Publications by authors named "Afshin Hatami"

32 Publications

A novel pathogenic RHOA variant in a patient with patterned cutaneous hypopigmentation associated with extracutaneous findings.

Pediatr Dermatol 2022 Mar 17;39(2):281-287. Epub 2022 Feb 17.

Division of Pediatric Dermatology, Department of Pediatrics, Sainte-Justine University Hospital Centre, University of Montreal, Montreal, Canada.

RHOA-related neuroectodermal syndrome is characterised by linear skin hypopigmentation along Blaschko's lines associated with alopecia, leukoencephalopathy, facial and limb hypoplasia, and ocular, dental, and acral anomalies. Herein, we report a patient with patterned cutaneous hypopigmentation with a similar phenotype due to a novel postzygotic RHOA variant (c.210G>T; p.Arg70Ser). This illustrates that the complexity of the orchestration of morphogenesis and organogenesis can be affected by different variants in the same gene.
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http://dx.doi.org/10.1111/pde.14923DOI Listing
March 2022

Pediatric Cutaneous Hematologic Disorders: Cutaneous Lymphoma and Leukemia Cutis: Experience of a Tertiary-Care Pediatric Institution and Review of the Literature.

J Cutan Med Surg 2022 Feb 15:12034754221077694. Epub 2022 Feb 15.

Department of Pathology, Sainte-Justine University Hospital Center, University of Montreal, Montreal, Quebec, Canada.

Background: Cutaneous hematologic malignancies are rare in children, and the literature about them is still sparse.

Objective: The purpose of our study was to report our experience with pediatric cases of cutaneous hematologic disorders and describe their clinical and histological features.

Methods: Data were retrospectively collected from the histopathologic database of the CHU Sainte-Justine, University of Montreal, Montreal, Canada. All patients up to 18 years of age with a diagnosis of a primary cutaneous lymphoma (including lymphomatoid papulosis), secondary cutaneous lymphoma or cutaneous manifestations of leukemia, followed from 1980 to 2019 at our center were reviewed.

Results: Thirty-six patients were included. Age at presentation ranged from birth to 18 years of age (mean 7.83 ± 5.16; median 7.0). Ten different hematologic disorders were identified according to the WHO-EORTC classifications: lymphomatoid papulosis (10 cases), mycosis fungoides (6 cases), anaplastic large cell lymphoma (4 cases), pre-B acute lymphoid leukemia (5 cases), primary cutaneous marginal zone B-cell lymphoma (4 cases), primary cutaneous CD4medium T-cell lymphoproliferative disorder (1 case), extranodal NK/T-cell lymphoma (1 case), hydroa vacciniforme-like lymphoproliferative disorder (1 case), B-cell lymphoblastic lymphoma (1 case) and acute myeloid leukemia (3 cases).

Conclusion: The most common subtype of cutaneous hematologic disease in our single institution study was lymphomatoid papulosis (type A and type C), followed by mycosis fungoides. Recognition of this large clinical and histological spectrum by dermatologists is important because diagnosis is often established by biopsy of skin lesions, even in secondary cutaneous cases. Moreover, the clinicopathological correlation is of utmost importance for the final diagnosis of those pathologies.
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http://dx.doi.org/10.1177/12034754221077694DOI Listing
February 2022

Linear cutaneous lupus erythematosus in children-report of two cases and review of the literature: A case report.

SAGE Open Med Case Rep 2020 9;8:2050313X20979206. Epub 2020 Dec 9.

Division of Pediatric Dermatology, Centre Hospitalier Universitaire Sainte-Justine, University of Montreal, Montreal, QC, Canada.

Linear cutaneous lupus erythematosus is an unusual presentation of cutaneous lupus following Blaschko's lines. It is described mostly in children and young adults and is usually not associated with systemic involvement. We report two cases of linear cutaneous lupus erythematosus in children who significantly improved after treatment with hydroxychloroquine in combination with topical corticosteroids and tacrolimus. These rare cases underline the importance of including linear cutaneous lupus erythematosus in the differential diagnosis of blaschkoid inflammatory lesions.
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http://dx.doi.org/10.1177/2050313X20979206DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7731695PMC
December 2020

Isotretinoin for the treatment of severe acneiform eruptions associated with the MEK inhibitor trametinib.

