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PubMed1983

Diagnostic criteria in Sézary's syndrome: a multiparameter study of peripheral blood lymphocytes in 32 patients with erythroderma.

In order to define additional diagnostic criteria for the early diagnosis of Sézary's syndrome (SS), peripheral blood lymphocytes of 32 patients with erythroderma, including 8 patients with SS, 4 patients with erythrodermic mycosis fungoides, 14 patients with an erythroderma on the basis of atopic or chronic dermatitis, and 6 patients with erythrodermic psoriasis, were investigated by computer-assisted morphometry. The degree of nuclear indentation, expressed as the nuclear contour index (NCI), was measured on electron micrographs. The mean NCI and the percentages of cerebriform mononuclear cells (CMC), defined by a NCI greater than or equal to 6.5, were calculated. In addition, the percentages of lymphocytes, T and B cells, and the distribution of T-cell sub-populations as defined by Fc-receptors (T mu, T gamma) and monoclonal antibodies (OKT3, OKT4, OKT8, HLA-DR) were determined. Statistical analysis showed as most discriminating parameters for the differentiation between SS and benign forms of erythroderma: high percentages of lymphocytes (50% or more), an expanded OKT3+, OKT4+ population with an OKT4/OKT8 ratio greater than 10, a mean NCI value greater than or equal to 5.5, the presence of more than 20% CMC, as well as the presence of cells with a NCI greater than or equal to 11.5. The total leukocyte and lymphocyte counts, as well as the percentages of B, T, T mu, and T gamma cells had limited value for the early diagnosis of SS.

Aged↗
PubMed1994

An aggressive spindle cell squamous carcinoma arising in a patient with long-standing erythroderma.

Spindle cell squamous carcinoma (SCSC) of the left hand arising in a patient with long-standing erythroderma is reported. Histopathologically, spindle shaped atypical cells were observed neighboring the cells of well differentiated squamous cell carcinoma. These two types of tumor cells, spindle cells and well differentiated cells, were present side by side and merged into each other. The erythroderma had been present for over 20 years, and both clinical and histopathological findings suggested cutaneous T cell lymphoma, but were not diagnostic for mycosis fungoides, Sézary syndrome, or adult T cell lymphoma. Flow cytometry of peripheral blood cells showed a low CD4/CD8 ratio which suggested impaired T cell function. Multiple metastases of SCSC occurred in a short period and the patient died ten months after his first visit to us. The aggressive course of this case was unusual, and may be due to immunological abnormalities associated with the long standing erythroderma with impaired T cell function.

Aged↗
PubMed1993

The specificity of histopathology in erythroderma.

Conflicting views about the diagnostic value of skin biopsy in the investigation of erythrodermic patients are extant. The objective of the present study was to establish the frequency with which a correct diagnosis can be based on histopathological assessment alone. This was achieved by comparison of the "blinded" microscopic diagnosis with the final diagnosis (based on combined clinico-pathologic parameters and response to therapy). In a retrospective review of 56 skin biopsies from 40 patients with erythroderma, we found a positive correlation between the pathological diagnosis and the final diagnosis in 66%; furthermore, when the microscopic characteristics observed in different diagnostic categories were assessed, these proved to simulate those seen in conventional manifestations of the various underlying diseases but tended to be subtle in the setting of erythroderma. We conclude that, despite the homogeneity of the clinical expression of erythroderma, diagnostic histopathological features of the underlying disease are retained in the majority of cases.

Adult↗
PubMed1994

Histopathology in erythroderma: review of a series of cases by multiple observers.

This study examines the utility of objective histopathological studies in the evaluation of adult patients with erythroderma. A series of 56 skin biopsies, from 40 erythrodermic patients, was reviewed sequentially by 4 Canadian dermatopathologists who were unaware of clinical details of the cases. The final diagnosis (gold standard), in each instance, had already been determined by others, based on clinicopathologic data and response to therapy. Direct comparison revealed that the mean accuracy of the histopathological diagnoses was 53% (range: 48-66%), a favorable result in view of the difficulty of the task at hand. Additional points of information which evolved from the study are as follows: (i) identification, by microscopy alone, of spongiotic dermatitis, cutaneous T-cell lymphoma and psoriasis, as underlying causes of erythroderma was more successful than that of drug eruptions and pityriasis rubra pilaris; (ii) the epidermotropism which characterizes cutaneous T-cell lymphoma may be mistaken for inflammatory interface changes seen in drug eruptions and vice versa, thus constituting a pitfall in diagnosis; (iii) finally, it appears that submission of multiple simultaneous biopsies, rather than a single specimen, from patients with erythroderma would be likely to enhance the accuracy of histopathological diagnosis.

