Bilateral Acromial Dimples: A Case Report and Review of the Literature

Bilateral acromial dimples are uncommon in pediatric dermatology. They are usually found as a sporadic finding with limited clinical symptoms but may cause concern for parents. They can occur spontaneously or be inherited. Bilateral acromial dimples may occasionally be present as part of more complex syndromes. This article reports an 18‐month‐old girl with congenital bilateral acromial dimples and presents a classification of this unusual entity. A literature review of English and non‐English publications was performed. We report the second case of bilateral acromial dimples seen in the context of maternal cocaine use during pregnancy. Our case is probably sporadic and nonsyndromal. We suggest that bilateral acromial dimples be classified as syndromal or nonsyndromal and that nonsyndromal cases be subdivided into inherited and sporadic. Although bilateral acromial dimples can be seen in 18q deletion syndrome, Apert syndrome, Say syndrome, and a recently described syndrome in Brazil, our review of the literature does not support the association with trisomy 9 syndrome. Dermatologists need to be aware of this unusual cutaneous finding and potential syndromal associations. The significance of the association with fetal exposure to cocaine during pregnancy has yet to be determined. The biological mother was not available for assessment for bilateral acromial dimples.     

Incontinentia pigmenti in a Male Infant with Klinefelter Syndrome: A Case Report and Review of the Literature

Abstract: Incontinentia pigmenti, also known as Bloch–Sulzberger syndrome, is a hereditary, X‐linked dominant disorder characterized by abnormalities of skin, hair, teeth, eyes, and the central nervous system. It is classically considered a male‐lethal disorder leading to recurrent miscarriages of male fetuses. We report a rare case of a surviving baby boy with the classic clinical features of incontinentia pigmenti that can be explained by Klinefelter syndrome.     

Severe Febrile Mucha-Habermann''s Disease in Children: Case Report and Review of the Literature

Mucha-Habermann disease, or pityriasis lichenoides et varioliformis acuta, is usually a benign, papulosquamous, cutaneous disorder. It has also been reported in a severe form with fever and systemic symptoms both in children and adults. We report a 12-year-old boy with the febrile, ulceronecrotic type. A review of similar cases in the literature shows a 16% frequency of acute necrotic lesions, as well as rare complications such as fever, superinfected lesions, bacteremia (most often with Staphylococcus aureus), and rheumatologic manifestations such as arthritis and scleroderma. There is no definitive treatment, but tetracycline, erythromycin, methotrexate, and ultraviolet light are used most frequently. The most common histologic feature is mononuclear perivascular infiltrates. Mucha-Habermann disease can mimic other common entities such as varicella and insect bites.     

Cutaneous Sparganosis: A Case Report and Literature Review

A sixty‐nine‐year‐old male patient, without a significant prior medical history, presented with a rapidly enlarging, 2 cm non‐tender nodule on the right lower eyelid. The lesion persisted in spite of an incision and drainage. Following an excision, the histology revealed a diffuse infiltrate of atypical mononuclear cells within the dermis. The cells were large, monomorphic, with irregular to convoluted nuclei, prominent nucleoli, amphophilic cytoplasm. They were very mitotically active. Immunohistochemical stains for CD45, CD3, and CD30 were strongly positive. Pancytokeratin, CD20, and ALK (Anaplastic Lymphoma Kinase fusion protein) were negative. The histopathologic diagnosis was CD30 (Ki‐1) positive anaplastic large cell lymphoma. The margin was positive for involvement by lymphoma. The lesion was re‐excised with no evidence of residual involvement by lymphoma. A primary cutaneous anaplastic large cell lymphoma was favored over systemic involvement based on the clinical presentation of a single eyelid nodule and ALK negativity. He was referred to a hematology‐oncologist for further management. Anaplastic large cell lymphoma is a distinctive type of malignant lymphoma with a relatively favorable prognosis. It frequently involves the skin, however, to the best of our knowledge, presentation of this lymphoma type as a single lesion on the eyelid is extremely rare.     

