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PubMed2000

Unilesional mycosis fungoides: a study of seven cases.

BACKGROUND: Unilesional mycosis fungoides (MF) is a rare variant of cutaneous T-cell lymphoma (CTCL), characterized by a solitary lesion clinically and by histopathological features indistinguishable from those of MF, and typically having a benign course. OBJECTIVE: To describe the clinicopathological features of a series of patients with unilesional MF. METHODS: The records of cases of unilesional MF identified during a 10-year period in two medical departments were reviewed. RESULTS: There were 7 patients: 6 males, 1 female; mean age at the time of diagnosis: 32 years; age range: 12-58 years; 3 were below the age of 18 years. The mean pretherapy follow-up period was 9 years (range: 2-20 years). In 5 patients, the eruption consisted of a characteristic patch or plaque of MF located on the trunk or upper extremity; in 2 it was atypical - in 1, a hypopigmented patch, and in 1, a plaque indistinguishable from MF-associated follicular mucinosis. Histopathologically all the lesions exhibited features characteristic of MF, with CD3+ lymphoid cells. In 6 cases (with available fresh frozen tissue) there was a predominance of CD3+ CD4+ cells; in 1 of 5 there was deletion of CD7, and in 3 of 5 there was an overexpression of IL-2 receptor. T-cell receptor gamma gene rearrangement was found in 1 of 4 cutaneous lesions tested; in 2 cases it was found in the blood but not in the skin. Treatment modalities included localized electron beam, excision, topical nitrogen mustard or topical steroids and sunbathing, resulting in all cases in a sustained complete clinical response. In 1 patient, however, there were 2 local recurrences and in yet another patient there was distant cutaneous spread 3.5 years after therapy. CONCLUSIONS: Unilesional MF is a rare variant of CTCL, has heterogeneous clinical manifestations and can affect any age group, including children. The histopathological and immunophenotypical features are in general indistinguishable from those observed in multilesional MF. Although it is a unifocal event, there may occasionally be cutaneous spread with the appearance of noncontiguous lesions, even a long time after therapy. Whether all cases represent minimal-stage IA MF or whether some are actually T-cell pseudolymphoma remains to be clarified.

Adolescent↗

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↗

PubMed2002

Mycosis-fungoides-type cutaneous T cell lymphoma of the hands and soles: a variant causing delay in diagnosis and adequate treatment of patients with palmoplantar eczema.

BACKGROUND: The etiopathology of chronic eczematous lesions of the palms and/or soles remains elusive in a considerable proportion of patients. Accumulating evidence suggests that a rare variant of mycosis fungoides (MF)-type cutaneous T cell lymphoma (CTCL) restricted to the palms and/or soles may mimic common palmoplantar dermatoses. OBJECTIVE: In the present study, we analyzed the clinical and histological characteristics of 3 adult patients with preexisting nonclassified chronic palmoplantar eczema poorly responding to standard therapies. Palmar and/or plantar MF was eventually diagnosed. METHODS: The course of the disease, response to previous therapies and dermatological features are described, results of histochemical and immunohistochemical analyses are reported, including T cell receptor gamma gene rearrangement where obtainable. RESULTS: Onset of cutaneous lesions with broad clinical variation was experienced 2-10 years prior to diagnosis; conventional therapies led to short-time or partial remission only; except for 1 patient, the epidermotropic infiltrate was predominantly composed of CD4-positive cells; topical photochemotherapy seems to result in more durable responses. CONCLUSION: As therapeutic strategies for this disease variant differ from symptomatic standard treatment regimens, awareness of MF-type CTCL as a relevant differential diagnosis of palmoplantar eczema should be expanded to prevent delay in diagnosis and adequate therapy.

Aged↗

PubMed2003

Imiquimod induces complete clearance of a PUVA-resistant plaque in mycosis fungoides.

Imiquimod is a topical immune response modifier that binds to Toll-like receptors 7 and 8 and induces alpha-interferon. We locally applied imiquimod 5% cream (Aldara) daily for 2 weeks on a PUVA- and retinoid-resistant plaque on the face of a patient with mycosis fungoides. The diagnosis was based on clinical appearance, histology and molecular studies. Most of the disease manifestations showed a clear remission during PUVA therapy combined with low-dose retinoids. However, the plaque on the face was PUVA resistant. Local imiquimod treatment resulted in the complete clearance of the plaque. The patient has now been in complete remission for 12 months.

Adjuvants, Immunologic↗

PubMed2004

Mycosis fungoides progression and chronic solvent exposure.