Adult↗
PubMed2006

Generalized mucinosis in a patient with erythroderma.

We describe an 81-year-old Japanese patient with erythroderma overlapping with widespread and symmetrical deposits of mucin in the upper dermis. Clinically, the mucinous lesions on the nape and upper trunk were localized papular mucinosis. Histologically, there was a perivascular infiltrate of lymphohistiocytic cells mingled with plasma cells in the upper dermis but no sclerosis. Immunohistochemical staining revealed that more than 90% of these infiltrating plasma cells produced immunoglobulin lambda-chain. Both the erythroderma and generalized mucinosis responded to topical steroid and PUVA therapy. To the best of our knowledge, this is the first case of erythroderma accompanied by generalized mucinosis.

Administration, Cutaneous↗
PubMed1983

Generalized vitiligo after erythroderma.

We report a 65-year-old black man whose onset of an erythroderma was followed in 6 weeks by a generalized vitiligo. The etiology of the erythroderma was unclear, although a prostate adenocarcinoma was found on evaluation. This is to our knowledge the first patient reported with a vitiligo following exfoliative dermatitis, and the first report of two uncommon markers of internal malignancy (erythroderma and vitiligo) occurring with prostate adenocarcinoma in the same patient.

Adenocarcinoma↗
PubMed1989

Two cases of postoperative erythroderma.

Postoperative erythroderma is a rare disease of which the clinical course is almost always fatal. It is characterized by fever, skin rash, hepatic dysfunction and leukopenia, and many of the cases occur after cardiovascular surgery. We have experienced two cases of this disease which are suspected to be related to a large amount of blood transfusion. In the second case, we observed genetical mismatch of the HLA typing between the patient and his family members. This observation strongly suggests the possibility that postoperative erythroderma is a form of graft-versus-host (GVH) reaction induced by transfused lymphocytes. The reason why the patient's immunity is so suppressed as to cause GVH reaction is still unknown. Cardiopulmonary bypass and a large amount of fresh blood transfusion are suspected for immune suppression. The fact that postoperative erythroderma occurs almost exclusively in the Japanese cannot be fairly explained, either. Racial homogeneity in Japan might cause immune tolerance in blood transfusion.

Cardiovascular Diseases↗
PubMed1999

[Erythroderma and alcohol abuse].

We report on 7 patients with chronic alcohol abuse and ichthyosiform erythroderma. Dry skin (i.e. xerosis cutis) was a striking common feature in all patients who did not present any signs of fluid depletion. Two patients had exclusively exsiccational dermatitis; three had atopic disposition and one nummular eczema of the legs. In another patient a drug allergy could be demonstrated. Xerosis cutis seems to be an important precipitating aetiologic factor for erythroderma, and may be a consequence of alcohol-induced diuresis. All patients recovered rapidly under topical treatment with emollients and low potency steroids. We propose that ichthyosiform erythroderma be added to the known skin diseases caused by chronic alcohol abuse.

Aged↗
PubMed1999

Erythroderma in Thai patients.

Erythroderma is a clinical manifestation of dermatoses from different causes. Our objective was to determine its incidence, causes and clinicopathological features. Clinical, laboratory, and biopsy materials of 49 patients diagnosed as having erythroderma were reviewed. They were treated in our department over a 10-year period (1985 through 1994). The male-female ratio was 2:1. The mean age at diagnosis was 51.7 years. The most common causative factors were drugs (38.77%) and preexisting dermatoses (26.5%). Hepatomegaly, jaundice and abnormal liver function tests were found more commonly in the drug allergy group, while in cases with preexisting dermatoses nail involvement was a common finding. Clinicopathologic correlation in our study did not inform the etiology because it showed chronic nonspecific dermatitis or psoriasiform dermatitis, without any clue as to its origin. Drug-induced-erythroderma had an acute onset and a good prognosis with rapid resolution when the causative drug was withdrawn, while histopathology and laboratory findings were largely unrewarding.