Syringocystadenocarcinoma Papilliferum: A Case Report and Review of the Literature

Syringocystadenocarcinoma papilliferum (SCACP) is a rare malignant adnexal neoplasm, which is considered as a malignant counterpart of syringocystadenoma papilliferum (SCAP). Clinically, SCACP appears as a nodule, inflammatory plaque, or tumor. The lesion is usually covered with crusts, which are formed by secretion of the apocrine epithelial cells. Histologically, SCACP resembles SCAP, with cystic papillomatous invaginations connected to the skin surface by funnel-shaped structures lined by infundibular epithelium. The stroma of the tumor consists of a dense inflammatory infiltrate of plasma cells and lymphocytes. SCACP differs from SCAP in terms of the architectural and cytological features of the tumor cells, and is characterized by higher nuclear cytoplasmic ratio, nuclear irregularity, coarse chromatin, and increased mitotic activity. However, the immunohistochemical findings of SCACP vary. Since only 49 cases of SCACP have been reported in the English literature, the clinical and histologic characteristics of SCACP have not been fully established. Further studies on the diagnostic criteria for SCACP are warranted. Here, we report a rare case of SCACP and present a review of other relevant literature.     

Cockarde Nevus: A Case Report and Review of the Literature

Benign acquired melanocytic nevi usually display an even and uniform architecture and coloration easily recognized by the dermatologist. The cockarde nevus, a variant of the melanocytic nevus, is an exception. It displays a targetlike morphology with histologic characteristics of a compound nevus. We believe because the lesion has been rarely reported in the American literature, it is often mistaken in this country for a dysplastic nevus.     

淋巴瘤样丘疹病1例

患者男,27岁。躯干、四肢反复发生暗红色丘疹、结节、坏死、结痂4月余,无痛,偶有瘙痒。皮肤科情况:躯干、臀部及四肢暗红色丘疹、结节,部分中央坏死、破溃,结黑褐色厚痂,留有萎缩性瘢痕或色素沉着。皮损组织病理示:真皮结节状淋巴及大组织样细胞浸润,可见多数异型细胞,约占30%,少数细胞呈双核或多核;免疫组化染色示:CD3(+),CD4(+),CD30(+),CD68(+),CD8个别(+),CD20个别(+),Ki-67约10%(+)。诊断:淋巴瘤样丘疹病。     

皮肤浆细胞增生症1例及相关文献复习

皮肤浆细胞增生症是一种罕见的疾病,其特征主要是皮肤良性多克隆浆细胞浸润,通常与多克隆γ-球蛋白血症相关.其典型的临床表现为红褐色斑、斑块或结节,高γ-球蛋白血症,一半以上的患者伴有不同程度的淋巴结病.据以往文献报道,此病主要发生于日本中老年人群.截止到目前为止,关于中国人皮肤浆细胞增生症的报道尚少.在此报道52岁的中国女性浆细胞增生症1例,并复习以往相关文献,对其流行病学、临床特征、病理特点、病因病机、诊断、治疗及预后等进行总结分析.     

Nivolumab-Induced Alopecia Areata: A Case Report and Literature Review

Nivolumab (anti-PD-1) currently used in many cancers. With the usage of nivolumab increased, many cutaneous side effects were reported including maculopapular rash, lichenoid reactions, vitiligo, bullous disorders, psoriasis exacerbation, and alopecia areata (AA). Here, we report AA after nivolumab for treatment of hepatocellular carcinomas (HCC). A 55-year-old male presented with multiple hairless patch from 1 month ago. He suffered HCC and treated with nivolumab for 6 months after hepatectomy. He treated for hair loss with triamcinolone intra-lesional injection without improvement. We performed skin biopsy on the scalp. Histopathologic findings revealed decreased of hair follicles on the horizontal section with lymphocyte infiltration on the perifollicular area on the vertical section. Clinicopathologic findings were agreed with AA. Considering lack of previous history of AA and hairless patches with 6 months after nivolumab injection, we diagnosed him as nivolumab induced AA. Treatment included topical steroid, and minoxidil. No regrowth of hair was noted after 4 months of follow-up. Nivolimumab induced AA is rare side effect. Pathogenesis of nivolumab induced AA remain unclear. But our case is likely related to nivolumab, known to induce immune related adverse events, and given in the delay of a few months between introduction and the occurrence of the hair loss. Here, we reports nivolmumab induced AA; rare side effect.     

