Cutaneous mucormycosis in a patient with acute lymphocytic leukemia

We describe a patient with invasive necrotizing cutaneous mucormycosis caused by Rhizopus oryzae. The patient, who had been suffering from acute lymphocytic leukemia (ALL) for eight months, had erythema and necrosis surrounded by swelling on the dorsum of his left hand. Debridement was performed, and microscopic examination of the obtained specimens revealed mucormycosis. Because amphotericin B was ineffective, amputation at the left shoulder joint was performed. Bone marrow transplantation (BMT) was successfully carried out 22 days after surgery. However, the patient died 162 days after the BMT due to progression of the ALL. Patients such as the present one should be evaluated promptly by tissue biopsy and appropriate cultures, so that vigorous treatment can be started without delay. Where necessary, amputation should be performed.     

原发性皮肤毛霉病

报告1例原发性皮肤毛霉病.患者男,57岁.右前臂埋置静脉套管针17 d后局部出现红肿,迅速坏死并扩大.患者患2型糖尿病及慢性肾功能不全.皮肤科检查示右前臂大片坏死区,上覆黑色焦痂及渗出.取黑痂及渗液行真菌镜检,镜下可见大量粗大、无分隔、垂直分枝的菌丝,培养第2天即有棉花糖样菌落生长,初步诊断为皮肤毛霉病.皮损行组织病理检查,PAS染色示真皮及皮下组织可见较多粗大、无分隔菌丝,确诊为皮肤毛霉病.给予两性霉素B脂质体泵入、坏死组织清创术、术后外敷两性霉素B脂质体溶液及外用人成纤维细胞生长因子.患者伤口愈合良好,随访4个月无复发.     

Primary cutaneous mucormycosis caused by Mucor irregularis in an elderly person

Primary cutaneous mucormycosis is a rare but often lethal severe fungal infection, which usually occurs in immunocompromised patients. We report a case of primary cutaneous mucormycosis caused by Mucor irregularis in an elderly patient. Seven months after the surgical dissection of the involved skin, cutaneous mucormycosis recurred at the peripheral edge of the skin graft. Shortly subsequent to the administration of liposomal amphotericin B, the remaining skin lesion was excised again. M. irregularis is rarely but increasingly reported as a cause of mucormycosis in immunocompetent individuals, especially in Asian farmers. M. irregularis may be largely disseminated in the soils of Asia and thus the trivial trauma at the time of farm work may be a trigger for the onset. These cases tend to leave severe cosmetic damage even in healthy individuals, although the vital prognosis is not affected.     

多变根毛霉引起原发皮肤毛霉病1例

报告1例由多变根毛霉引起的皮肤毛莓病。患者男．33岁。右上肢外伤后弥漫性斑块伴肿胀、化脓7年余。免疫功能检测CD4^ T细胞略低(28％)，皮损组织病理检查示真皮中下层有炎细胞及多核巨细胞浸润，并见粗大较短的无隔菌丝经真菌培养菌种鉴定为多变根毛霉患者曾接受伊曲康唑、特比荼芬和氟康唑等抗真菌药物治疗．均无满意疗效．最后经用两性霉素B治疗痊愈．随访6个月未复发。     

The first case of cutaneous mucormycosis caused by Rhizopus azygosporus

A rapidly enlarging leg ulcer appeared in a 54-year-old woman with systemic lupus erythematosus receiving aggressive immunosuppressive therapy. Skin biopsy revealed proliferation of hyphae in the midst of a neutrophilic abscess. Culture yielded Rhizopus azygosporus. As no organ involvement was detected by thorough examination, the patient was diagnosed as having primary cutaneous mucormycosis. Although intravenous amphotericin B therapy seemed to be very effective, it had to be discontinued due to nephrotoxicity. She unfortunately died of subsequent disseminated fungal infection and cerebral infarction in which the primary cause could not be determined. Minimum inhibitory concentrations of several antifungal drugs to the isolate were examined and amphotericin B proved to be the only agent that may potentially reach the effective plasma concentration. This is the first case report of cutaneous mucormycosis caused by R. azygosporus.     

Impairment of Th cell response in Card9 knockout mice with cutaneous mucormycosis caused by Rhizopus arrhizus

Card9 is a signalling adaptor protein in the downstream of many innate pattern recognition receptors (PRRs) and exerts a significant role in antifungal immunity. To date, Card9 deficiency has been reported to be related to increased susceptibility to many fungal infections. In this study, we established mucormycosis murine model of Rhizopus arrhizus (R. arrhizus) using wild‐type (WT) mice and Card9 knockout (Card9^?/?) mice to investigate the antifungal effect of Card9 against R. arrhizus infection. Card9^?/? mice were more susceptible to R. arrhizus infection than WT mice, which could be related to the impaired NF‐κB pathway activation, local cytokine production and Th cell responses in Card9^?/? mice.     

Cutaneous mucormycosis

The patient, a 59-year-old male, had been diagnosed as having alcoholic hepatopathy 20 years previously. He noticed localized swelling on his left leg after fishing in a river. The patient was diagnosed as having cutaneous mucormycosis upon histological and mycological examination of the skin. Gradual improvement of the symptoms was noted four weeks after administration of itraconazole and fruconazole; these were discontinued after five months.     

