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PubMed1985

Clonal rearrangements of T-cell receptor genes in mycosis fungoides and dermatopathic lymphadenopathy.

Histologic diagnosis of mycosis fungoides may be difficult, especially in lymph nodes that show changes frequently associated with chronic skin disease. As an alternative approach to diagnosis, we have analyzed the configuration of DNA for the beta T-cell receptor genes in biopsy tissues from 14 patients with mycosis fungoides. Clonal rearrangements of these genes were found in each specimen tht contained histologically unambiguous mycosis fungoides. Clonal rearrangements were also found in seven of nine lymph nodes removed from patients with mycosis fungoides and considered histologically to contain only benign lymphadenopathy. Matching rearrangements of beta T-cell receptor genes were detected in benign lymph nodes and histologically involved tissues when paired specimens were available from the same cases. Our findings provide molecular evidence for the clonal T-cell origin of mycosis fungoides and indicate the high incidence of extracutaneous disease in patients with palpable lymphadenopathy. In addition, this study demonstrates that the detection of rearranged T-cell receptor genes can be a sensitive and practical method for the diagnosis and characterization of T-cell neoplasms.

Clone Cells↗
PubMed1997

The polymerase chain reaction in the diagnosis of early mycosis fungoides.

The earliest or patch stage of mycosis fungoides may present diagnostic difficulty both clinically and pathologically. The present study of the polymerase chain reaction (PCR) as a diagnostic tool in early mycosis fungoides was therefore undertaken, using a rapid PCR method for the detection of gamma- and beta-chain T-cell receptor (TCR) gene rearrangements in routine formalin-fixed, paraffin-embedded histological sections. Forty-two biopsies were studied from 26 patients with mycosis fungoides. Twenty-three skin biopsies with a clinicopathological diagnosis of early, or patch stage, mycosis fungoides were investigated. Of these, 18 (78 per cent) showed TCR-gamma or both beta- and gamma-chain TCR gene rearrangements. TCR gene rearrangements were shown in seven of the 14 plaque stage lesions (50 per cent) and also in the single case of tumour stage disease. Where gene rearrangements were identified, these were identical in all biopsies from an individual patient, irrespective of the site of the lesion, the disease stage, or the time lapse between the biopsies. The PCR is therefore a highly sensitive technique, which can be performed on routine pathological material, in cases where the diagnosis of early mycosis fungoides cannot be made with certainty on conventional histopathological and immunohistochemical grounds.

Adult↗
PubMed1989

Combined total body X-ray irradiation and total skin electron beam radiotherapy with an improved technique for mycosis fungoides.

Twelve consecutive patients with advanced stage mycosis fungoides (MF) were treated with combined total body X ray irradiation (TBI) and total skin electron beam radiotherapy (EBRT). Six had generalized plaque disease and dermatopathic nodes, three had tumor stage disease and node biopsy positive for mycosis fungoides, and three had erythroderma/Sezary syndrome. The treatment regimen consisted of split course total body X ray irradiation, given in twice weekly 15 cGy fractions to 75 cGy, then total skin electron beam radiation therapy given in once weekly 400 cGy fractions to a total dose of 2400 cGy. Underdosed areas and areas of greatest initial involvement were boosted 400 cGy twice weekly for an additional 1200 cGy. This was followed by a second course of total body X ray irradiation, to a total dose of 150 cGy. The total skin electron beam radiotherapy technique is a modification of an established six position EBRT technique for mycosis fungoides. Measurements to characterize the beam with and without a lexan scattering plate, demonstrated that the combination of no-plate beams produced better dose uniformity with a much higher dose rate. This improved technique is particularly advantageous for elderly and/or frail patients. Nine (75%) of the 12 patients achieved complete response (CR). The other three had significant improvement with greater than 80% clearing of their disease and resolution of symptoms. All six patients with generalized plaque disease achieved complete response and remained free of disease from 2 to 16 months. Two of three node positive patients also achieved complete response; one, with massive biopsy-documented mycosis fungoides nodal disease and deep open tumors, remained relapse-free over 2 years. Only one of the three patients with erythroderma/Sezary syndrome achieved a complete response, which was short lived. Therapy was well tolerated. No significant hematological toxicity occurred. Although total body X ray irradiation and total skin electron beam radiotherapy produced excellent palliation of patients with advanced stage mycosis fungoides, new strategies to provide more effective systemic treatment are needed.

