Imiquimod:A potential option for inhibition of repigmentation of congenital melanocytic nevus after laser ablation

Congenital melanocytic nevi(CMN) are common skin tumors. Large and specially located nevi cannot be completely removed by surgery, posing the risks of both cosmetic deformities and potential malignancy.Nonsurgical treatments, such as laser therapy and physical dermabrasion, can overcome the limitations of surgery;however, the high rate of repigmentation remains an unresolved global challenge. We conducted a self-controlled observational study of a patient with a nevus on the chest. Two areas of the lesion were treated with an Er:YAG laser and 5% imiquimod cream was applied to one of these areas. After nearly 7-months of follow-up, we observed a significant difference in color between the two areas, suggesting that topical imiquimod may inhibit repigmentation and significantly enhance the effectiveness of laser treatment.

Blue rubber blister nevus syndrome: A case report

BACKGROUND Blue rubber blister nevus syndrome(BRBNS)is a congenital,rare disease charac-terized by venous malformations of the skin and internal organs,affecting all systems throughout the body.The pathogenesis is unknown.There is no con-sensus on the treatment of BRBNS.Most of the previously reported cases were mild to moderate with a good prognosis,and this case was a critically ill patient with severe gastrointestinal hemorrhage,disseminated intravascular coagulation(DIC),and severe joint fusion that was different from previously reported cases.CASE SUMMARY An 18-year-old man with early onset of BRBNS in early childhood is reported.He presented with recurrent melena and underwent malformed phlebectomy and partial jejunectomy and ileal resection.The patient had melena before and after surgery.After active treatment,the patient's gastrointestinal bleeding improved.This was a case of atypical BRBNS with severe gastrointestinal bleeding and severe joint fusion,which should be differentiated from other serious joint lesions and provide clinicians with better understanding of this rare disease.CONCLUSION This case of critical BRBNS with gastrointestinal hemorrhage,DIC and severe joint fusion provides further understanding of this rare disease.

Giant Cerebriform Nevus Cell Nevus of the Scalp: A Case Report

Giant cerebriform nevus cell nevus of the scalp is an extremely rare form of congenital melanocytic nevus. Giant cerebriform nevus of the scalp has a major psychosocial impact because of its unsightly appearance with fetid maceration. We report the case of a 35-year-old woman who had a painless, malodorous swelling of the cerebriform scalp measuring 20 × 17 cm in diameter with a wide base of insertion at the occipital level adhering to the deep planes. The excision associated with a skin plasty was carried out. Histology concluded that there was a giant cerebriform naevo-cellular nevus of the scalp.

Blue Nevus Hidden within the Nevus of Ota

A 3-year-old boy presented with bluish patch and scattered blue spots on the left side of his face.After several sessions of laser treatment,the azury patch in the periorbital area became even darker.Histopathology showed many bipolar,pigment-laden dendritic cells scattered in the papillary and upper reticular dermis.Immunohistochemically,these cells were positive for S100,SOX-10,melan-A,P16,and HMB-45.The positive rate of Ki-67 was less than 5%.Finally,the lesion was diagnosed with nevus of Ota concurrent with common blue nevus.Therefore,for cases of the nevus of Ota with poor response to laser treatment,the possible coexisting diseases should be suspected.

History and prospect of melanocytic nevus treatment:A bibliometric analysis

Background:Melanocytic nevus is mainly treated by complete or partial removal.However,predicting the risk of malignant transformation of melanocytic nevi and which treatment patients should receive,surgical or nonsurgical management,to gain the best results and aesthetic outcomes is controversial.Methods:Global literature on melanocytic nevus treatment,published between 1997 and 2022,was scanned using the Web of Science Core Collection database.Microsoft Office Excel,CiteSpace V,VOSviewer,Scimago Graphica,Bibliometrix,and Biblioshiny packages in R were used for the bibliometric analysis to summarize the leading countries,institutions,professors,and research trends in this field.Results:This study included 1723 articles.Publications and citations exhibited positive trends over the past 20 years.The United States had the most productive organizations and publications in the comprehensive worldwide cooperation network,and China was recently one of the most active major participants.Professor Giovanni Pellacani,whose H-index,G-index,and M-index ranked first in this field,founded a virtual biopsy using reflectance confocal microscopy.In addition,Krengel and Kinsler contributed significantly to diagnosing and treating melanocytic nevi.The top 25 keywords in recent years were mostly about the mechanisms and risk factors for the malignant transformation of nevi.Conclusion:The future trend for melanocytic nevi treatment is to specify genotype-phenotype and genotypeoutcome correlations,choose proper therapy to reduce the risk of malignant transformation,and simultaneously achieve the best aesthetic outcomes.

