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PubMed2000

Invisible mycosis fungoides: A diagnostic challenge.

We describe a 76-year-old woman who presented 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 biopsies 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↗
PubMed1982

Mycosis fungoides presenting as areas of hypopigmentation: a report of three cases.

In three patients with histologically documented mycosis fungoides, all or most of the lesions were hypopigmented. Unsuspected microscopic foci of mycosis fungoides were found in a purely macular hypopigmented area in one of these patients who had been treated with vitiligo 15 years earlier. Two of the patients were black, and one was Latin American. Repigmentation in all patients followed treatment with carmustine (BCNU) and mechlorethamine.

Adult↗
PubMed1982

Second cutaneous malignancies in patients with mycosis fungoides treated with topical nitrogen mustard.

Previous reports of skin cancer occurring in mycosis fungoides patients who have been treated with topical nitrogen mustard have been of carcinomas occurring predominantly on sun-exposed skin. Four of twenty-nine mycosis fungoides patients in our clinic treated with topical nitrogen mustard developed six skin cancers on sun-protected skin. Four lesions were squamous cell carcinomas and two were basal cell carcinomas. Few other risk factors for the development of skin cancer were present in our patients. These cases support the hypothesis that topical nitrogen mustard is a carcinogen or tumor promote and emphasize the need for careful examination of the skin of patients treated with topical nitrogen mustard.

Administration, Topical↗
PubMed1982

Hyperkeratotic mycosis fungoides restricted to the palms.

The case of a 45-year-old Latin American man, who presented to the Dermatology Clinic with a 6-month history of hyperkeratotic lesions confined to the palms and the palmar aspects of the digits of both hands, is discussed. Biopsy of these lesions revealed the classic histologic findings of mycosis fungoides. The clinical and histologic differential diagnosis of mycosis fungoides is considered.

Biopsy↗
PubMed1983

Necrotizing vasculitis within cutaneous lesions of mycosis fungoides.

Mycosis fungoides is a T cell lymphoma with a predilection for cutaneous involvement. This paper describes the clinical manifestations and histopathologic features of a case of mycosis fungoides with necrotizing vasculitis localized to the lesions of cutaneous lymphoma. Elevated levels of circulating immune complexes were found in this patient. The large numbers of perivascular malignant helper T lymphocytes may have induced immunoglobulin synthesis, resulting in the formation of these complexes followed by deposition in vessel walls and subsequent necrotizing vasculitis. Possible alternative mechanisms include the presence of anti-T cell antibodies, or cytotoxic effector cells.

Adult↗
PubMed1986

Histopathologic studies in Sézary syndrome and erythrodermic mycosis fungoides: a comparison with benign forms of erythroderma.

Histologic sections from eleven patients with Sézary syndrome were reviewed and compared with those of four patients with erythrodermic mycosis fungoides and twenty-four patients with a benign form of erythroderma, including fifteen patients with chronic dermatitis, four with a generalized drug eruption, and five with an erythrodermic psoriasis. The most important discriminating histologic feature in patients with Sézary syndrome was the presence of a monotonous bandlike or perivascular infiltrate in the papillary dermis, mainly composed of large cerebriform-mononuclear cells, as seen in seven of eleven Sézary syndrome patients. Pautrier's microabscesses were observed in seven of eleven Sézary syndrome patients, two of four patients with erythrodermic mycosis fungoides, but not in any of the patients with a benign form of erythroderma; their presence was therefore considered a reliable criterion in differentiating erythrodermic cutaneous T cell lymphoma from benign forms of erythroderma. However, features of chronic dermatitis were often found superimposed on those of Sézary syndrome and were even predominating in four of eleven Sézary syndrome patients. Moreover, four patients with a benign form of erythroderma showed a histologic picture suggestive of cutaneous T cell lymphoma. Therefore, in dubious cases repeated skin biopsies, additional investigations of lymph nodes and peripheral blood, and careful follow-up are mandatory for the achievement of a correct diagnosis.

Adult↗
PubMed1987

Combined total body electron beam irradiation and chemotherapy for mycosis fungoides.

