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PubMed1995

Erythroderma: analysis of 247 cases.

The profile of 247 patients with erythroderma during a 23 year period from January, 1962 through March, 1985, with a follow-up period ranging from 1 to 26 years were analysed. The patients presented with diffuse erythema, scaling and pruritus of more than 2 months' duration, and the age ranged from 16 to 60 years. Psoriasis was the most frequent underlying disease with an estimated frequency of 44.9%, the reaction to the use of drugs appeared in 7.3% of total cases and association with reticulosis showed a frequency of 4.1%. The cause of the erythroderma could not be determined in 29.2% of the cases. Six differences in terms of underlying diseases were not observed. One or more skin biopsies along with clinical findings were diagnostic or suggestive of the underlying disease in 63.6% of the cases. Repeated skin biopsies are recommended as the best method for etiologic diagnosis of erythroderma. At P = 0.05 significance level, masculine/feminine ratio of 2:1 was found. The question arises whether causal agent of erythroderma may not be somehow related to different exposure by sex to environmental antigens.

Adolescent↗
PubMed2005

Exfoliative dermatitis. Erythroderma can be a sign of a significant underlying disorder.

64-year-old man presented with a 3-week history of a diffuse, pruritic rash that had started on his trunk and then spread to his entire cutaneous surface, including the palms of his hands and soles of his feet. Physical examination revealed widespread fine scaling and diffuse erythema. Generalized lymphadenopathy was noted. No fever, hair loss, onycholysis, or nail shedding was detected. The patient had neither a personal history of skin disorders or, specifically, atopic eczema or psoriasis nor a family history of eczema or psoriasis. He also had no history of malignancy and was taking no medications. The patient's complete blood cell count with differential was unremarkable. He was treated with moisturizers, topical corticosteroids, and antihistamines and was advised to avoid possible irritants. One week later, the patient returned because of a worsening of his erythroderma. He also reported malaise and chills. Three 4-mm biopsy specimens were obtained from representative areas (ie, back, arm, and abdomen), and a 2-week course of oral corticosteroids was prescribed. The erythroderma greatly improved but worsened shortly after the steroid dose was tapered. The specimens showed psoriasiform hyperplasia with features suggestive of psoriasis vulgaris. The patient was treated with 25 mg of oral acitretin once a day. His erythroderma slowly resolved over 6 months, at which time the acitretin dose was tapered. The patient reported no recurrence of the erythroderma.

Dermatitis, Exfoliative↗
PubMed2005

Usefulness of cutaneous T-cell clonality analysis for the diagnosis of cutaneous T-cell lymphoma in patients with erythroderma.

CONTEXT: Demonstration of a dominant T-cell clone in skin biopsy specimens by a molecular assay constitutes an additional diagnostic criterion to differentiate cutaneous T-cell lymphomas (CTCLs) from inflammatory dermatoses. OBJECTIVE: To determine which patients, depending on their clinical presentations, could most benefit from a cutaneous T-cell clonality analysis in addition to histopathologic analysis for the diagnosis of CTCL. DESIGN: Comparison of sensitivity and specificity of histopathologic analysis and a combination of this method and the detection of a T-cell receptor gamma chain gene rearrangement by polymerase chain reaction denaturing gradient gel electrophoresis performed on skin biopsy specimens obtained at initial presentation. PATIENTS: One hundred forty consecutive patients were classified into 4 groups, depending on their clinical presentation: (1) eczematous patches suggestive of early-stage mycosis fungoides (MF) (IA and IB of the TNM classification) (n = 42); (2) plaques, nodules, or tumors that arise on or are associated with plaques suggestive of late-stage MF (IIB and III of the TNM classification) (n = 16); (3) erythroderma (n = 50); and (4) nodules or tumors that arise in normal skin, suggestive of non-MF CTCL (n = 32). RESULTS: When compared with histopathologic examination, the addition of clonality analysis increased the sensitivity of CTCL diagnosis in all groups of patients except those with cutaneous lesions suggestive of late-stage MF, because the diagnosis was made based on histopathologic analysis alone in 100% of these cases. The main increase in sensitivity of CTCL diagnosis was observed in patients with erythroderma: 62% with histopathologic analysis alone to 87% with the combination of both methods (P = .04). Diagnostic specificity of molecular assays decreased from 100% to 76% (P = .01) in patients with patch lesions and from 100% to 70% (P = .04) in patients with nodules that occurred in normal skin due to the detection of a T-cell clone in 6 patients with follicular mucinosis without a histologic pattern of MF and in 5 of 20 cases of T-cell pseudolymphoma (25%), respectively. In contrast, a T-cell clone was not detected in the 34 patients with erythroderma of inflammatory origin. CONCLUSION: Polymerase chain reaction analysis of cutaneous T-cell clonality could be useful for the diagnosis of CTCL in patients who present with erythroderma.