Plantar Spitz Nevus Resembling Malignant Melanoma: Description of Immunohistochemical Findings

Spitz nevus (SN) is predominantly distributed throughout the lower extremities, while an acral location is rare. Since SN occasionally resembles the clinicopathological presentation of malignant melanoma (MM), it presents a diagnostic challenge, especially on glabrous skin. Past reports suggest that several genetic aberrations are associated with specific clinicopathological subtypes of melanocytic tumors. Immunohistochemistry can provide a clue to the presence or absence of a molecular aberration typical of Spitz tumors. We describe a case of a plantar SN with genetic analysis, including anaplastic lymphoma kinase (ALK), ROS proto-oncogene 1 (ROS1), BRAF (V600E) protein, and BRCA1-associated protein-1 (BAP1). However, we were not able to detect a characteristic gene aberration. To the best of our knowledge, no genetic aberrations in plantar SN cases have been reported. A comprehensive understanding of tumor genomics is expected to play an essential role in the classification of melanocytic tumors. Further genetic research on plantar SN is required to establish new criteria for distinguishing between SN and MM.

Blue rubber bleb nevus syndrome:A case report and literature review

Blue rubber bleb nevus syndrome(BRBNS)is a rare disease characterized by multiple venous malformations and hemangiomas in the skin and visceral organs.The lesions often involve the cutaneous and gastrointestinal systems.Other organs can also be involved,such as the central nervous system,liver,and muscles.The most common symptoms are gastrointestinal bleeding and secondary iron deficiency anemia.The syndrome may also present with severe complications such as rupture,intestinal torsion,and intussusception,and can even cause death.Cutaneous malformations are usually asymptomatic and do not require treatment.The treatment of gastrointestinal lesions is determined by the extent of intestinal involvement and severity of the disease.Most patients respond to supportive therapy,such as iron supplementation and blood transfusion.For more significant hemorrhages or severe complications,surgical resection,endoscopic sclerosis,and laser photocoagulation have been proposed.Here we present a case of BRBNS in a 45-year-old woman involving 16sites including the scalp,eyelid,orbit,lip,tongue,face,back,upper and lower limbs,buttocks,root of neck,clavicle area,superior mediastinum,glottis,esophagus,colon,and anus,with secondary severe anemia.In addition,we summarize the epidemiology,clinical manifestations,diagnosis,differential diagnosis and therapies of this disease by analyzing all previously reported cases to enhance the awareness of this syndrome.

Endoscopic therapy for esophageal hematoma with blue rubber bleb nevus syndrome

A 57-year-old woman previously diagnosed with blue rubber bleb nevus syndrome(BRBNS) reported hematemesis. BRBNS is a rare vascular anomaly syndrome consisting of multifocal hemangiomas of the skin and gastrointestinal(GI) tract but her GI tract had never been examined. An upper gastrointestinal endoscopy revealed a large bleeding esophageal hematoma positioned between the thoracic esophagus and the gastric cardia. An endoscopic injection of polidocanol was used to stop the hematoma from bleeding. The hematoma was incised using the injectionneedle to reduce the pressure within it. Finally, argon plasma coagulation(APC) was applied to the edge of the incision. The esophageal hematoma disappeared seven days later. Two months after the endoscopic the rapy, the eso phage alulcerhealed and the hemangioma did not relapse. This rare case of a large esophageal hematoma originating from a hemangioma with BRBNS was treated using a combination of endoscopic therapy with polidocanol injection, incision, and APC.

Radiofrequency ablation micro-dissecting of eyelid nevus with XL-RFA device under operating microscope

AIM: To study the effect of an innovative micro-dissection procedure by radiofrequency ablation (MRA) in removing eyelid nevus.METHODS: Fifty-six consecutive outpatients with eyelid nevus were treated with MRA using a monopolar device. The effect of MRA was determined after following-up for 6mo to 5y.RESULTS: Fifty-two cases (52 eyes, 92.9%) were cured once, and 4 cases (4 eyes, 7.1%) received second treatment for small residual. All cases healed well after surgery, with no pigmentation, no scars, no loss of eyelashes, no deformation of eyelid margin. There was no visual impairment after healing.CONCLUSION: MRA of eyelid nevus using the XL-RFA device is highly efficient without significant complications.

