Clinicopathological aspects and proviral load of adulthood infective dermatitis associated with HTLV-1: Comparison between juvenile and adulthood forms.

BACKGROUND: Infective dermatitis associated with human T-cell lymphotropic virus type-1 (HTLV-1), (IDH), is a chronic eczema occurring in HTLV-1 infected children. Rare cases of adulthood IDH have been reported and no study until now aimed to compare juvenile and adulthood IDH. METHODOLOGY/PRINCIPAL FINDINGS: Twelve cases of adulthood IDH followed for a mean time of 7.5 years were analyzed according to clinicopathological and molecular aspects, comparing them to juvenile IDH cases. Diagnosis was based on the modified major criteria used for juvenile IDH. Proviral load (PVL) assessment was performed by real-time PCR technique. Adulthood IDH presented similar clinicopathological and molecular aspects compared to juvenile IDH. The morphology of lesions and areas of involvement were similar, except for the involvement of the ankles and inframammary folds in the adulthood form. HTLV-1 associated myelopathy/tropical spastic paraparesis (HAM/TSP) occurred in six adulthood IDH patients, with almost equal frequency. However, at least in two patients, HAM/TSP appeared prior to IDH, differently from what was observed in juvenile IDH. CONCLUSIONS/SIGNIFICANCE: Adulthood IDH is similar to juvenile IDH according to clinicopathological aspects and PVL levels. Therefore, the same modified major diagnostic criteria for juvenile IDH can be applied to both forms.

Parvovirus B19 and in situ immune response in eczema and psoriasis skin lesions of patients from the Brazilian Amazon region.

Parvovirus B19 (PVB19) is a virus found in the skin that causes asymptomatic infections and can exist in the host for long periods to time. The virus induces a local inflammatory response and is associated with the development of arthritis and other autoimmunes diseases. Parvovirus B19 DNA was investigated by PCR in the skin of 20 patients with psoriasis and 20 patients with eczema. Additionally, immunohistochemistry was used to characterize the expression of cytokines in these lesions. The sociodemographic variables were similar in the two groups studied. Psoriasis vulgaris was the most common clinical type in men (50%) and women (80%) (p = 0.0106). Comorbidities were observed in most patients with psoriasis (75%), with an OR of 14 (p = 0.0068). Another important finding was the high prevalence (50%) of psychiatric disorders in patients with psoriasis (OR = 16, p = 0.0218). Only two patients (10%) with psoriasis were positive for PVB19. Comparison of cytokine expression showed the same cytokine profile in the two groups (p > 0.05). However, expression of TNF-α tended to be higher in psoriasis patients. There was no significant positivity for PVB19 in the two groups studied. Immunohistochemistry showed higher expression of TNF-α in psoriasis lesions compared to the eczema group.

Case for diagnosis. Infective dermatitis associated with HTLV-1: differential diagnosis of atopic dermatitis.

Infective dermatitis associated with HTLV-1 (IDH) is the main cutaneous marker of HTLV-1 infection. This disease occurs primarily in children and should be differentiated from other eczemas, especially from atopic dermatitis. The largest series of IDH are from Jamaica and Brazil. There are an estimated 15 to 20 million infected people in the world, and Brazil is one of the endemic regions. Studies suggest that IDH in children may be a marker for the development of T-cell leukemia/lymphoma (ATL) or myelopathy associated with HTLV-1/tropical spastic paraparesis (HAM / TSP) in adulthood.

Case for diagnosis. Infective dermatitis associated with HTLV-1: differential diagnosis of atopic dermatitis

Abstract: Infective dermatitis associated with HTLV-1 (IDH) is the main cutaneous marker of HTLV-1 infection. This disease occurs primarily in children and should be differentiated from other eczemas, especially from atopic dermatitis. The largest series of IDH are from Jamaica and Brazil. There are an estimated 15 to 20 million infected people in the world, and Brazil is one of the endemic regions. Studies suggest that IDH in children may be a marker for the development of T-cell leukemia/lymphoma (ATL) or myelopathy associated with HTLV-1/tropical spastic paraparesis (HAM / TSP) in adulthood.

A case of human to human transmission of orf between mother and child.

Orf is caused by a parapoxvirus. In adults, it is commonly associated with specific occupations, whereas cases in children tend to be associated with household or recreational exposure. Spontaneous recovery usually occurs within 6 weeks. Infection in humans is believed to be through exposure to an infected animal or fomite. We present a case of a 13-month-old boy who was exposed to orf through his mother, a farmer, who had contracted the disease through administering medication to an infected animal. We believe that this may represent only the fifth case of human to human transmission of orf reported in the literature.