Clone Cells↗
PubMed2003

[Erythroderma and multiple cutaneous necrosis revealing a dermatomyositis].

INTRODUCTION: We report an original case of dermatomyositis associated with neoplasia, which initial clinical expression was erythroderma and multiple cutaneous necrosis. OBSERVATION: A 64-year-old patient was admitted at hospital for erythroderma. He had a diffuse and inflammatory erythema with thrill, periorbital oedema, periungueal telangiectasia and epidermal necrosis. Physical examination also revealed symmetric proximal muscle weakness as well as hepatomegaly. There were biological signs of myolysis. Complementary investigations revealed a liver carcinoma with lung metastasis. The patient first underwent topical corticosteroid treatment, which provided partial improvement of the clinical and biological signs of disease. Thereafter he was treated with prednisone and tamoxifen. Death occurred at home 4 months after the diagnosis. DISCUSSION: Diagnosis of dermatomyositis was definitely set according to the criteria of Bohan and Peter. Epidermal necrosis, present in our observation, occur classically in dermatomyositis where they are a predictive factor of association with neoplasia. Other recognized predictive factors are the age of the patient and persistent itching. Erythroderma linked to dermatomyositis is a very unusual event: 4 cases have been reported in the literature, only one of which was associated with cancer. On the other hand, numerous cases of diffuse erythema were reported, which are close to erythroderma. It is not possible to set out that this clinical form is a factor of bad prognosis of dermatomyositis. As a matter of fact, in the published cases, one does not find more frequent deceases, pejorative signs or increased frequency of association with neoplasia.

Dermatitis, Exfoliative↗
PubMed1990

[Studies on so-called "postoperative erythroderma": report of four cases].

"Postoperative erythroderma", the pathogenesis of this disease have solved as graft-versus-host disease (GVHD) due to blood transfusion, is fatal and impossible to cure for the time being. Therefore the prevention against the disease is very important. One woman and three men who underwent an operation and blood transfusion at our department died of this disease. They fell into high fever on 11-13 days, erythroderma on 12-16 days, liver dysfunction on 14 days, and leukocytopenia on 17-19 days, after surgery and transfusion. Eventually, they all suffered from thrombocytopenia, diarrhea, renal dysfunction, and sepsis which led to death. The clinical course, macroscopic and microscopic findings of them coincided with those of GVHD. Since 1989, we have tried following methods for prevention of postoperative erythroderma: Reducing blood transfusion, especially fresh blood and fresh thrombocyte plasma, by using predeposited autologous blood, autologous washed erythrocytes collected from the operative area before and after extracorporeal circulation (ECC), concentrated residual blood from the ECC using a hemoconcentrater, and 1,500 rad of cobalt-irradiation of fresh blood, fresh thrombocyte plasma, and blood collected within 7 days prior to the transfusion. Postoperative erythroderma has not been experienced by introduction of these methods since 1989.

Aged↗
PubMed1994

Persistent light reaction with erythroderma caused by musk ambrette: a case report.

