down with carbon decelerators of appropriate thickness. The results in 73 patients treated by electrons are analysed. The early and late side-effects are mentioned and the necessity for investigating other forms of treatment is stressed. It is concluded that at the present time electron therapy is the most satisfactory form of treatment for mycosis fungoides.

Adult↗

PubMed1991

Mycosis fungoides: cutaneous T-cell lymphoma.

Most patients with mycosis fungoides are between 40 and 60 years of age. The disease has three clinical stages: (1) the premycotic, or patch, stage, consisting of macular, scaling, faint pink to red pruritic patches, usually on unexposed surfaces; (2) the mycotic, or plaque, stage, consisting of reddish, purple-brown plaques, often annular in shape and symmetric in distribution, and (3) the tumor stage, consisting of red-brown to violaceous, dome-shaped, firm tumors with a predilection for the face and body folds. The Sézary syndrome is a leukemic variant. Treatment depends on the extent of disease and includes topical or systemic chemotherapy, radiotherapy and psoralen plus long-wave ultraviolet light therapy.

Adult↗

PubMed2003

Detection of telomerase activity in patients with mycosis fungoides.

OBJECTIVES: To detect telomerase activity in patients with mycosis fungoides (MF) and to study the role of telomerase in the tumorigenesis of MF. METHODS: The technique of PCR-ELISA was employed to detect telomerase activity in 35 patients with various stages of MF. RESULTS: 92.3% tumor stage of MF, 78.6% plaque stage of MF and 75.0% patch stage of MF had positive telomerase activity. The control samples had no telomerase activity. Telomerase activity in tumor stage of MF was significantly higher than that in plaque stage, while the latter was higher than that in patch stage. Telomerase activity was correlated with the stage of MF. CONCLUSION: High level of telomerase activity frequently occurred in patients with MF, suggesting that telomerase might play an important role in the tumorigenesis of MF and is a useful marker for the diagnosis of MF possibly.

Humans↗

PubMed1992

Establishment of two continuous T-cell strains from a single plaque of a patient with mycosis fungoides.

From a plaque biopsy of a patient with mycosis fungoides, two different continuous cell lines were established by including both IL-2 and IL-4 in the culture medium. Both continuous cell lines appeared with characteristic chromosome markers after approximately 40 cell population doublings. The initial karyotype recognized in T cells from the skin biopsy was 46,XY and the karyotypes of the continuous cell strains were 46,XY, -18, + i(18q) and another with multiple chromosome aberrations as described in Sezary T-cell leukemia. Phenotyping with monoclonal antibodies and T-cell receptor analysis indicates that the latter cell strain represents a minority of T-cells in the plaque. Due to its many chromosomal aberrations it probably represents the malignant cell, which may be a central cell in the immune stimulation taking place in the skin.

Aged↗

PubMed1980

Immunologic evaluation in mycosis fungoides. Before and after PUVA therapy.

Eight patients with mycosis fungoides (seven in plaque stage and one with tumors) were successfully treated with methoxalen-ultraviolet-A (PUVA) photochemotherapy. A selective immunity evaluation had been carried out prior to starting treatment and was repeated sevedn to 33 months (mean, 22 months) later. The results of pre-PUVA and post-PUVA therapy evaluation were compared. The delayed hypersensitivity responses improved in all eight patients, and four patients who were anergic before treatment manifested a normal response after PUVA therapy. The T lymphocyte counts were reduced in four patients before PUVA therapy (mean +/- SD, 66.72% +/- 9.78%) and in seven patients after PUVA (mean +/- SD, 54.43% +/- 10.86%; P = .03). The results of mitogen stimulation studies with phytohemagglutinin, pokeweed mitogen, and concanavalin A were equivocal. The levels of serum immunoglobulins were not affected. One patient had a positive antinuclear factor test of low titer.

Adult↗

PubMed2000

Exposure at home to airborne concentrations of nitrogen mustard during topical application for the treatment of mycosis fungoides: a case study.

BACKGROUND: Although effective at treating mycosis fungoides (MF), nitrogen mustard (HN(2)) creates potential exposure risks to those who administer it, including health care workers and family members. OBJECTIVE: The main objective was to examine the potential for nontarget individuals to be exposed to HN(2) vapors during and shortly after treatment with HN(2) in a home environment. METHODS: Air concentrations of HN(2) were measured during the topical treatment of MF in a patients home. RESULTS: The results demonstrate that eye and mucous membrane irritation may occur at HN(2) levels commonly encountered during the treatment of MF in the home, hospital or health clinic. Because no exposure thresholds have been developed for HN(2), the exposure thresholds of a surrogate chemical (sulfur mustard) were used. CONCLUSIONS: The study findings show that eye and mucous membrane irritation may occur at HN(2) levels below the exposure thresholds of the surrogate chemical. Recommendations for controlling exposures to HN(2) in the home are given.

