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PubMed1996

Clinical stage IA (limited patch and plaque) mycosis fungoides. A long-term outcome analysis.

OBJECTIVES: To study the long-term results of treatment of patients with stage IA mycosis fungoides and analyze the factors related to disease progression and the effect of initial therapy on survival and freedom from relapse. DESIGN: A single-center, 32(1/2)-year, retrospective cohort analysis. SETTING: Private referral medical center. PATIENTS: One hundred twenty-two patients with clinical stage IA (T1, N0, M0) mycosis fungoides. MAIN OUTCOME MEASURES: Long-term actuarial survival and freedom-from-relapse results as calculated by the technique of Kaplan-Meier. RESULTS: The long-term (30-year) survival of patients with stage IA mycosis fungoides is similar to the expected survival of a race-, age-, and sex-matched control population. The median survival of this group has not been reached at 32(1/2)-years. Eleven patients (9%) who progressed to more advanced disease had a lower complete response rate to initial therapy than did other patients (36% vs 82%) and an older mean age than did other patients with T1 disease (61 vs 48 years, P < .05). Only 3 (2%) of 122 patients died of disease. Among stage IA patients who achieved a complete response, 25% are relapse free at 10 years. Patients who received total skin electron beam therapy (n = 34) had a more favorable freedom-from-relapse outcome than those treated with topical mechlorethamine hydrochloride (nitrogen mustard) (n = 73, P < .05). No significant difference was seen in the long-term survival between the 2 treatment groups. CONCLUSIONS: Patients with clinical stage IA mycosis fungoides treated at Stanford University do not have an altered life expectancy. Fewer than 10% progressed to more advanced stages and few died of disease. Although the response rate to total skin electron beam therapy was superior to that of topical mechlorethamine, the longterm survival results were similar. Topical mechlorethamine is a cost-effective and convenient therapy for patients with limited patch and plaque mycosis fungoides.

Actuarial Analysis↗
PubMed1981

Mycosis fungoides: diagnostic criteria based on quantitative electron microscopy.

Quantitative electron microscopy can provide objective measurements of nuclear atypia in lymphoid cells in skin biopsies during early stages of infiltration of the skin as part of inflammatory or neoplastic processes. This potentially provides a method for establishing reproducible criteria for determining whether an individual skin biopsy represents mycosis fungoides or is unlikely to represent mycosis fungoides. To investigate such criteria, quantitative electron microscopic studies of skin biopsies from 109 patients were performed. Seventy-seven patients had benign disorders, 16 patients had early mycosis fungoides, and 16 patients had controversial lesions. Measurements were made of lymphocyte nuclear profile perimeter and areas in electron micrographs. A nuclear contour index was calculated. On the basis of the mean degree of nuclear convolution alone, mycosis fungoides was not adequately distinguished from benign disorders. Better discrimination was achieved when the proportion of cells with a anuclear contour index of 9.0 or more was considered as an additional actor. When statistical methods were used to select discriminating factors, the best electron microscopic discrimination between benign and mycosis fungoides groups occurred when the proportion of cells with a nuclear contour index of 7 or more and the proportion of cells with a nuclear profile area greater than 30 sq. micrometer. were used together. Nomograms are presented which demonstrate the use of these criteria for a given case.

Aged↗
PubMed1998

Radiotherapy for unilesional mycosis fungoides.