Persistent light reaction is an uncommon type of photodermatitis caused mainly by musk ambrette, a synthetic fragrance material commonly used in foods and cosmetics. Erythrodermic persistent light reaction is rare. We report a case of erythroderma with underlying persistent light reaction due to musk ambrette. A 71-year-old man showed a photodermatitis that waxed and waned for five years before it became more persistent and finally evolved into erythroderma. Positive results of a photopatch test to musk ambrette and a low minimal erythema dose to ultraviolet B were noted. A biopsy specimen of the erythrodermic lesion revealed spongiotic dermatitis. The erythroderma and photodermatitis responded to systemic steroids and psoralen/ultraviolet A therapy (total dose: 90 J/cm2). We suggest that persistent light reaction be included in the differential diagnosis of erythroderma.

Aged↗
PubMed1994

Acute erythroderma as an exclusion criterion for idiopathic CD4+ T lymphocytopenia.

BACKGROUND: Idiopathic CD4+ T lymphocytopenia is defined as a CD4+ T lymphocytopenia of less than 0.3 x 10(9)/L that is not associated with human immunodeficiency virus, other immunodeficiency, or immunosuppressive therapy. The associated clinical course and laboratory findings are variable. We describe a subset of patients whose peripheral CD4+ T-lymphocytopenia was transient, and suggest a pathomechanism for this phenomenon. OBSERVATIONS: We describe three patients with cutaneous T-cell lymphoma, atopic dermatitis, or psoriasis in whom acute erythroderma was concomitant with a peripheral CD4+ T lymphocytopenia that normalized after resolution of the erythroderma. Immunoperoxidase staining of skin biopsy specimens and quantitative estimation of CD4+ T lymphocytes in the cutaneous and peripheral blood compartments demonstrated that the peripheral CD4+ T lymphocytopenia in these cases most probably resulted from sequestration of CD4+ T lymphocytes in the skin. The skin of an erythrodermic patient appears capable of sequestering 10(10) to 10(11) CD4+ T lymphocytes, whereas the peripheral blood compartment contains in the range of 10(9) CD4+ T lymphocytes. CONCLUSIONS: We propose that CD4+ T lymphocytopenia can occur as a result of acute erythroderma of multiple causes and that acute erythroderma associated with transient CD4+ T lymphocytopenia be considered as an exclusion criterion for idiopathic peripheral blood CD4+ T lymphocytopenia.

Adult↗
PubMed1996

[Pigmented erythroderma in AIDS. 5 cases].

INTRODUCTION: We report five cases of pigmented erythroderma occurring during AIDS, noteworthy for its unusual hyperpigmented feature, its advent at the terminal stages of AIDS, and an CD8 cells dermal infiltrate. PATIENTS AND METHODS: It is a retrospective study of five patients infected with HIV: a woman infected by transfusion and four homosexual men, average 55 years old. No one was intravenous drug user. They were all severely immunocompromised; HTLV I/II serology was negative. Skin biopsies were studied with light microscopy (Hematoxylin-eosin) and immunohistochemical studies were performed on frozen sections. RESULTS: The patients had an erythroderma of particular interest because of the associated hyperpigmentation, the severe repercussion (pruritus, weight loss), and the difficulty in treating (except systemic corticosteroids). The histology demonstrated a mononuclear dermal lymphocytic infiltrate, without epidermotropism and atypical cytonuclear feature. The phenotype of the infiltrate was uniformly of the suppressor-cytotoxic subset (CD8+, CD4-). COMMENTS: Our cases are like those previously described as "Pseudo-Sezary", mimicking a lymphoma during AIDS. Numerous factors are probably the cause of this hyperpigmented erythroderma: HIV, CD8 cells... CONCLUSIONS: This severe skin disease, complicating AIDS, seems very particular, but not yet clearly defined. In practice, the problem remains the treatment of this severe erythroderma, because only the systemic corticosteroids are effective, but this is debatable during the treatment of AIDS.

Acquired Immunodeficiency Syndrome↗
PubMed1996

[Anesthetic management of a patient with erythroderma].