Administration, Topical↗

PubMed2006

Mycosis fungoides: is it a Borrelia burgdorferi-associated disease?

Mycosis fungoides (MF) is the most frequently found cutaneous T-cell lymphoma with an unknown aetiology. Several aetiopathogenetic mechanisms have been postulated, including persistent viral or bacterial infections. We looked for evidence of Borrelia burgdorferi (Bb), the aetiologic agent of Lyme disease (LD), in a case study of MF patients from Northeastern Italy, an area with endemic LD. Polymerase chain reaction for the flagellin gene of Bb was used to study formalin-fixed paraffin-embedded lesional skin biopsies from 83 patients with MF and 83 sex- and age-matched healthy controls with homolocalised cutaneous nevi. Borrelia burgdorferi-specific sequence was detected in 15 out of 83 skin samples of patients with MF (18.1%), but in none out of 83 matched healthy controls (P<0.0001). The Bb positivity rates detected in this study support a possible role for Bb in the aetiopathogenesis of MF in a population endemic for LD.

Adult↗

PubMed1979

A novel lymphocyte differentiating factor in serum of patients with mycosis fungoides and Sezary syndrome.

Sera from 13 patients with mycosis fungoides and 2 with Sezary syndrome were tested for activity that induces lymphocyte differentiation. Induction of Thy-1.2 antigen and surface immunoglobulin were used, respectively, to measure T- and B-cell differentiation. The indicator cells were null lymphocytes from the spleens of congenitally athymic nude mice. Normal serum induced some T-cell but no B-cell differentiation. The T-cell-inducing activity was ascribed to thymic hormone and declined with advancing age. A totally different pattern emerged with patient serum. T-cell-inducing activity was significantly more active than in normal serum (p less than 0.001). This activity did not decline with advancing age and was not inhibited by a concentration of ubiquitin, which blocks nonspecific beta-adrenergic induction. B-cell-inducing activity was also present. This novel serum factor (or factors) is a potent inducer of T- and B-lymphocyte differentiation and is associated with neoplastic lymphoproliferation of the T-cell series.

Adult↗

PubMed1998

Antitumor activity of DAB389IL-2 fusion toxin in mycosis fungoides.

BACKGROUND: DAB389IL-2 is a novel fusion toxin that retargets the cytotoxic A-chain of diphtheria toxin to interleukin-2 (IL-2) receptor-expressing tumors. OBJECTIVE: The purpose of this phase I trial was to study the toxicity, maximum tolerated dose, and clinical efficacy of DAB389IL-2 in IL-2 receptor expressing lymphoproliferative malignancies, including cutaneous T-cell lymphoma. METHODS: DAB389IL-2 was administered intravenously daily for 5 days every 3 weeks. Dose escalation occurred between patient groups. Patients were monitored for laboratory and clinical toxicity, kinetics, immune response, and clinical efficacy. RESULTS: Thirty-five patients with cutaneous T-cell lymphoma (including 30 patients with mycosis fungoides) were treated. Previously, conventional therapy had not worked for 34 of the patients. Thirteen patients (37%) achieved an objective response, including a complete response in five patients (14%). Complete response was achieved in patients with extensive erythroderma and tumor stage mycosis fungoides. Adverse events consisted of reversible fever/chills, hypotension, nausea/vomiting, and elevation of hepatic transaminase. Doses of less than 31 microg/kg per day were well tolerated. Clinical responses were observed at all dose levels. CONCLUSION: DAB389IL-2 is well tolerated at doses of less than 31 microg/kg per day, and it induced clinical responses in previously treated mycosis fungoides, providing evidence for the antitumor activity of this molecule.

Adult↗

PubMed2000

Detection of TCR-gamma gene rearrangements in early mycosis fungoides by non-radioactive PCR-SSCP.

Early mycosis fungoides (MF) can mimic numerous benign inflammatory dermatoses on routine histological examination. In this study, a recently developed non-radioactive polymerase chain reaction-single strand conformational polymorphism (PCR-SSCP) technique was used to assess T-cell clonality in paraffin-embedded skin biopsies clinically and pathologically suspicious for early MF. Non-radioactive PCR-SSCP is a simple, sensitive, reproducible and rapid procedure requiring minimal instrumentation. DNA was extracted from 22 skin biopsies of 20 patients with suspected patch stage MF and 15 skin biopsies of inflammatory dermatoses. V gamma1-8, V gamma9, V gamma10, V gamma11 and J gamma1/J gamma2 consensus primers were used for T-cell receptor (TCR)-gamma gene rearrangement amplification. PCR products were analyzed by non-radioactive SSCP. Clonal TCR-gamma gene rearrangements were detected in 17 of 22 (77%) suspected MF specimens. Clonal SSCP banding patterns were different among individual patients. In addition, identical banded patterns were demonstrated in serial skin biopsies from the same patient. No dominant T-cell clones were found in the inflammatory dermatoses studied. Therefore, non-radioactive PCR-SSCP is a useful molecular diagnostic tool for assessment of T-cell clonality in paraffin-embedded specimens suspicious for early MF. The SSCP imprint of PCR products is specific for each TCR-gamma gene rearrangement, and may be used to evaluate concurrent/recurrent disease in individual patients.