JAAD Case Rep 2020 Oct 23;6(10):1056-1058. Epub 2020 Jul 23.

Division of Pediatric Dermatology, CHU Sainte-Justine, University of Montreal, Montreal, Canada.

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http://dx.doi.org/10.1016/j.jdcr.2020.07.021DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7509361PMC
October 2020

Kaposiform hemangioendothelioma with overlapping features of rapidly involuting congenital hemangioma and a delayed complication of necrotizing fasciitis.

Pediatr Dermatol 2020 Sep 16;37(5):902-906. Epub 2020 Jul 16.

Dermatology Division, CHU Sainte-Justine, Montreal, Canada.

We report the case of a male infant born at term with kaposiform hemangioendothelioma (KHE) of the right forearm and coagulopathy. Our case was unusual as it involuted leaving subcutaneous atrophy and prominent veins, which are more commonly observed in rapidly involuting congenital hemangioma. At 3 years of age, the child developed recurrent superficial thrombophlebitis localized to the area where the KHE had regressed. Subsequently, he developed necrotizing fasciitis and thrombotic veins in the same location and group A streptococcal septic shock.
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http://dx.doi.org/10.1111/pde.14262DOI Listing
September 2020

Skin Manifestations in Pediatric Patients Treated With a TNF-Alpha Inhibitor for Inflammatory Bowel Disease: A Retrospective Study [Formula: see text].

J Cutan Med Surg 2020 Jul/Aug;24(4):333-339. Epub 2020 Jun 11.

Division of Dermatology, CHU Sainte-Justine, University of Montreal, QC, Canada.

Background: Tumor necrosis factor (TNF) alpha inhibitors (anti-TNF) are effective in the treatment of inflammatory bowel disease (IBD) as well as psoriasis. Their increasing use has raised the identification of cutaneous side effects (CSEs). Evidence in children is limited.

Objectives: The objective of this study is to describe CSEs of anti-TNF treatment in a pediatric population with IBD.

Methods: This is a retrospective single-center study of children with IBD under anti-TNF treatment between 2013 and 2016. A total of 40 patients with CSEs related to anti-TNF were referred to our pediatric dermatology clinic. A control group was randomly selected from patients receiving anti-TNF for IBD, who were referred to the dermatology clinic for other conditions unrelated to anti-TNF.

Results: Of 343 patients with IBD, 40 (11.3%) presented CSEs potentially related to the treatment. No differences in sex, age, and underlying disease were found between those with and without CSEs. The most frequent CSEs were psoriasiform eruptions (41%) which were more exudative than usual, located especially in skin folds and on the scalp; skin infections (20%); and eczematous eruptions (10%). Only 5% of patients changed or discontinued the current anti-TNF because of CSEs.

Conclusion: This is one of the largest pediatric cohorts of IBD patients with CSEs. Psoriasiform eruptions were the most common CSEs, with predilection for skin folds and scalp, and frequent superimposed bacterial infection. Topical and/or systemic antibiotics were required in addition to topical corticosteroids in 25% of patients. The rate of discontinuation of anti-TNF therapy due to CSEs was low.
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http://dx.doi.org/10.1177/1203475420917387DOI Listing
June 2021

Patterned cutaneous hypopigmentation phenotype characterization: A retrospective study in 106 children.

Pediatr Dermatol 2019 Nov 30;36(6):869-875. Epub 2019 Jul 30.

Division of Pediatric Dermatology, Department of Pediatrics, Sainte-Justine University Hospital Centre, University of Montreal, Montreal, Quebec, Canada.

Background: Cutaneous patterned hypopigmentation's phenotype is highly variable and may be associated with extracutaneous anomalies.

Objective: We evaluated the phenotypic and clinical characteristics of patients with cutaneous patterned hypopigmentation to determine whether certain patterns were more likely to be associated with underlying anomalies.

