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PubMed2001

Are alcohol intake and smoking associated with mycosis fungoides? A European multicentre case-control study.

The incidence of mycosis fungoides (MF) is low, and the aetiology of the disease is unknown. The aim of this study was to investigate whether wine consumption protects against the disease and whether smoking constitutes a risk factor. This paper is part of the European Rare Cancers Study that tries to determine the risk factors for seven selective rare cancers, including mycosis fungoides, involved in the development of cancer. A multicentre case-control study was conducted in six European countries. Only incident cases with confirmed histology were included in the analysis which include a total of 76 cases of MF and 2899 controls. Wine intake had no protective effect; on the contrary the consumption of more than 24 g of alcohol per day was associated with a high risk of MF (odds ratio (OR)=3.02, 95% confidence interval (CI), 1.34-6.79), after adjusting for centre, country, age, sex and education. There was a dose-dependent increase in the risk of MF with increased smoking habits, albeit the observed trend was not statistically significant. A combined exposure to high tobacco and alcohol use yielded a significantly increased risk factor for MF (P=0.0073). Alcohol intake was associated with MF.

Adult↗
PubMed2000

Expression of VCAM-1, ICAM-1, E-selectin, and P-selectin on endothelium in situ in patients with erythroderma, mycosis fungoides and atopic dermatitis.

BACKGROUND: Erythroderma may result from different causes. At present it is unclear whether the patho-mechanisms that lead to these different types of erythroderma are identical or different. Adhesion molecules and their ligands play a major role in endothelial-leukocyte interactions, which affect the binding, transmigration and infiltration of lymphocytes and mononuclear cells during inflammation, injury, or immunological stimulation. The aim of this study was to investigate the adhesion molecule expression on endothelial cells in erythroderma in situ. METHODS: Snap-frozen skin biopsy specimens from 23 patients with erythroderma were studied. Eight had idiopathic erythroderma, 5 erythrodermic atopic dermatitis, 4 Sézary syndrome and 6 had erythroderma from miscellaneous causes. As a control we studied skin specimens from 10 patients with mycosis fungoides, 5 patients with atopic dermatitis and 5 healthy non-atopic volunteers. To determine adhesion molecule expression on endothelial cells in situ, sections were immuno-histochemically double stained with biotinylated Ulex Europaeus agglutinin 1 as a pan-endothelial cell marker, and for the adhesion molecules VCAM-1, ICAM-1, E-, and P-selectin. All double- and single-stained blood vessels in the dermis were counted. RESULTS: Mean endothelial expression in erythroderma was as follows: VCAM-1 51.4%, ICAM-1 70.1%, E-selectin 43.5%, and P-selectin 52.6%. There was no statistical difference between different groups of erythroderma. Mean expression of all adhesion molecules tested, was in Sézary syndrome higher than in mycosis fungoides albeit not significant. In erythrodermic atopic dermatitis only VCAM-1 expression was significantly higher than in lesional skin of atopic dermatitis. No differences were observed in expression of the other three adhesion molecules. CONCLUSIONS: There is no difference regarding adhesion molecule expression on endothelial cells between different types of erythroderma.

Cell Adhesion Molecules↗
PubMed2001

Follicular mycosis fungoides: a case report and review of the literature.

BACKGROUND: Follicular mycosis fungoides is an unusual variant of mycosis fungoides (MF). Unlike classic MF where atypical lymphocytes show a predilection for the epidermis (epidermotropism), follicular MF displays a malignant lymphocytic infiltrate tropic for hair follicles (folliculotropism). This malignant lymphocytic infiltrate results in follicular disruption typically manifesting clinically as plaques, comedones and follicular papules. METHODS: This report describes a 40-year-old patient with follicular MF presenting as nodules on the face and chest. Histologic examination of the patient's biopsy revealed a folliculocentric infiltrate of atypical lymphocytes with sparing of the epidermis. RESULTS AND CONCLUSIONS: Our case is discussed in the context of previously reported cases of follicular MF; we also include a review of all cases of follicular MF published to date that meet the strict criterion for this diagnosis.

