Klippel–Trenaunay syndrome is a rare congenital malformation predominantly affecting lower limb. In most cases, it is characterized by a classic triad of cutaneous capillary malformation (port-wine stain), lymphatic and venous abnormalities, in association with variable soft tissue and bone overgrowths. We describe a 48-year-old male presenting on the genitalia several whitish vesicles discharging a milky fluid compatible with chyle. Extensive radiology workup revealed pelvic megalymphatic malformations. Pelvic lymphatic ligations and bleomycin sclerotherapy only allowed a partial improvement. Given the high potential of recurrence, the patient will soon undergo a genetic evaluation for PIK3CA gene mutation and may need further systemic treatment with Sirolimus. As this scrotal chylous effusion in the setting of Klippel–Trenaunay syndrome is rare and highly affects the quality of life, we wanted to raise awareness of this entity and its management.

Corresponding Author: Alexandre Lemieux, Department of Medicine, Division of Dermatology, Centre Hospitalier de l’Université de Montréal, 1000 rue SaintDenis, Montréal, QC, Canada. Email: alexandre. lemieux. 2@ umontreal. ca Journal of Cutaneous Medicine and Surgery 2022, Vol. 26(1) 103–104 © The Author(s) 2021 Article reuse guidelines: sagepub. com/ journalspermissions DOI: 10. 1177/ 1203 4754 2110 35098 journals. sagepub. com/ home/ cms Superiority of Rituximab Over Mycophenolate Mofetil in the Treatment of Pemphigus Vulgaris: A Step Further in Its Approval as a FirstLine Treatment?

Immune checkpoint inhibitor therapy nowadays became a treatment for a wide range of cancers, and may be responsible for various dermatologic adverse effects, including bullous eruptions. In our report, we present a case of late-onset immunotherapy-induced eruption in a 62-year-old woman treated with anti-programmed cell death-L1 agent durvalumab for metastatic squamous cell carcinoma. Diagnosed as lichenoid dermatitis upon initial presentation, this eruption evolved into necrotic bullous dermatitis after several weeks of phototherapy, with histology and direct immunofluorescence study favoring lichen planus pemphigoides. Thus, this case may be regarded as durvalumab-induced lichenoid dermatitis with phototherapy-triggered progression to necrotic lichen planus pemphigoides-like eruption. The patient's eruption responded to oral prednisone and immunotherapy interruption. Interestingly, durvalumab reintroduction in this patient led to recurrent lichenoid dermatitis without bullous component. This case of immunotherapy skin toxicity is rather distinctive by its clinical and histopathologic features, with phototherapy as an additional triggering factor.

Therapeutic Plasma Exchange in Refractory VITT Three patients with severe manifestations of VITT after Covid-19 vaccination had a poor response to initial therapy with anticoagulants and intravenous immune globulin. Their conditions improved after therapeutic plasma exchange was initiated, and they were able to leave the hospital.

A 66-year-old woman presented to the hospital with cutaneous necrosis of her right ankle and foot. Her symptoms began immediately after an intra-articular injection of hyaluronic acid for ankle osteoarthritis, which was performed 6 days before. Histopathology showed an intra-vascular hyaluronic acid embolus. The initial treatment approach was conservative, but the patient's clinical state degraded. She was thus treated with sub-cutaneous hyaluronidase, the enzyme that degrades hyaluronic acid, which yielded a moderate improvement even though it was administered 22 days after the initial hyaluronic acid injection. Although hyaluronic acid embolism and subsequent cutaneous necrosis are well-known complications of dermal fillers, there are few reported cases of embolism following intra-articular injection. To our knowledge, this is the first time hyaluronidase has been used in this setting.

Treatment of moderate-to-severe psoriasis in patients with HIV infection is a clinical challenge. We present the case of a patient with a longstanding history of well-controlled HIV. He had failed topical management, and his hypertriglyceridemia made use of acitretin potentially unsafe. He was unable to regularly attend a phototherapy unit. Physical examination revealed 12% total body surface area involvement with a Psoriasis Area Severity Index (PASI) of 10.2. His Dermatology Quality of Life Index (DLQI) was 20. After 3 months of apremilast treatment, his PASI decreased to 4.1. After 7 months, his PASI decreased to 2.7 and his DLQI to 1. Two years later, his PASI score was 2.4, with a stable CD4 count of 1200 cells/mm3 and an undetectable viral load. There were no serious opportunistic infections or laboratory abnormalities. To our knowledge, this represents the second reported case of psoriasis treatment with apremilast in a patient with HIV.

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.

Type II diabetes progresses with inadequate insulin secretion and prolonged elevated circulating glucose levels. Also, pancreatic islets isolated for transplantation or tissue engineering can be exposed to glucose over extended timeframe. We hypothesized that isolated pancreatic islets can secrete insulin over a prolonged period of time when incubated in glucose solution and that not all islets release insulin in unison. Insulin secretion kinetics was examined and modeled from single mouse islets in response to chronic glucose exposure (2.8‐20 mM). Results with single islets were compared to those from pools of islets. Kinetic analysis of 58 single islets over 72 h in response to elevated glucose revealed distinct insulin secretion profiles: slow‐, fast‐, and constant‐rate secretors, with slow‐secretors being most prominent (ca., 50%). Variations in the temporal response to glucose therefore exist. During short‐term (<4 h) exposure to elevated glucose few islets are responding with sustained insulin release. The model allowed studying the influence of islet size, revealing no clear effect. At high‐glucose concentrations, when secretion is normalized to islet volume, the tendency is that smaller islets secrete more insulin. At high‐glucose concentrations, insulin secretion from single islets is representative of islet populations, while under low‐glucose conditions pooled islets did not behave as single ones. The characterization of insulin secretion over prolonged periods complements studies on insulin secretion performed over short timeframe. Further investigation of these differences in secretion profiles may resolve open‐ended questions on pre‐diabetic conditions and transplanted islets performance. This study deliberates the importance of size of islets in insulin secretion. © 2018 American Institute of Chemical Engineers Biotechnol. Prog., 34:1059–1068, 2018

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.

