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PubMed1987

Oral retinoids in mycosis fungoides and Sézary syndrome: a comparison of isotretinoin and etretinate. A study from the Scandinavian Mycosis Fungoides Group.

Thirty-nine patients with mycosis fungoides in various stages or Sézary syndrome were treated with isotretinoin and 29 with etretinate as single drug therapy. Complete remission within 2 months was obtained with isotretinoin in 8 cases (21%) and partial remission in another 15 cases (38%). Etretinate induced complete remission in 5 cases (21%) and partial remission in 11 (46%). Only 1 case with Sézary syndrome went into partial remission. The first sign of remission occurred in 2 to 4 weeks. During continued treatment remissions could not always be maintained. Isotretinoin and etretinate were considered to be of equal potency in the treatment of mycosis fungoides.

Drug Eruptions↗
PubMed1979

Mycosis fungoides plaque stage treated with topical nitrogen mustard with and without attempts at tolerance induction: report from the Scandinavian mycosis fungoides study group.

The Scandinavian Mycosis Fungoides Study Group has treated 21 patients with mycosis fungoides in plaque stages with topical, whole-body application of nitrogen mustard, 20 mg in 40 ml water per square metre. Ten patients were treated after previous attempts at intravenous tolerance induction ad modum van Scott & Kalmanson and eleven without. Complete remission was initially achieved in 10 patients and partial remission in 9 patients. Contact dermatitis to nitrogen mustard developed in 2/10 after tolerance induction and in 1/11 without tolerance induction. It is concluded that topical, whole-body application of nitrogen mustard gives high remission rates. In this series, however, many relapses occurred, due to inadequacy of the maintenance treatment. Tolerance induction has not been found of any value.

Administration, Topical↗
PubMedDate not supplied

Mycosis fungoides and pregnancy.

Mycosis fungoides is a cutaneous T-cell lymphoma, a subgroup of non-Hodgkin's lymphomas, characterized by skin infiltration and occasionally systemic involvement. The association of pregnancy and mycosis fungoides has not been described previously. A case of mycosis fungoides, stage IVb, in a pregnant woman is reported. Prior to pregnancy, the patient received adriamycin, cyclophosphamide, vincristine prednisolone (CHOP) and bleomycin and total body irradiation. Around the concepcional period she presented a cutaneous relapse palliated with photon radiotherapy. No obstetrics complications were observed during gestation. At 39 week's gestation a cesarean section was performed and a healthy 2900 g boy was delivered. Mycosis fungoides did not worsen during pregnancy and postpartum period. In conclusion mycosis fungoides did not adversely affect pregnancy outcome and gestation did not worsen the malignancy course. This case report may be valuable in managing patients with mycosis fungoides who are currently pregnant or are contemplating pregnancy.

Adult↗
PubMed1999

Unilesional follicular mycosis fungoides.

Follicular mycosis fungoides (FMF) is an unusual clinical and histological variant of MF, characterized by selective involvement of hair follicles by atypical lymphocytes. We describe a female patient who had follicular papules located only on the medial aspect of her right thigh. To the best of our knowledge, this patient represents the first reported case of unilesional FMF.

Diagnosis, Differential↗
PubMedDate not supplied

Chromosomes and B and T cells in mycosis fungoides.

Because mycosis fungoides (MF) and Sézary syndrome (SS) share several features, some investigators have considered them to be different stages of the same disease. Others view them as separate entities. Cytogenetic studies in four typical MF patients showed that the chromosomal abnormalities were different from those reported in SS patients, and the abnormalities were found with almost equal frequency in both B and T cells. In addition, we found the frequencies of the T cells in six of the eight peripheral blood determinations in three patients were within normal range. These observations support the possibility that MF and SS are two separate disease entities.

Aged↗
PubMed2002

Ichthyosiform mycosis fungoides.