Methods: The charts of 106 children with cutaneous patterned hypopigmentation were reviewed retrospectively (2007-2018) at Sainte-Justine University Hospital Centre, in Montreal, Canada. Retrieved information included sex, age at diagnosis, phototype, pattern, and distribution of the cutaneous lesions and the presence of extracutaneous findings. Data were recorded on a software tool which collects and analyzes phenotypic information.

Results: The predominant types of cutaneous patterned hypopigmentation were along Blaschko's lines in narrow (38.7%) and broad bands (53.8%). Mixed patterns were observed in 22.5% of children. The anterior trunk and posterior trunk were most frequently affected (69% and 56%, respectively). Extracutaneous involvement, especially neurological and developmental, was present in 28.3% of patients and was significantly associated with ≥ 4 involved body sites.

Conclusion: Distribution and types of cutaneous patterned hypopigmentation were not predictive of extracutaneous findings, with the exception of multiple sites involvement and possibly centrofacial location and blocklike lesions. Follow-up until school entry should help identify subtler associated extracutaneous anomalies.
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http://dx.doi.org/10.1111/pde.13913DOI Listing
November 2019

Silver-coated textiles in hidradenitis suppurativa: A case report.

SAGE Open Med Case Rep 2019 2;7:2050313X19845212. Epub 2019 May 2.

Centre Hospitalier Universitaire Sainte-Justine, Division of Dermatology, Department of Pediatrics, University of Montreal, Montreal, QC, Canada.

Hidradenitis suppurativa is a chronic and debilitating skin disease of apocrine gland-bearing areas. The mainstay of treatment usually includes topical and systemic antibiotics. These agents can be used as monotherapy or combination therapy. The therapeutic role of functional textiles with antimicrobial activity has been recently emerging in the treatment of other skin diseases such as atopic dermatitis and epidermolysis bullosa. The pathologic processes involved in the development of atopic dermatitis and hidradenitis suppurativa are still incompletely understood, but these two diseases share some similarities including bacterial proliferation and chronic inflammation. We report the case of a 14-year-old boy with hidradenitis suppurativa that has been successfully treated with silver-coated textiles. To the best of our knowledge, this article is the first to report the benefits of silver-coated textiles in the treatment of hidradenitis suppurativa.
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http://dx.doi.org/10.1177/2050313X19845212DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6498763PMC
May 2019

Non-involuting congenital hemangiomas (NICH) with postnatal atypical growth: A case series.

Pediatr Dermatol 2019 Jul 29;36(4):466-470. Epub 2019 Apr 29.

Division of Dermatology, Department of Pediatrics, Sainte-Justine University Hospital Center, University of Montreal, Montreal, Quebec, Canada.

Background: Non-involuting congenital hemangiomas (NICH) are fully formed vascular tumors at birth, with a distinctive clinical, radiologic, and histopathological profile, and classically lack expansion or involution over time. We describe a series of NICH cases with atypical postnatal growth.

Methods: The authors retrospectively analyzed all NICH cases diagnosed from 2007 to 2017. We reviewed charts and photographic databases from our Vascular Anomalies Clinic. We included in the study all NICH with an atypical postnatal growth. Clinical data, imaging, and histopathology were analyzed.

Results: Eighty cases of NICH were identified. Nine presented with atypical postnatal growth after a stable period, at ages from 2 to 10 years (mean: 5.3 years). Two patients had associated pain; 5 patients showed new red papules on the surface of the lesion; 2 reported bleeding from the papules; and 1 developed a pyogenic granuloma. All patients had Doppler ultrasound and/or MRI compatible with NICH, and a confirmatory biopsy was performed in 4 cases. In treatment, 2 patients received endovascular embolization, and one required further surgery.

Conclusions: Non-involuting congenital hemangiomas (NICH) may develop significant postnatal growth over time (10% in our series), requiring closer follow-up for longer periods. The development of red papules, pyogenic granulomas, and superficial bleeding may be observed. Since this is a small series, we were not able to establish risk factors for NICH with postnatal growth.
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http://dx.doi.org/10.1111/pde.13837DOI Listing
July 2019

Multiple superficial oral mucoceles after Mycoplasma-induced mucositis.