Adult↗
PubMed1998

[Granulomatous mycosis fungoides histologically simulating cutaneous sarcoidosis].

BACKGROUND: Granulomatous mycosis fungoïdes is an uncommon mycosis fungoïdes. We report a misleading case initially thought to be cutaneous sarcoidosis. CASE REPORT: A 34-year-old man developed stationary erythematous plaques on the trunk and members. Pathology reported a sarcoidal aspect. No extracutaneous sarcoidal lesions were found. The lesions resolved with puvatherapy. Five years later, the patient developed a voluminous skin tumor in the scapular area. Pathology reported granulomatous mycosis fungoides. Radiotherapy was given. Other plaques developed on the skin with the same histological aspect as initially and disappeared after local applications of chlormethine. During the clinical course, the patient developed visceral localizations of mycosis fungoides in the abdomen and pelvic nodes which responded to polychemotherapy. DISCUSSION: Histologically, granulomatous mycosis fungoïdes is defined as the association of epidermotropic T-cell lymphoma with epitheloid and giant cell dermal granulomas and no necrosis. Variants may be misleading when the epitheloid and giant cell aspect predominates, as in our observation. An analysis of 28 reported cases showed no disinction between the clinical features. Clinical course or treatment for granulomatous fungoides mycosis or classical fungoid mycosis. Extracutaneous sarcoidosis is rarely associated. Most likely, there is a tissue reaction to the lymphona.

Adult↗
PubMed2003

Chemokine receptor expression on neoplastic and reactive T cells in the skin at different stages of mycosis fungoides.

We analyzed the expression of 13 chemokine receptors in mycosis fungoides, in order to assess the contribution of chemotaxis to the pathogenesis of the disease. Material from skin biopsies of six patients with early disease and six patients at the tumor stage of mycosis fungoides was analyzed by immunohistochemistry and partly also by flow cytometry. The receptors CCR1, CCR2, CCR3, CCR5, CCR6, CXCR1, CXCR2, CXCR5, and CX3CR1 were rarely and inconsistently detected in lesional skin and thus their participation in mycosis fungoides could largely be ruled out. In contrast, CCR4, CXCR3, and CXCR4 were substantially expressed on both mycosis fungoides cells and the surrounding reactive T cells in the early patch and plaque stages of the disease, indicating an involvement of these chemokine receptors in the disease process. In the tumor stage of mycosis fungoides, we interestingly observed a loss of a relevant chemokine receptor in four out of six patients. In three patients CXCR3 and in one patient CCR4 was absent on tumor mycosis fungoides cells, whereas the reactive T cells showed normal levels of expression. Within these samples, tumor mycosis fungoides cells exhibited high levels of CCR7, a chemokine receptor central for the entry of T cells to lymphatic tissue. Taken together, our data suggest that the loss of one or more of the chemokine receptors involved in the homing of the mycosis fungoides cells to the skin may trigger the latent potential of these cells to metastasize into regional lymphatic tissue.

Adult↗
PubMed1987

Mycosis fungoides beginning in childhood and adolescence.

Mycosis fungoides is a form of cutaneous T cell lymphoma usually occurring in mid to late adulthood. We report 12 patients with mycosis fungoides whose eruption began before age 20 years, including one patient with histologic documentation at 5 years of age. The onset of mycosis fungoides during the first two decades may be more common than is generally recognized and should be included in the differential diagnosis of chronic dermatoses in children and adolescents.

Adolescent↗
PubMedDate not supplied

A case of hypopigmented mycosis fungoides in a young Caucasian boy.