Biopsy↗

PubMed1995

Distinctive dendritic cell subsets expressing factor XIIIa, CD1a, CD1b and CD1c in mycosis fungoides and psoriasis.

The papillary dermis of psoriasis and mycosis fungoides (MF) lesions is characterized by prominent collections of cells with dendritic morphology. Immunophenotypically distinct populations of cutaneous dendritic cells have been identified as CD1a+, FXIIIa-Langerhans cells (LC) and CD1a-, FXIIIa+ dermal dendritic cells (DDC). In this study, antibodies against the human CD1 cluster of antigens (i.e. CD1a, CD1b and CD1c) and the DDC marker (FXIIIa) were used to further characterize the subsets of dendritic cells in normal skin as compared to neonatal foreskin, psoriasis and MF by both immunoperoxidase and double immunofluorescence techniques. Normal skin and foreskin epidermis and dermis contained few CD1b+ or CD1c+ cells along with normal numbers of CD1a+ LC and FXIIIa+ DDC. Both MF and psoriasis were characterized by CD1a+ cells in the epidermis and dermis. FXIIIa+ cells were greatly expanded in the upper dermis of MF lesions and to a lesser degree in psoriasis as has been previously described by our group. MF contained significantly increased epidermal and dermal CD1b+ (15.7/5 high power fields [HPF] and 59.7/5 HPF respectively) and CD1c+ dendritic cells (33.8/5 HPF and 95.9/5 HPF respectively), while in psoriasis these cells were not statistically different from normal skin. Double immunofluorescence studies revealed that some (< 25%) FXIIIa+ cells co-expressed CD1b and CD1c in MF > psoriasis > foreskin, while FXIIIa+ DDC never co-expressed CD1a. Thus, in contrast to normal skin in which epidermal or dermal dendritic cells rarely express CD1b and CD1c antigens, these members of the CD1 family are upregulated on both LC and DDC in benign and malignant inflammatory states.(ABSTRACT TRUNCATED AT 250 WORDS)

Antigens, CD1↗

PubMed2005

Durable clinical, cytogenetic, and molecular remissions after allogeneic hematopoietic cell transplantation for refractory Sezary syndrome and mycosis fungoides.

PURPOSE: Sezary syndrome (SS) and tumor-stage mycosis fungoides (MF) are generally incurable with currently available treatments. We conducted a retrospective study to evaluate the outcome of allogeneic hematopoietic stem-cell transplantation (HSCT) in this patient population. PATIENT AND METHODS: From August 1996 through October 2002, eight patients with advanced MF/SS underwent allogeneic HSCT at our institution. All patients were heavily pretreated, having failed a median number of seven prior therapies (range, five to 12). Clonal T-cell populations in peripheral blood or bone marrow were detectable by polymerase chain reaction analyses of T-cell receptor gamma-chain gene rearrangements in six patients and cytogenetics in three patients. The conditioning regimen included total-body irradiation and cyclophosphamide (n = 3), busulfan and cyclophosphamide (n = 1), and the reduced-intensity regimen of fludarabine and melphalan (n = 4). Allogeneic hematopoietic stem cells were obtained from HLA-matched siblings (n = 4) and unrelated donors (n = 4). RESULTS: All patients achieved complete clinical remission and resolution of molecular and cytogenetic markers of disease within 30 to 60 days after HSCT. Two patients died from transplantation-related complications; graft-versus-host disease (GVHD; n = 1) and respiratory syncytial virus pneumonia (n = 1). With a median follow-up of 56 months, six patients remain alive and without evidence of lymphoma. CONCLUSION: Our results suggest that allogeneic HSCT from both HLA-matched sibling and unrelated donors can induce durable clinical, molecular, and cytogenetic remissions in patients with advanced cutaneous T-cell lymphoma that is refractory to standard therapies.

Adult↗

PubMed2005

Survival of mycosis fungoides in patients in the Southeast of England.

