A case of radiotherapy-induced bullous pemphigoid with unusually delayed onset: A case report.

Bullous pemphigoid is an auto-immune blistering disease that generally affects older patients. Radiotherapy is one of the many triggering factors that have been described. Time to disease onset is variable; cases have been described during the course of radiotherapy while others have occurred up to 9 years later. We report a case of localized bullous pemphigoid on an irradiated site with unusual late presentation, 25 years after radiotherapy for left breast cancer. The pathophysiology of radiation-induced bullous pemphigoid is not clear, but the concept of an immunocompromised district seems to be a plausible explanation for the delayed onset of the disease.

A case of dermatomyositis with anti-TIF1γ antibodies revealing isolated para-aortic lymphadenopathy metastatic recurrence of endometrial cancer: A case report.

Dermatomyositis is an inflammatory myopathy presenting with characteristic cutaneous eruption and may be accompanied by proximal muscle weakness. Dermatomyositis may represent a paraneoplastic syndrome in 15%-25% of cases and has rarely been associated with endometrial cancer. Herein, we report a case of dermatomyositis with anti-TIF1γ antibodies as the first clinical manifestation revealing isolated para-aortic lymphadenopathy metastatic recurrence of endometrial cancer after 4 years of remission. Interestingly, dermatomyositis rash completely resolved after lymphadenectomy. This case highlights the importance of early dermatomyositis diagnosis, thorough cancer screening, and that cancer treatment may, in some patients, foster dermatomyositis remission.

Trichodysplasia spinulosa in a renal transplant patient: A case report of successful treatment.

Trichodysplasia spinulosa is a rare cutaneous disease caused by the trichodysplasia spinulosa-associated polyomavirus. It occurs more frequently in immunocompromised patients, particularly in solid organ transplants. A few successful treatments have been described in the literature. In our report, we present a biopsy-proven trichodysplasia spinulosa case in a kidney transplant recipient who rapidly responded to a reduction of his immunosuppressive therapy.

Multifocal cocaine-induced pyoderma gangrenosum: A report of two cases and review of literature.

Pyoderma gangrenosum is often associated with a systemic disease. Cocaine-induced pyoderma gangrenosum, most probably caused by levamisole, has been described recently and typically presents as multiple, large cribriform ulcers. Peri-nuclear antineutrophil cytoplasmic antibody is the most common serological finding. A strong counseling for cocaine cessation, combined with wound care and immunosuppressive therapy, is the mainstay of treatment. We present two cases of cocaine-induced pyoderma gangrenosum and correlate their findings with the typical clinical, histological and serological presentation.

Basal cell carcinoma stroma as a mimicker of lymphatic invasion: A case report.

Basal cell carcinoma metastases are rare. We report a case of an 83 year-old male with recurrent cutaneous infiltrative basal cell carcinoma on the right vertex with tumor aggregates of infiltrative basal cell carcinoma in the lymphatic vessels of the superficial dermis strongly suspected on pathology slide evaluation during Mohs micrographic surgery. D2-40 immunohistochemical stain allows the detection of lymphatic invasion by highlighting endothelial cells. Further histologic review and D2-40 stains performed on the paraffin-embedded sections did not reveal an endothelial cell lining in favor of lymphatic invasion. What was identified as lymphatic invasion was the result of a peculiar basal cell carcinoma stroma. Correct identification of basal cell carcinoma stroma as a mimicker of lymphatic invasion would prevent potentially invasive investigation as well as associated costs. This is of great importance as the prognostic and therapeutic implications can be of significant value for the patient.

Refractory extensive vulvar high-grade squamous intraepithelial lesion in an immunosuppressed transplant patient treated with topical cidofovir: A case report.

High-grade squamous intraepithelial lesion of the vulva is a premalignant condition which may be especially resistant to treatments among immunosuppressed patients. We present our experience with the use of topical cidofovir in a refractory case of extensive vulvar high-grade squamous intraepithelial lesion in a 37-year-old transplant patient. Eighteen cycles of cidofovir over a 2-year period led to a sustained significant improvement, mainly of the mucosal lesions and was well tolerated. To our knowledge, we have not seen this therapy described in transplant patients with extensive high-grade squamous intraepithelial lesion.

Impacts of Environmental Changes on Well-Being in Indigenous Communities in Eastern Canada.