BACKGROUND: Mycosis fungoides (MF) is a skin malignancy of T helper lymphocytes with a wide clinical spectrum. Among the atypical variants of MF, there is an ichthyosis-like presentation. However, to date, only 1 case of ichthyosiform MF has been reported. OBJECTIVE: Our goal was to summarize the clinical characteristics and course, and the pathological, immunohistochemical and molecular genetic findings on 4 patients with ichthyosiform MF. METHODS: A retrospective study was conducted. RESULTS: The 4 patients represented 1.8% of the 221 patients with MF seen by us since 1975. None progressed to systemic disease in up to 12 years (median, 10 years) after the onset of the cutaneous manifestations. Interestingly, skin lesions typical of so-called follicular MF (FMF) were associated in 3 of 4 cases, whereas cutaneous manifestations of classic MF were absent in all 4 patients. CONCLUSION: Ichthyosiform MF represents a rare variant within the clinicopathologic spectrum of MF usually featuring a benign course and a tendency to be associated with lesions of FMF but not with lesions of classic MF.

Adult↗
PubMed1994

Proliferative activity of epidermal and dermal T lymphocytes in mycosis fungoides.

BACKGROUND: Mycosis fungoides (MF) is histologically characterized by an accumulation of T lymphocytes (T cells) in both epidermis and dermis. However, the mechanism of the formation of Pautrier's microabscess or the maintenance of the epidermal T cells has not yet been clarified. OBJECTIVE: The rate of proliferation of these T cells was investigated. METHODS: The formalin-fixed, paraffin-embedded sections from 9 cases of patch/plaque stage MF were immunostained with proliferating cell nuclear antigen (PCNA). RESULTS: The PCNA-positive T cell nuclei were found both in the epidermis (including Pautrier's microabscesses) and dermis in all the specimens. The mean PCNA indices of the T cells were 20.2 +/- 10.6% (mean +/- SD) within the epidermis and 16.8 +/- 6.5% in the dermis, which was not statistically significantly different (p > 0.05). The ratio of the PCNA index between the epidermis and dermis was 1.3 +/- 0.6, which was not apparently correlated with the duration of the disease (8.4 +/- 5.1 years). CONCLUSION: These findings suggested that in situ proliferation might contribute to the epidermal T cell population resulting in the formation of Pautrier's microabscesses and an increase in dermal T cell volume during the course of the disease.

Abscess↗
PubMed2006

Presence of circulating CCR10+ T cells and elevated serum CTACK/CCL27 in the early stage of mycosis fungoides.

PURPOSE: Mycosis fungoides (MF), a common type of cutaneous T cell lymphoma with an indolent clinical course, has the characteristic that malignant T cell clones are recruited into the skin from the early disease stages. The mechanisms of recruitment have been suggested from our knowledge of various chemokine-chemokine receptor interactions. Recently, CCR10 and CTACK/CCL27 were proposed to play a role in the recruitment of other types of cutaneous T cell lymphoma. We examined the expression of CCR10 in peripheral blood and serum CTACK/CCL27 levels in patients with MF. EXPERIMENTAL DESIGN: Eighteen patients with MF, six patients with atopic dermatitis, and nine healthy volunteers were enrolled in our investigation. We investigated the differences in CCR10+ CD4+ expression in peripheral blood mononuclear cells by flow cytometry. Serum CTACK/CCL27 levels were determined using a CTACK/CCL27 ELISA assay kit. RESULTS: The number of circulating CCR10+ CD4+ cells was significantly higher in MF peripheral blood than in controls, even during the early stages. In lesional MF skin, infiltrating tumor cells also showed extensive expression of CCR10. The serum level of CTACK/CCL27 was higher in patients with MF than normal controls, but no statistical difference was found compared with atopic dermatitis patients. CONCLUSIONS: CCR10-CTACK/CCL27 interactions between circulating T cells and keratinocytes would seem to play an important role in the pathophysiology of MF from the early disease stages.

Adult↗
PubMed2006

A correlation between acute phase proteins and cytokines in patients suffering from mycosis fungoides.

BACKGROUND: Mycosis fungoides (MF) is the most common form of cutaneous T-cell lymphoma and cytokines are involved in the pathogenesis of the disease. To evaluate whether the serum levels of sIL- 2R, IL-4, IL-5, IL-10, and INF-gamma reflect the processes in various stages of MF, to measure the acute phase protein concentrations in the serum of the patients, and to determine the glycosylation profile of two proteins. MATERIAL AND METHODS: Serum samples were obtained from 52 patients with MF and from 20 healthy subjects. Concentrations of interleukins IL-4, IL-5, IL-10, INF-gamma, and sIL-2R were measured using ELISA tests. Concentrations of proteins were measured using Laurell rocket electrophoresis. RESULTS AND DISCUSSION: An inflammatory reaction exists in all MF patients because the concentrations of acute phase proteins were increased. In more advanced stages of MF, intensive activation of T cells is present, as reflected by the sIL-2R levels and Th2 cytokines outweighing the main Th1 cytokine (IFNgamma). Increased levels of sIL-2R, IL-4, and IL-10 accompanied the increase in concentrations of acute phase proteins and a decrease in their reactivity with Con A. It appears that the increase of IL-4, IL10, and sIL-2R may be due to the activation of T-helper lymphocytes.