播散性皮肤组织胞浆菌病1例及文献复习

患者女,31岁。多发性骨关节疼痛伴发热8月,多发性皮肤脓肿4月。脓液及组织真菌培养可见双相真菌荚膜组织胞浆菌,经rDNA的ITS区鉴定该菌与荚膜组织胞浆菌荚膜变种高度同源。诊断:播散性皮肤组织胞浆菌病。予口服伊曲康唑0.2g(2次/d)和5-氟胞嘧啶1g(3次/d)治疗,5个月后患者病情明显好转。并对相关文献进行复习。     

Congenital Dermatofibrosarcoma Protuberans: A Case Report and Literature Review

Congenital dermatofibrosarcoma protuberans (DFSP) is an extremely rare skin tumor that is commonly misdiagnosed, or is often diagnosed long after the initial presentation. Although many cases of DFSP are diagnosed in adulthood, there are some differences between adult DFSP and congenital DFSP. We report a case of congenital DFSP that was initially misdiagnosed as a simple vascular lesion. The delay in diagnosis led to the considerable growth of the lesion, such that a huge scar was left after the surgical treatment. The major differences between adult and congenital DFSP are discussed through a literature review. Clinicians should be aware of the characteristics of congenital DFSP, to reduce misdiagnosis and the delay of diagnosis from the initial presentation.     

Lichen Nitidus Presenting with Nail Changes—Case Report and Review of the Literature

Lichen nitidus of the nail is rare and can precede the onset of skin lesions. Delayed diagnosis is common. We present an unusual case of lichen nitidus–associated nail changes that preceded the onset of skin lesions in a 4‐year‐old Indian girl. We also conduct a review of six other cases of lichen nitidus with nail involvement from the English‐language literature. Clues to the diagnosis of lichen nitidus include violaceous or pigmentary changes of the nail fold and subtle lichenoid papules on the affected digits. Lichen nitidus of the nails appears to be less severe than nail changes of lichen planus and is generally self‐limiting. Understanding the natural history of lichen nitidus of the nails will help physicians better counsel patients and their families.     

Congenital Lupus with Multiorgan Involvement: A Case Report and Review of Literature

Neonatal lupus erythematosus (NLE) is an autoimmune disease caused by transplacental antibodies that can damage fetal tissue and cause various findings. With the exception of congenital heart block, which can be easily recognized at birth because of neonatal cardiac monitoring during the delivery and immediately after birth, most cases of NLE are recognized within days to weeks of life, but fewer than 10 cases with findings present at birth have been reported. We report the case of a newborn with extensive cutaneous eruption at the time of birth and multisystemic involvement, including hematologic, respiratory, hepatic, and neurologic involvement.     

Neutrophilic Eccrine Hidradenitis: A Case Report and Review of the Literature

Neutrophilic eccrine hidradenitis (NEH) is a recently recognized dermatosis occurring in patients receiving chemotherapy for a variety of malignancies. We report the second pediatric case. An 11-year-old boy with non-Hodgkin's lymphoma developed widespread erythematous papulopustules beginning two days after receiving high-dose cytarabine (Ara-C) in preparation for a bone marrow transplant. The lesions spontaneously regressed in two weeks. Histologic examination revealed a neutrophilic infiltrate around and within the eccrine ducts and secretory coils. Bacterial, fungal, and viral cultures were negative. These findings are characteristic of NEH. The condition should be differentiated from infectious dermatoses that may require treatment.     

Follicular Porokeratosis at alae Nasi; A Case Report and Short Review of Literature

Porokeratosis are disorders of keratinization, characterized histologically by a parakeratotic column, the cornoid lamellae, and clinically by a distinct peripheral ridge. Porokeratosis usually occurs on the trunk or extremities, and facial lesions are rare. Follicular involvement, identified by follicular localization of cornoid lamellae, is uncommon and described only in association with disseminated superficial actinic porokeratosis (DSAP) and porokeratosis Mibelli (PM). A 34-Year- old woman is presented with asymptomatic 1 cm plaque lesion with hyperkeratotic ridge and depressed center on her nose since three years. Histopathological examination of a biopsy revealed cornoid lamellae confined exclusively to the follicle.     