Climate change and natural resource exploitation can affect Indigenous people's well-being by reducing access to ecosystem services, in turn impeding transmission of traditional knowledge and causing mental health problems. We used a questionnaire based on the Environmental Distress Scale (EDS) and the Connor-Davidson Resilience Scale (CD-RISC-10) to examine the impacts of environmental changes on 251 members of four Indigenous communities in the eastern Canadian boreal forest. We also considered the potential mitigating effects of sociodemographic characteristics (i.e., age, gender, parenthood, and time spent on the land) and protective factors (i.e., health, quality of life, resilience, life on the land, life in the community, and support from family and friends). Using linear regression, model selection, and multi-model inference, we show that the felt impacts of environmental changes increased with age but were lower for participants with higher quality of life. The effect of resilience was opposite to expectations: more resilient participants felt more impacts. This could be because less resilient individuals ceased to go on the land when environmental changes exceeded a given threshold; thus, only the most resilient participants could testify to the impacts of acute changes. Further research will be needed to test this hypothesis.

Statin-associated anti-HMGCR immune-mediated necrotizing myopathy with dermatomyositis-like features: A case report.

Anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase (HMGCR) immune-mediated necrotizing myopathy is a subtype of idiopathic inflammatory myopathy which may be associated with statin exposure. It presents with severe proximal muscle weakness, high creatine kinase levels and muscle fiber necrosis. Treatment with intravenous immunoglobulins and immunosuppressants is often necessary. This entity is not commonly known among dermatologists as there are usually no extramuscular manifestations. We report a rare case of statin-associated anti-HMGCR immune-mediated necrotizing myopathy with dermatomyositis-like cutaneous features. The possibility of anti-HMGCR immune-mediated necrotizing myopathy should be considered in patients with cutaneous dermatomyositis-like features associated with severe proximal muscle weakness, highly elevated creatine kinase levels and possible statin exposure. This indicates the importance of muscle biopsy and specific autoantibody testing for accurate diagnosis, as well as significant therapeutic implications.

Improvement in Hailey-Hailey disease with a combination of low-dose naltrexone and oral magnesium chloride: A case report.

Hailey-Hailey disease is a rare autosomal dominant acantholytic disorder due to mutation in the ATP2C1 gene and presents with flaccid blisters in intertriginous regions. Its chronic and relapsing course may negatively impact patients' quality of life. Multiple medical and interventional treatments have been described with various efficacy. Low-dose naltrexone and oral magnesium chloride represent emerging treatments. Sustained improvement in Hailey-Hailey disease has been reported with the former in case series, while others have shown variable results. Oral magnesium chloride has been reported in four patients with possible results after 2-4 weeks. Two recent cases suggest that the combination of both treatments may have a synergistic effect. Herein, we present a 63-year-old woman with long-standing and recurrent bilateral inguinal Hailey-Hailey disease who significantly improved with low-dose naltrexone and oral magnesium chloride, representing the third case described with this combination.

Toxic epidermal necrolysis associated with pembrolizumab.

INTRODUCTION: Stevens-Johnson syndrome and toxic epidermal necrolysis are severe cutaneous drug eruptions characterized by epidermal detachment. Pembrolizumab is a monoclonal antibody that binds to the programmed death-1 receptor, and it has been associated with numerous cutaneous adverse side-effects, including Stevens-Johnson syndrome. CASE REPORT: We describe a 63-year-old male with metastatic lung adenocarcinoma who developed a rapidly progressing maculopapular rash three days after a first dose of pembrolizumab. On day 16, the rash affected more than 80% of body surface area with detachment of large sheets of necrolytic epidermis in 30-40% of body surface area. However, the patient only presented with mild mucosal involvement. Histopathologic examination of a skin biopsy showed a subepidermal blister with overlying prominent full thickness epidermal keratinocytic necrosis and a superficial perivascular infiltrate of lymphocytes. A toxic epidermal necrolysis secondary to pembrolizumab was then diagnosed. Management and outcome: In addition to supportive cares, the patient received corticosteroids and cyclosporine. The patient responded rapidly to the immunosuppressant therapy, and nearly complete re-epithelialization was achieved 24 days after the start of the reaction. DISCUSSION: In our review of the literature, 15 other cases of Stevens-Johnson syndrome/toxic epidermal necrolysis were reported with programmed death-1/programmed cell death ligand-1 inhibitors. To our knowledge, this is the first case of toxic epidermal necrolysis secondary to pembrolizumab published in the literature. The American Society of Clinical Oncology guidelines suggest that cyclosporine, in addition to corticosteroids, be initiated when toxic epidermal necrolysis is suspected. Clinicians should be aware of this rare dermatological emergency with the increasing use of pembrolizumab in oncology.