Pediatr Dermatol 2018 Jul 15;35(4):e210-e211. Epub 2018 May 15.

Division of Dermatology, Department of Pediatrics, Centre Hospitalier Universitaire de Ste-Justine, University of Montreal, Montreal, QC, Canada.

Oral mucoceles are lesions arising mainly from the minor salivary glands and are thought to occur after excretory duct trauma. We report a case of multiple superficial oral mucoceles on a child's labial mucosa after Mycoplasma pneumoniae mucositis. Mucoceles can mimic persistent or recurrent stomatitis and lead to potential errors in management.
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http://dx.doi.org/10.1111/pde.13515DOI Listing
July 2018

Emergence of African species of dermatophytes in tinea capitis: A 17-year experience in a Montreal pediatric hospital.

Pediatr Dermatol 2018 May 24;35(3):323-328. Epub 2018 Mar 24.

Department of Social and Preventive Medicine, Faculty of Medicine, Laval University, Quebec, QC, Canada.

Background/objectives: An increase in dermatophyte infections caused by African species is reported in countries receiving African immigrants. Our goal was to determine the epidemiologic and clinical characteristics of tinea capitis in children infected with African species of dermatophytes in Montreal, Canada.

Methods: Demographic and clinical data from medical records of children infected with African species of dermatophytes were retrieved retrospectively (2000-2016) at Sainte-Justine University Hospital Center.

Results: In Montreal, the number of tinea capitis cases caused by African species of dermatophytes increased sixfold over 17 years. African immigrant children (84%), men and boys (61%), and preschoolers (2-5 years old) (51%) were the most frequently affected in our 315 cases. Family contamination was frequent (45%). Referring physicians prescribed systemic antifungal treatment in 39% of cases and pediatric dermatologist consultants in 90%. Treatment failure to oral terbinafine occurred in 39% of Microsporum audouinii infections.

Conclusion: In Montreal, there was a significant increase in tinea capitis caused by African species of dermatophytes. Microsporum audouinii is highly transmissible and often resistant to oral terbinafine. Recognizing tinea capitis trends in a given environment will improve patient care.
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http://dx.doi.org/10.1111/pde.13446DOI Listing
May 2018

Pilomatricoma Associated with Kabuki Syndrome.

Pediatr Dermatol 2017 Jan 25;34(1):e26-e27. Epub 2016 Oct 25.

Division of Dermatology, Department of Paediatrics, Centre Hospitalier Universitaire Sainte-Justine, Université de Montreal, Montreal, Canada.

We report three cases of pilomatricomas associated with Kabuki syndrome (KS), supporting the hypothesis proposed of an association between pilomatricomas and KS and suggesting a noncoincidental association, because the Wnt pathway mutations involved could affect both morphogenesis and tumorigenesis in these patients.
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http://dx.doi.org/10.1111/pde.13014DOI Listing
January 2017

Aortic Dilatation Associated With a De Novo Mutation in the SOX18 Gene: Expanding the Clinical Spectrum of Hypotrichosis-Lymphedema-Telangiectasia Syndrome.

Can J Cardiol 2016 Jan 13;32(1):135.e1-7. Epub 2015 Apr 13.

Cardiovascular Genetics, Department of Pediatrics, CHU Sainte-Justine, Université de Montréal, Montréal, Québec, Canada. Electronic address:

Background: We report a 13-year-old female patient followed since birth for multiple rare congenital defects, including hypotrichosis, telangiectasia, and severe dilatation of the ascending aorta.

Methods: Comprehensive phenotype assessment throughout childhood included repeated echocardiographic measurements, evaluation of renal function, and immunohistochemical analysis of skin biopsy samples. Whole-exome sequencing was performed for the patient and both unaffected parents.

Results: We identified a novel de novo mutation in the transcription factor SOX18 (c.481C>T:p.Gln161*) in the patient, which was absent in all unaffected family members. Echocardiography revealed early onset and progressive dilatation of the ascending aorta. Skin biopsy results confirmed the defects of the blood vasculature in the presence of intact lymphatic vessels. Assessment of renal function did not show any signs of renal problems or renal failure in the patient.