We gave anesthesia to a patient with erythroderma for distal partial gastrectomy. Erythroderma is an inflammatory disorder in which generalized erythema and scaling occur, either as a specific phase of processes such as T cell lymphoma, as a manifestation of underlying malignancy or in the absence of preexisting diseases. Severe cases of erythroderma may involve disturbances in the cardiovascular, thermoregulatory and metabolic systems which make the management of general anesthesia difficult. Systemic scale also causes a difficulty in placing ECG monitor electrodes and endotracheal tube. In the present case, dyspnea and asthmatic bronchitis occurred postoperatively. Therefore, in a patient with erythroderma, we recommend careful systemic management throughout the perioperative period.

Adenocarcinoma↗
PubMedDate not supplied

Severe congenital generalized exfoliative erythroderma in newborns and infants: a possible sign of Netherton syndrome.

We examined skin biopsy specimens from 17 of 19 newborns or infants with generalized ichthyosiform, exfoliative, seborrheic, or psoriasiform erythroderma. The specimens showed similar characteristic but nonspecific and therefore, at first sight, uninformative histologic features. Morphologically, the skin was affected overall with a persistent outbreak of eczema-like eruptions of subacute or chronic dermatitis. Pronounced dermal inflammatory processes were obvious by their perivascular and interstitial presence as well as exocytosis of lymphocytes, macrophages, and neutrophils. Epidermal barrier function was Impaired by the highly suppressed terminal differentiation, with thin or in part completely absent stratum corneum, decrease of keratin filaments, decrease or lack of keratohyalin granules, and of keratinosomes containing stacks of lipid membranes. As a result, the formation and discharge of epidermal barrier lipids from the keratinosomes that normally provide intercellular lamellar sheets at the granular-horny layer interface contributing to the epidermal barrier, was highly disturbed. The concomitant loss of water, electrolytes, and proteins by fluid exudation caused the patients severe metabolic problems and recurrent infections. The suspicion of Netherton syndrome was eventually confirmed in 18 patients by light microscopic demonstration of bamboo hairs (trichorrhexis invaginata), mostly from the scalp, but also in vellus hairs and eyelashes. Atopy actually belongs to the symptom triad defining Netherton syndrome and is, in our opinion, primarily responsible for the pathologic events within the skin and of the keratinizing parts of the growing hair shafts. Differential expression of the atopic condition determines the appearance of the keratinization disorder of the skin, namely, severe, generalized, exfoliative erythroderma or milder forms of ichthyosis linearis circumflexa Comèl. Retinold treatment seems to be contraindicated in these conditions since their biopharmacologic effects involve suppression of terminal differentiation, which is the proper pathognomonic event. In six patients the condition had a fatal course within months because of hypernatremia, recurrent infections, failure to thrive, and sepsis. Our aim is to call attention to and reaffirm that in congenital or early infantile cases of generalized exfoliative erythroderma. Netherton syndrome should be suspected as the underlying disease.

Child, Preschool↗
PubMed1990

[Non-bullous erythroderma (congenital) ichthyosiforme with perinuclear shells].

A congenital dermatosis with an unusual course is described in a 5-year-old girl. During the first months of life she was found to have erythrokeratodermatic plaques, which developed into a non-bullous (congenital) ichthyosiform erythroderma (CIE) within 1 year. Since then, the clinical picture has remained stable. Histologically, the upper epidermal layers showed ballooning and vacuolization of the keratinocytes, glycogen storage, and focal parakeratotic keratinization. Conspicuous ultrastructural alterations consisted in the formation of shells of fine-filamentous, cross-linked material of unknown nature, binuclear keratinocytes, perinuclear edema formation, and the occurrence of lipid droplets in the horny layer. The n-alkane value was within the normal range. Our histological and ultrastructural findings are largely in accordance with those in a case of "congenital reticular ichthyosiform erythroderma". The disorder seems to represent a special genotype in the heterogeneous group of the non-bullous CIEs. We suggest naming it "non-bullous (congenital) ichthyosiform erythroderma with perinuclear shells".

Cell Nucleus↗
PubMed1985

Heterogeneity in autosomal recessive ichthyosis. Clinical and biochemical differentiation of lamellar ichthyosis and nonbullous congenital ichthyosiform erythroderma.