Congenital sideroblastic anemia associated with B cell immunodeficiency, periodic fevers, and developmental delay: A case report and review of mucocutaneous features.

This is a 40-year-old woman with sideroblastic anemia with B cell immunodeficiency, periodic fevers, and developmental delay syndrome, who has genital and extragenital lichen sclerosus on the abdomen and the upper back that have become erythematous and painful during febrile episodes. This report summarizes the published cases of sideroblastic anemia with B cell immunodeficiency, periodic fevers, and developmental delay and highlights associated mucocutaneous features.

Cutaneous squamous cell carcinoma arising in hidradenitis suppurativa: A case report.

We present a case of a 64-year-old man who presented with a rapidly growing tumor in the left buttock and intergluteal cleft area, which was affected by hidradenitis suppurativa. The patient was on tumor necrosis factor-alpha inhibitors for hidradenitis suppurativa for 2 years prior to the development of the mass. Initial biopsy of the mass showed a well-differentiated squamous cell carcinoma with spindle cells and positive epithelial immunomarkers. Subsequent excisional biopsy of the tumor showed an infiltrating poorly differentiated squamous cell carcinoma composed of islands of atypical sarcomatoid spindle cells. Squamous cell carcinoma arising in hidradenitis suppurativa is a rare complication which may occur secondary to chronic inflammation and epidermal hyperproliferation in hidradenitis suppurativa-affected areas.

Exaggerated Arthropod Bite Reaction During Pregnancy: A Case Report.

INTRODUCTION: The diagnosis of exaggerated bite reactions is based on the clinical and pathological characteristics of the lesions. These reactions can be an indicator of impending immune suppression. METHODS: The authors report the case of a 35-year-old pregnant woman who presented with a pruriginous vesicular and pustular eruption over her thighs and buttocks. The clinical and pathological findings were compatible with an exaggerated bite reaction. The patient did not report any severe or exaggerated reaction to insect bites in the past. It was her first episode. CONCLUSION: Exaggerated bite reactions have been described with hematological malignancies, mostly chronic lymphocytic leukemia. In our literature review, we did not find any reports of severe local bite reactions occurring during pregnancy. We hypothesize that the changes in the immune system during pregnancy might explain the development of exaggerated bite reactions in our patient.

Use of bilaminar grafts as life-saving interventions for severe burns: A single-center experience.

BACKGROUND: Skin coverage remains a significant hurdle in large-sized burns. Recent advances have allowed to grow Bilaminar Cultured Skin Autografts (BCSGs) from patients' own donor sites. The aim of this study was to report long-term outcomes in patients with large-sized burns having received BCSGs. METHODS: Nine patients received BCSGs from January 2010 to May 2015. Except one patient who died during hospitalization, all patients were contacted. Four agreed to partake in the study. Patients were tested with the Vancouver Scar Scale (VSS), QuickDASH questionnaire and Burn Specific Health Scale (BSHS). Incisional biopsies of BCSGs were compared with patients' autografts. RESULTS: From nine patients, mean age was 40 years and mean TBSA was 70.3%. For the four patients included, score averaged was 2.25 on the VSS, 29.5 on QuickDASH, 36/36 for psychosocial items and 63/84 for functional abilities on the BSHS. Compared with autografts, BCSGs demonstrated better pliability VSS and functionality. Biopsies showed no evidence of malignancy or atypical changes, but areas of hyperpigmentation. CONCLUSION: This is the first report investigating the long-term outcome of a newly developed BCSG. BCSGs demonstrated comparable results with patients' autografts, functional outcomes on self-reported questionnaires and excellent psychological states. Precaution given the extensive unexpected hyperpigmentation must be taken and a randomized controlled study is underway.

An interesting case of pyoderma gangrenosum with immature hystiocytoid neutrophils.

We present a unique case of a 36-year-old male who developed more than 20 pyoderma gangrenosum (PG) ulcers showing on histopathology a dense inflammatory infiltrate composed of histiocytoid mononuclear immature cells with a strong positivity for myeloperoxidase and Leder stain, suggesting a myeloid lineage in the absence of a concomitant myeloproliferative disorder. Histiocytoid Sweet syndrome (SS) is now recognized as a histological subtype of SS. Although PG and SS belong to the spectrum of neutrophilic diseases, to the best of our knowledge, this is the first case of a "Histiocytoid pyoderma gangrenosum" encompassing immature granulocytes in the absence of leukemia cutis.