Conclusions: The genetic finding of a pathogenic SOX18 mutation enabled the diagnosis of the rare hypotrichosis-lymphedema-telangiectasia syndrome in our patient. The identification of a novel stop gain mutation in the SOX18 gene in association with dilatation of the aorta highlights the importance of this gene during the development of the circulatory system. Our study highlights the importance of whole-exome sequencing in the rapid identification of genes and gene mutations involved in rare conditions and thus expanding the knowledge and spectrum of clinical manifestations associated with them.
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http://dx.doi.org/10.1016/j.cjca.2015.04.004DOI Listing
January 2016

Diseases of abnormal sensitivity to cold in children on psychostimulant drugs.

J Cutan Med Surg 2015 Mar-Apr;19(2):121-4. Epub 2015 Mar 11.

Pediatric Dermatology Division and Department of Pharmacy, CHU Ste-Justine, Montreal, PQ.

Background: Oral psychostimulant (PS) drugs, the pharmacologic treatment of choice for attention-deficit/hyperactivity disorder (ADHD), have been associated with diseases of abnormal sensitivity to cold (DASC) such as Raynaud phenomenon and acrocyanosis.

Objectives: In a cohort of pediatric patients with DASC, we sought to identify prevalence and clinical features of patients on PS drugs.

Methods: A 6-year retrospective chart review (2005-2011) of Ste-Justine University Hospital Center DASC patients with and without exposure to PS drugs was performed. Clinical data were analyzed with descriptive statistical methods.

Results: Of 43 patients with DASC, 11 (25%) were exposed to PS drugs. In this group males were overrepresented, there was no evidence of collagen vascular diseases, serologic findings were not significant and the mean duration of PS intake was of 2.5 years. DASC age of onset was similar in both exposed and nonexposed patients. The incidence of more than one DASC type was greater in teenager patients with a positive family history of autoimmune and/or collagen vascular diseases.

Limitations: This study is limited by its small population size, short follow-up period and its retrospective nature.

Conclusion: Physicians should be aware of PS drugs as possible triggers for DASC.
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http://dx.doi.org/10.2310/7750.2014.14052DOI Listing
September 2015

Pigmented purpuric dermatosis: clinicopathologic characterization in a pediatric series.

Pediatr Dermatol 2015 May-Jun;32(3):358-62. Epub 2015 Feb 18.

Department of Dermatology, Centre Hospitalier Universitaire Ste-Justine, University of Montreal, Montreal, Quebec, Canada.

Pigmentary purpuras (PPs) are a group of chronic disorders of unknown origin seldom described in children. With this study we sought to better characterize PP eruptions, including clinical evolution and management. A retrospective chart review from 2003 to 2013 querying characteristics of children with a biopsy-proven diagnosis of PP in the Centre Hospitalier Universitaire Ste-Justine dermatology clinic (Montreal, Quebec, Canada) was performed. Follow-up was obtained through telephone interviews. Descriptive statistical analysis was used. Of the 17 subjects, 8 were male and the mean age of onset was 9 years. PP was asymptomatic in 11 patients, pruritic in 3, and of cosmetic concern in 3. Schamberg's disease was the most frequent subtype in 12 cases. Resolution of PP was found in 13 cases with a median duration of less than 1 year (range 6 months-9 years). Five patients experienced spontaneous clearing without treatment, and improvement was observed in 75% of cases treated with topical corticosteroids and 100% with narrowband ultraviolet B (nbUVB). No associated disease, significant drug exposure, or contact allergens were found. Those findings support that PPs in children are idiopathic, chronic eruptions that can benefit from watchful waiting, although topical corticosteroids or nbUVB are may be useful if the patient or family desires faster resolution. This study was limited by its small size, its retrospective nature, and selection and recall bias.
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http://dx.doi.org/10.1111/pde.12519DOI Listing
April 2016

Diseases of abnormal sensitivity to cold in children on psychostimulant drugs.

