Hypopigmented mycosis fungoides: report of five cases with ultrastructural observations

Breathnach, S.M.; McKee, Phillip H.; Smith, N.P.

doi:10.1111/j.1365-2133.1982.tb11678.x

Cited by 77 publications

(63 citation statements)

References 14 publications

(12 reference statements)

Supporting

Mentioning

Contrasting

Order By: Relevance

“…1 In this context, hypopigmentation during flares of erythrodermic cutaneous T-cell lymphoma has been purported to result from reaction of cytotoxic T-cells. 12 On ultrastructural examination, damaged melanosomes 10 and a decreased number of melanosomes 11 have been observed in the lesional skin. The pathogenesis of hypopigmentation in vitiligo is also not completely understood; suggested mechanisms include autoimmune, neural, biochemical, oxidative stress, autocytotoxic, viral, and melanocyte detachment.…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Decreased CD117 expression in hypopigmented mycosis fungoides correlates with hypomelanosis: lessons learned from vitiligo

et al. 2006

View full text Add to dashboard Cite

Hypopigmented mycosis fungoides is an uncommon clinical variant of cutaneous T-cell lymphoma. We hypothesized that hypomelanosis in hypopigmented mycosis fungoides may have a similar mechanism as in vitiligo, a condition in which it is believed that alterations in expression of CD117 (stem cell factor receptor/KIT protein) on epidermal melanocytes and abnormal interactions between melanocytes and surrounding keratinocytes may play a pathogenic role. To test the hypothesis that similar mechanisms might also explain hypopigmentation in hypopigmented mycosis fungoides, skin specimens from five cases each of hypopigmented mycosis fungoides and vitiligo were studied immunohistochemically for immunophenotype of the infiltrating cells, CD117 (expressed by epidermal melanocytes), and pan melanoma cocktail of antigens (gp100, tyrosinase, and MART-1) expression; cases of conventional mycosis fungoides and normal skin were studied in parallel as controls. Our findings confirm a predominance of CD8 þ neoplastic T cells in hypopigmented mycosis fungoides. Similarly, the epidermal lymphocytic infiltrate in vitiligo was also composed of CD8 þ cytotoxic T cells, in contrast to an epidermal infiltrate composed of CD4 þ T cells in conventional mycosis fungoides. The average number of epidermal CD117 expressing cells followed the same pattern of decreased expression in hypopigmented mycosis fungoides as in vitiligo, whereas the levels in conventional mycosis fungoides were higher, and similar to that observed in normal skin. Furthermore, a decreased number of melanocytes per high-power field of the length of the biopsy was present in hypopigmented mycosis fungoides and vitiligo, as compared with either conventional mycosis fungoides or normal skin, suggesting a correlation between decreased expression of CD117 and decreased number of melanocytes. We propose that decreased expression of CD117 and its downstream events in melanocytes may be initiated by cytotoxic effects of melanosomal-antigen-specific CD8 þ neoplastic T lymphocytes, resulting in destabilization of CD117 and leading to dysfunction and/or loss of melanocytes in the epidermis of hypopigmented mycosis fungoides.

show abstract

Section: Discussionmentioning

confidence: 99%

“…[6][7][8][9] The mechanism of hypomelanosis in hypopigmented mycosis fungoides is as yet unclear. Damaged melanosomes 10 and a decreased number of melanocytes 11,12 have been reported in hypopigmented mycosis fungoides lesions.…”

mentioning

confidence: 99%

Decreased CD117 expression in hypopigmented mycosis fungoides correlates with hypomelanosis: lessons learned from vitiligo

et al. 2006

View full text Add to dashboard Cite

show abstract

“…None of the 45 patients with childhood-onset hypopigmented disease reported in the current series (n ϭ 8) or elsewhere in the literature (n ϭ 37) has experienced progression beyond Stage IB disease. 7,[12][13][14][15][16][17][18][19][20][21][22] This subgroup of hypopigmented childhood-onset MF appears to have an excellent prognosis.…”

Section: Clinical Featuresmentioning

confidence: 99%

Outcome in 34 patients with juvenile‐onset mycosis fungoides

Wain

Orchard

Whittaker

et al. 2003

Cancer

136

View full text Add to dashboard Cite

New methodology for the stereoselective synthesis of trisubstituted olefins is presented. The use of ortho‐diphenylphosphanyl benzoate (o‐DPPB) as a directing leaving group for copper‐mediated allylic substitution with Grignard reagents allowed for the stereoselective construction of a wide range of E olefins, without the need for an adjacent electron‐withdrawing group. Our modular three‐step approach toward trisubstituted alkenes commenced with geminal α‐methylene aldehydes. Addition of an organometallic reagent and introduction of the o‐DPPB group by esterification was followed by the o‐DPPB‐directed copper‐mediated allylic substitution with a Grignard reagent to furnish stereodefined trisubstituted olefins. Additionally, incorporation of a stereocenter from the chiral pool allowed the preparation of an enantiomerically pure olefin that bore three alkyl substituents in high E/Z selectivity.

show abstract

“…Two groups of patients were compared: (1) MF patients diagnosed in our Clinic from January 1975 to August 1997, in whom the first histological diagnosis was performed after 20 years of age (252 cases; median age at diagnosis: 61 years, range: 24–91) and (2) the cases of early-onset MF (first histological diagnosis before 20 years of age) reported in the literature from January 1975 to August 1997 [3, 4, 5, 6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16, 17, 18, 19, 20, 21]plus the 7 herein reported. SS cases reported in the literature [26, 27]with histological diagnosis before 20 years of age were not included in this study.…”

Section: Methodsmentioning

confidence: 99%

Mycosis fungoides in Patients under 20 Years of Age: Report of 7 Cases, Review of the Literature and Study of the Clinical Course

et al. 1999

View full text Add to dashboard Cite

Background: Mycosis fungoides (MF) is rare in young patients. Its clinical behavior is still uncertain, as some reports have suggested that it has a more aggressive course than does the adult-onset type. Aim: To ascertain if early-onset MF represents a heterogeneous group of cutaneous T cell lymphomas. Materials and Methods: Clinical, immunohistopathological and follow-up data of early-onset (<20 years of age) MF cases reported in the literature (n = 42) plus 7 described herein were compared with those of a cohort of adult-onset MF patients (n = 252) diagnosed at our institution since 1975. Results: The majority of the 49 early-onset MF patients had patch-plaque stage disease at diagnosis. Ten had hypopigmented lesions. The predominant phenotype was CD3+ CD4+CD7–CD8–. Seven patients had a stage progression, 6 with extracutaneous involvement. Five- and 10-year survival rates were 93 and 74%, respectively. Conclusions: No statistically significant differences were found in the disease course between early- and adult-onset MF.

show abstract

Hypopigmented mycosis fungoides: report of five cases with ultrastructural observations

Cited by 77 publications

References 14 publications

Decreased CD117 expression in hypopigmented mycosis fungoides correlates with hypomelanosis: lessons learned from vitiligo

Decreased CD117 expression in hypopigmented mycosis fungoides correlates with hypomelanosis: lessons learned from vitiligo

Outcome in 34 patients with juvenile‐onset mycosis fungoides

Mycosis fungoides in Patients under 20 Years of Age: Report of 7 Cases, Review of the Literature and Study of the Clinical Course

Contact Info

Product

Resources

About