Since 1979 a protocol of total body electron beam therapy (3,600 rads; 6 MeV), followed by six monthly cycles of chemotherapy (doxorubicin, 30 mg/M2 given intravenously once monthly, and cyclophosphamide, 100 mg/M2 given orally each day for 14 days), has been used to treat fifty patients with mycosis fungoides (primarily Stages I and II). A group of twenty-four patients, treated by identical high-dose electron beam therapy alone, served as control subjects. Actuarial analysis by the Kaplan-Meier method and statistical analysis by the generalized Wilcoxon test of Gehan demonstrated a significant difference (p = 0.008) in the probability of Stages I and II patients' remaining in complete clinical remission when combination therapy was compared with high-dose electron beam therapy alone. No statistically significant difference was demonstrated in patients in Stages III and IV mycosis fungoides. Although 60% of patients were in "complete clinical remission," the longest follow-up being 75 months, all continued to show karyotypic abnormalities of circulating lymphocytes, and 70% had intermittently and abnormally elevated blood levels of Sézary cells.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed1987

Electron microscopic and immunolabeling studies of the lesional and normal skin of patients with mycosis fungoides treated by total body electron beam irradiation.

Biopsy specimens were taken from lesional and normal skin of nine patients with mycosis fungoides before and after total body electron beam therapy. By electron microscopy, lesional skin had one and one-half to ten times as many epidermal Langerhans cells and indeterminate cells as did the normal skin. In successfully treated lesional skin 1 month after the end of electron beam therapy, the density of epidermal Langerhans cells and indeterminate cells had decreased markedly. In incompletely resolved lesions, Langerhans cells and indeterminate cells were still at pretreatment levels. Epidermal T6 and Ia antigens showed the same pattern of response. Epidermal cell suspensions from lesional and normal skin before and after electron beam therapy were assayed for epidermal thymocyte activating factor. The values of production of this factor did not correlate with the source of the epidermal cells, response to therapy, or the patient's disease course. Skin lesions resembling xerosis and parapsoriasis and histologically lacking the criteria for mycosis fungoides appeared during clinical remissions. These nonspecific skin lesions had densities of epidermal Langerhans cells, indeterminate cells, and T6-positive and Ia-positive cells comparable to levels found in pretreatment lesional skin.

Antibodies, Monoclonal↗
PubMed1988

Topical mechlorethamine therapy for early stage mycosis fungoides.

One hundred seventeen patients with mycosis fungoides were treated with topical mechlorethamine hydrochloride. The probability of achieving a clinically apparent remission within 2 years of therapy was 75.8% in patients with stage I disease, 44.6% in patients with stage II disease, and 48.6% in patients with stage III disease. Patients with stage I disease achieved complete remission sooner (median, 6.5 months) than patients with stage II (median, 41.1 months) or stage III (median, 39.1 months) disease. The median time to relapse was 44.5 months. Sixty-eight patients (58.1%) developed a delayed hypersensitivity reaction, but only one patient had to discontinue therapy as a consequence. No appreciable differences were seen in the probability to achieve complete remission or time to complete remission as stratified by gender, substage, or the development of a delayed hypersensitivity reaction. Survival analysis revealed that the probability of surviving at 5 years was 89% for all patients. These findings compare favorably with results with other treatments for early stage mycosis fungoides.

Administration, Topical↗
PubMed1989

Unilesional mycosis fungoides: a distinct entity.

Four cases of solitary lesions showing the histologic features of mycosis fungoides are described. Three patients had an erythematous scaling plaque, whereas the fourth had a solitary nodule. The lesion was located on the arm in two patients, on the foot in one patient, and on the back in one patient. No preceding factors were noted, and no lymphadenopathy or other significant illness was found on examination. Histologic examination demonstrated features of a cutaneous T cell lymphoma with epidermotropism. Monoclonal markers on frozen and paraffin sections revealed a predominant T cell infiltrate. Three patients were treated with local excision. The remaining patient was treated with radiotherapy. The lesions have not recurred, and no new lesions have developed after individual follow-up of 7 months, 7 1/2 months, 13 years, and 18 years. These cases appear to be unique reports of solitary mycosis fungoides behaving in a biologically benign manner. The relationship to Woringer-Kolopp disease and lymphomatoid papulosis is discussed.