J Cutan Med Surg 2014 Oct;18(5):1-4

Background: Oral psychostimulant (PS) drugs, the pharmacologic treatment of choice for attention-deficit/hyperactivity disorder (ADHD), have been associated with diseases of abnormal sensitivity to cold (DASC) such as Raynaud phenomenon and acrocyanosis.

Objectives: In a cohort of pediatric patients with DASC, we sought to identify prevalence and clinical features of patients on PS drugs.

Methods: A 6-year retrospective chart review (2005-2011) of Ste-Justine University Hospital Center DASC patients with and without exposure to PS drugs was performed. Clinical data were analyzed with descriptive statistical methods.

Results: Of 43 patients with DASC, 11 (25%) were exposed to PS drugs. In this group males were overrepresented, there was no evidence of collagen vascular diseases, serologic findings were not significant and the mean duration of PS intake was of 2.5 years. DASC age of onset was similar in both exposed and nonexposed patients. The incidence of more than one DASC type was greater in teenager patients with a positive family history of autoimmune and/or collagen vascular diseases.

Limitations: This study is limited by its small population size, short follow-up period and its retrospective nature.

Conclusion: Physicians should be aware of PS drugs as possible triggers for DASC.
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October 2014

Quality of life in pediatric patients before and after cosmetic camouflage of visible skin conditions.

J Am Acad Dermatol 2014 Nov 11;71(5):935-40. Epub 2014 Sep 11.

Division of Pediatric Dermatology, Department of Pediatrics, Centre Hospitalier Universitaire Sainte-Justine, University of Montreal, Montreal, Quebec, Canada.

Background: Visible vascular and pigmentary conditions have a negative impact on children's and adolescents' quality of life (QoL). We sought to quantitate the effect of visible skin anomalies and their camouflage on QoL.

Methods: In all, 41 patients, 5 years of age and older, were taught to use cosmetic camouflage. QoL was assessed using the Children's Dermatology Life Quality Index (CDLQI) before and 6 months after the intervention. Satisfaction and use were evaluated after 1 and 6 months.

Results: Baseline QoL scores revealed a small impact of vascular anomalies (CDLQI score 4.2) and a small to moderate effect of pigmentary anomalies (CDLQI score 6.1). Six months after the intervention, QoL improved in the study population as a whole (CDLQI score 5.1 vs 2.1, P<.001), with significant improvements documented for facial lesions and vascular malformations. Cosmetic camouflage was well tolerated and patients with pigmentary anomalies were more likely to continue using the products.

Limitations: Limitations include small study population, few male patients, cultural influences not addressed, and limited range of conditions.

Conclusions: Children and teenagers with visible vascular and pigmentary anomalies experience an impairment of QoL that is abrogated by introduction to use of cosmetic camouflage.
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http://dx.doi.org/10.1016/j.jaad.2014.07.029DOI Listing
November 2014

Expression of the senescence marker p16INK4a in skin biopsies of acute lymphoblastic leukemia survivors: a pilot study.

Radiat Oncol 2013 Oct 31;8:252. Epub 2013 Oct 31.

Centre de recherche du CHU Sainte-Justine, Montréal, Québec, Canada.

Background: Most childhood cancer survivors will develop ionizing radiation treatment-related health conditions that, in many instances, resemble age-associated pathologies. Treatment-induced premature senescence could be an underlying mechanism.

Findings: Here we wanted to know whether the expression of p16INK4a, a senescence/aging biomarker, is increased in skin biopsies of acute lymphoblastic leukemia survivors (ALL), previously exposed to chemotherapy and radiation therapy. Several years post-treatments, we found p16INK4a mRNA levels are 5.8 times higher in scalp skin biopsies (targeted by cranial irradiation therapy) compared to buttocks skin biopsies (n = 10, p = 0.01).

Conclusions: These results demonstrate for the first time that premature senescence is induced in pediatric cancer survivors and that p16INK4a expression could be used as a potential biomarker in this population.
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http://dx.doi.org/10.1186/1748-717X-8-252DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3827993PMC
October 2013

Anti-IgE therapy and severe atopic dermatitis: a pediatric perspective.