Chronic rhinosinusitis (CRS) is a frequent chronic condition, which has origins in complex interactions between genetic, immunological and microbial factors. The role of auto-immunity in CRS remains unclear, although recent studies have started to emerge in CRS patient refractory to maximal medical management. We discuss the possible auto-immunity link between CRS and other skin diseases, in particular acquired bullous dermatoses, and review the current evidence. We raise additional considerations for auto-immunity from both research and clinical standpoints.

Background: Doxorubicin is an antineoplastic agent frequently used in diverse cancer regimens. Cutaneous adverse effects have frequently been reported with its use. However, a flagellate-like dermatitis is not mentioned in the literature.Objective: The investigators report a case of toxic erythema of chemotherapy with a flagellate pattern induced by doxorubicin.Methods and Results: A 75-year-old woman with endometrial cancer received doxorubicin as part of her treatment. After her third cycle, she presented a pruritic vesiculobullous eruption, with linear elements that left hyperpigmented streaks on follow-up. A biopsy was compatible with a drug eruption.Conclusion: Doxorubicin is a well-known cause of toxic erythema of chemotherapy. As seen in this patient, the investigators suggest that it also be added to the list of causes of flagellate dermatosis.

Chronic rhinosinusitis (CRS) is a frequent chronic condition, which has origins in complex interactions between genetic, immunological and microbial factors. The role of auto-immunity in CRS remains unclear, although recent studies have started to emerge in CRS patient refractory to maximal medical management. We discuss the possible auto-immunity link between CRS and other skin diseases, in particular acquired bullous dermatoses, and review the current evidence. We raise additional considerations for auto-immunity from both research and clinical standpoints.

International Journal of DermatologyVolume 51, Issue 11 p. 1363-1365 Case report Lues maligna and Bell’s palsy: report of a case in a immunocompetent host Janie Bertrand MD, Janie Bertrand MD Division of Dermatology, Department of MedicineSearch for more papers by this authorLouise G. Labrecque MD, Louise G. Labrecque MD Departments of Microbiology and Infectious DiseasesSearch for more papers by this authorAnnie Belisle MD, Annie Belisle MD Pathology, CHU Montreal, University of Montreal, Montreal, CanadaSearch for more papers by this authorBenoît Côté MD, Benoît Côté MD Division of Dermatology, Department of MedicineSearch for more papers by this author Janie Bertrand MD, Janie Bertrand MD Division of Dermatology, Department of MedicineSearch for more papers by this authorLouise G. Labrecque MD, Louise G. Labrecque MD Departments of Microbiology and Infectious DiseasesSearch for more papers by this authorAnnie Belisle MD, Annie Belisle MD Pathology, CHU Montreal, University of Montreal, Montreal, CanadaSearch for more papers by this authorBenoît Côté MD, Benoît Côté MD Division of Dermatology, Department of MedicineSearch for more papers by this author First published: 16 October 2012 https://doi.org/10.1111/j.1365-4632.2011.05169.xCitations: 3 Janie Bertrand, mdDivision of Dermatology Department of Medicine CHU Montreal, Hôpital Saint-Luc 1058, Saint-Denis Montreal (Quebec) H2X 3J4 Canada E-mail: [email protected] Conflicts of interest: None. Read the full textAboutPDF ToolsRequest permissionExport citationAdd to favoritesTrack citation ShareShare Give accessShare full text accessShare full-text accessPlease review our Terms and Conditions of Use and check box below to share full-text version of article.I have read and accept the Wiley Online Library Terms and Conditions of UseShareable LinkUse the link below to share a full-text version of this article with your friends and colleagues. Learn more.Copy URL Share a linkShare onEmailFacebookTwitterLinkedInRedditWechat References 1 Public Health Agency of Canada. Report on Sexually Transmitted Infections in Canada: 2008, Syphilis (Treponema pallidum). Available at: http://www.publichealth.gc.ca [Accessed 13 November 2010]. 2 D’Amico R, Zalusky R. A case of lues maligna in a patient with acquired immunodeficiency syndrome (AIDS). Scand J Infect Dis 2005; 37: 697–700. 3 Watson KM, White JM, Salisbury JR, et al. Lues maligna. Clin Exp Dermatol 2004; 29: 625–627. 4 Bayramgürler D, Bilen N, Yildiz K, et al. Lues maligna in a chronic alcoholic patient. J Dermatol 2005; 32: 217–219. 5 Hoffman UB, Hund M, Bröcker EB, et al. “Lues maligna” in a female patient with diabetes. J Dtsch Dermatol Ges 2005; 3: 780–782 (In German). 6 Tucker JD, Shah S, Jarell AD, et al. Lues maligna in early HIV infection case report and review of the litterature. Sex Transm Dis 2009; 36: 512–514. 7 Williams EY, Bowie-Elder Z. Bell’s palsy. A new sign. J Natl Med Assoc 1970; 62: 153–155. 8 Davis LE, Sperry S. Bell’s palsy and secondary syphilis: CSF spirochetes detected by immunofluorescence. Ann Neurol 1978; 4: 378–380. Citing Literature Volume51, Issue11November 2012Pages 1363-1365 ReferencesRelatedInformation