Nonbullous congenital ichthyosiform erythroderma (CIE) and classic lamellar ichthyosis (LI) can be distinguished by clinical, histopathologic, and biochemical findings reported herein. Whereas all patients with CIE (n = 12) had fine white scales, they were heterogeneous with respect to degree of erythroderma, ectropion, and prognosis. In contrast, all patients with LI (n = 6) had large, dark, platelike scales, severe ectropion that showed no improvement with age, and minimal erythroderma. Although the stratum corneum of patients with LI was two to three times thicker than that of those with CIE, the latter group demonstrated more acanthosis, parakeratosis, hypergranulosis, and less prominent dermal capillaries. Studies of scale-lipid content have indicated biochemical correlates of this clinical heterogeneity. These clinical, histologic, and biochemical findings provide useful guidelines to differentiate CIE from LI and strongly suggest that autosomal recessive primary ichthyosis comprises two distinct diseases.

Adolescent↗
PubMed1988

Benign and malignant forms of erythroderma: cutaneous immunophenotypic characteristics.

In order to determine if immunohistologic features are useful in distinguishing benign from malignant types of erythroderma, we studied the immunophenotype of lesional T cells in 20 patients (8 mycosis fungoides/Sézary syndrome, 12 benign) and found them to be generally similar. In all cases, the majority of T cells were Leu-1+, Leu-4+, and Leu-5+, as is typical of mature T cells. Although in most cases a majority of Leu-3+ (helper/phenotype) T cells were present, in 2 there was a majority of the Leu-2+ (cytotoxic/suppressor) subset and in 12 others, a significant minority (20%-40%) of these cells. Low percentages of Leu-2+ cells (less than or equal to 10%), resulting in high Leu-3+/Leu-2+ ratios, did not distinguish benign from malignant erythroderma. Leu-8 antigen deficiency was common in both mycosis fungoides/Sézary syndrome and benign cases (62% vs 75%, respectively). In contrast, Leu-9 antigen deficiency was present in only one patient in each group. The lack of combined Leu-8/9 antigen deficiency in our patients may be due to a heavy inflammatory T cell component, obscuring the antigen deficiencies seen in most nonerythrodermic mycosis fungoides cases. We conclude that immunophenotypic studies with the use of the current antibody panel show many similarities between benign and malignant forms of erythroderma, as well as some minor differences that may prove diagnostically useful if corroborated by future studies.

Antibodies, Monoclonal↗
PubMed1996

Erythroderma after autologous bone marrow transplantation modified by administration of cyclosporine and interferon gamma for breast cancer.

BACKGROUND: Allogeneic graft-versus-host disease is associated with decreased rates of tumor relapse. The addition of interferon gamma to cyclosporine, given to induce graft-versus-host disease after autologous bone marrow transplantation, increases the extent of the cutaneous eruption. OBJECTIVE: Our purpose was to describe the clinical and histologic cutaneous changes in 10 patients with breast cancer who received interferon gamma to potentiate graft-versus-host disease after autologous bone marrow transplantation modified by cyclosporine. METHODS: Ten women receiving autologous bone marrow transplantation modified by the administration of cyclosporine and interferon gamma were observed clinically with sequential biopsy of the skin weekly and at the time of cutaneous eruptions. RESULTS: Erythroderma (stage 3) developed in five women after the first or second administration of interferon gamma. At least on skin biopsy specimen from 7 of the 10 women showed grade 2 changes of graft-versus-host reaction, including all patients with erythroderma. Epidermal intercellular edema was prominent in these specimens with expression of keratinocyte HLA-DR and intercellular adhesion molecule 1. Induction of keratinocyte HLA-DR and intercellular adhesion molecule 1 expression was not observed in specimens from normal skin during administration of interferon gamma. CONCLUSION: This protocol causes a more widely distributed cutaneous eruption, including erythroderma (50%), than autologous bone marrow transplantation and cyclosporine administration alone (3%). Whether it will affect survival is unknown.

Adult↗
PubMed2001

Heteroduplex analysis of T-cell receptor gamma gene rearrangement as an adjuvant diagnostic tool in skin biopsies for erythroderma.