The effect of repeated exposure to specific chemicals on the initiation or progression of mycosis fungoides (MF) remains unsettled. A patient with low-grade patch stage MF progressively developed MF plaques restricted to his arms, and a tumour on his right thigh. These areas were subject to repeated exposure to solvents. His thigh was indeed in close contact with his trousers pocket where he used to store a wiping rag drenched into white spirit and cellulosic thinner. Immunophenotyping these lesions revealed a dense LCA+, CD2+, CD3+, CD4+, CD5+, CD7+, CD45+, CD45RO+ T-cell infiltrate admixed with many factor XIIIa+ dendrocytes. T-cell receptor rearrangement analysis identified a monoclonal T-cell infiltrate. An internal work-up remained negative. Stopping further solvent exposure failed to improve his condition. Oral corticotherapy combined with low-dose interferon-alpha2a halted disease progression. This observation suggests that long-term solvent exposure may trigger MF and hasten its progression from the patch stage to the plaque and tumour stages.

Adrenal Cortex Hormones↗

PubMed2004

Fas gene mutations in mycosis fungoides: analysis of laser capture-microdissected specimens from cutaneous lesions.

Fas (APO-1/CD95) is a transmembrane protein which mediates programmed cell death (apoptosis). Cells with a mutated Fas gene are resistant to apoptosis and thus accumulate in lesional tissues. This might provide a basis for the development of neoplasias. Genomic DNA selectively obtained from Pautrier's microabscesses in 16 cases of mycosis fungoides (MF) using a laser capture microdissection method was analyzed. Fas gene mutations were detected in 3 of 16 cases of MF (18.8%); 1 was silent and 2 were missense mutations located in exon 9. One of the 2 missense mutations involved the death domain of the Fas gene, which is essential for apoptotic signal transduction. The missense mutations resulted in the substitution of Ala with Asp at codon 220 and Ile with Thr at codon 314. Mouse T cell lymphoma cells transfected with mutant genes were resistant to apoptosis induced by the anti-Fas antibody, indicating that the missense mutations found in MF were loss-of-function mutations, thus causing the accumulation of cells in the cutaneous lesions. These findings suggest that the accumulation of lymphoid cells with Fas mutations provides, in part, a basis for the development or maintenance of MF.

Adaptor Proteins, Signal Transducing↗

PubMed2005

Ichthyosiform mycosis fungoides: report of a case associated with IgA nephropathy.

We report a case of ichthyosiform mycosis fungoides (MF) associated with IgA nephropathy. Histological examination showed a dense atypical lymphocytic infiltrate admixed with epithelioid cells and giant cells in the dermis associated with the features of epidermotropism and folliculotropism. Reported cases of ichthyosiform MF are reviewed and histopathological characters of ichthyosiform MF are summarized. We suggest a histiocyte/dendritic-cell-rich infiltrate, or granulomatous features of infiltrate may be another characteristic of ichthyosiform MF. This case was associated with IgA nephropathy, which is uncommon. Such a presentation has never been reported in the literature.

Adrenal Cortex Hormones↗

PubMed2005

Mycosis fungoides in European Russia: no antibodies to human T cell leukemia virus type I structural proteins, but virus-like sequences in blood and saliva.

OBJECTIVE: Mycosis fungoides (MF) is the most frequent form of cutaneous T cell lymphoma (CTCL). Human T cell leukemia virus type 1 (HTLV-1) involvement in MF progression is a matter of debate. The goal of the investigation was to search for HTLV-1 markers in a group of MF patients from a nonendemic area to HTLV-1. MATERIALS AND METHODS: Fifty MF patients and 60 healthy donors from Moscow and the Moscow region were examined for HTLV-1 markers by Western blot, PCR, nested PCR, PCR/Southern hybridization, TaqMan real-time PCR and sequencing. RESULTS: Plasma samples from MF patients were repeatedly negative for antibodies to HTLV-1 structural proteins. HTLV-1 tax-related sequences (corresponding to the second exon) were found in blood from 20 of 50 MF patients and in 3 of 5 saliva specimens. Three of 8 sequenced tax-like amplimers were identical and 5 of 8 contained 1-2 substitutions. tax transcripts and antibodies to p40(tax) were detected in some 'PCR-tax'-positive MF patients. Defective HTLV-1 genomes were demonstrated in 2 of 50 MF patients. Phylogenetic analysis of the defective genome 5'-LTR sequence revealed a relationship with HTLV-1a sequences from the transcontinental subgroup of HTLV-1. CONCLUSIONS: HTLV-1 tax-like sequences were revealed in blood and for the first time in saliva from MF patients living in an HTLV-1 nonendemic region. Expression of tax-like sequences was confirmed by both reverse transcription PCR and Western blot.

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↗

PubMed1981

Neurological sequelae in mycosis fungoides. Report of a case.

Central nervous system (CNS) involvement in mycosis fungoides (MF) usually occurs in the presence of active skin lesions and infiltration of many visceral organs. A case of MF is presented, associated with a neurological syndrome, characterized by ataxia and motor-neuron disease. Autopsy revealed widespread infiltration of the CNS by MF, in the presence of only one minimal skin lesion and with no involvement of visceral organs. The importance of being aware of this discrepancy is stressed in the light of the possibility of radiotherapy in CNS involvement.

Aged↗

PubMed1983

Monoclonal antibody-defined peripheral blood T lymphocyte subpopulations in patients with mycosis fungoides.