类风湿性嗜中性皮炎1例并文献复习

报告1例类风湿性嗜中性皮炎。患者女,56岁,躯干四肢红斑丘疹脓疱伴痛痒8个月余。组织病理:浅表结痂,轻度角化过度,局部表皮轻度增生,可见少量角化不良细胞,表皮下脓肿形成,真皮浅中层见较弥漫中性粒细胞浸润,并可见少量嗜酸性粒细胞浸润,血管内皮肿胀伴中性粒细胞浸润,未见明确血管炎改变。诊断:类风湿性嗜中性皮炎。结合病例及相关文献,对此少见病发病机制、临床及病理、治疗等进行综合分析。     

Anterior Cervical Hypertrichosis: A Case Report and Review of the Literature

Anterior cervical hypertrichosis (ACH), or “hairy throat,” is a rare form of localized hypertrichosis that refers to the presence of a tuft of terminal hair on the anterior neck. Only 40 cases of ACH have been reported in the literature. Although it is usually an isolated finding, it may be associated with systemic disorders such as neurologic abnormalities (peripheral neuropathy, developmental delay, mental retardation), ophthalmologic disorders (optic atrophy, chorioretinal changes), hallux valgus, and dorsal hypertrichosis. Thus it is strongly advised to take a thorough family history and to perform clinical examinations and investigations (neurologic and ophthalmologic examination, electromyography, X‐ray of the feet) in all patients with ACH to exclude possible associated abnormalities. We report the case of a 7‐year‐old Italian girl who presented with this condition as an isolated finding.     

臂桡侧瘙痒症1例及文献复习

目的报道1例臂桡侧瘙痒症,并回顾文献,学习该病的临床特征、影像学变化及治疗,以提高临床医生对该病的认识。方法分析1例臂桡侧瘙痒症患者的临床表现、影像学检查及治疗预后,并复习近年国内外相关文献。结果老年男性患者,近1年无明显诱因出现双侧前臂瘙痒,夜间较重,不能忍受。皮肤科情况:双前臂外侧散在暗红色粟粒大小坚实丘疹,大片褐色色素沉着。患者既往颈椎病多年,颈椎X光片示颈椎退变,C5-C6椎间隙狭窄。颈部核磁共振成像(MRI)示C3-C7多节段椎间盘突出,椎管狭窄,以C5-C6为著。诊断:臂桡侧瘙痒症。治疗:0.1%他克莫司软膏(商品名:普特彼软膏)2次/d外用。2周后瘙痒明显减轻,1个月后皮疹消失,无瘙痒感,停药,3个月随访暂无复发。结论臂桡侧瘙痒症发病可能与颈椎退变相关,治疗可选择外用他克莫司软膏。     

An Indian Boy with Griscelli Syndrome Type 2: Case Report and Review of Literature

Griscelli syndrome 2 is a rare autosomal recessive disorder of pigmentary dilution of hair, skin, splenohepatomegaly, pancytopenia, immune and neurologic dysfunction. Clinical course is characterized by recurrent infection triggered by uncontrolled T-lymphocyte and macrophage activation, called hemophagocytic syndrome. Since the primary presentation is with depigmented hair, we attempt to highlight diagnostic difficulties in such cases in developing countries like ours where pigmentary changes in hair and skin are commonly attributed to severe malnutrition. We also evaluated phenotype of all 10 cases of genotype (c.C550T; p.R184X), collected from published literature worldwide and emphasize the potential role of above mutation as hotspot in Southeast Asian region.     

Periocular Granuloma Annulare: A Case Report and Review of Literature

Granuloma annulare (GA) is a granulomatous dermatosis that rarely presents on the face and is extremely uncommon in the periocular region. We report our experience with the presentation and management of GA lesions on the eyelids of a 17‐year‐old girl. We performed a review of published literature and identified 13 cases of pediatric periocular GA. One additional case was identified upon review of all pediatric GA cases at the Cleveland Clinic Foundation. Review of these cases suggests that periocular GA is a benign condition that spontaneously regresses within a few months. GA nodules have a predilection for the upper eyelids. A greater incidence is noted in African American children. Awareness of the self‐resolving nature of this condition can prevent unnecessary surgical excisions in affected children.