Endoscopic treatment of blue rubber bleb nevus syndrome in a 4-year-old girl with long-term follow-up:A case report

BACKGROUND Blue rubber bleb nevus syndrome(BRBNS)is a rare vascular disease,difficult to diagnose and choose a treatment method,especially in young children.There are several limiting factors to the use of enteroscopy for diagnostics and treatment in pediatric patients,in general.The literature on BRBNS cases is limited and presents various therapeutic approaches.CASE SUMMARY We present here a case of BRBNS involving a 4-year-old female,whose intestinal venous lesions were successfully treated by endoscopic sclerotherapy and aethoxysklerol foam.Skin lesions,typical for BRBNS,appeared on the 8th d of the child’s life and their number increased over the next several months.The child also experienced episodes of critical decrease in hemoglobin level(by as much as 52 g/L)for several years,requiring iron supplementation and several blood transfusions.Video capsule endoscopy revealed numerous vascular formations in the small bowel.The combined findings of gastrointestinal venous formations and skin lesions prompted BRBNS diagnosis.Single-balloon enteroscopy was used to perform sclerotherapy,with aethoxysklerol foam.A positive effect was observed within 19 mo of follow-up.We continue to monitor the patient’s hemoglobin level,every 2 wk,and it has remained satisfactory(>120 g/L).CONCLUSION Endoscopic sclerotherapy can be effective in the clinical management of gastrointestinal manifestations of BRBNS in young children.

Ocular leech infestation initially misdiagnosed as conjunctival pigmented nevus

Dear sir,Iam Dr.Wen-Wei Li,from the Department of Ophthalmology,Zhejiang Provincial People’s Hospital,Hangzhou,China.I write to present a case of ocular leech infestation initially misdiagnosed as conjunctival pigmented nevus.Although human interaction with leeches is not uncommon

Follow the nevus: the cost-utility of monitoring for growth of choroidal nevi

AIM: To develop a model to evaluate the cost-utility of choroidal nevi monitoring recommendations with varying clinical risk factors.METHODS: A Markov model was created to evaluate the cost-utility in cost per quality-adjusted life-year($/QALY) for monitoring patients with choroidal nevus. This probabilistic model was applied both to a hypothetically monitored and unmonitored group of patients beginning at different ages and with varying clinical risk factors of the nevus. Duration of screening was modeled for the remainder of the patients’ life expectancy. Best available clinical data on the prevalence and incidence of choroidal nevi/melanoma, and relative risk of nevus transformation were combined with the initial and downstream costs of screening, downstream costs of melanoma-related mortality, and QALY saved by monitoring, to estimate the best monitoring regimen. Main outcome measures were average $/QALY saved by consensus recommended monitoring scenarios for the duration of a patient’s remaining life expectancy in comparison with no follow-up, and the cost-utility of modified regimens. RESULTS: The $/QALY of the recommended monitoring scenarios varied substantially based on nevus clinical risk factors, patient age, frequency of follow-up, and objective testing utilized. The $/QALY for the recommended monitoring scenario of a flat nevus without risk factors in a 60-yearold patient was $77 180. The $/QALY for monitoring a nevus with 3 clinical risk factors in a 60-year-old patient was $85 393. The $/QALY values for differently-aged patients were larger, and intermediate degrees of risk factors for nevus growth varied, depending largely upon the specifics of the modeled monitoring scenarios.CONCLUSION: The average $/QALY of currently recommended monitoring scenarios fall within economically acceptable standards and could provide insight for formulating appropriate clinical strategies. Cost-utility could be enhanced by targeting higher risk groups and considering less frequent monitoring for the lower risk groups.

Coexistence of esophageal blue nevus, hair follicles and basaloid sqamous carcinoma:A case report

We present the case of a 57-year-old man who underwent esophagectomy for esophageal carcinoma found at barium meal and gastroscopic examination. He was diagnosed as esophageal basaloid squamous carcinoma （BSC） and gastric stromal tumor, which were associated with focal proliferation of melanocytes/ pigmentophages and hair follicles in esophageal mucosa. Melanocytic hyperplasia （melanocytosis） has previously been recognized as an occasional reactive lesion, which can accompany esophageal inflammation and invasive squamous carcinoma. The present case is unusual because of its hyperplasia of not only melanocytes but also hair follicles. To our knowledge, this is the first report of esophageal blue nevus and hair follicle coexisting with BSC.

Sirolimus treatment for neonate with blue rubber bleb nevus syndrome:A case report

BACKGROUND Blue rubber bleb naevus syndrome(BRBNS)is a rare disease that usually presents with multiple venous malformations in the skin and gastrointestinal tract.Lesions located in the gastrointestinal tract always result in chronic gastrointestinal bleeding and severe anemia.The successful management of BRBNS with sirolimus had been reported in many institutions,due to its impact on signaling pathways of angiogenesis.However,the experience in treatment of neonates with BRBNS was limited.CASE SUMMARY A 38-day-old premature female infant born with multiple skin lesions,presented to our center complaining of severe anemia and hematochezia.Laboratory examination demonstrated that hemoglobin was 5.3 g/dL and contrast-enhanced abdominal computed tomography showed multiple low-density space-occupying lesions in the right lobe of the liver.She was diagnosed as having BRBNS based on typical clinical and examination findings.The patient was treated by transfusions twice and hemostatic drugs but symptoms of anemia were difficult to alleviate.A review of BRBNS case reports found that patients had been successfully treated with sirolimus.Then the patient was treated with sirolimus at an average dose of 0.95 mg/m2/d with a target drug level of 10-15 ng/mL.During 28 mo of treatment,the lesion was reduced,hemoglobin returned to normal,and there were no adverse drug reactions.CONCLUSION This case highlights the dosing regimen and plasma concentration in neonates,for the current common empiric dose is high.

