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PubMed2001

Mycosis fungoides bullosa: report of a case and review of the literature.

Mycosis fungoides, the most common type of cutaneous T-cell lymphoma, can manifest in a variety of clinical and histologic forms. Presentation with vesiculobullous lesions is extremely rare. We report the ninth documented case of mycosis fungoides bullosa in which other concomitant autoimmune blistering diseases were ruled out by negative immunofluorescence. All previously reported cases in the world literature since the first in 1887 are reviewed. We recommend the following defining criteria for the disease: (1) clinically apparent vesiculobullous lesions, with or without typical mycosis fungoides lesions (patches, plaques, tumors); (2) typical histologic features of mycosis fungoides (atypical lyphoid cells, epidermotropism, Pautrier's microabscesses) with intraepidermal or subepidermal blisters; (3) negative immunofluorescence (both direct and indirect, if possible) to rule out concomitant autoimmune bullous diseases; (4) negative evaluation for other possible causes of vesiculobullous lesions (eg, medications, bacterial or viral infection, porphyria, phototherapy).

Aged↗

PubMed1997

Folliculotropic mycosis fungoides with large-cell transformation presenting as dissecting cellulitis of the scalp.

Follicular mycosis fungoides (MF) is a rare variant of cutaneous T-cell lymphoma (CTCL) in which malignant lymphocytes preferentially infiltrate hair follicles. This report describes a patient with follicular mycosis fungoides presenting in a manner similar to dissecting cellulitis of the scalp with nonhealing, draining nodular lesions. Follicular mucinosis associated with folliculotropic mycosis fungoides resulted in follicular disruption and deep dissecting cellulitis. Large-cell transformation of CTCL was present in the initial diagnostic scalp and axillary lymph node specimens. The patient died from progressive CTCL 9 months following initial diagnosis despite electron beam radiation, topical mechlorethamine, interferon-alpha, and systemic chemotherapy. This case indicates that large-cell transformation of folliculotropic mycosis fungoides is an aggressive form of CTCL, and that folliculotropic mycosis fungoides can give rise to lesions which resemble dissecting cellulitis of the scalp. Upregulation of intercellular adhesion molecule-1 (ICAM-1) on follicular epithelium adjacent to lymphocyte function-associated antigen-1 (LFA-1)-positive folliculotropic lymphoma cells in this report provides insight into lymphocyte homing mechanisms in folliculotropic MF.

Adult↗

PubMed1976

Lymphocyte abnormalities in mycosis fungoides.

Fourteen patients with clinical and histological evidence of mycosis fungoides have undergone a series of immunological tests. Significant findings include the presence of low numbers of E and EAC rosette-forming cells in the peripheral circulation of the mycosis fungoides patients. Elevated levels of IgE were seen in five of the mycosis fungoides patients, and the mean IgE level was significantly higher than in the control series. Possible explanations of these findings are considered.

Adult↗

PubMedDate not supplied

Cytokine profile of patients with mycosis fungoides and the immunomodulatory effect of AS101.

Cytokines are known to play a major role in the pathogenesis of mycosis fungoides, a cutaneous malignant neoplasm of CD 4 T cells. In the present study, we investigated the effect of AS101, a tellurium-based compound with immunomodulating properties, on the pattern of lymphokine production by peripheral blood mononuclear cells (PBMCs) from patients with mycosis fungoides. PBMCs were isolated from 35 patients with mycosis fungoides stage IA and IB before initiation of treatment and from 20 healthy sex and age-matched controls. Unstimulated and phytohaemagglutinin-stimulated PBMCs were tested with and without the addition of AS101. The production of interferon-gamma, interleukin 2 (IL-2), IL-2 receptor (IL-2R), interleukin 5 (IL-5) and interleukin 10 (IL-10) was determined by enzyme-linked immunosorbent assays. The effects of AS-101 on mycosis fungoides PBMCs were compared to those of healthy donor PBMCs. Significantly higher levels of IL-2R, IL-5 and IL-10 and significantly lower levels of interferon-gamma were found in the patients compared to the controls. There was no significant difference between the groups in the production of IL-2. AS101 inhibited the production of IL-2R, IL-5 and IL-10 and induced a significant increase in IL-2 levels in the mycosis fungoides PBMCs. These findings may have important clinical implications for the possible therapeutic benefit of AS101 in mycosis fungoides.

Adjuvants, Immunologic↗

PubMed1997

[Unilesional plaque-type mycosis fungoides: 3 cases].

