Cases reported "Hypopigmentation"

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1/18. Leukoderma following occupational allergic contact dermatitis.

    We describe 2 cases of occupational allergic contact dermatitis followed by leukoderma. The 1st case was a 49-year-old wood machinist who developed leukoderma in areas of contact dermatitis involving his lips, neck, hands and forearms and was found to be allergic to colophony and pine sawdust on patch testing. The 2nd case involved a 44-year-old man who worked as an epoxy applicator in a ceramics factory. He developed depigmentation in areas of contact dermatitis involving his face, hands, forearms, back, thighs and legs and was found on patch testing to be allergic to epoxy resin. The appearance of contact leukoderma may be indistinguishable from idiopathic vitiligo. However the prognosis for repigmentation may be better in contact leukoderma than in idiopathic vitiligo.
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2/18. A case of hypopigmented mycosis fungoides.

    We experienced a 26-year-old woman in whom widespread hypopigmented macules and patches developed as the initial clinical feature of mycosis fungoides. Histopathological examination confirmed the diagnosis. The patients was treated with PUVA, and the lesion progressively disappeared within five months.
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3/18. Cutis tricolor: congenital hyper- and hypopigmented lesions in a background of normal skin with and without associated systemic features: further expansion of the phenotype.

    The term cutis tricolor describes the uncommon co-existence of congenital hyper- and hypopigmented macules, in close proximity to each other, in a background of normal skin so far seen in a 17-year-old patient with various other congenital defects. The suggested explanation for this phenomenon is allelic twin spotting. We report on two boys, aged 6 and 11 years, with an unusual combination of three different degrees of pigmentation, one of whom had in addition, psychomotor delay, dysmorphic features, musculoskeletal abnormalities and subcortical and periventricular white matter high signal lesions on brain neuroimaging. In both cases a search for mosaicism in peripheral blood lymphocytes and cultured fibroblasts was negative. In contrast to the previously reported case, the two children had large streaks or patches of hyper- and hypopigmented skin lesions, in close proximity to each other, involving large areas of the body. The rest of the skin had a normal intermediate pigmentation. CONCLUSION: This combination of three degrees of pigmentation in association with systemic defects in one child and the lack of such association in the other confirms and further expands the clinical phenotype of cutis tricolor.
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4/18. Phylloid hypomelanosis is closely related to mosaic trisomy 13.

    Phylloid hypomelanosis is a distinct type of pigmentary mosaicism characterized by congenital hypochromic macules resembling a floral ornament with various elements such as round or oval patches, macules resembling the asymmetrical leaves of a begonia, or oblong lesions. A review of cases with documentation of cytogenetic findings showed that aberrations involving chromosome 13 were present in 5 out of 6 patients. Examination of blood lymphocytes revealed a 46, XX/47 XX, 13 or 46, XX/47, XX, der (13) mosaic in three of these cases and a karyotype 46, XX, t(13;13) in the other two cases. cytogenetic analysis of skin fibroblasts showed chromosomal mosaicism in 4 of the 5 patients. In the remaining case, a chromosome 13 translocated on 13 was found in 100% of blood lymphocytes and skin fibroblasts, suggesting that mosaicism involving chromosome 13 may have developed in the melanocyte system. In conclusion, contrasting with hypomelanosis of Ito which is a cutaneous sign of many different states of mosaicism, phylloid hypomelanosis seems to originate preponderantly from a mosaic state involving chromosome 13. Future case reports may help to delineate further the significance of this relationship.
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5/18. Depigmented genital extramammary Paget's disease: a possible histogenetic link to Toker's clear cells and clear cell papulosis.

    BACKGROUND: The histogenesis of extramammary Paget's disease (EMPD) is still controversial. Benign pagetoid cells of the nipple first described by Toker and the similar clear cells found in white maculopapules of clear cell papulosis (CCP) have been proposed to be potential precursor cells giving rise to EMPD and primary intraepidermal Paget's disease in the nipple. The observation of a rare case of depigmented EMPD provided us with a chance to examine further the interesting Toker's clear cell/CCP hypothesis. methods: We performed pathologic studies, including Fontana-Masson stain and immunostaining for AE1/AE3 and S100P, on a new case of depigmented EMPD manifesting a 4 x 3 cm hypopigmented-depigmented patch on the root of the penis. RESULTS: The lesion showed extensive intraepithelial proliferation of atypical pagetoid cells with markedly reduced epidermal melaninization but nearly normal numbers of melanocytes. The tumor cells were strongly positive for AE1/AE3 by immunostaining. Some tumor cells displayed tadpole-like morphology resembling the pagetoid cells of CCP. Such morphology was not observed in two random examples of non-depigmented genital EMPD. CONCLUSIONS: The findings of tadpole-shaped pagetoid cells and depigmentation in the present case suggest that depigmented EMPD may be histogenetically related to CCP. Depigmented EMPD should be considered in the differential diagnosis of vitiligo, depigmented mycosis fungoides and lichen sclerosus located along the milk line.
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6/18. Phylloid pattern of pigmentary disturbance in a case of complex mosaicism.