Administration, Topical↗
PubMed1998

Mycosis fungoides presenting as keratosis lichenoides chronica.

We report a patient with a 17-year history of reticulated keratotic papules on the trunk and limbs, and telangiectatic eruption on the face, in whom the diagnosis of keratosis lichenoides chronica was first established. However, the biopsies showed an epidermotropic infiltrate of small irregular CD4+ lymphocytes, and detection of a T-cell clone in the lesions by polymerase chain reaction confirmed the diagnosis of mycosis fungoides. Thus, keratosis lichenoides chronica can be an unusual and potentially misleading presentation of mycosis fungoides.

Adult↗
PubMed2000

Pityriasis lichenoides-like mycosis fungoides in children.

We report three children with clinical features of pityriasis lichenoides (scaly red to brown papules and macules) in whom there were histopathological findings of mycosis fungoides (disproportionate epidermotropism, Pautrier's microabscesses, and wiry and coarse collagen bundles). Immunohistochemical staining revealed a prevalence of T lymphocytes in the infiltrate. T-cell receptor gene rearrangement analysis in lesional skin demonstrated rearrangement of the gamma chain in all cases. Human T-cell lymphotropic virus type 1 serology was negative in the two patients in whom this test was performed. Thus, lesions resembling pityriasis lichenoides can be an unusual and potentially misleading presentation of mycosis fungoides.

Child↗
PubMed2000

Mycosis fungoides and chronic lymphocytic leukaemia--composite T-cell and B-cell lymphomas presenting in the skin.

Composite lymphomas involving cutaneous B-cell and T-cell lymphomas are very uncommon. We report here the unique circumstance of a patient with mycosis fungoides (primary cutaneous T-cell lymphoma) who later developed chronic lymphocytic leukaemia (B-cell lymphoproliferation, B-CLL), which presented in the skin (leukaemia cutis) as a composite lymphoma affecting an earlobe. The presence of both lymphoproliferative disorders was confirmed with immunophenotyping and the finding of both immunoglobulin gene rearrangements and T-cell receptor gene rearrangements in the ear and the same T-cell receptor gene rearrangement in a plaque lesion of mycosis fungoides on the arm.

Aged↗
PubMed2001

Interleukin-15 expression in cutaneous T-cell lymphoma (mycosis fungoides and Sézary syndrome).

BACKGROUND: Cytokines are of potential importance in the pathogenesis of cutaneous T-cell mediated disorders, including cutaneous T-cell lymphoma (CTCL). OBJECTIVES: To compare interleukin (IL)-15 expression in certain inflammatory cutaneous diseases, with that in CTCL (mycosis fungoides and Sézary syndrome). METHODS: IL-15 mRNA and protein expression were examined by in situ hybridization and immunohistochemistry, respectively, on formalin-fixed, paraffin-embedded biopsies of normal human skin, atopic dermatitis, psoriasis, parapsoriasis and CTCL. RESULTS: Despite similar expression of IL-15 mRNA, we found differences in IL-15 protein expression between normal human skin, atopic dermatitis and psoriasis on the one hand, and parapsoriasis and CTCL on the other. IL-15 protein expression was not detected in normal human skin, atopic dermatitis or psoriasis, but was detected, mainly at low levels but in a few patients at higher levels, in epidermal keratinocytes in parapsoriasis, mycosis fungoides and Sézary syndrome. CONCLUSIONS: Induction of keratinocyte IL-15 expression appears to be a feature of CTCL. The factors stimulating such an expression remain unknown.

Antineoplastic Agents↗
PubMed2001

T-cell depletion and autologous stem cell transplantation in the management of tumour stage mycosis fungoides with peripheral blood involvement.