Hypopigmented mycosis fungoides is a variant of mycosis fungoides characterized by the presence of hypopigmented patches as the sole manifestation of the disease. It has been described almost always in young black or dark-skinned patients. The only white patient described was a 64-year-old woman who not only had hypopigmented lesions, but also nodular lesions with lymphadenopathy. We describe hypopigmented lesions arising in a white boy 12 years of age, born in northern Italy, without any foreign ancestors. The microscopic alterations, with epidermotropism, the immunologic markers, the negativity of T-cell receptor gene rearrangement, and the good response to PUVA therapy correspond to the main findings in black patients with this disease. Long-term follow-up of these patients is important to obtain better knowledge of the natural history of the disorder. Hypopigmented mycosis fungoides must now be included in the differential diagnosis of hypopigmented macular lesions not only in black or dark-skinned patients but also in white patients.

Antigens, CD↗
PubMed1984

Death due to splenic rupture in suppressor cell mycosis fungoides: a case report.

A case of mycosis fungoides in which pathologic rupture of the spleen led to intraperitoneal hemorrhage and death is described. To our knowledge, splenic rupture has not been reported previously as a cause of death in mycosis fungoides. Immunologic studies demonstrated that the neoplastic cell was a suppressor/cytotoxic T-cell. In most cases of mycosis fungoides or the Sézary syndrome, the neoplastic cell has been a helper/inducer T-cell. This case was very aggressive clinically with prominent visceral involvement and suggests that mycosis fungoides may be clinically diverse as well as immunologically heterogeneous.

Aged↗
PubMed2001

[Follicular mycosis fungoides (FMF): a rare disease].

Follicular mycosis Fungoides (FMF) was first described in 1924. Since the first description, 22 patients with this special form of mycosis fungoides have been published. Clinical features include epidermal cysts as well as follicular papules, nodules and hyperkeratoses. FMF can be confused with acneiform dermatoses. In most cases, infiltrated plaques typical for cutaneous T cell lymphoma are also present. Histology shows a monomorphic CD4+ T cell infiltrate. Treatment and prognosis are similar of those of classical mycosis fungoides. We present one patient with FMF and review the literature of all published cases.

Acitretin↗
PubMed1996

Detection of low-level tumor cells in allergic contact dermatitis induced by mechlorethamine in patients with mycosis fungoides.

Two patients with histologically proven mycosis fungoides, a malignancy of phenotypically mature T cells, received a topical challenge with mechlorethamine to areas of clinically uninvolved skin to exclude possible hypersensitivity reactions to this chemotherapeutic agent. In both patients, allergic contact dermatitis (ACD) developed at the sites of the application and resolved completely after withdrawal of mechlorethamine. The lesions were biopsied and analyzed for the presence of clonal T-cell receptor (TCR)-gamma gene rearrangements using two polymerase chain reaction (PCR)-based assays involving denaturing gradient gel electrophoresis (PCR/DGGE) and ribonuclease protection analysis (PCR/RPA). The former method has a clonal detection threshold of 10(-3)-10(-2), while the latter has a sensitivity of 10(-5). In both cases, the ACD lesions were polyclonal by PCR/DGGE. In contrast, PCR/RPA detected tumor-specific TCR-gamma gene rearrangements in these same lesions. This indicates that the ACD lesions contained tumor cells at a density within the 10(-5)-10(-2) range. Analysis of peripheral blood mononuclear cells from both patients failed to detect the malignant clone and showed the same result as blood from four normal individuals. The normal skin from one skin patient also lacked detectable TCR-gamma gene rearrangements. These results indicate that mycosis fungoides tumor are present within ACD lesions induced in mycosis fungoides patients and that this phenomenon does not appear to be due to the ubiquitous presence of detectable levels of these tumor cells in the blood or skin. These findings might be explained by nonspecific recruitment of malignant T cells to sites of local inflammation mediated by non-neoplastic antigen-specific T cells. Alternatively, they might be due to the local proliferation of very rare tumor cells in apparently normal skin in response to cytokines generated during the ACD reaction. In either case, the present study offers evidence that the malignant cells in myosis fungoides retain at least some capability of responding in vivo to physiologic stimuli.

Adult↗
PubMed2000

Fibromucinous T-cell lymphoma: a new clinicopathologic variant of mycosis fungoides?