PURPOSE: To evaluate the treatment outcome and natural history of patients with the diagnosis of unilesional mycosis fungoides, treated according to a prospective radiotherapy protocol in our institution since July 1975. METHODS AND MATERIALS: A total of 325 patients with the diagnosis of mycosis fungoides have been referred to the Department of Radiation Oncology at Allegheny University of Health Sciences from July 1975 through September 1996. Of these, 18 patients (5%) were classified as having unilesional mycosis fungoides and were irradiated with a curative intent using local electron fields. One patient received 22 Gy; 1 patient received 40 Gy, and the rest of the patients 30.6 Gy. Daily fractions ranged from 1.8 to 2.0 Gy. Treatments prior to radiation consisted of topical steroids and/or antifungal creams in the majority of patients, with temporary partial responses. One patient had received 2 years of topical mechlorethamine (HN2) and another patient had received topical carmustine solution (BCNU) without response prior to irradiation. RESULTS: The responses were measured clinically; posttreatment skin biopsy was not performed routinely unless there was clinical evidence of disease persistence. Complete response rate was 100%; all treated lesions cleared completely within 4 to 8 weeks after the completion of radiation. With a median follow-up of 43 months (range 12 to 240 months), 2 relapses have occurred, 2 and 71 months after the completion of radiation. Both relapses were confined to the skin and were remote from the original site. Both relapses responded to topical application of HN2. There have been no recurrences in the irradiated field nor systemic dissemination. No long-term side effects were found related to treatment, and all the patients are currently alive and without evidence of disease. Actuarial relapse-free and overall survival at 10 years are, respectively, 86.2% and 100%. CONCLUSION: Unilesional mycosis fungoides has a long natural history, is possibly the earliest manifestation of a malignant process, and local treatments, including local radiotherapy, result in long-term disease-free intervals and, possibly, cure. Total skin electron beam radiotherapy is not indicated for this disease entity.

Adult↗
PubMed2004

Mycosis fungoides and the Sézary syndrome.

PURPOSE OF REVIEW: Mycosis fungoides and the Sézary syndrome represent a heterogeneous group of good-to-intermediate-risk non-Hodgkin lymphomas that have recently been identified as distinct histopathologic and clinical entities by the World Health Organization and European Organization for Research on the Treatment of Cancer lymphoma classification systems. Significant progress has been made in identifying and categorizing patients based on clinical prognostic factors, but there is little information regarding the etiology, molecular biology, and molecular genetics of these diseases. This review outlines recent advances in clinical diagnosis and prognosis as well as novel therapeutic approaches. RECENT FINDINGS: A number of reports have further defined clinical prognostic subgroups among early-stage patients and those with circulating Sézary cells. The recent availability and demonstrated efficacy of the oral RXR retinoid, bexarotene, has altered the treatment paradigm of early-stage patients who would not otherwise be exposed to systemic therapies. Novel targeted agents and receptor-directed therapies, including the fusion toxin, denileukin diftitox, histone deacetylase inhibitors, and novel nucleoside analog therapies, have demonstrated promising activity and are undergoing further clinical evaluation. The evolution of immunotherapy has been augmented by studies demonstrating the efficacy of peptide-loaded dendritic cells as well as the use of photopheresis to generate an anti-idiotype cytotoxic T-cell response. SUMMARY: This review will enumerate the most recent findings with respect to clinical staging, prognosis, and treatment of patients with mycosis fungoides and the Sézary syndrome. Novel treatment options will be reviewed and treatment paradigms will be outlined.

Anticarcinogenic Agents↗
PubMed1976

Monocytopoiesis in chronic eczematous diseases, psoriasis vulgaris, and mycosis fungoides.

Monocytopoiesis and blood monocytes were examined in 8 patients with disseminated chronic eczematous diseases, 8 patients with disseminated psoriasis vulgaris, and 8 patients with mycosis fungoides in plaque or tumor stage. Monocytopoiesis was moderately stimulated in all these patients. The stimulation manifested itself by: (1) a rise in relative number of promonocytes in bone marrow in all patients with eczema, in 1 out of 8 patients with psoriasis, and in 7 out of 9 examinations in patients with mycosis fungoides; (2) a rise in [3H]thymidine labeling indices of medullar promonocytes (8/8 eczema, 7/7 psoriasis, 8/9 mycosis fungoides); and (3) a rise in the naphthol-AS-D-chloroacetate esterase activity of blood monocytes, indicating premature monocyte marrow egress (3/5 eczema, 7/8 psoriasis, 9/9 mycosis fungoides). In eczema and psoriasis the mean enhancement of monocytopoietic activity was similar but less pronounced than in mycosis fungoides. In the latter disease there was no correlation between measured parameters and visible skin lesions. The results were interpreted as indicative of increased monocyte consumption by pathologic, immunologic, and/or inflammatory processes.

Adult↗
PubMed2006

Retrospective 5-year review of 131 patients with mycosis fungoides and Sézary syndrome seen at the National Skin Centre, Singapore.