Varicella infection is not associated with increasing prevalence of eczema: a U.S. population-based study.

BACKGROUND: Chickenpox infection early in childhood has previously been shown to protect against the development of childhood eczema in line with the hygiene hypothesis. In 1995, the American Academy of Pediatrics recommended routine vaccination against varicella zoster virus in the United States. Subsequently, rates of chickenpox infection have dramatically decreased in childhood. OBJECTIVES: We sought to understand the impact of declining rates of chickenpox infection on the prevalence of eczema. METHODS: We analysed data from 207 007 children in the 1997-2013 National Health Interview Survey. One-year prevalence of eczema and 'ever had' history of chickenpox were analysed. Associations between chickenpox infection and eczema were tested using survey-weighted logistic regression. The impact of chickenpox on trends of eczema prevalence was tested using survey logistic regression and generalized linear models. RESULTS: Children with a history of chickenpox compared with those without chickenpox had a lower prevalence [survey-weighted logistic regression (95% confidence interval, CI)] of eczema [8·8% (8·5-9·0%) vs. 10·6% (10·4-10·8%)]. In pooled multivariate models controlling for age, sex, race/ethnicity, household income, highest level of household education, insurance coverage, U.S. birthplace and family size, eczema was inversely associated with chickenpox [adjusted odds ratio (95% CI), 0·90 (0·86-0·94), P < 0·001]. The prevalence of eczema significantly increased over time (Tukey post-hoc test, P < 0·001 for comparisons of survey years 2001-13 vs. 1997-2000, 2008-13 vs. 2001-04 and 2008-13 vs. 2005-07). In multivariate generalized linear models, the odds of eczema was not associated with chickenpox in 2001-13 (P ≥ 0·06). CONCLUSIONS: These findings suggest that lower rates of chickenpox infection secondary to widespread vaccination against varicella zoster virus are not contributing to higher rates of childhood eczema in the U.S.

HTLV-1-associated infective dermatitis demonstrates low frequency of FOXP3-positive T-regulatory lymphocytes.

BACKGROUND: Human T-lymphotropic virus (HTLV)-1-associated infective dermatitis (ID) is a rare severe chronic eczema, considered as a harbinger for the development of cutaneous adult T-cell leukemia/lymphoma (ATLL) and/or HTLV-1-associated myelopathy (HAM)/tropical spastic paraparesis (TSP). The pathogenesis of ID remains unclear. High numbers of peripheral blood CD4+ CD25+ FoxP3+ regulatory T cells (Tregs) have been reported in ATLL and HAM/TSP. OBJECTIVE: To investigate the status of Tregs, unknown to date, and the histopathological features of ID. METHODS: We studied 16 skin biopsies from 15 Peruvian adults and children with ID by immunohistochemistry. RESULTS: Histopathological patterns were seborrheic dermatitis-like and lichenoid. Intraepidermal lymphocytes were conspicuous. The infiltrate was composed of a CD3+ T cell infiltrate with a predominance of CD8+ over CD4+ cells. CD4+ CD25+ FoxP3+ Tregs were rare and their numbers were significantly lower than those reported in other inflammatory dermatoses. CONCLUSION: Tregs have an essential role in maintaining immune homeostasis of skin. Treg dysregulation ends in severe clinical manifestations. The clinical presentation of ID, with lesions resembling those seen in patients with atopic dermatitis and with mutations in the FoxP3 gene, is in agreement with a common Treg-deficient skin environment in these disorders, possibly secondary to HTLV-1 infection.

Infective dermatitis associated with HTLV-1 mimics common eczemas in children and may be a prelude to severe systemic diseases.

Infective dermatitis associated with human T-cell lymphotropic virus type 1 (HTLV-1) (IDH) is a chronic dermatitis that has been observed in a variable proportion of HTLV-1-infected children. IDH may serve as an early clinical marker for HTLV-1 infection and an indicator of increased risk for developing other HTLV-1-associated conditions. Factors that lead only some infected children to develop IDH are poorly understood. The variable clinical presentation of IDH, in particular its chronicity, the morphology and distribution of the lesions, and its clinical resemblance to other cutaneous inflammatory conditions, make it necessary to distinguish it from other common dermatoses.

"Eczema coxsackium" and unusual cutaneous findings in an enterovirus outbreak.