Aged↗
PubMed1997

Lichenoid changes in mycosis fungoides.

BACKGROUND: The lichenoid tissue reaction (LTR) is present in a variety of dermatoses. Numerous histologic variants of mycosis fungoides (MF) have also been described. OBJECTIVE: Our purpose was to analyze a subset of patients with mycosis fungoides (MF) who had a prominent LTR at the time of presentation. METHODS: Clinical and pathologic features were evaluated in 12 patients with MF and an LTR. RESULTS: The primary complaint was intense pruritus (in 10 of 12 patients). An accelerated course was noted in one half of the patients including four with Sézary syndrome. Four patients died: two with Sézary syndrome and two with extensive patch/plaque MF. Lymphocyte atypia, prominent basal cell layer epidermotropism, and the presence of plasma cells and eosinophils may help to distinguish lichenoid MF from lichen planus. CONCLUSION: A subset of patients with MF present with lichenoid changes. The pathologic features may have a striking resemblance to lichen planus. Our findings suggest that lichenoid changes in MF tend to be associated with intense pruritus and may connote a poor prognosis.

Adult↗
PubMed1998

Unusual presentation of mycosis fungoides as pigmented purpura with malignant thymoma.

Thymoma is known to be associated with many lymphoreticular and nonlymphoreticular tumors. A woman who presented with patch stage mycosis fungoides with skin lesions resembling a pigmented purpura was found to have an anterior mediastinal mass on routine staging evaluation. A core needle biopsy of the mass revealed a lymphocyte predominant malignant thymoma. Review of the literature reveals many diseases with dermatologic manifestations associated with thymoma; however, malignant thymoma coexisting with mycosis fungoides has not previously been reported.

Adult↗
PubMed1999

Prognostic factor analysis in mycosis fungoides/Sézary syndrome.

BACKGROUND: The influence of prognostic factors on survival in patients with mycosis fungoides/Sézary syndrome (MF/SS) is less well described than for other lymphomas. OBJECTIVE: Our purpose was to evaluate the prognostic value of diverse clinicopathologic and laboratory characteristics in patients with MF/SS. METHODS: All 115 patients with MF/SS seen at the Mycosis Fungoides clinic at M. D. Anderson Cancer Center during the study period who had slides available for pathologic review were analyzed. Univariate and multivariate methodologies were used. RESULTS: Age (> or = 60 years; P = .0002), advanced stage (P < 10(-5)), tumor (T3) stage disease (P < or = 10(-5)), lymphadenopathy (P = .006), bone marrow infiltration (P = .03), high lactate dehydrogenase (LDH; P = .0002), high beta2-microglobulin (> 2 mg/L; P = .009), and transformation to large-cell lymphoma (P = .004) were significant prognostic factors in the univariate analysis and correlated with a poorer survival. The outcome of patients staged as IIB was significantly worse than that of those staged as I or IIA or III (P < .001) and was comparable to that of the patients staged as IV (P = .8). In the multivariate analysis, the factors selected include stage (I to IIA and III vs IIB and IV; P < .0001), LDH (P = .006), and age (> or = 60 vs < 60 years; P = .02). The actuarial median survival of patients with advanced stage, high LDH, or age 60 years or more was 2.5 to 3.5 years, whereas that of patients without any of these parameters was more than 13 years. CONCLUSION: Our results suggest that patients with MF/SS who are staged as IIB or IV, who have a high LDH, or who are 60 years of age or older have an aggressive course and poor survival.

Bone Marrow↗
PubMed1991

Mycosis fungoides and Sézary syndrome in Japan. Clinicopathologic study of 107 autopsy cases.