BACKGROUND: The clinical course of unilesional plaques with a histological diagnosis of mycosis fungoides is generally benign. This uncommon situation raises the question of nosology among epidermotropic T-cell proliferations. CASE REPORT: Two women (age 65 and 70 years, cases 1 and 2) and one man (age 66 years, case 3) each had a well-limited unique oval-shaped plaque measuring 1 to 3 cm in diameter for several months. The localization of the erythematous, squamous plaques with minimal infiltration and little or no pruritus remained unchanged in the dorsal area. Histology reported mycosis fungoides: dense superficial dermal infiltration in band attached to the epidermis with a clear lower limit, formed by small and medium-sized atypical lymphocytes, sometimes with cerebriform nuclei. Epidermotropism on isolated cells or theques was noted. There was no spongiosis. Immunolabeling demonstrated predominance of CD4+ cells and tumoral infiltration uptake of anti-V beta 8 monoclonal antibodies in the two cases examined. The lesion totally regressed in case 1 after local application of carmustine with no recurrence after 5 years. Patient 2 declined treatment and no change was observed 5 years later. Surgical exeresis was performed in case 3 without recurrence 3 years later. DISCUSSION: No histological feature or T-cell immunophenotype distinguishes this clinical entity from classical mycosis fungoides. As no other long-term lesions developed, our three cases would argue for a benign course of what is probably a variety of mycosis fungoides. Unilesional plaque mycosis fungoides must be lymphomatoid contact dermatitis. Woringer-Kolopp disease differs by its nearly exclusive epidermotropic distribution.

Aged↗

PubMed1991

Cell-bound IgE and increased expression of Fc epsilon-receptors on dendritic cells in cutaneous infiltrates of mycosis fungoides.

Skin biopsies of 31 non-atopic patients, 20 with mycosis fungoides, six with psoriasis and five with contact dermatitis, and of five non-atopic healthy controls were compared for the presence of cell-bound IgE and vacant IgE binding sites. IgE+ cells were demonstrated in the cutaneous infiltrate of nine (45%) patients with mycosis fungoides, two (33%) with psoriasis and one (20%) with contact dermatitis. Following pre-incubation of skin sections with IgE myeloma protein to saturate vacant IgE-binding sites, 14 out of 16 patients (88%) with stage I mycosis fungoides, five (83%) patients with psoriasis and one (20%) with contact dermatitis showed an increase in the number of IgE+ cells. While cell-bound IgE was positively related to serum IgE levels the expression of IgE-binding sites was not. All IgE+ cells were HLA-DR+ dendritic cells identified as either macrophages (CD68+, CD14+) or Langerhans cells (CD1+). Skin biopsies of non-atopic healthy controls or clinically uninvolved skin in mycosis fungoides had neither any IgE+ cells nor any vacant binding sites. Inhibition studies with IgG1, IgG4 and IgE myeloma proteins as well as with several enzymatic fragments of IgE demonstrated that IgE interacted with Fc epsilon-receptors through isotype-specific structures on the Fc epsilon-fragment. Four anti-CD23 monoclonal antibodies, however, were unable to stain vacant Fc epsilon-receptors nor could they block IgE-binding. We hypothesize that locally-secreted lymphokines, like IL-4 or interferon-gamma, induce Fc epsilon-receptors on dendritic cells in the cutaneous infiltrate and that these receptors become occupied in parallel with elevated serum IgE levels.

Antigens, CD↗

PubMed1991

Coexisting mycosis fungoides and Hodgkin's disease as a composite lymphoma: a case report.

Within the past few years, an increasing number of reports of Hodgkin's disease following the diagnosis of, and frequently coexisting with, mycosis fungoides have appeared. Previously, Hodgkin's disease found in the lymph nodes of the patient diagnosed as mycosis fungoides was considered as a transformed form of the mycosis fungoides. But, now it has been proven that Hodgkin's disease and mycosis fungoides are histologically and immunohistochemically distinct disease entities. We report a well-documented case of a man who developed Hodgkin's disease and mycosis fungoides simultaneously as a composite lymphoma. Our case emphasizes the importance of considering the diagnosis of another lymphoma in patients with mycosis fungoides who have lymphadenopathy. The cutaneous mycosis fungoides and the Hodgkin's disease should be treated as an independent disease.

Adult↗

PubMed2004

Milia in regressing plaques of mycosis fungoides: provoked by topical nitrogen mustard or not?

OBJECTIVE: To report three cases of mycosis fungoides with milia formation in the regressing lesions. PATIENTS AND SETTING: Dermatology clinic of a university hospital (referral center). Three patients with mycosis fungoides with body surface involvement of 10% in one case (stage IIb) and exceeding 30% in two cases (stages IIb and III). All patients were treated with photochemotherapy and topical nitrogen mustard ointment in a concentration of 0.01%. After approximately 3 months multiple milia erupted on regressing plaques. RESULTS: The presence of milia was evident and was confirmed by histopathology. Regression of mycosis fungoides was noted in these plaques both clinically and in comparison with the pretreatment histologic appearance. Two of the patients showed a histological picture of follicular mucinosis. CONCLUSIONS: We do not know the significance of milia in mycosis fungoides (MF). However, we suggest that follicular rupture or a degenerative process might result in milia formation.