Acute-Phase Proteins↗
PubMed1998

Prognostic significance of a polymerase chain reaction-detectable dominant T-lymphocyte clone in cutaneous lesions of patients with mycosis fungoides.

Although mycosis fungoides (MF) is considered to be an indolent lymphoma, survival is highly influenced by TNM stage. At diagnosis, most MF patients present with early stage disease and a high probability of long-term survival. Treatment is generally directed towards skin lesions, and achievement and duration of complete responses are variable. A dominant T-cell clone is detectable in the cutaneous lesions of 60% of patients. The aim of this study was to determine whether the presence of a T-cell clonal population influences the clinical course of the disease after topical therapy. Cutaneous biopsies from 68 patients were histologically diagnosed as MF and T-cell clonality was analyzed by in vitro amplification of TCR-gamma chain gene rearrangements (polymerase chain reaction gamma [PCRgamma]). After a median follow-up of 48 months, response to treatment was clinically assessed. Age, sex, duration of symptoms before diagnosis, type of cutaneous lesions (T stage), TNM stage, and PCRgamma were evaluated as predictive factors of response to treatment in univariate and multivariate analyses. Univariate analysis demonstrated that T1 cutaneous lesions (P = .05) and PCRgamma negativity (P = .007) were associated with a higher complete remission rate. Using multivariate analysis, T stage (relative risk, 3.13; P = .06) and PCRgamma (relative risk, 4.4; P = .01) remained independent significant predictive parameters of response. In conclusion, T stage and cutaneous PCRgamma at diagnosis are the two predictive parameters of treatment response for MF. Therefore, the cutaneous PCRgamma findings should be considered in the analysis of future therapeutic trials.

Administration, Cutaneous↗
PubMed2006

CD4/CD8 double-negative epidermotropic cutaneous T-cell lymphoma: an immunohistochemical variant of mycosis fungoides.

BACKGROUND: Mycosis fungoides (MF) is an epidermotropic cutaneous T-cell lymphoma in which the tumor cells express a mature T-helper memory phenotype, ie, CD3(+), CD4(+), CD8(-), CD45RO(+), with a T-cell receptor (TCR) of the alpha/beta heterodimer. A minority of patients have an unusual immunohistochemical profile consisting of a CD4(-), CD8(+) mature T-cell phenotype. An aberrant CD4/CD8 double-negative (DN) immunophenotype in patients with early MF has rarely been reported. OBJECTIVES: We sought to evaluate the frequency of CD4/CD8 DN immunophenotype in patients with early MF, and to study their clinical, histopathologic, and immunohistochemical features, and the course of their disease. METHODS: Our departmental archives were searched for patients with early-stage MF and CD4/CD8 DN immunophenotpye. RESULTS: Of the 140 patients with early MF immunophenotyped in our laboratory, 18 (12%) showed CD4 and CD8 expression in less than 10% of their intraepidermal T cells on fresh-frozen and paraffin-embedded samples. The group included 13 male and 5 female patients; 14 adults and 4 children; and 15 Jews and 3 Arabs. In all, 8 had classic MF and 10 had unusual clinical variants (5 hypopigmented, 3 localized, 1 ichthyosiform, 1 purpuric). All received skin-targeted therapies and all had an indolent course (mean follow-up 3.5 years). Histopathology revealed early MF. Results of immunohistochemical analysis of the intraepidermal lymphocytes were as follows: CD3(+), CD4(-), CD8(-) in all patients; CD7(-) in all of 17; CD45RO(+) in 15 of 16; T-cell-restricted intracellular antigen-1(+) in 11 of 15; CD30(+) in 2 of 16; and CD56(+) in 2 of 16. A betaF1(+)/delta(-) phenotype, indicating a TCR of the alpha/beta heterodimer, was found in 8 of 16; betaF1(-)/delta(+) phenotype, indicating a TCR of the gamma/delta heterodimer, in 1 of 16; betaF1(-)/ delta(-) in 5 of 16; and no determinable phenotype in 2 of 16. The TCR gamma gene was clonally rearranged in 10 of 16 patients. LIMITATION: This was a single-center case series. CONCLUSIONS: There is a subgroup of patients with early MF that exhibit a CD4/CD8 DN immunophenotype. In our region, this aberrant immunophenotype is not as rare as reflected in the literature, is overrepresented in the unusual clinical variants of MF, and does not seem to have prognostic significance. Like CD4(+) MF, the tumor cells represent memory T cells and in many cases express alpha/beta TCR, but unlike CD4(+) MF, they have a mostly cytotoxic phenotype. We suggest that CD4/CD8 DN MF should be recognized as another immunohistochemical variant of this lymphoma.