Adolescent↗
PubMed2000

[Icthyosiform erythroderma: atypical manifestation of sarcoidosis].

BACKGROUND: Specific cutaneous manifestations of sarcoidosis can be extremely varied. We report one observation of cutaneous sarcoidosis with an exceptional clinical presentation: icthyosiform erythroderma. CASE REPORT: A 33-year-old woman had a dry icthyosiform erythroderma associated with arthralgia and muscular deficit of the pelvic belt in a context of fever and weight loss. Skin biopsy revealed the presence of an epithelioid giant-cell granuloma. This granuloma was also found in the salivary glands and in the bronchial mucosa. The chest X ray showed diffuse reticularis opacities. The diagnosis of sarcoidosis was retained. The patient was treated with oral prednisone (0.75 mg/kg/day). Total skin clearing was obtained after 15 days. DISCUSSION: This observation points out the heterogeneous nature of clinical manifestations of cutaneous sarcoidosis. The icthyosis erythroderma form is exceptional. Correct diagnosis is difficult and must be based on clinical, histological, radiological and biological criteria of sarcoidosis.

Adult↗
PubMed1992

[Erythroderma induced by teicoplanin].

We report the case of a 38-year old man who developed erythroderma during treatment with teicoplanin for staphylococcal septicaemia. The clinical symptoms, which included high fever, shock and erythroderma, were suggestive of staphylococcal toxic shock. After teicoplanin was withdrawn and replaced by vancomycin the disease followed a favourable course. The reintroduction test was positive. This case illustrates the problems associated with the diagnosis of erythroderma in intensive care patients and confirms that it is possible to prescribe vancomycin in cases with allergic reaction to teicoplanin.

Adult↗
PubMed2003

[Anesthetic management of a patient with non-bullous congenital ichthyosiform erythroderma].

Non-bullous congenital ichthyosiform erythroderma is a disorder in the cornification of the skin, histopathologically characterized by hyperkeratosis. Previous reports mentioned that the fixation of endotracheal tubes and ECG electrodes to the skin was likely to be infirm in the patients with this disorder, and that the patients' body temperatures were easily affected by the environment. A 3-year-old girl with non-bullous congenital ichthyosiform erythroderma underwent two operations separately under general anesthesia. We used Hollister Skin Gel to fix the endotracheal tube. This gel helped prevent the irritation associated with the application and removal of adhesives. The patient became severely hypothermic during the first operation. Fortunately, the intraoperative body temperature could be maintained at a normal level during the second operation with the use of a forced-air warming system. Careful perioperative consideration is required for the fixation of endotracheal tubes and the management of body temperature in patients with non-bullous congenital ichthyosiform erythroderma.

Anesthesia, General↗
PubMed1991

[Erythroderma "en nappes claires" as a marker of metastatic kidney cancer. Lasting, successful treatment with rIFN-alpha-2a].

Two years after surgical removal of a renal cell carcinoma, a 70-year-old male patient developed generalized erythroderma and diffuse alopecia within a few days, which could not be further classified as a definite entity. The clinical picture showed well circumscribed roundish areas free of erythema, simulating normal skin (" nappes claires"). The skin disease was found to be resistant to systemic corticosteroids and oral retinoids, whereas in the laboratory work-up multiple lung metastases of the renal cell carcinoma were shown by computer tomography. The clinical picture and the course of the disease suggested a close relationship to the metastasizing visceral tumour, and the erythroderma was seen as a cutaneous marker of the internal neoplasia. A therapeutic trial with recombined interferon alpha-2a led to complete healing of the erythroderma within a few weeks and long-lasting stabilization of the metastasizing visceral tumour over 18 months.

Aged↗
PubMed1986

Expansion of large granular lymphocytes in psoriatic erythroderma. A case report.