We report a case of mycosis fungoides associated with extensive dermal fibrosis and mucin deposition. The patient developed indurated plaques with diffuse tightening of the skin reminiscent of the sclerosing disorder scleromyxedema, which was later associated with nodules and lymphadenopathy. Skin biopsies showed diffusely thickened collagen bundles in the dermis and mucin deposition with a dense infiltrate of atypical lymphocytes with an immunophenotypic pattern indicative of mycosis fungoides. In our opinion, these clinical and histopathologic features are unusual for mycosis fungoides and can be construed as a distinct fibromucinous variant. Alternatively, this may represent a fibrosing reaction pattern similar to that described with systemic T- and B-cell lymphomas or a variety of inflammatory disorders.

Antigens, Neoplasm↗
PubMed1982

Brain biopsy in the diagnosis of cerebral mycosis fungoides.

A case of cerebral mycosis fungoides co-existing with progressive multifocal leucoencephalopathy presented with dementia. Brain biopsy established the diagnosis of mycosis fungoides after cerebrospinal fluid examinations and computerised tomographic scanning of the brain produced non-specific abnormalities.

Biopsy↗
PubMed1980

Familial mycosis fungoides revisited.

Familial examples of mycosis fungoides are rarely seen; to my knowledge, only two carefully documented examples have been reported. This report uniquely involves a father and daughter. The disease onset in the daughter occurred 24 years after the father's death from mycosis fungoides.

Aged↗
PubMed1981

Demethylchlortetracycline and griseofulvin as examples of specific treatment for mycosis fungoides.

Long-term control of mycosis fungoides in the tumour stage was achieved in one patient by daily demethylchlortetracycline therapy. On two occasions widespread nodules and tumours completely involuted within 1 month of initiating this therapy. A second patient showed complete resolution of his plaque stage mycosis fungoides after the institution of oral griseofulvin therapy for a chronic fungus infection. The disease reappeared upon stopping the griseofulvin and involuted upon its resumption. The observations are interpreted as evidence that reduction or eradication of a persistent bacterial or fungal antigen may have a remarkable ameliorative effect in selected mycosis fungoides patients.

Demeclocycline↗
PubMed2005

[Follicular mycosis fungoides, comedo-like and cystic].

Follicular mycosis fungoides is an infrequent variant of mycosis fungoides. It has classically been defined by the presence of an atypical lymphoid infiltrate around and in the follicular epithelium with little or no epidermotropism, and no follicular mucin deposits. The fact that there are cases with epidermal involvement and/or follicular mucinosis means that some uniform diagnostic criteria are necessary. We describe two cases of follicular mycosis fungoides with follicular mucinosis and with varying degrees of associated epidermotropism.

Adult↗
PubMed1983

Mycosis fungoides: manifestations on computed tomography.

Mycosis fungoides is a malignant lymphoma of the skin that causes intense erythema, plaques, edema, and induration. The primary computed tomographic findings of mycosis fungoides, i.e., skin thickening, tumorous infiltration, edema of the soft tissues, and lymphadenopathy, are presented in two patients.

Edema↗
PubMed1977

DNA content of mycosis fungoides cells.

DNA content of mycosis infiltrate cells was measured in 5 patients with the Feulgen cytophotometric method in the plaque and tumor stages. In addition, the infiltrate cells were differentiated cytochemically into histiocytes and atypical lymphoid cells with NaF-sensitive naphthol-AS-D-acetate esterase. In no case was an aneuploid stem line demonstrated. However, increasing duration of the tumor stage was associated with a larger proportion of tetraploid and octoploid cells. The DNA histograms also exhibited a local proliferation of atypical lymphoid cells. This proliferation was arrested by cytostatic therapy. Comparison with semi-thin-sections of tumor tissue showed that the mycosis fungoides cells are atypical lymphoid cells. These DNA measurements do not contradict the concept of limited aneuploidy, as reported in cytogenic studies. Thus mycosis fungoides fits in with the DNA distribution pattern in the group of lymphomas.

Adult↗