Adolescent↗
PubMed1983

Mycosis fungoides of the mastoid, middle ear, and CNS. Literature review of mycosis fungoides of the CNS.

Facial nerve paralysis developed in a man with tumor-stage mycosis fungoides (MF). Mastoidectomy disclosed that MF had involved the mastoid and middle ear. Meningeal lymphoma, confirmed by the finding of Sézary cells in the CSF, was subsequently established. Autopsy disclosed MF lymphoma in the leptomeninges, medulla, spinal cord, and cranial nerves. A unique feature was the formation of a communicating hydrocephalus. Case reports of 23 patients with MF of the CNS, including 21 autopsies, are reviewed. Practically all had tumor-stage or erythrodermic MF. Atypical mononuclear cells were found ante mortem in the CSF in eight patients. In contrast to other CNS lymphomas, bone marrow involvement was uncommon. Cranial, especially facial, nerve paralyses were often premonitory signs of meningeal lymphomas. Patients with MF having such symptoms should have cytologic examination of the CSF.

Adult↗
PubMed1980

Combination chemotherapy in the tumour stage of mycosis fungoides with cyclophosphamide, vincristine, vp-16, adriamycin and prednisolone (cop, chop, cavop): a report from the Scandinavian mycosis fungoides study group.

Combination chemotherapy with cyclophosphamide, vincristine, VP-16, adriamycin and prednisolone was given in 9 cases of mycosis fungoides in the tumor stage; 3 of these patients received COP, one received CHOP, and 5 CAVOP. Complete remission was obtained in one case and partial remission in 5, the response rate thus being 6/9. It was impossible, however, to maintain remission. The treatment was reduced or withdrawn owing to toxic effects in 4 cases. These forms of combination chemotherapy are beneficial in non-Hodgkin lymphomas but do not seem to be of value in the tumour stage of mycosis fungoides.

Adult↗
PubMed2005

[Hypopigmented mycosis fungoides].

INTRODUCTION: Hypopigmented mycosis fungoides is a rare clinical form of the disease, first described in 1978. Since then, only a hundred odd cases have been documented. CASE REPORT: A young 19 year-old African woman had presented with hypochromatic macules since the age of 9 and for which the diagnostic enquiry had finally concluded in hypopigmented mycosis fungoides. DISCUSSION: The particularities of this form of mycosis fungoides, grade I according to the TNM classification, are principally its onset in black-skinned persons or of Asian origin, and the age of early onset with a predominance of pediatric cases. Its course is indolent for several years and thus source of delayed diagnosis. The differential diagnoses to be evoked are basically vitiligo, achromate eczematides and parapsoriasisis. The post-inflammatory depigmentation, frequent in black-skinned subjects, is only an eliminating diagnosis. Its treatment is that of classical Grade I mycosis fungoides: topical mechlorethamine, phototherapy and topical corticosteroids.

Adult↗
PubMed2006

Molecular staging of lymph nodes from 60 patients with mycosis fungoides and Sézary syndrome: correlation with histopathology and outcome suggests prognostic relevance in mycosis fungoides.