    Among the various types of pigmentary disturbances associated with mosaicism, the phylloid pattern (Greek phyllon = leaf, eidos = form) is characterized by multiple leaf-like patches reminiscent of an art nouveau painting. The number of cases displaying this unusual pattern is so far limited. We describe a phylloid pattern of hypomelanosis in a 3-year-old girl with multiple congenital anomalies including microcephaly, midfacial hypoplasia, cleft lip, coloboma, posteriorly rotated ears, pectus carinatum, and pronounced mental and physical retardation. In addition, this child had oval or oblong patches of hyperpigmentation involving the trunk in a horizontal arrangement dissimilar from the phylloid hypomelanotic pattern. In peripheral blood lymphocytes a karyotype 46,XX,-13, t(13q;13q) was consistently found, whereas cultured skin fibroblasts showed a complex form of mosaicism comprising three different abnormal cell lines (46,XX,-13, t(13q;13q)/45,XX,-13/45,XX,-13, frag). This case provides further evidence that the phylloid pattern represents a separate category of pigmentary disturbance to be distinguished from other types of cutaneous mosaicism such as the lines of Blaschko or the checkerboard arrangement.
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7/18. Hypopigmented mycosis fungoides.

    We report the case of a 25-year-old Jamaican woman with hypopigmented mycosis fungoides. She first developed a hypopigmented patch on her arm at the age of 11 years. Further lesions developed on the trunk and limbs over a period of 10 years. The lesions were completely impalpable. skin biopsy showed an infiltrate of atypical lymphocytes, some with cerebriform nuclei, suggesting a diagnosis of mycosis fungoides. The lesions cleared with puva therapy.
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8/18. melanoma-associated leukoderma.

    A case is presented of a man with a history of melanoma treated with sentinel lymphadenectomy and interferon therapy, who subsequently developed diffuse hypopigmented patches thought to be consistent with a diagnosis of melanoma-associated leukoderma. Clinically, melanoma-associated leukoderma is a diffuse macular hypomelanosis or depigmentation, which often develops at sites distant to the location of the primary melanoma. The leukoderma may be hypomelanotic and mottled or depigmented and milk-white. Spontaneous repigmentation may occur. A recent study has demonstrated that T cells involved in the destruction of neoplastic melanocytes are identical clones of those that accumulate in melanoma-associated leukoderma.
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9/18. Linear basaloid follicular hamartoma on the Blaschko's line of the face.

    We report a 5-year-old girl with localized basaloid follicular hamartoma. The lesion consisted of a linear, hypopigmented, atrophic patch on the right lateral side of nose. Of great interest is that the linear distribution of lesion may follow the Blaschko's line of the face.
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10/18. Bilateral acute depigmentation of the iris.

    PURPOSE: To report on five cases of unusual bilateral stromal depigmentation of the iris and pigment dispersion masquerading as uveitis. methods: We describe the clinical features of five consecutive patients who presented with acute depigmentation of the iris stroma between June and October 2003. RESULTS: Four patients were female, one was male. Age at presentation ranged from 15 to 25 years. Presenting symptoms were sudden-onset ocular discomfort and red eye in four patients and change of eye color in one patient. All patients had bilateral involvement, with a symmetrical diffuse depigmentation of the iris stroma in three cases and patchy areas of iris depigmentation in two. Other common features were mild ciliary injection (seven eyes), Krukenberg spindle (seven eyes), circulating pigment in the anterior chamber (eight eyes), and heavy pigment deposition in the angle (ten eyes). No eyes had iris transillumination defects, inflammatory keratic precipitates or inflammatory cells in the anterior chamber. Systemic laboratory work-up was unrewarding in all cases, and PCR analysis of the aqueous humor for HSV1 and 2 was negative in one patient. Four patients were treated with a short course of topical corticosteroids and three with oral acyclovir. One patient was lost to follow-up. The remaining four patients were followed up for 6-19 months with a stable clinical picture. CONCLUSION: In contrast to pigment dispersion syndrome, pigment seemed to be released from iris stroma in the five cases described here. Although patchy depigmentation of the iris resembled the lesions seen in herpetic iridocyclitis in two of the patients, symmetrical bilateral involvement and lack of intraocular inflammation were the differentiating features. The patients described here could represent a new entity or an unusual presentation of herpetic eye disease.
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