Administration, Cutaneous↗

PubMed2005

Bath-water PUVA therapy with 8-methoxypsoralen in mycosis fungoides.

PUVA therapy is widely used for early stage mycosis fungoides. While the efficacy of PUVA with oral 8-methoxypsoralen (8-MOP) is well documented, the use of its topical variation, bath-water PUVA therapy with 8-MOP has not been studied. The purpose of this study was to assess the effect of 8-MOP bath-water PUVA therapy in adult patients with early stage mycosis fungoides. We retrospectively evaluated the outcomes of bath-water delivery of 8-MOP (1 mg l(-1)) in 16 patients with early stage mycosis fungoides. In all patients complete response was achieved after a mean duration of 63 days requiring 29 treatments and a mean cumulative UVA dose of 33 J cm(-2). The time to relapse after complete clinical clearance was 45.6 (+/-9.2) weeks. In comparison, oral PUVA therapy with 8-MOP resulted in complete response after 64.5 days (25.8 treatments) with a mean relapse-free period of 30 (+/-3.5) weeks. We conclude that bath-water PUVA therapy with 8-MOP is a valuable photo-therapeutic alternative, which should be considered for patients in whom systemic psoralen cannot be used.

Administration, Cutaneous↗

PubMed2007

Mycosis fungoides--a review of the management of 28 patients and of the recent literature.

BACKGROUND: Mycosis fungoides is an uncommon cutaneous T-cell lymphoma characterized by malignant monoclonal proliferation of T-helper lymphocytes. Its course is variable with a potential for lymphatic and hematogenous involvement. We report the investigations, staging, treatment, follow-up, and outcome of 28 patients. This is the first such study reported from Ireland. METHODS: Twenty-eight patients with mycosis fungoides (14 women, 14 men; average age, 52.5 years) were reviewed over 12 years in the dermatology clinic which assesses an average of 4500 patients per year. All mycosis fungoides patients were referred from their family physicians. The diagnosis was made in all cases from a combination of clinical findings, histology, and immunohistochemistry. TNM staging revealed 11 patients at diagnosis stage IA (T1), 12 at stage IB (T2), four at stage IIB (T3), and one at stage III (T4). RESULTS: The usual male preponderance was not found. Eight patients needed multiple biopsies to establish the diagnosis. Detailed investigations were not useful in the early stages. Patients were followed up over a 12-year period. Thirteen patients died as a result of cutaneous lymphoma. Two patients with stage IA disease progressed rapidly and died, a feature reported in only 10% of patients at this stage. Five patients showed unusual features, including a long history prior to presentation, the development of the rarely reported bullous mycosis fungoides, and aggressive disease beginning at a young age. CONCLUSIONS: Mycosis fungoides is rare; we reviewed 28 patients over 12 years. The prognosis is poor at the later stages; 13 patients died. Two patients who died were unusual in that they rapidly progressed from stage IA disease; however, in the majority of patients with this stage, the prognosis is excellent. Detailed investigations were unhelpful in early stage disease. Close clinical follow-up is essential to identify disease progression.

Adult↗

PubMed2000

Granulomatous mycosis fungoides. Report of two cases and review of the literature.

BACKGROUND: Granulomatous mycosis fungoides is an extremely rare type of cutaneous T-cell lymphoma. Two cases are described and checked for clinical, histological and therapeutic differences to other variants of mycosis fungoides. CASE REPORTS: Case 1: a 52-year-old patient with livid plaques covering the whole integument. Histological findings showed granulomas with multinuclear giant cells in addition to a malignant lymphohistiocytic infiltrate (monodonal T-cell receptor (TCR)-gamma rearrangement). Despite various chemotherapeutic regimens, progression to tumour stage was observed. Case 2: an 88-year-old man with plaques and ulcerating tumours on the trunk and head. Histological findings showed malignant T-lymphocyte infiltrate (monoclonal TCR-gamma rearrangement) and granulomas with multinuclear foreign-body giant cells. Complete regression of all lesions was achieved using both local psoralen-ultraviolet A and electron radiotherapy. CONCLUSIONS: The diagnosis of a granulomatous mycosis fungoides depends exclusively on the histological demonstration of granulomas. Distinct clinical characteristics are not present. Apart from granuloma formation, no other noticeable histological features are evident. The presence of granulomas in mycosis fungoides does not have prognostic implications, as cases with aggressive, but also with a prolonged course have been described.

