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1/36. Striae and acne following cardiac surgery in a child.

    We report a 13-year-old girl with extensive striae and an acneiform eruption following surgery for complex congenital heart disease. These findings were associated with elevated serum and urinary cortisol levels with loss of diurnal rhythm. The resolution of the eruption and the fading of her striae coincided in time with normalization of her blood parameters on day 72 postoperatively. We conclude that the cause of steroid excess in our patient was stress induced by the cardiac surgery and a complicated and protracted postoperative course. To our knowledge, this is the first report in the English language literature of skin changes due to endogenous hypercortisolaemia caused by intense physical and emotional stress.
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2/36. methotrexate-induced papular eruption in patients with rheumatic diseases: a distinctive adverse cutaneous reaction produced by methotrexate in patients with collagen vascular diseases.

    BACKGROUND: In the past few years, low doses of methotrexate have been used for treatment of patients with rheumatoid arthritis and other collagen vascular diseases, mainly as an immunosuppressive and corticosteroid-sparing drug. Several cutaneous adverse reactions have been described in association with methotrexate therapy. OBJECTIVE: We describe the clinical and the histopathologic features of distinctive cutaneous lesions that appeared in 4 patients with acute bouts of collagen vascular diseases who were receiving methotrexate therapy. methods: We clinically and histopathologically evaluated cutaneous lesions caused by methotrexate therapy in 4 patients, 2 with systemic lupus erythematosus, 1 with rheumatoid arthritis, and 1 with Sharp syndrome. RESULTS: Clinically, lesions consisted of erythematous indurated papules most commonly located on proximal areas of the extremities. Histopathologic examination of these papules showed an inflammatory infiltrate mainly composed of histiocytes interstitially arranged between collagen bundles of the dermis, intermingled with few neutrophils. In some foci of deeper reticular dermis, small rosettes composed of clusters of histiocytes surrounding a thick central collagen bundle were seen. Cutaneous lesions showed a direct chronologic relationship with methotrexate therapy, and they disappeared when the drug was tapered or withdrawn and corticosteroids were increased. CONCLUSION: patients receiving low doses of methotrexate for acute bouts of collagen vascular diseases may experience characteristic cutaneous lesions with distinctive clinical and histopathologic findings shortly after methotrexate administration. We discuss the differential diagnosis with other dermatoses showing similar histopathologic findings that have been described in patients with collagen vascular diseases.
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3/36. panniculitis mimicking lupus erythematosus profundus: a new histopathologic finding in malignant atrophic papulosis (Degos disease).

    We present the case of a 57-year-old woman with a 1-year history of a cutaneous eruption clinically and histopathologically characteristic of Degos disease. In one of the two cutaneous biopsy specimens taken from the trunk lesions, the histopathology consisted of necrosis and sclerosis of the subcutaneous lobules, a finding that to our knowledge has not been previously described in the cutaneous lesions of Degos disease. Two months after cutaneous biopsies were taken, the patient developed ptosis and an episode of acute abdominal pain; intestinal perforation and many characteristic lesions of visceral Degos disease involving the entire small bowel were noted at laparotomy.
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4/36. Generalized eruptive keratoacanthoma (Grzybowski variant).

    We describe a patient with generalized eruptive keratoacanthoma (KA) of Grzybowski showing the characteristic features of this extremely rare condition. Since the first description by Grzybowski in 1950, only 28 additional cases (including the present one) have been reported. This variety of KA most commonly affects patients during the fifth to seventh decade of life and appears as a generalized eruption of hundreds to thousands of follicular papules. The small pruriginous papules often have a keratotic centre and show microscopic features of KA. Marked facial involvement is characteristic and can lead to masked facies with ectropion, as in our patient. The course of the disease is chronic and the response to therapy is poor.
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5/36. Clonal CD8 infiltration of the skin in common variable immunodeficiency: a prelymphomatous stage?

    common variable immunodeficiency is a heterogeneous syndrome characterized by hypogammaglobulinemia and recurrent bacterial infections. patients with this syndrome also have an increased incidence of autoimmune disease and malignancy, most notably lymphoproliferative disorders. The treatment for common variable immunodeficiency is supported by the use of intravenous infusion of immunoglobulins that allows for control of the disease and avoidance of recurrent opportunistic infections. This report describes a young patient having common variable immunodeficiency who presented with a clonal CD8( ) lymphocytic infiltration of the skin clinically manifesting as a widespread papulonodular eruption and resolving with intravenous immunoglobulin therapy. We speculate that such a unique cutaneous presentation may represent a prelymphomatous condition because of the development of an autonomous T-suppressor/cytotoxic cell clone, possibly also responsible for the impaired immunoglobulin production.
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6/36. Benign cutaneous Degos' disease: a case report with emphasis on histopathology as papules chronologically evolve.