We reviewed 111 clinical and pathological materials of patients who had been entered as mycosis fungoides (MF) or Sézary syndrome (SS) in the Annuals of the Pathological Autopsy Cases in Japan during the 29-year-period since the Annuals' inception in 1958. One hundred and seven patients were classified. Of these, 64 were in Group 1 (mycosis fungoides, MF), 23 in Group II (Sézary syndrome, SS), and 20 in Group III (non-Hodgkin's lymphoma, NHL). The clinicopathologic features of Groups I and II (MF, SS) were similar to those of cutaneous T cell lymphoma (CTCL) in Europe and the United States. Those of Group III (NHL) were indistinguishable from primary cutaneous non-Hodgkin's lymphoma. It was suggested that some patients with adult T cell leukemia/lymphoma (ATLL) with skin lesions used to be misdiagnosed as MF or SS in earlier years when ATLL was still unknown. It was concluded that CTCL in Japan has clinicopathologic features quite like those of CTCL in Europe and the United States and that they are probably the same disease.

Adolescent↗
PubMed1993

Pulmonary involvement by mycosis fungoides. A case showing angiocentric lesions and granulomas.

We describe an unusual case of pulmonary involvement by mycosis fungoides in a 31-year-old woman. Disseminated disease had developed despite cutaneous irradiation and systemic combination chemotherapy. Microscopy of biopsied tumour deposits in the lung showed a polymorphous, angiocentric lymphoid infiltrate, including atypical cells with convoluted nuclei which displayed a T-cell phenotype. Numerous epithelioid granulomas were also present. These histological appearances represent an unusual pattern of pulmonary involvement by disseminated mycosis fungoides.

Adult↗
PubMed1999

Total skin electron beam therapy with or without adjuvant topical nitrogen mustard or nitrogen mustard alone as initial treatment of T2 and T3 mycosis fungoides.

PURPOSE: To compare the efficacy of total skin electron beam therapy (TSEBT) with or without adjuvant topical nitrogen mustard (+/- HN2) with topical nitrogen mustard (HN2) alone as initial management of T2 and T3 mycosis fungoides (MF). METHODS AND MATERIALS: A retrospective analysis of 148 patients presenting to Stanford from January, 1970 through January, 1995 within 4 months of pathologic diagnosis of MF. Fifty-five patients with T2 and 27 with T3 disease received TSEBT +/- HN2. Fifty-four patients with T2 and 12 with T3 disease received HN2 alone. Boosts with radiotherapy were usually administered to cutaneous tumors of patients with T3 disease. RESULTS: TSEBT +/- HN2 yielded significantly higher complete response (CR) rates than did HN2 alone in patients with T2 and T3 disease (76% vs 39%, p = 0.03 for T2, and 44% vs 8%, p < 0.05 for T3, respectively). In T2 disease, treatment with adjuvant HN2 was associated with a longer freedom from relapse following TSEBT when compared to observation following a CR to TSEBT (p = 0.068). However, no significant differences in survival were observed for different management programs for T2 or T3 disease. In T2 disease, both TSEBT and HN2 were as effective as salvage therapy as when utilized as initial therapy. However, salvage therapy in T3 disease was rarely effective. Limited tumor involvement in T3 disease did not correlate with improved survival compared to more generalized tumorous disease. MF contributed to 27% and 68% of deaths in patients with T2 and T3 disease, respectively. CONCLUSION: Because of high response rates, management of significantly symptomatic or extensive T2 MF should include TSEBT, and adjuvant HN2 should be administered after a CR to TSEBT. Patients with T2 disease who fail TSEBT or HN2 can be salvaged with the other modality. TSEBT is also an effective treatment for T3 disease. The small subset of patients with limited T3 disease may also be treated with HN2 and local radiotherapy to the tumors. Further investigations are necessary to improve the overall outcome for T3 mycosis fungoides.

Administration, Topical↗
PubMed1995

Symptomatic intracranial involvement by mycosis fungoides.

Involvement of the central and peripheral nervous system by mycosis fungoides is rare and usually occurs in the presence of widespread cutaneous and visceral disease. We describe a patient with mycosis fungoides, previously confined to the skin, who developed intracranial parenchymal disease with no evidence of systemic involvement.

Central Nervous System Diseases↗