Aged↗

PubMed1975

Sea-blue histiocytes in mycosis fungoides.

A patient with tumor-stage mycosis fungoides and lymph node involvement had sea-blue (ceroid-containing) histiocytes in her bone marrow. Leukopenia, eosinophilia, and a mild type II beta-hyperlipoproteinemia were also features. Sea-blue histiocytes were demonstrated in the skin after the mycosis fungoides infiltrate had cleared with treatment. There is a possible interrelationship between the sea-blue histiocytes, the abnormal mycosis cells in the skin, and lipid metabolism.

Adult↗

PubMed1982

Spinal cord compression in mycosis fungoides.

Symptomatic involvement of the central nervous system (CNS) with mycosis fungoides is rare, and usually characterized by involvement of meninges. We describe a patient with long-standing mycosis fungoides who developed acute spinal cord compression. Since tumor-related spinal cord compression requires early intervention for a successful outcome, it should be recognized as an additional form of CNS mycosis fungoides.

Humans↗

PubMed1996

Atypical cutaneous changes after topical treatment with nitrogen mustard in patients with mycosis fungoides.

Side effects in the treatment of mycosis fungoides with topical nitrogen mustard include allergic contact dermatitis, hyperpigmentation, urticaria, and erythema multiforme-like dermatitis. We reviewed biopsy specimens from 10 patients with mycosis fungoides who were treated with topical nitrogen mustard for 10-76 months. There was no history of oral psoralen with long-wave UV radiation treatment, radiotherapy, or systemic chemotherapy. Control biopsies taken from erythematous or poikilodermatous patches on the trunk or proximal extremities showed epidermal and dermal changes associated with cytologic atypia that were not present before treatment. These changes included slight epidermal hyperplasia with foci of flat rete ridges, atypical keratinocytes with large nuclei, mostly in the lower portion of the epidermis; suprabasal mitotic figures; a few dyskeratotic cells, focal vacuolar alteration of the epidermal basal layer; increased number of slightly enlarged junctional melanocytes; melanophages in the papillary dermis; dilated blood vessels lined by plump, atypical endothelial cells; and large fibroblasts with atypical nuclei. These atypical histologic changes resemble, in part, those described in association with systemic chemotherapeutic agents, such as etoposide, busulfan, and bleomycin. We conclude that topical nitrogen mustard should be added to the list of chemotherapeutic agents that can produce atypical histologic changes in the skin.

Administration, Topical↗

PubMedDate not supplied

Hypopigmented mycosis fungoides associated with atopy in two children.

Mycosis fungoides is very rare in children. Hypopigmented lesions of this disease are usually observed in dark-skinned individuals and often show a T supressor CD8(+) phenotype. Two Caucasian children with predominantly hypopigmented lesions of mycosis fungoides are presented here. Atopy was a concomitant feature in both.

Child, Preschool↗

PubMed1998

Mycosis fungoides with involvement of the oral cavity.

Mycosis fungoides rarely involves the oral cavity. To our knowledge only 29 cases of oral cutaneous T-cell lymphoma have been described up to 1994. This report presents a case of mycosis fungoides with involvement of the oral cavity in a 57-year-old man who died from septicemia 7 months after the onset of oral involvement.

Diagnosis, Differential↗

PubMed2005

Pseudotumorous folliculotropic mycosis fungoides.

Fungating nodules and infiltrated plaques are usually equated with advanced tumor stage mycosis fungoides. We report an 85-year-old man who presented in this way but multiple skin biopsies revealed that the bulk of his nodules were due to marked follicular hyperplasia as a result of folliculotropic mycosis fungoides. This clinical presentation may be best described as a pseudotumorous form of mycosis fungoides dominated by follicular epithelial hyperplasia rather than lymphocytic proliferation characteristic of true tumor stage disease. Similar presentations have been described as a verrucous and hyperplastic variant of mycosis fungoides due to the presence of prominent epidermal hyperplasia.

Aged↗

PubMed1991

Intraocular involvement with subretinal pigment epithelium infiltrates by mycosis fungoides.

We report a case of intraocular mycosis fungoides in a 48-year-old man. The patient presented with decreased visual acuity, white subretinal lesions, and vitritis. Post-mortem histopathology revealed malignant T cell infiltrates consistent with mycosis fungoides in the retina, vitreous, and between the retinal pigment epithelium (RPE) and Bruch's membrane Focal atrophy of the RPE, along with the sub-RPE infiltrates, correlated with the clinically visible fundus lesions.

Bruch Membrane↗