Trichilemmomal Horn Arising from a Nevus Sebaceus and Pilar Cyst: A Case Report and Review of the Literature

Nevus sebaceus can give rise to multiple neoplasms, some of which have the potential to become malignant. We describe the occurrence of a previously unreported combination of a cutaneous horn proliferating from a trichilemmoma overlying a multilocular pilar cyst and nevus sebaceus. An elderly woman with a several years’ history of multiple scalp subcutaneous nodules and an enlarging cutaneous horn presented to our clinic. The nodule underlying the large exophytic horn was tender and occasionally bled. The lesion was removed by excisional biopsy due to concern for malignancy. Histologically, a verrucous trichilemmoma was identified overlying irritated pilar cysts and a nevus sebaceus. Phosphatase and tensin homolog (PTEN) mutations are frequently found in trichilemmomas and HRAS mutations in nevus sebaceus. Mammalian target of rapamycin (mTOR) is a part of the final common pathway for HRAS and PTEN raising the therapeutic question if rapamycin can be used to treat these neoplasms.

Long term clinical and dermoscopic follow-up of a child with a Spitz nevus

Background: Spitz nevus is uncommon, benign melanocytic neoplasm that may show some clinical, dermoscopical and histological features of melanoma. It occurs often in childhood, but may appear also in early adulthood. Rare congenital cases have been reported in literature. It is frequently located on the face and the lower extremities, but in some cases may appears on the trunk. Methods: We report a case of a 9-years-old girl presented to our Dermatology Unit because of the presence of a pigmented lesion on her right leg, 4 mm in diameter, which was clinically and dermoscopically diagnosed as Spitz nevus. We described the clinical and dermoscopic features that we observed every six months, over 11 years follow-up period. Objective: Our observation show that the globular, the starburst, the homogeneous patterns and diffuse brown colour with diffuse hypopigmented areas may be the different expression that correspond to possible evolutionary phases of pigmented Spitz nevus.

Reed Nevus (Pigmented Spindle Cell Nevus) in an 11-Month-Old Japanese Infant

Reed nevus (pigmented spindle cell nevus) is a nevus with the histopathological features of abundant melanin, monomorphic spindle-epithelioid melanocytes, spindle cell nests confined within the epidermis and papillary dermis, and occasional upward interepidermal progression of melanocytes. Here, we reported an unusual case of Reed nevus in an 11-month-old Japanese male infant. Infantile Reed nevus should be included as a differential diagnosis for nevus pigmentosus in infancy.

Intracranial Intermediate-Grade Melanocytic Neoplasm: Case Report Associated with Nevus of Ota

Melanocytic lesions of the CNS are rare tumours originating from melanocytes that are present in the leptomeninges. They consist of a spectrum of pigmented tumours ranging from melanocytoma to melanoma. A small group of these tumours have histopathological features between those of a benign melanocytoma and a malignant melanoma;these present as intermediate grade melanocytic neoplasms. Naevus of Ota is a blue hyperpigmented dermal lesion characterized by increased number of melanocytes in the distribution of ophthalmic and maxillary divisions of the trigeminal nerve. The association of an intracranial intermediate-grade melanocytic neoplasm with a nevus of Ota is extremely rare, with only 2 cases reported in the literature to date. As a result, their behavior and progression are still poorly understood. We present the first case of a familial naevus of Ota associated with intermediate-grade melanocytic neoplasm.

Perianal verrucous epidermal nevus masquerading as warts

Verrucous epidermal naevus(VEN) is a rare form of epidermal naevus. We present a case of VEN occurring over the perianal region of a 7-year-old boy. The lesion was initially thought to be an area of chronic dermatitis; however it was refractory to treatment. Histopathology confirmed the diagnosis of VEN. VEN in the inguinogenital region may be misdiagnosed as flexural psoriasis, genital warts or sexual abuse. This is what precisely happened to our patient for a duration of more than 1 year.

IRIS NEVUS SYNDROME (REPORT OF 9 CASES)

This paper reports on 9 rases of iris nevus syndrome diagnosed clinically, eight of which were confirmed by microscopy. The clinicopathologic features, etiology and pathogenesis of iris nevus syndrome are discussed. It was verified that iris nevus syndrome and other clinical variations of iridocorneal endothelial(ICE) syndrome had the same characteristic corneal endothelial defect. Also this syndrome was considered to he related to Chandler's syndrome Clinically.