OBJECTIVE: To characterize the atypical cutaneous presentations in the coxsackievirus A6 (CVA6)-associated North American enterovirus outbreak of 2011-2012. METHODS: We performed a retrospective case series of pediatric patients who presented with atypical cases of hand, foot, and mouth disease (HFMD) from July 2011 to June 2012 at 7 academic pediatric dermatology centers. Patients were included if they tested positive for CVA6 or if they met clinical criteria for atypical HFMD (an enanthem or exanthem characteristic of HFMD with unusual morphology or extent of cutaneous findings). We collected demographic, epidemiologic, and clinical data including history of skin conditions, morphology and extent of exanthem, systemic symptoms, and diagnostic test results. RESULTS: Eighty patients were included in this study (median age 1.5 years, range 4 months-16 years). Seventeen patients were CVA6-positive, and 63 met clinical inclusion criteria. Ninety-nine percent of patients exhibited a vesiculobullous and erosive eruption; 61% of patients had rash involving >10% body surface area. The exanthem had a perioral, extremity, and truncal distribution in addition to involving classic HFMD areas such as palms, soles, and buttocks. In 55% of patients, the eruption was accentuated in areas of eczematous dermatitis, termed "eczema coxsackium." Other morphologies included Gianotti-Crosti-like (37%), petechial/purpuric (17%) eruptions, and delayed onychomadesis and palm and sole desquamation. There were no patients with serious systemic complications. CONCLUSIONS: The CVA6-associated enterovirus outbreak was responsible for an exanthem potentially more widespread, severe, and varied than classic HFMD that could be confused with bullous impetigo, eczema herpeticum, vasculitis, and primary immunobullous disease.

Infective dermatitis associated with human T-cell lymphotropic virus type 1: evaluation of 42 cases observed in Bahia, Brazil.

BACKGROUND: Infective dermatitis associated with human T-cell lymphotropic virus type 1 (HTLV-1; IDH) is a chronic recurrent eczema affecting HTLV-1-infected children. The epidemiological and dermatological characteristics of IDH are described, and their principal diagnostic criteria are reevaluated. METHODS: Forty-two patients were included: 40 patients serologically positive for HTLV-1 and 2 seronegative patients who tested positive in polymerase chain reaction (PCR) assays. RESULTS: The mean age at onset of the disease was 2.6 ± 2.4 years (range, 2 months-11 years). The mean duration of breast-feeding was 24.2 months. The lesions were erythematous, scaly, and crusted, always affecting the scalp and retroauricular regions. Crusting of the nostrils was observed in 64.3% of the patients. Of the 36 patients followed up, 23 had the active disease. The age at which IDH disappeared in the others was 10-20 years. CONCLUSIONS: The onset of IDH may occur earlier than reported in the literature. The scalp and retroauricular regions are always affected, and lesions are invariably present in ≥3 areas. Crusting of the nostrils cannot be considered an obligatory factor for the diagnosis of IDH. The recurring nature of IDH was a characteristic found in all cases. Patients with classic IDH lesions who are serologically negative should be investigated by PCR. Therefore, the indispensable criteria for diagnosis are (1) presence of erythematous-scaly, exudative, and crusted lesions involving ≥3 areas, including the scalp and retroauricular regions; (2) recurring nature of the lesions; and (3) a finding of HTLV-1 infection by serology or molecular biology.

Successful treatment of young-onset adult T cell leukemia/lymphoma and preceding chronic refractory eczema and corneal injury by allogeneic hematopoietic stem cell transplantation.

Only some carriers of human T cell lymphotropic virus type I (HTLV-1) develop adult T cell leukemia/lymphoma (ATLL) after a long latency period, and an association has been reported between chronic refractory eczema, known as infective dermatitis, and young-onset ATLL. A 25-year-old female developed ATLL and underwent allogeneic hematopoietic stem cell transplantation (HSCT) in non-remission. She had chronic refractory eczema and corneal injury at the onset of ATLL. Remission of ATLL was achieved, and the HTLV-1 proviral load decreased after HSCT. In addition, her pre-existing eczema and corneal injuries almost disappeared. More than a year has passed since the transplantation was performed, and she has had no recurrence of either ATLL or lesions in the skin and eye. Her clinical course suggests a possible association between skin and eye lesions and HTLV-1 infection. Changes in the immunological condition after HSCT might play a key role. Special attention is needed when HTLV-1 carriers develop eye or skin lesions.