J Am Acad Dermatol 2013 Nov;69(5):832-834

Department of Pediatrics, Service of Allergy and Clinical Immunology, Centre Hospitalier Universitaire Sainte-Justine, Montréal, Quebec, Canada. Electronic address:

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http://dx.doi.org/10.1016/j.jaad.2013.05.035DOI Listing
November 2013

Late rebound of infantile hemangioma after cessation of oral propranolol.

Pediatr Dermatol 2013 Sep-Oct;30(5):587-91

Division of Dermatology, Department of Pediatrics, CHU Sainte-Justine, University of Montreal, Quebec, Canada.

Propranolol has become the first line of treatment for infantile hemangiomas (IHs), with a high response rate, but rebound growth after cessation of propranolol has been reported, primarily in the first year of life. We sought to determine the frequency and associated factors leading to late regrowth after successful treatment at an age when the proliferative phase has usually ceased. We retrospectively reviewed the clinical charts, serial photographs, and radiologic images of children with rebound IH occurring after the age of 15 months after a successful course of oral propranolol averaging 2.6 mg/kg/day (range 2-3 mg/kg/day). Thirteen (10 female, 3 male) of 212 patients (6%) treated with oral propranolol since 2008 were evaluated. The mean age at the start of treatment was 5.3 months (range 1.8-13 months), and an average of 10.3 months (range 4.5-16 months) of treatment was given. It took an average of 5.3 months (range 1-13.8 months) for a significant rebound to appear. Late rebound after successful propranolol indicates a prolonged proliferation phase of IH even after 15 months of age. This is compared with previous reports of rebound, which occurred primarily in infants younger than 1 year old. Late proliferation can occur in localized, small, mixed, and deep IH, even after several months of a positive response to propranolol. A second course of propranolol readily controlled the recurrence.
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http://dx.doi.org/10.1111/pde.12190DOI Listing
April 2014

Therapy of ulcerated hemangiomas.

J Cutan Med Surg 2013 Jul-Aug;17(4):233-42

Department of Family Medicine, McGill University, Canada.

Background: Cutaneous ulceration is the most common complication of infantile hemangiomas (IHs) seen in a pediatric dermatology practice.

Objective: The most effective treatments in our experience are compared to those in the current literature.

Methods: The study was a retrospective chart review of therapy of 169 ulcerated IHs at a tertiary care pediatric hospital and a literature review.

Results: Combination therapy was the rule. Local wound care was required in all, pain management in 72%, pulsed dye laser in 42%, infection control in 38%, diminution of the hemangioma through systemic therapy in 36%, and suppression of bleeding in 2%.

Limitations: A retrospective review compared to a case-control study has inherent bias. In addition, our cases were all at a tertiary referral center.

Conclusion: All ulcerated IHs benefit from local barrier creams or dressings. Pulsed dye laser, antibiotics, topical morphine 0.1% in hydrogel, topical becaplermin, and, most importantly, systemic therapy (especially propranolol) to reduce the hemangioma may be useful.
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http://dx.doi.org/10.2310/7750.2012.12037DOI Listing
September 2013

Propranolol in the treatment of problematic infantile hemangioma: review of 35 consecutive patients from a vascular anomalies clinic.

J Cutan Med Surg 2012 Sep-Oct;16(5):317-23

Department of Medicine, CHU, Montreal, Canada.

Background: Propranolol, a nonselective β-blocker, has been reported as efficient for controlling the growth of complicated infantile hemangiomas (IHs). No uniformly accepted protocol exists regarding the administration of oral propranolol for IH.

Objective: We sought to share our experience using propranolol for problematic IH and to evaluate the efficacy of this treatment modality.

Methods: A retrospective chart review analysis was performed for 35 consecutive children treated with propranolol as an oral solution on an outpatient basis in our dermatology/vascular anomalies clinic. A protocol was established with the help of our pediatric cardiologists, including pretreatment electrocardiography and echocardiography. Medical photographs taken after 2 months of treatment were rated by two independent evaluators.

Results: We treated 31 girls and 4 boys with a median age of 3.5 months. Rapid improvement was reported in the first days of treatment in 34 patients. Mean improvement after 2 months was 61.5%. No serious adverse effects were reported.