A psoriasis patient developed erythroderma after the withdrawal of a self-administered chronic topical glucocorticoid therapy. A marked expansion of cells with the morphological and phenotypic features of large granular lymphocytes was noticed in peripheral blood. Functional investigations revealed that these cells responded poorly to polyclonal activators and exhibited antibody-dependent cellular cytotoxicity and natural killer activity. Blood abnormalities completely subsided in about two months in the absence of any cytostatic therapy and coincided with the recovery from the erythroderma and the spreading of classical psoriatic plaque lesions. This excluded an underlying malignant process. This patient represents the first report of a previously undescribed immunological disorder in psoriatic erythroderma.

Adult↗
PubMed1975

3H-thymidine labelling of epidermis and dermal infiltrate in psoriatic erythroderma.

The comparative pattern of 3H-thymidine (3H-T) pulse labelling in lesions of psoriatic erythroderma (3 cases) and of psoriasis vulgaris (20 cases) has been studied by means of radioautography. In the epidermis, no difference was noted between the two types of lesions concerning the epidermal 3H-T labelling index (L.I.) and the localization of labelled cells. The 3H-T L.I. of "round" cells infiltrating the dermis was much higher in lesions of psoriatic erythroderma (4.03 +/- 0.56%) than in lesions of psoriasis vulgaris (0.62 +/- 0.21%), and the difference is highly significant (p less than 0.0001). The findings indicate that most of these labelled cells could well be lymphoblasts, suggesting that an immunological process is possibly involved in the pathogenesis of psoriatic erythroderma.

Dermatitis, Exfoliative↗
PubMed1976

[Congenital psoriasiform erythroderma with bullous eruptions].

A 39 years old female patient with congenital erythroderma is reported. The condition was considered as the non bullous type of Brocq's congenital ichthyosiform erythroderma until recently. The patient was admitted as she had generalized erythema, desquamation and oedema, pyrexia and oliguria. The scales were fine and small, quite different from those of lamellar ichthyosis. The patient developed a recidivant pustular eruption on palms, face and trunk. So the diagnosis was changed to congenital psoriasiform erythroderma and this was supported by histopathological examination of both scaling and pustular lesions. There was a remarkable improvement on amethopterin therapy. The case is discussed and the relevant litterature is reviewed.

Adult↗
PubMed1998

Ichthyosiform erythroderma and cardiomyopathy: report of two cases and review of the literature.

We report two children with ichthyosiform erythroderma who at the ages of 9 weeks and 8 years, respectively, developed dilated cardiomyopathy, which was fatal in one and required heart transplantation in the other. A link between these conditions is considered likely, either as a primary genetic syndrome or secondary to micronutrient deficiency and/or infection. Owing to its insidious onset, cardiomyopathy may be overlooked, or symptoms attributed to the other conditions such as severe infections and failure to thrive that are common in these patients. We therefore recommend that children with congenital erythroderma are monitored closely, clinically and with echocardiography, for cardiomyopathy.

Cardiomyopathy, Dilated↗
PubMed2001

Ultrastructural features resembling those of harlequin ichthyosis in patients with severe congenital ichthyosiform erythroderma.

Congenital ichthyoses are a group of heterogeneous disorders of cornification. Autosomal recessive congenital ichthyosis (ARCI) can be clinically subdivided into congenital ichthyosiform erythroderma and lamellar ichthyosis. Ultrastructurally, ARCI is classified into four groups: ichthyosis congenita (IC) types I-IV. The genetic background of the ARCI disorders is heterogeneous, but only one disease gene, transglutaminase 1, has been detected so far. We describe six patients with severe congenital ichthyosis from six different Scandinavian families. They could not be classified ultrastructurally into the four IC groups because of atypical findings of electron microscopy. These included abnormal lamellar bodies, alterations in keratohyalin, remnant organelles and lipid inclusions in the upper epidermal cells, which resembled the ultrastructural findings of harlequin ichthyosis (HI), although the HI phenotype was not present at birth. Some clinical features, such as thick scales, erythroderma, alopecia and ectropion were common to all patients. Ichthyosis was usually accentuated in the scalp and four patients had clumped fingers and toes. None of the patients carried the transglutaminase 1 mutation. We conclude that ultrastructural findings resembling those detected in previous HI cases (type 1 and 2) can also be found in patients who do not have classic clinical features of that rare ichthyosis. This may be due to lack of specificity of ultrastructural markers for HI or to its clinical heterogeneity.

Adolescent↗