河南不规则毛霉致原发性皮肤毛霉病一例

【摘要】 患者女,50岁。右上肢皮肤结节、斑块、红肿、溃烂6年,右臀部斑块2年,右面颊斑块1年。曾单独口服特比萘芬、伊曲康唑治疗无效。右上肢及臀部皮损组织病理及PAS染色均可见真皮内宽大呈直角分支菌丝。真菌培养及分子生物学鉴定为不规则毛霉。联合伊曲康唑和特比萘芬治疗2个月仍无效,改为静脉滴注两性霉素B（总量1 060 mg）治疗,患者右面颊、右臀部斑块肿胀逐渐消退,右上肢包括右手背斑块、红肿、坏死、黑痂消退,皮损愈合成瘢痕,取右上肢皮肤组织再次行真菌培养阴性。随访1年未复发。     

原发于头皮的皮肤淀粉样变一例

原发性皮肤淀粉样变皮损常见于双侧胫前、背部及大腿,也可见于阴茎及面颈部,原发于头皮者少见,诊治具有一定的难度。现报道1例我科诊治原发于头皮的皮肤淀粉样变患者。给予0.1%维A酸外用头皮,治疗2个月,皮损变化不明显,瘙痒缓解,目前进一步随访中。     

不规则毛霉致皮肤型毛霉病一例

患者,女,65岁。反复左上肢结节、斑块、流脓伴疼痛7年,累及右上肢1年。2型糖尿病病史3年。左手背皮损组织病理示表皮棘层肥厚伴角化不全,真皮全层多灶肉芽肿形成伴淋巴细胞、浆细胞浸润,胶原纤维变性,并累及皮下脂肪;真菌培养示白色绒毛状菌落生长,镜下表现为粗大、无分隔菌丝,可见圆形孢子囊;宏基因组测序示不规则毛霉。诊断为不规则毛霉致皮肤型毛霉病。     

伊曲康唑治疗1例原发性皮肤毛霉病

应用伊曲康唑治疗１例罕见的由冻土毛霉黄色（或土黄）型所致的原发性皮肤毛霉病,并对其治疗前后的临床、真菌学及组织病理改变进行了观察。临床、真菌学痊愈。斯皮仁诺对毛霉目某些真菌是敏感的,对原发性皮肤毛霉病的治疗是安全有效的。     

原发性皮肤弥漫性大B细胞淋巴瘤一例

患者,男,51岁。左小腿红斑、结节、斑块伴疼痛3个月。组织病理及免疫组化确诊为原发性皮肤弥漫性大B细胞淋巴瘤,腿型。     

Primary cutaneous nocardiosis due to Nocardia nova in a healthy woman

We describe a 68-year-old healthy woman with an unusual skin manifestation of mycetoma. A dark reddish erythematous plaque was present on the dorsum of the left hand for over 10 years. A skin biopsy revealed sulphur granules in the abscess, and Nocardia nova was identified from the specimen. Treatment with dapsone resulted in significant improvement. We review the clinical features of cutaneous N. nova infections.     

Self‐resolving superficial primary cutaneous mucormycosis in a 7‐week‐old infant

A 7‐week‐old girl, born at 30 weeks' gestational age, presented to clinic for evaluation of a crop of vesicular lesions that were noted after removal of a bandage that had been in place for 4 days. A punch biopsy of the lesion revealed fungal elements that were later identified as Rhizopus spp. The lesion began to self‐resolve, and no further treatment was needed, with full resolution of the lesion by 1 month after presentation. Clinicians should be aware of the variable presentations of mucormycosis and consider fungal infection in the differential diagnosis when evaluating vulnerable patients with skin eruptions.     

Primary cutaneous marginal zone B-cell lymphoma in a patient with chronic lymphocytic leukaemia

Primary cutaneous marginal zone B-cell lymphoma (PCMZL) is a low-grade malignant lymphoma that presents in the skin with no evidence of extracutaneous localization at diagnosis. We present an 80-year-old woman with B-cell chronic lymphocytic leukaemia (CLL) who developed multifocal PCMZL lesions 14 months after CLL diagnosis. PCMZL was clonally similar to the original bone marrow (BM) CLL cells. The specific translocation t(14;18) (q32;q21) with breakpoints in IGH and BCL2 loci was found in a skin specimen, but was absent in BM and peripheral blood (PB) cells. In contrast, a 13q deletion was found in BM and PB CLL cells. The patient was treated with chlorambucil and complete response of PCMZL was achieved. To our knowledge this is the first patient with CLL in whom PCMZL has been diagnosed.     

Primary cutaneous carcinosarcoma: Dermoscopic and immunohistochemical features

A 73-year-old man presented a 9-month history of an enlarging nodule on his right temple. Dermoscopy revealed a non-pigmented lesion with ulceration, fibrosis and pale globules. An excisional biopsy was carried out and histology showed a biphasic tumour with a basal cell carcinoma like epithelial component and a dermal undifferentiated sarcoma, with pleomorphic spindle cells and numerous osteoclast-like giant cells. Based on immunohistochemistry findings, a diagnosis of primary cutaneous carcinosarcoma was made and the patient underwent wide local excision.     

Primary cutaneous aggressive epidermotropic CD8(+) cytotoxic T-cell lymphoma with atypical presentation

Primary cutaneous aggressive epidermotropic CD8(+) cytotoxic T-cell lymphoma is characterized by a proliferation of epidermotropic CD8(+) cytotoxic T cells and an aggressive clinical behavior. Patients present with localized or disseminated eruptive papules, nodules and tumors. We report a case of primary cutaneous aggressive epidermotropic CD8(+) cytotoxic T-cell lymphoma with unusual clinical manifestation. The lesion occurred as multiple brownish macules and flat-topped papules on the hands, feet and face in a 25-year-old woman.