Chemotherapy‐induced eccrine squamous syringometaplasia in an infant

There are few reports of chemotherapy‐induced eccrine squamous syringometaplasia in children. We report the first case of an infant developing this condition after treatment with busulfan, fludarabine, and antithymocyte globulin in preparation for bone marrow transplantation. Twenty‐eight days after transplantation, the infant developed faintly erythematous papules and plaques on the bilateral axillae, inguinal folds, and sites of adhesives. Punch biopsy revealed eccrine glands with dyskeratotic cells and focal squamous metaplasia consistent with chemotherapy‐induced eccrine squamous syringometaplasia.     

Necrotizing eccrine squamous syringometaplasia presenting as acral erythema

Acral erythema (AE) and eccrine squamous syringometaplasia (ESS) are distinctive clinico-pathological entities described in patients receiving chemotherapy for malignancies, mainly myelogenous leukemia. In no case of chemotherapy-induced AE have histopathologic changes of ESS been described. We report a patient with acute myelogenous leukemia who had typical AE due to cytarabine with histopathologic ESS. The latter findings in a case of AE may be a useful diagnostic clue for distinguishing chemotherapy-induced AE from palmo-plantar erythema of graft-vs-host reaction.     

Herpetic syringitis associated with eccrine squamous syringometaplasia in HIV-positive patients

Herpetic syringitis has been described as a rare manifestation of herpes virus infection in patients with an immunodeficiency, usually secondary to human immunodeficiency virus (HIV) infection. Eccrine squamous syringometaplasia (ESS) is an infrequent alteration of the eccrine duct epithelium reported in association with several conditions, including chronic ulcers, inflammatory processes, and patients receiving chemotherapy. The association of herpetic syringitis with ESS has not been reported before. We identified 3 cases of herpetic syringitis associated with ESS in patients with the acquired immunodeficiency syndrome. In 2 of 3 cases the signs of herpetic syringitis were limited to the meta-plastic duct epithelium, but in 1 case there were also herpetic alterations without ESS.
The histological features of herpetic infection in HIV-positive patients may be atypical and lack the typical epidermal alterations, observing only an extensive epidermal necrosis. In those cases, the alterations of the eccrine ducts may be a diagnostic clue in the diagnosis of herpetic infection. ESS of the ductal epithelium is probably secondary to the herpetic infection, although it might also stimulate the extension of the herpetic infection. Futher studies are needed to elucidate the association of ESS and herpes virus infection.     

Recall phenomenon with the unusual presence of eccrine squamous syringometaplasia

Summary The recall phenomenon is an inflammatory reaction limited to a previously X-irradiated field when the patient is treated months or years later with certain drugs. Only a few cases have been reported in dermatological journals. We report a patient with lymphoma who was treated with low-dose abdominal irradiation and high-dose irradiation to the knees and who, 2 months laler, when chemotherapy was started, developed a pronounced inflammatory reaction limited to the areas treated with high-dose irradiation. A skin biopsy specimen showed features of radiation damage, marked epidermal changes and extensive eccrine squamous syringometaplasia. This case of the recall phenomenon is of interest because we have found that there is apparently a radiation dose threshold for this event, and because this is the first report of its association with eccrine squamous syringometaplasia.     

Syringomatous hyperplasia and eccrine squamous syringometaplasia associated with benoxaprofen therapy

Two patients are described, each of whom developed multiple eruptive lesions on sun-exposed areas within days after starting benoxaprofen therapy. The lesions were 2- to 5-mm papules suggestive of syringomas, and microscopically showed squamous metaplasia of eccrine ducts and focal necrosis of eccrine keratinocytes as well as prominent eccrine hyperplasia. In both cases, the lesions resolved completely after benoxaprofen therapy was discontinued. A theoretical basis for these findings is discussed.     

A case of bleomycin-induced acral erythema (AE) with eccrine squamous syringometaplasia (ESS) and summary of reports of AE with ESS in the literature

Chemotherapy-induced acral erythema (AE) is primarily induced by hydroxyurea, methotrexate, and cytarabine, although there are rare reports of AE induced by combination chemotherapy containing bleomycin. It is thought that the accumulation of chemotherapeutic drugs in eccrine glands may cause eccrine squamous syringometaplasia (ESS), which is characterized by metaplasia and focal necrosis of the epithelium of the eccrine duct. ESS is occasionally detected in conjunction with AE, but such occurrences are relatively uncommon. This is the first report of AE with ESS induced by the administration of bleomycin alone. We also provide a summary of past cases of AE with ESS in the literature.     

Tularemia‐induced erythema multiforme minor in an 11‐year‐old girl

Tularemia is a rare and potentially life‐threatening infection caused by the highly infectious gram‐negative coccobacillus Francisella tularensis. We present the case of an 11‐year old girl who presented with erythema multiforme minor in the setting of an indolent but progressive soft tissue infection and was found to have tularemia. We review the role of dermatologists in identifying the features of and complications associated with this rare zoonosis and discuss the potential effect of climate change on its incidence.     

Case of severe bullous erythema including intertrigo‐like eruptions with angioedema induced by pegylated liposomal doxorubicin

Pegylated liposomal doxorubicin (PLD) is an anthracycline anticancer agent used in ovarian cancer and a form of doxorubicin enclosed in pegylated liposomes. There are only a few reports on intertrigo‐like eruptions caused by PLD. We describe the first case of severe bullous erythema, including intertrigo‐like eruptions with angioedema, induced by PLD in Japan. We present the case of a 53‐year‐old woman who was diagnosed with stage IIIC ovarian cancer. After receiving three cycles of PLD, the patient developed swelling of the upper lip and painful erythema with blisters and erosions on the axilla, upper back, flank and wrists. The patient was diagnosed with angioedema and severe skin lesions, including intertrigo‐like eruptions induced by PLD. Although treatment with oral prednisolone and topical steroids was effective against these eruptions, the administration of PLD was discontinued because of its ineffectiveness against the primary disease. Several risk factors, such as obesity, perspiration and racial differences, may contribute toward a severe manifestation such as that seen in our patient. Moreover, our case was the first accompanied by angioedema. The mechanism of coexistence of intertrigo‐like eruptions and angioedema is not clear; further studies are required to clarify the pathological mechanism of intertrigo‐like eruptions.