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Scleredema Adultorum Of Buschke: A Case With Multiple Causes

机译：Buschke成年巩膜水肿：多原因病例

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Background: Scleredema adultorum of Buschke is an uncommon skin condition that may accompany following diabetes mellitus, proceeding of infection or may be associated with monoclonal gammopathy,. Our case had it with multiple other underlying causes.Observation: This is report of a case with scleredema of Buschke with history of eight months. The patient had no history of a definite diabetes mellitus, but only an impaired glucose tolerance test. Hyper IgG gammopathy with increasing of ASO titer in association with a history of preceding chronic infection of both legs following accidental fractures since of two years ago were seen also. Histological findings of a biopsy specimen from involved neck skin showed marked thickening of the reticular dermis and collagen fibers has been broaden and separated by clear spaces which showed positive reaction with alcian blue. Anti streptolysin O titer in his serum level was more than 1000 UI/ml (international union / per milliliter). Glucose tolerance test (2 hours after oral eating 70 gram anhydrous glucose powder) showed impaired (>200mg/dL) in three times doing exam, but glycosylated hemoglobin A-1c was as 4.5 as in goal limitation (action suggested >6.4).Results: Therapeutic plan included oral penicillin and cyclosporine and azathioprin that were not significantly effective during the last 7 months, but the patient feels mild improvement in the last month. Conclusion: This is an uncommon case of progressive scleredema adultorum of Buschke in a man with multiple underlying causes with mild impaired glucose tolerance test, increased G type of immunoglobulin (IgG) of 1942 mg/dl (normal range =700-1600mg/dl), and increased serum level of ASO titer, as well as history of chronic over imposing infections on both leg fractures and after operations. Case Report A 32-year old man is referred to dermatology clinic due to bilaterally hardening of neck distributed to both shoulders following of appear of some sparse papules and pustules on the surface of skin. He noted hardening and tightness of the skin firstly over his posterior neck and upper back area approximately 3 moths before referring to this center. Woody sclerosing edema of upper proximal limbs which has been appeared at these site and progressed to anterior chest wall and symmetrically on both arms, These findings in our case were typically in favor of scleredema adultorum (Buschke type) (Fig 1).
机译：背景：Buschke的成年人硬皮病是一种罕见的皮肤病，可能伴随糖尿病，感染的进行或与单克隆性丙种球蛋白病相关。我们的病例有其他许多潜在原因。观察：这是一例伴有8个月病史的Buschke巩膜水肿的报告。该患者没有明确的糖尿病病史，只有糖耐量测试受损。自两年前以来，还发现伴随ASO滴度增加的高IgG丙种球蛋白病，以及先前因意外骨折导致双腿慢性感染的历史。从受累颈部皮肤的活检标本的组织学发现表明，网状真皮明显增厚，胶原纤维被加宽并被透明空间隔开，透明空间显示出与阿尔辛蓝呈阳性反应。他的血清抗链球菌溶血素O滴度超过1000 UI / ml（国际联盟/每毫升）。葡萄糖耐量试验（口服70克无水葡萄糖粉口服2小时后）在3次检查中显示受损（> 200mg / dL），但糖化血红蛋白A-1c达到目标限制的4.5（行动建议> 6.4）。 ：治疗计划包括口服青霉素，环孢素和硫唑嘌呤，在最近7个月内效果不显着，但患者在上个月感觉有所改善。结论：这是在男性中发生多种原因的轻度葡萄糖耐量测试受损，G型免疫球蛋白（IgG）升高的1942 mg / dl（正常范围= 700-1600mg / dl）的男性中常见的Buschke进行性硬化性水肿的罕见病例。 ，并增加血清ASO滴度水平，以及腿部骨折和术后长期过度感染的病史。病例报告一名32岁的男子因皮肤表面出现一些稀疏的丘疹和脓疱而双侧分布在双肩的颈部变硬而被转到皮肤科门诊。他指的是先在后颈部和上背部上方约3个月的飞沫中发现皮肤变硬和紧绷，然后再指向该中心。上肢近端的木质硬化性水肿已出现在这些部位，并逐渐发展到前胸壁并对称地伸在两臂上。在我们的病例中，这些发现通常有利于成年巩膜水肿（Buschke型）（图1）。

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