Nine patients with tumour stage mycosis fungoides (MF) have been entered into a pilot study of T-cell depletion and autologous stem cell transplantation (SCT). Eight patients had detectable rearrangements of the T-cell receptor (TCR) gamma-gene demonstrated by polymerase chain reaction (PCR)/single-stranded conformation polymorphism (SSCP) in the peripheral blood. The median age was 47 years and the median duration of disease before SCT was 61 months; Peripheral blood progenitor cells were mobilized using high-dose etoposide (1.6 g/m2) and granulocyte colony-stimulating factor (G-CSF). The apheresis products underwent rigorous T-cell depletion with immunomagnetic methods. Double CD34-positive and CD4/CD8-negative selection achieved a median reduction of 3.89 log of T cells. All nine patients have been transplanted. Conditioning included carmustine (BCNU), etoposide and melphalan (BEM) in seven patients and total body irradiation plus etoposide or melphalan in two. Eight patients engrafted promptly and one patient died of septicaemia. All survivors entered complete remission. Seven patients have relapsed at a median of 7 months (2-14) post SCT. However, most patients have relapsed into a less aggressive stage, which has responded to conventional therapy. Four out of seven evaluable patients had detectable TCR rearrangements in the T-cell depleted graft. A T-cell clone was also detected in the peripheral blood before relapse in four cases. Autologous SCT is feasible, safe and can result in complete remission in a significant proportion of patients with tumour stage mycosis fungoides. Despite a short relapse-free survival, most patients achieved good disease control at the time of relapse.

Adult↗
PubMed2002

Coexistence of CD30-positive anaplastic large cell lymphoma and mycosis fungoides.

Primary cutaneous T-cell lymphomas, including lymphomatoid papulosis, mycosis fungoides and CD30+ anaplastic large cell lymphoma (ALCL) overlap clinicopathologically and form part of a spectrum of lymphoproliferative disorders. There have been several case reports of these diseases coexisting. We describe a 59-year-old Korean man who presented with a recurrent, solitary CD30+ ALCL of 25 years' duration as well as patch stage mycosis fungoides of 11 years' duration. Such occurrences may represent different clinical manifestations of the same clonal T-cell abnormality, and provide further insight into the pathogenesis of these related disorders.

Humans↗
PubMed2002

Mycosis fungoides involving the oral cavity.

Mycosis fungoides is a malignant T-cell lymphoproliferative disease with a predilection for cutaneous involvement. Extracutaneous disease is uncommon and oral mucosal involvement is rare. We describe a case of mycosis fungoides involving the hard palate treated with radiotherapy. The relevant literature on this topic is reviewed.

Aged↗
PubMed2002

Infrequent Fas mutations but no Bax or p53 mutations in early mycosis fungoides: a possible mechanism for the accumulation of malignant T lymphocytes in the skin.

Mycosis fungoides (MF) is the most frequent manifestation of cutaneous T cell lymphoma but its cause and pathophysiology remain unclear. Because progression of lesions is characteristically slow, we hypothesized that mycosis fungoides originates from an accumulation of lymphocytes due to defective apoptosis of skin homing T lymphocytes. In this study, we investigate possible alterations of three molecules regulating apoptosis, i.e., Fas antigen, Bax, and p53, at the genomic level in skin lesions from 44 patients with MF, as Fas mediates one of two major pathways for apoptosis of activated T cells. Fas mutations were found in six patients using a polymerase chain reaction and single-strand conformational polymorphism method followed by cloning and sequencing of abnormal polymerase chain reaction products. The mutations predict for defective transmission of the death signal in three cases. Immunohistochemistry demonstrated the lack of Fas protein expression on dermal lymphocytes in one case with Fas gene mutation predicting for a truncated death domain, whereas Fas protein was expressed by dermal lymphocytes in the other investigated cases. By contrast, no mutations of Bax or p53 were found, whereas immunohistochemistry demonstrated increased p53 expression in the nucleus of basal keratinocytes above the neoplastic infiltrate in some MF cases. These results support the hypothesis that Fas defects may play a role in the pathogenesis of MF.

Apoptosis↗
PubMedDate not supplied

Hypopigmented mycosis fungoides in childhood and adolescence.

We present a case of purely hypopigmented mycosis fungoides of 8-years duration in an 18-year-old woman who responded readily to psoralen plus ultraviolet A (PUVA) treatment. The literature pertaining to hypopigmented mycosis fungoides is reviewed.