BACKGROUND: Mycosis fungoides (MF) is the most common skin lymphoma. The aetiology of MF remains unknown, and no therapy has to date significantly altered patient survival. OBJECTIVE: The present study examines trends in survival of MF patients in a well-defined population-based disease group, namely patients registered over a 40-year period at the Thames Cancer Registry, Southeast England. METHODS: The Thames Cancer Registry is a population-based registry, covering a population of approximately 14 million people. Data were taken from the Surveillance, Epidemiology and End Results cancer registry programme and the National Centre for Health Statistics. The database was used to identify cases of MF diagnosed between 1961 and 2000. A total of 985 records were identified, 821 (83%) of which had complete information on age, sex, year of diagnosis and area of residence. The observed and relative survivals of patients diagnosed during the periods 1971-1975, 1981-1985 and 1991-1995 were examined over a 5-year period of follow-up, using the relsurv Stata program to perform Cox proportional hazard analysis. RESULTS: A total of 821 MF eligible patients were available with a median follow-up of 4.3 years and a maximum follow-up of 30 years. The overall 5-year relative survival rate was 80%, and there was marked improvement between 1971 and 1981. The prognostic factors leading to a significantly poorer survival were high age, male sex, the presence of the Sézary syndrome, the use of hormone treatment and radiotherapy. CONCLUSIONS: A statistically significantly better survival over the last 20 years was found. The prognosis is generally good for most patients but not all. The best survival was seen for the female patients under 45 years of age without the presence of the Sézary syndrome. This difference in survival may be partly due to a difference in the disease stage or different treatment, or to both.

Age Factors↗

PubMed1998

Mycosis fungoides in young patients: clinical characteristics and outcome.

BACKGROUND: Mycosis fungoides (MF) can begin as early as the first decade of life. Few studies have reviewed MF in younger patients and none has been large enough to assess prognosis and outcome. OBJECTIVE: We reviewed the clinical characteristics, prognosis, factors related to disease progression, and therapy in patients with MF younger than 35 years of age. METHODS: Fifty-eight patients were entered into this retrospective cohort analysis. RESULTS: Significantly fewer patients with MF who are younger than 35 years presented with erythroderma (T4) and more with limited patch/plaque (T1) disease than older patients. Duration of skin disease before diagnosis of MF did not vary between the two groups. The long-term survival of younger patients with MF is significantly decreased when compared with a race-, age-, and sex-matched control population (p < 0.001). Disease-specific survivals (DSS) of younger and older patients are similar, but young patients show a slight but significantly better overall DSS (p < 0.02). However, DSS comparison of generalized patch/plaque (T2) and tumor stage (T3) patients with MF showed no significant difference between young and old patients (p=0.47, p=0.59). Patient age was not a significant predictor of survival when controlled for T-stage. Sixteen of 58 young patients with MF have died, 13 because of MF (22%), compared with 138 of 500 older patients (28%) who died as a result of MF. All younger patients with MF who progressed had at least T2 disease at presentation. Fifty of 56 young patients with MF and T1-T3 disease were treated initially with total skin electron beam or topical nitrogen mustard. The response to therapy was similar in younger and older patients with MF. CONCLUSION: T1 disease is more common and T4 disease is unusual in young patients with MF compared with an older population of patients with MF. Young patients with T1 disease, all of whom were treated with either topical nitrogen mustard or total skin electron beam therapy, or both therapies, showed no disease progression. Overall, young patients with MF showed slightly better DSS, but this was because of differences in stage distribution.

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↗

PubMedDate not supplied

[Immunological studies on patients with mycosis fungoides].

Recently, much has been published on the immunological status of patients affected with various lymphomas. In the particular case of Mycosis fungoide, there was no general agreement on the immunological status of the corresponding patients. In fact, López Borrasca et al, found severe depression of cellular immunity in such patients. On the contrary, Blaylock and Clendenning found very little change in cellular immunity, but a very high serum-IgA. We want to offer our experience on this problem with the immunological survey of four patients with the Alibert-Bazin-form of Mycosis fungoide. The following tests were performed on each patient: a) Intracutaneous test with candidina, PPD and other bacterial antigens. b) Sensitization to a concentrate solution of dinitrochlorobencene (DNCB). c) Lymphocyte Transformation Test (LTT), with phytohemagglutinin as mitogen. d) Quantitative determination of IgG, IgM, IgA and beta1C, with the radial immunodiffusion technique (Mancini et al.). e) Agar immunoelectrophoresis. The following results were obtained: 1) The cellular immunity was markedly depressed in the four patients when any of a, b or c-test was performed. 2) All the patients showed very high levels of serum IgA, 150% higher than control. The reason for this is unknown. On the contrary, IgG in serum was less elevated and IgM and beta1C serum levels were normal. 3) No monoclonal bands were found in any case (immunoelectrophoresis). 4) No definite conclusions could be reached due to the limited number of cases, but the uniformity of results should encourage to carry this work further.

Angioedema↗