Unilesional Follicular Mycosis fungoides

Marzano, Angelo Valerio; Berti, Emilio; Lupica, L.; Alessi, Elvio

doi:10.1159/000018232

Cited by 24 publications

(30 citation statements)

References 6 publications

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“…Of note are the 3 previously reported cases of unilesional MF in the head area [4, 8, 9]. There is only one previous report of folliculotropic MF of a localized nature in the literature [12]; in this case, as in ours, there was no evidence of mucin deposit, and this is in keeping with multilesional MF, in which cases indistinguishable clinically from follicular mucinosis but without evidence of mucin deposit have been reported [20]. …”

Section: Discussionsupporting

confidence: 85%

“…The few reports appearing in the literature since 1981 [2, 3, 4, 5, 6, 7, 8, 9, 10, 11, 12, 13], usually using the term ‘unilesional MF’ [3, 4, 5, 6, 7, 8, 9, 12], indicate that this variant of MF is rare, presenting as an isolated patch or plaque, generally indistinguishable clinically and histopathologically from typical MF. It follows a benign course for long periods without any treatment, and the initial reports stressed that there had been no case of disease recurrence after curative-intent treatment after variable follow-up periods [1, 2, 3, 4, 5, 6, 7].…”

Section: Introductionmentioning

confidence: 99%

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Unilesional Mycosis fungoides

Hodak¹,

Phenig²,

Amichai

et al. 2000

Dermatology

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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 γ 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.

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Section: Discussionsupporting

confidence: 85%

Section: Introductionmentioning

confidence: 99%

Unilesional Mycosis fungoides

Hodak¹,

Phenig²,

Amichai

et al. 2000

Dermatology

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“…10 Since the first report in 1981, 8 approximately 128 cases of solitary MF have been described, usually termed 'unilesional MF'. 5,6,[9][10][11][12][13][14][15][16][17][18][19][20][21][22][23][24][25] Solitary MF can affect any age group and has been reported in children. 17,19 The mean age of our patients was 47 years, with one case occurring in a 14-year-old girl.…”

Section: Discussionmentioning

confidence: 99%

Solitary mycosis fungoides: A distinct clinicopathologic entity with a good prognosis

Ally¹,

Pawade²,

Tanaka³

et al. 2012

Journal of the American Academy of Dermatology

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“…The most noteworthy clinical finding in our series is the association between ichthyosiform manifestations and skin lesions typical of so-called FMF, an unusual clinicopathologic variant of MF characterized by selective involvement of hair follicles by the atypical T lymphocytes [1, 2, 3, 4]. Such skin lesions, including comedo-like lesions and epidermal cysts, alopecia and follicular keratotic papules, were seen in 3 of 4 patients, whereas cutaneous manifestations of classic MF were absent in all 4 cases.…”

Section: Discussionmentioning

confidence: 99%

“…Cutaneous manifestations typically comprise patches, plaques and tumors. Atypical variants include, among others, follicular [1, 2, 3], unilesional [4], bullous [5], hypopigmented [6]and invisible [7]MF. To the best of our knowledge, only one case of MF presenting solely with the clinical features of acquired ichthyosis has been reported [8].…”

Section: Introductionmentioning

confidence: 99%

Ichthyosiform Mycosis fungoides

et al. 2002

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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.

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Unilesional Follicular Mycosis fungoides

Cited by 24 publications

References 6 publications

Unilesional Mycosis fungoides

Unilesional Mycosis fungoides

Solitary mycosis fungoides: A distinct clinicopathologic entity with a good prognosis

Ichthyosiform Mycosis fungoides

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