BACKGROUND: Histological evidence of lymph node involvement is associated with a poor prognosis in patients with cutaneous T-cell lymphoma (CTCL). OBJECTIVES: To determine whether T-cell receptor (TCR) gene analysis is of prognostic relevance in CTCL. METHODS: TCR gene analysis was performed on lymph node specimens from 60 patients with mycosis fungoides (MF) and Sézary syndrome (SS) using a highly sensitive polymerase chain reaction (PCR)/single-strand conformational polymorphism analysis and results were correlated with skin, overall clinical and histological lymph node stages. RESULTS: The frequency with which a T-cell clone was detected in lymph node samples from patients with MF increased with skin stage, overall clinical stage and with the degree of histological involvement: six of 19 patients with uninvolved lymph nodes or limited histological involvement (LN0-2) and 13 of 14 patients with advanced histological involvement (LN3-4) had a detectable T-cell clone. In SS, 22 of 27 patients had a detectable lymph node T-cell clone. The clonal patients had a poorer prognosis than nonclonal patients (median survival from biopsy of > 72 months vs. 16 months for MF and 41.5 vs. 16.5 months for SS). Regression analysis confirmed that TCR gene analysis identifies a group of MF patients with a worse prognosis (P = 0.013). However, the molecular lymph node stage did not provide independent prognostic information in this cohort of patients in multivariate analysis. CONCLUSIONS: Molecular staging in MF and SS using a PCR-based method for TCR gene analysis provides additional information to histological examination. Specifically, this study identified a group of MF patients with early lymph node involvement with a poorer prognosis. However, a larger prospective study of patients with MF and early histological lymph node involvement is required to confirm whether molecular staging of lymph nodes provides independent prognostic information in a multivariate model.

Adolescent↗
PubMed1972

Mycosis fungoides.

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Mycosis Fungoides↗
PubMed2003

Mycosis on mycosis fungoides: zoophilic dermatophytosis selectively superimposed on pre-existing cutaneous T-cell lymphoma (mycosis fungoides) plaques.

We report a case of tinea corporis caused by a cattle-derived strain of Trichophyton mentagrophytes in a 44-year-old male affected by cutaneous T-cell lymphoma (CTCL, so-called mycosis fungoides). Fungal colonization of glabrous skin was strictly confined within pre-existing lymphomatous plaques. Either oral itraconazole or griseofulvin, or topical terbinafine were ineffective until the patient, who was treated with systemic retinoids and interferon-alpha for his CTCL, was shifted from leucocyte to lymphoblastoid interferon. The hypothesis that a local immunodisturbance could be responsible for the selective superimposition of tinea on CTCL lesions ('mycosis on mycosis'), and that such an immunodisturbance could be partially corrected by the interferon switch is discussed.

Antineoplastic Agents↗
PubMed1993

Transformation of mycosis fungoides: T-cell receptor beta gene analysis demonstrates a common clonal origin for plaque-type mycosis fungoides and CD30+ large-cell lymphoma.

It is well recognized that patients with classical mycosis fungoides (MF) may develop a large-cell lymphoma (LCL), a phenomenon known as "transformation." An unresolved issue regarding the transformation of MF is whether MF and LCL represent two separate lymphomas or whether they are derived from the same T-cell clone. We report the clinicopathologic, immunophenotypic, and immunogenotypic analysis of MF and LCL in a white male. He developed a rash at age 51 that was diagnosed at age 56 as clinical stage IA patch/plaque MF. After topical nitrogen mustard and total skin electron beam therapy for progressive generalized CD3+CD4+ patch/plaque lesions, he developed nodules of Ki-1+ (CD30+) T-LCL at age 72. Southern blot analysis of DNA digested with Bg/II or BamHI and probed with a T-cell receptor (TCR)-beta gene J beta 1/J beta 2 probe showed a single, identical rearranged band in both the MF and LCL skin lesions that had been obtained 4 years apart. V beta gene family--specific gene amplification assays demonstrated dominant V beta 6 PCR products in both types of lesions. These PCR products and lesional cDNA exhibited a monoclonal pattern when amplified with consensus TCR-beta gene VDJ joint primers and electrophoresed under conditions that allowed the resolution of small differences in size. Furthermore, sequence analysis of the V beta 6 PCR products amplified from both the MF and LCL lesions showed an identical nucleotide sequence involving V beta 6.4, D beta 1.1, J beta 1.2, and C beta 1. These findings indicate that both the MF and the LCL in this patient arose from the same T-cell clone and that these diseases developed at a stage in the clone's differentiation subsequent to rearrangement of the TCR-beta gene.

Aged↗