BACKGROUND: Erythroderma, defined as red skin covering most of the body surface often accompanied or followed by exfoliation, is the clinical manifestation of at least six different underlying etiologies with allergic or irritant contact dermatitis, atopic/asteotic dermatitis, pityriasis rubra pilaris (PRP), psoriasis, and seborrheic dermatitis accounting for the majority of cases. Approximately 10% of cases are due to adverse drug reactions with roughly another 10% due to cutaneous T-cell lymphoma (CTCL), predominantly mycosis fungoides, or leukemia. It is clear from multiple studies that the clinical diagnosis of the underlying entity is often difficult, as these diseases can present in a very similar fashion. A skin biopsy is usually employed in this setting as a diagnostic tool. However, the histopathologic diagnosis of the underlying cause is complicated by the subtlety of the distinguishing histologic features. In this situation, an ancillary technique demonstrating the presence of a monoclonal T-cell proliferation could help to rule in or out CTCL in cases that clinically and histopathologically do not allow a definitive diagnosis. METHODS: We retrospectively studied 25 biopsies from sixteen patients who presented to the Stanford Dermatology Clinic with erythroderma. We examined the specimens morphologically and analyzed the gamma chain of the T-cell receptor (TCR- gamma) by polymerase chain reaction (PCR) followed by heteroduplex analysis for clonality. We then correlated the results of our PCR and heteroduplex analyses with the patients' clinical outcomes. RESULTS: Four biopsies, from three patients, contained clonal TCR-gamma rearrangements; the four biopsies, all of which were equivocal histologically, correlated to diagnoses of mycosis fungoides (MF) or Sézary syndrome (SS). Twenty-one biopsies contained polyclonal T-cell populations. Eighteen of these biopsies represent patients with inflammatory dermatoses. Three of these biopsies, all of which were taken from a single patient, correlate to a diagnosis of MF. CONCLUSION: TCR-gamma PCR heteroduplex analysis seems to represent an important adjuvant diagnostic tool that, used in conjunction with histopathology and clinical history, could help to clarify the underlying etiology of erythroderma.

Aged↗
PubMed2001

Adolescent-onset ichthyosiform-like erythroderma with lichenoid tissue reaction: a new entity?

A patient with severe ichthyosiform erythroderma and lichenoid histological changes is presented. We discuss the clinical and histological differential diagnosis, including lupus erythematosus, lichenoid drug eruption, lichen planus, graft-versus-host disease, lymphoma, keratosis lichenoides chronica, Netherton's syndrome and ichthyosiform erythroderma. None of these is consistent with the features in our case, which may represent either a hitherto unreported form of ichthyosiform erythroderma or possibly a new entity.

Adolescent↗
PubMed2001

Bullous and non-bullous ichthyosiform erythroderma associated with generalized pustular psoriasis of von Zumbusch type.

Bullous ichthyosiform erythroderma (BIE) and non-bullous ichthyosiform erythroderma (NBIE) are rare congenital ichthyoses. Generalized pustular psoriasis (GPP) of von Zumbusch type is a rare and severe form of psoriasis marked by desquamative and pustular erythroderma associated with fever and altered general conditions. We report two adults with an ichthyosis typical of BIE in the first case and NBIE in the second, without any previous history of psoriasis, who presented with a severe and relapsing GPP of von Zumbusch type. Using current knowledge of the genetic relationship between psoriasis and congenital ichthyoses, we discuss the possibility of a common physiopathological link between congenital ichthyoses and GPP, and examine the possible therapeutic problems resulting from this pathological association, especially in BIE.

Adult↗
PubMed2003

Erythroderma: an unusual presentation of pulmonary tuberculosis.

We report an elderly Chinese man who presented with erythroderma. He was treated with topical corticosteroids and emollients, but did not improve. Chest X-ray revealed cavitation in the right upper pulmonary lobe, and laryngeal swab culture was positive for Mycobacterium tuberculosis complex. Polymerase chain reaction performed on a skin biopsy was positive for M. tuberculosis DNA. Within a month of starting antituberculous medication, his erythroderma had almost completely resolved. Pulmonary tuberculosis should be included in the list of differential diagnosis for erythroderma.

Aged↗