Adolescent↗
PubMed2000

Mycosis fungoides d'embleé: CD30-negative cutaneous large T-cell lymphoma.

The d'embleé variant of mycosis fungoides initially described patients with a rapid onset of tumors without progression through patch- and plaque-stage disease. We report a case of a patient with the clinical presentation of mycosis fungoides d'embleé and correlate the histologic and immunophenotypic data with those of a more updated classification scheme.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed2002

Invisible mycosis fungoides: a diagnostic challenge.

We describe a 76-year-old woman who had persistent generalized pruritus as the only cutaneous manifestation of a cutaneous T-cell lymphoma (mycosis fungoides). No cutaneous lesions were observed throughout the patient's course. Skin biopsy specimens obtained from normal-looking pruritic skin revealed a discrete perivascular lymphocytic infiltrate in the upper dermis and focal intraepidermal clusters of atypical lymphoid cells (Pautrier's microabscesses). PCR analysis of TCR-gamma gene disclosed a monoclonal T-cell rearrangement. Sequencing of the PCR monoclonal product identified the J(8)V(2)C(2) TCR gene rearrangement. This observation illustrates the existence of a peculiar and exceedingly rare form of mycosis fungoides characterized only by persistent pruritus unresponsive to several therapeutic approaches. The diagnostic difficulties of this rare variant are stressed.

Aged↗
PubMed2002

Cold urticaria in a patient with mycosis fungoides.

We report what we believe to be the first documentation of a patient with both cold urticaria and mycosis fungoides. The patient described a marked worsening of his long-standing lesions of mycosis fungoides at the same time as the onset of cold sensitivity. We believe this suggests a possible association between these 2 rare diseases.

Adult↗
PubMed2003

A complete and durable response to denileukin diftitox in a patient with mycosis fungoides.

We describe a patient with stage IA mycosis fungoides treated with denileukin diftitox as part of a randomized phase III trial. The patient had a rapid and complete response and has remained in remission for more than 5 years without maintenance therapy. Although denileukin diftitox is not usually considered for patients with early-stage mycosis fungoides, our experience provides anecdotal evidence that fusion toxin therapy may be worth considering as a treatment for patients with stage I disease.

Aged↗
PubMed2003

Purely follicular mycosis fungoides without mucinosis: report of two cases with review of the literature.

Follicular lesions can either associate with typical patch-/plaque-type mycosis fungoides or, more rarely, be the only clinical manifestation of the disease. We describe 2 adult patients who presented with alopecia and disseminated follicular erythematous papules, and comedones and cysts, respectively. In both patients, histology showed a folliculotropic infiltrate of atypical lymphocytes that spared the epidermis, in the absence of follicular mucinosis. Molecular genetic analysis confirmed the oligo/monoclonal nature of the T-cell infiltrate. Reported cases of purely follicular mycosis fungoides without mucinosis are reviewed.

Aged↗
PubMed2003

Folliculotropic mycosis fungoides with central nervous system involvement: demonstration of tumor clonality in intrafollicular T cells using laser capture microdissection.

BACKGROUND: Folliculotropic mycosis fungoides (MF) is a rare variant of cutaneous T-cell lymphoma, MF type, characterized by atypical lymphocytes preferentially infiltrating the hair-follicle epithelium relative to the epidermis. OBSERVATIONS: We describe a rare case of folliculotropic MF involving the central nervous system. This is also the first case in which laser capture microdissection was used to show that the atypical lymphocytes within the hair-follicle epithelium were part of the same tumor clone present in other tissue compartments. CONCLUSIONS: In reviewing the literature describing atypical lymphocytes infiltrating hair-follicle epithelium relative to the epidermis, we encourage the use of the term folliculotropic mycosis fungoides. Our case also supports previous findings that central nervous system involvement can occur in advanced MF. The successful procurement and analysis of atypical lymphocytes from hair-follicle epithelium by laser capture microscopy ushers in a new era in molecular diagnostics.

Aged↗