A total of 131 new cases of mycosis fungoides and Sézary syndrome were diagnosed clinically and histopathologically at our centre over a 5-year period. There were 87 males and 44 females with a mean age of 36.3 years (range 3-87 years) and no racial predilection. Of the 62 patients (47.3%) with classical mycosis fungoides, the majority were male (male : female = 4.2:1). There was one patient with Sézary syndrome. Patients aged older than 50 years were more likely to present with a longer duration of symptoms and advanced disease. In contrast to classical mycosis fungoides, the 47 patients diagnosed with hypopigmented mycosis fungoides had early stage disease, were younger, and no gender predilection was noted. The mean duration of follow up was 19.7 months (range 0.2-54.8 months). Complete remission was achieved in 24.7% and 53.8% of patients followed up at 1 and 3 years, respectively, using skin-directed and systemic treatment modalities appropriate for the stage of disease. There were five patients with progressive disease and three patients with advanced disease who died from disease-related complications. The most significant prognostic factors for 1-year and 3-year outcomes were the patient's duration of symptoms and stage of disease at presentation.

Adolescent↗
PubMed1990

Coexisting follicular mucinosis and mycosis fungoides--a case report.

This is a case report of a patient with mycosis fungoides coexisting with follicular mucinosis. The simultaneous occurrence of these two conditions has been documented. There is controversy as to whether mycosis fungoides in patients with follicular mucinosis arises de novo or whether follicular mucinosis can evolve into mycosis fungoides. The relationship between the two conditions is discussed.

Adult↗
PubMed1992

Mycosis fungoides of the larynx. Report of two cases and review of the literature.

Involvement of the larynx by mycosis fungoides is extremely rare with only three reported clinical cases in the English-language literature. We present two patients with laryngeal mycosis fungoides, one of whom presented with vocal cord paresis (progressing to paralysis) as the initial clinical manifestation of laryngeal involvement. Our clinical findings and the observations from the three previous case reports suggest that laryngeal mycosis fungoides has a predilection for the arytenoids, aryepiglottic folds, and the laryngeal surface of the epiglottis. Laryngeal involvement, like other forms of visceral dissemination, appears to manifest clinically in the terminal stages of the disease. The natural history, clinical features, histopathology, and treatment of mycosis fungoides are reviewed and the etiopathology of the vocal cord paralysis is described.

Aged↗
PubMed1995

[Hypopigmented mycosis fungoides].

INTRODUCTION: Isolated areas of hypopigmented skin can be a manifestation of mycosis fungoides. This is a rare clinical form with a particular clinical course and histology which has been described only in black subjects. CASE REPORT: Over a 10-year period, a 28-year-old woman from Mali developed non-infiltrated hypopigmented and discretely squamous macules involving the entire body. The diagnosis of mycosis fungoides was made on pathology evidence of epidermotropic lymphocyte infiltration with a few small nests. PUVA led to rapid regression of the lesions. DISCUSSION: Hypopigmented mycosis fungoides has been described in young black subjects (mean age 19 years). The diagnosis is usually made late because benign skin affection is usually the first diagnosis (pityriasis alba, pityriasis versicolor). The pathogenesis of these hypopigmented lesions is not known. PUVA appears to be effective in most cases.

Adult↗
PubMed1997

Immunohistochemical expression of IL-10 in mycosis fungoides.

Immunoreactivity of the cytokine IL-10 has been investigated in situ in mycosis fungoides (MF). Expression of IL-10 was detected using immunohistochemistry in skin biopsies (n = 8) and T-cell lines (n = 2) from mycosis fungoides patients. IL-10 positivity was seen in the dermal cell infiltrates and in T-cell lines in mycosis fungoides. The dermal IL-10 reaction in a skin biopsy from an active lesion in MF indicates the possibility for disease progression.

Adult↗
PubMed2006

Clinicopathological spectrum of mycosis fungoides type cutaneous T-cell lymphoma.