A panel of monoclonal antibodies has been used to identify peripheral blood lymphocyte subpopulations in 13 patients with mycosis fungoides and in 13 normal subjects. An increase in the T6+ population and a decrease in the T8+ population were noticed in our patients. Their T4/T8 ratio was considerably augmented. In 4 patients reevaluated after treatment, marked changes in the relative proportions of T lymphocyte subsets had occurred. The possible pathogenetic significance of these immunological alterations is discussed.

Adolescent↗

PubMed1997

Extracorporeal photochemotherapy induces a significant increase in CD36+ circulating monocytes in patients with mycosis fungoides.

BACKGROUND: In patients with Sezary syndrome undergoing extracorporeal photochemotherapy it is currently thought that photodamage of a congruent number of pathogenic circulating T cells is a necessary condition for a specific anticlonal response to be induced against these cells by the immune system. However other mechanisms of action, such as the induction of release by photoirradiated monocytes of some cytokines, are thought to cooperate in the induction of a therapeutic response. OBJECTIVE: We investigated the effects of extracorporeal photochemotherapy on peripheral blood mononuclear cells in seven stage Ib cutaneous T cell lymphoma patients. METHOD: Samples of peripheral blood were taken before starting extracorporeal photochemotherapy (baseline), and before the 3rd and 6th cycle of treatment, and then incubated with specific monoclonal antibodies. RESULTS: The results showed a significant increase of CD8+ and CD36+ and a significant decrease of CD25+ in all the treated patients. CONCLUSION: Our results show that extracorporeal photochemotherapy is able to induce an increase of CD36+ cell in peripheral blood of patients with mycosis fungoides in its early stages. This subset of monocytes could be involved in the complex mechanism of action of extracorporeal photochemotherapy.

Aged↗

PubMed1996

Granulomatous mycosis fungoides presenting as sarcoidosis.

This report describes the case of a woman who developed cutaneous granulomatous infiltration during childhood that was initially considered to be sarcoidosis and, after 27 years of evolution, was determined to be caused by granulomatous mycosis fungoides transforming to Ki-1-positive anaplastic large-cell lymphoma of T-cell type. We discuss the differential diagnosis, especially granulomatous slack skin and the interest of immunohistochemical and genotypic studies in this case.

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↗

PubMed1993

Mycosis fungoides and eruptive epidermoid cysts: a unique response of follicular and eccrine structures.

A case of a patient who developed simultaneously mycosis fungoides (MF) and multiple, tiny eruptive cutaneous cysts on the face, neck and upper part of the trunk is reported. Histologically and immunohistochemically MF infiltrate was recognized not only in the upper part of the dermis and in the epidermis but also around and within the walls of cystic lesions. Furthermore, the eccrine structures were also involved by MF infiltrate. We emphasize that MF infiltrate can affect the follicular and eccrine structures inducing the formation of keratinous cysts. The clinical appearance of the facial lesions may be confused with Favre-Racouchot disease.

Aged↗

PubMed1991

Follicular mucinosis associated with mycosis fungoides.

We report a patient who developed erythematous indurated plaques with alopecia on the face and multiple well-demarcated infiltrated scaling lesions on the trunk and extremities. Skin biopsy showed a mucinous degeneration of the follicular outer root sheaths and sebaceous glands consistent with the diagnosis of follicular mucinosis. A histologic examination of a specimen from the left forearm showed microscopic changes of mycosis fungoides. The present case confirms the need of multiple skin biopsies in the evaluation of patients with follicular mucinosis.

Aged↗

PubMed1990

T cell receptor gene rearrangement analysis in mycosis fungoides and disseminated lymphocytoma cutis.

A cDNA from the CT beta 2 region of the T cell receptor gene was used as a probe to investigate the clonal composition of T cells in the skin lesions of 3 patients with mycosis fungoides (MF) and 1 patient with disseminated lymphocytoma cutis (DLC). All of these skin lesions showed helper/inducer T cell dominant infiltration. Clonal T cell populations, as evidenced by rearranged DNA bands, were demonstrated in all the skin lesions of the 3 patients with MF, but not in the lesion of the patient with DLC. The patients with MF appeared to have the same gene rearrangement pattern in DNA obtained from separate lesions, providing evidence that the T cells in both sites were derived from the same clone. Our results show that this gene rearrangement analysis helps to diagnose difficult cases of lymphoproliferative disorders.

Adult↗

PubMed1987

Association of vesiculobullous eruptions with mycosis fungoides.

Mycosis fungoides (MF) is, on extremely rare occasions, associated with vesiculobullous eruptions. This report describes clinical, histological and immunohistochemical findings of a case of MF with vesiculobullous lesions. Characteristic features of our case included: bullous lesions evolving rapidly in 12-24 h with severe itch on erythematous and normal-appearing skin; a positive Nikolsky sign; an intraepidermal blister; direct and indirect immunofluorescence stainings were all negative with IgG, IgA, IgM and C3, and atypical lymphocytes in the infiltrates were of helper/inducer T cell type.

Blister↗