    The following case report details a 53-year-old man with a 6-year history of the benign cutaneous or skin-limited form of Degos' disease. Clinically, the patient demonstrated a diffuse eruption of papules on the upper trunk and arms. Many papules demonstrated the classic porcelain-white centers characteristic of Degos' disease, but others exhibited different clinical morphologies that corresponded to the evolutionary stages of papules originally described by Degos. Over the course of several clinic visits, the patient underwent a total of 5 punch biopsies, the histologies of which were correlated with their clinical morphologies. Early papules were skin-colored and demonstrated a superficial and deep perivascular, periadnexal, and perineural chronic inflammatory cell infiltrate associated with interstitial mucin deposition. The overlying epidermis showed a mild vacuolar interface reaction and the histologic appearances at this early stage resembled tumid lupus erythematosus. Fully developed papules were raised with umbilicated porcelain-white centers and a surrounding erythematous rim. Histologically these exhibited a prominent interface reaction with squamatization of the dermo-epidermal junction, melanin incontinence, epidermal atrophy, and a developing zone of papillary dermal sclerosis that resembled the early stages of lichen sclerosus et atrophicus in miniature. These interface reactions were invariably confined to the central portion of the punch biopsy specimen, corresponding to the central porcelain-white area seen clinically. Additional features of fully developed papules included a prominent lymphocytic vasculitis affecting venules, a mild periadnexal infiltrate of neutrophils and/or eosinophils, and interstitial mucin deposition. In late-stage papules, the porcelain-white areas were better developed and the lesion flattened. Histologically, the degree of inflammation was generally sparse and the overall picture mirrored the classic histologic description of Degos' disease with a central roughly wedge-shaped zone of sclerosis surmounted by an atrophic epidermis and hyperkeratotic compact stratum corneum. These late-stage papules closely resembled a miniaturized version of fully developed lichen sclerosus et atrophicus confined to the center of the punch biopsy specimen.
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7/36. acute generalized exanthematous pustulosis with erythema multiforme-like lesions.

    acute generalized exanthematous pustulosis (AGEP) resembles generalized pustular psoriasis, but may manifest targetoid lesions, purpura, and blisters in addition to pustules. We describe a case of AGEP with erythema multiforme (EM)-like features in a 35-year-old woman who presented with acute onset of high fever and a strikingly polymorphic eruption consisting of numerous tiny pustules on erythematous bases, marked facial edema, oral and genital erosions, targetoid vesicular and purpuric lesions, pustules in string-of-pearl configuration and ring-like vesicles. The histology revealed, in addition to subcorneal pustules, vacuolar interface dermatitis with involvement of eccrine glands, and microabscesses in pilosebaceous structures. Systemic corticorsteroid and antibiotics were initiated, resulting in rapid resolution without recurrence. Recognition of EM-like lesions on a background of generalized pustular eruption could facilitate the diagnosis of AGEP and the institution of appropriate treatment.
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8/36. Imatinib-induced acute generalized exanthematous pustulosis (AGEP) in two patients with chronic myeloid leukemia.

    Imatinib mesylate blocks bcr/abl kinase activity effectively, and thus is a promising drug in philadelphia chromosome positive leukemias. While under imatinib treatment high hematological and cytogenetic response rates could be observed, usually only mild non-hematological side-effects like skin rash, edema, and muscular cramps occur. Here we report two severe cases of acute generalized exanthematous pustulosis due to imatinib. In both patients the generalized pustular eruptions could be observed 12 wk after initiation of imatinib treatment. Numerous microbiological investigations excluded an infectious etiology, and histopathology of cutaneous lesions was consistent with acute generalized exanthematous pustulosis. Accordingly, withdrawal of imatinib led to a restitutio at integrum of the integument. Our report confirms another single observation of acute generalized exanthematous pustulosis in chronic myeloid leukemia under imatinib therapy, and confirms that this is a rare but proven adverse effect of imatinib.
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9/36. Kikuchi-Fujimoto disease with papulopustular skin manifestations.

    Kikuchi-Fujimoto disease commonly presents with cervical lymphadenopathy accompanied by fever, myalgia, neutropaenia, and rarely cutaneous eruption. Most cutaneous lesions present as erythematous macules, papules, plaques, nodules, or ulcers on the upper part of the body such as trunk, upper extremities, and face. We present a case of Kikuchi-Fujimoto disease with a papulopustular eruption on the whole body including the lower extremities.
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10/36. Generalized pustules in a healthy woman.

    A healthy 47-year-old woman developed diffuse pustules and edema of the skin after exposure to diltiazem and cephalexin. Bacterial, fungal and viral cultures were sterile suggesting a noninfectious etiology. A skin biopsy showed spongiosis, subcorneal collections of neutrophils, papillary dermal edema and a superficial perivascular mixed cell infiltrate. The clinical and histopathologic findings were consistent with acute generalized exanthematous pustulosis (AGEP). The patient was treated with supportive care and the pustular dermatitis cleared. AGEP is a rare complication of drug therapy and should be considered in the differential diagnosis of patients presenting with acute onset pustular dermatitis. Drug reactions are an uncommon and unpredictable complication of medical therapy. Cutaneous drug reaction rates occur with a frequency of 1% to 8% and can be higher for certain classes of drugs. They can range from mild morbilliform eruptions to more severe forms such as drug-hypersensitivity syndrome, toxic epidermal necrolysis or anaphylaxis. Acute generalized exanthermatous pustulosis (AGEP) is a rare presentation of a drug reaction and can be difficult to distinguish from other pustular dermatoses. Herein we review a case of AGEP and include a discussion of salient clinical and histological features of AGEP.
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