OBJECTIVE: To determine the clinical, histological, and immunophenotypic characteristics of mycosis fungoides type cutaneous T-cell lymphoma. DESIGN: Descriptive study. PLACE AND DURATION OF STUDY: This study was conducted from January 2000 to December 2004 at the Department of Dermatology, Military Hospital and the Department of Dermatopathology, Armed Forces Institute of Pathology, Rawalpindi. MATERIALS AND METHODS: The medical case records of patients with mycosis fungoides diagnosed during the period of study were surveyed. Data was collected pertaining to patient s characteristics, clinical descriptions, histopathological features, immunophenotypic analysis and stage of disease at the time of diagnosis. RESULTS: A total of 33 cases of mycosis fungoides were diagnosed between the years 2000 and 2004. There were 24 male and 9 female patients with male to female ratio of 2.6:1 The age ranged from 24 to 68 years and the duration of disease prior to diagnosis varied between 2 to 36 months. The number of skin biopsies performed for definite diagnosis ranged from 01 to 5. The various clinical presentations recorded in these patients were hypopigmented patches in 7 (21.3%), infiltrated papules and plaques in 6 (18.2%), erythroderma in 5 (15.2%), psoriasiform lesions in 3 (9%), and nodular lesions in 3 (9%) patients. There were 2 (6%) cases respectively of noduloulcerative, ichthyosiform and poikilodermatous lesions, and 1(3%) case each of follicular, morphoea-like and purpuric skin lesions. The predominant histological features were lymphocytic infiltrate in the upper dermis, epidermotropism, haloed lymphocytes in epidermis, Pautrier s microabscesses, and interface dermatitis. The immunohistochemical studies (n=12) showed predominantly T helper cell immunophenotype (CD3+, CD45RO+) in 11(92%) cases and T suppressor cell immunophenotype (CD3+, CD8+) in 1(8%) patient. CONCLUSION: The mycosis fungoides type cutaneous T-cell lymphoma has a wide clinicopathological spectrum. In a clinically non-specific dermatosis, a high index of suspicion and a regular follow-up may eventually lead to the definite diagnosis.

Adult↗
PubMed2000

Detection of a peripheral blood T cell clone is an independent prognostic marker in mycosis fungoides.

UNLABELLED: T cell receptor gene analysis is a sensitive method for assessment of peripheral blood involvement in mycosis fungoides. This study uses polymerase chain reaction/single-strand conformational polymorphism (PCR/SSCP) analysis of the T cell receptor gamma gene and relates the results to skin stage and outcome in mycosis fungoides. Seventy-five peripheral blood samples from 66 patients were obtained from 1990 onwards and subjected to PCR/SSCP. Both Southern blot analysis and PCR/SSCP analysis were performed on 63 samples from 56 patients. Fourteen patients had T1 disease (12 IA, two IIA), 20 T2 (14 IB, five IIA, one IVA), 29 T3 (24 IIB, two IVA, three IVB, two patients tested at both T2 and T3), and five T4 (all III). The percentage of positive samples was higher with PCR/SSCP than with Southern blot analysis (29 of 63 vs eight of 63 samples, p < 0.001), and the percentage of positive samples increased with each stage (21% at T1, 35% at T2, 58% at T3, and 71% at T4). Proportional hazards analysis corrected for age, skin, and lymph node stage showed that the presence of a peripheral blood clone is associated with a worse outcome (p = 0.03, CI 1.1-6.03). These results indicate that the presence of a peripheral blood clone is an independent prognostic variable in patients with mycosis fungoides after correcting for age, skin, and lymph node stage, and that peripheral blood involvement is present in a large proportion of patients with early stage mycosis fungoides. KEYWORDS: polymerase chain reaction/single-strand conformational polymorphism/T cell receptor gene rearrangement. J Invest Dermatol 114:117-121, 2000

Biomarkers↗
PubMed1996

Pilotropic cutaneous T-cell lymphoma without mucinosis. A variant of mycosis fungoides? French Study Group of Cutaneous Lymphomas.

BACKGROUND: In the course of mycosis fungoides, pilofollicular manifestations without mucinosis (papules, keratoses, comedones, or epidermal cysts) are rare (15 cases reported). Therefore, histological and clinical diagnoses may be difficult. The clinical course and histopathological and immunohistochemical findings in 9 patients are described. OBSERVATIONS: Pilofollicular lesions were present at the onset (n = 3), before (n = 3), or during a relapse of mycosis fungoides (n = 3). Comedones and epidermal cysts were most frequent (n = 5). They disappeared with lymphoma therapy (n = 4), therapy with isotretinoin (n = 3), or spontaneously (n = 1), or they persisted (n = 1). Clues to the histopathological diagnosis consisted of pilotropism of the infiltrate with minor alteration of the hair follicle walls. The infiltrate was monomorphous and composed of sezariform CD4+ lymphocytes. Pilotropic or peripilofollicular infiltrates, or the absence of infiltrate, were detected in consecutive biopsy specimens obtained from the same patient. The keratinocyte expression of intercellular adhesion molecule type 1 was observed in the hair follicle bulb in front of the pilotropic infiltrate but not in the epidermis. No staining was observed in biopsy specimens of 6 of 7 patients with follicular mucinosis, of folliculitis lesions, or of normal hair follicles. CONCLUSIONS: Our findings indicate the role of adhesion molecules in pilotropism leading to mechanical obstruction of the follicle by tumoral cells followed by hyperkeratosis and cyst formation. It remains to be determined if the expression of intercellular adhesion molecule type 1 is the cause or the consequence of pilotropism. By becoming more aware of it, this variant of mycosis fungoides is probably not so rare.

Adult↗
PubMed1984

[Change in the turnover of HLA ABC molecules in circulating T lymphocytes in mycosis fungoides].

The turnover of HLA ABC molecules at T and B lymphocyte surface was analyzed in five cases with mycosis fungoides and six healthy controls. The patients had only skin lesions and are staged from T1 to T3 and No, Bo and Mo. The turnover was analyzed by mean of the decrease of the lysis by complement on sensitisized cells with anti HLA ABC antibodies that were incubated for progressive times at 37 degrees C. The results show a largest turnover for the HLA ABC molecules all T cells surface from mycosis fungoides that was significant (PWilcoxon less than 0.01). The turnover of B cells surface was not different from mycosis fungoide and healthy controls. The observed phenomenon was not specific for mycosis fungoide or T cells because previously has been shown in others lympho-proliferative and autoimmune diseases. The authors suggest that the analyzed T lymphocyte, morphologically normal is functionally altered and the behavious could be a metabolic phenotypic marker for the T cell before his coming at skin lesions.

Adult↗
PubMed1992

Adenosine deaminase activity in sera of patients with psoriasis, mycosis fungoides and adult T cell leukemia.

Adenosine deaminase activities in sera were measured in 18 psoriatic patients, 8 mycosis fungoides patients, and 9 patients with adult T cell leukemia. Adenosine deaminase activity in the sera of the psoriatic patients showed no significant increase. An elevated adenosine deaminase activity was observed in 7 of the 8 patients with mycosis fungoides and 8 of the 9 patients with adult T cell leukemia. After chemotherapy, adenosine deaminase activity in serum of acute adult T cell leukemia was reduced. Adenosine deaminase activity in the sera of 2 patients with smoldering adult T cell leukemia was more elevated, with exacerbation of the disease. It is difficult to grade the extension of the tumors in plaque stage mycosis fungoides and smoldering adult T cell leukemia. To know the progression of the disease is critical in determining its management. These results indicate that adenosine deaminase activity in serum is one of the reliable indicators for the grading of mycosis fungoides and adult T cell leukemia.

Adenosine Deaminase↗
PubMed1993

Follicular mycosis fungoides. A clinical and histologic variant of cutaneous T-cell lymphoma: report of two cases.

We report two cases of mycosis fungoides with marked, pleomorphic follicular manifestations. Follicular hyperkeratosis, comedo-like lesions, acquired epidermal cysts, and patchy alopecia developed in various locations in both patients. Findings of histopathologic and immunohistochemical studies showed atypical CD4+ T lymphocytes infiltrating the follicles without follicular mucinosis. Focal expression of intercellular adhesion molecule type 1 was observed within the cyst walls. These findings suggest that the follicular lesions were specific for mycosis fungoides. These manifestations represent a distinct clinical and histologic form of mycosis fungoides. This variant probably accounts for cases of mycosis fungoides with clinically suspected alopecia mucinosa in which follicular mucinosis cannot be histologically proved.

Aged↗
PubMed1984

Monoclonal gammopathy and mycosis fungoides. Report of four cases and review of the literature.

Of four patients with mycosis fungoides and monoclonal gammopathy, one died of multiple myeloma that developed 4 years after the mycosis fungoides. The other three had monoclonal gammopathy of undetermined significance. This association does not appear to occur with a high frequency in patients with mycosis fungoides than in the general population. However, it demonstrates that B cell proliferation can occur in patients with primary cutaneous T cell disease.

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