Conclusion: Propranolol was effective in controlling the proliferative phase of problematic IH. It was well tolerated in our study. Outpatient treatment is possible if parents follow strict guidelines. Propranolol should be a first-line treatment for problematic IH in carefully selected patients.
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http://dx.doi.org/10.1177/120347541201600508DOI Listing
December 2012

Connective tissue nevi in children: institutional experience and review.

J Am Acad Dermatol 2012 Nov 26;67(5):890-7. Epub 2012 Jun 26.

Department of Pediatrics, Division of Dermatology, Centre Hospitalier Universitaire Sainte-Justine, University of Montreal, Montréal, Québec, Canada.

Background: Connective tissue nevi (CTN) are circumscribed hamartomas of the skin in which there is an abnormal mixture of normal components of the dermis that may be sporadic or associated with syndromes such as Buschke-Ollendorff, tuberous sclerosis, and Proteus.

Objective: We sought to specify the clinical and histologic features of CTN in childhood and to propose a diagnostic approach and updated classification.

Methods: This was a retrospective study in a tertiary pediatric outpatient population, accessing clinical and histopathological records.

Results: We classified 114 cases of CTN from 1980 to 2008.

Limitations: The majority of cases were confirmed by histopathological examination. Therefore, our series excludes many CTN that were not biopsied. In addition, follow-up was variable.

Conclusion: Our series demonstrates the usefulness of a modified classification for CTN. Biopsy should be done when clinical diagnosis is uncertain, or in multiple lesions. When biopsy is performed it should include normal-appearing skin for comparison and, in Buschke-Ollendorff syndrome, limited anterior-posterior x-rays of the hands, wrists, feet, ankles, knees, and pelvis instead of a full skeletal survey.
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http://dx.doi.org/10.1016/j.jaad.2012.01.036DOI Listing
November 2012

Propranolol in the treatment of problematic infantile hemangioma: review of 35 consecutive patients from a vascular anomalies clinic.

J Cutan Med Surg 2012 Mar-Apr;16(2):115-21

Division of Dermatology, Department of Medicine, CHU University of Montreal, Montreal, QC.

Background: Propranolol, a nonselective β-blocker, has been reported as efficient for controlling the growth of complicated infantile hemangiomas (IHs). No uniformly accepted protocol exists regarding the administration of oral propranolol for IH.

Objective: We sought to share our experience using propranolol for problematic IH and to evaluate the efficacy of this treatment modality.

Methods: A retrospective chart review analysis was performed for 35 consecutive children treated with propranolol as an oral solution on an outpatient basis in our dermatology/vascular anomalies clinic. A protocol was established with the help of our pediatric cardiologists, including pretreatment electrocardiography and echocardiography. Medical photographs taken after 2 months of treatment were rated by two independent evaluators.

Results: We treated 31 girls and 4 boys with a median age of 3.5 months. Rapid improvement was reported in the first days of treatment in 34 patients. Mean improvement after 2 months was 61.5%. No serious adverse effects were reported.

Conclusion: Propranolol was effective in controlling the proliferative phase of problematic IH. It was well tolerated in our study. Outpatient treatment is possible if parents follow strict guidelines. Propranolol should be a first-line treatment for problematic IH in carefully selected patients.
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http://dx.doi.org/10.2310/7750.2011.10117DOI Listing
August 2012

Propranolol versus prednisone in the treatment of infantile hemangiomas: a retrospective comparative study.

Pediatr Dermatol 2011 Nov-Dec;28(6):649-654. Epub 2011 Oct 13.

Division of Dermatology, Department of Pediatrics, CHU Sainte Justine, University of Montreal, Montreal, Canada.

The goal of the current study was to compare the clinical effectiveness of oral propranolol with that of oral prednisone in the treatment of infantile hemangiomas (IH). Patients treated for IH with oral propranolol were retrospectively matched with patients treated with oral prednisone according to type, location, and size of the IH and age at start of treatment. Response to treatment was evaluated by rating serial medical photographs taken 1, 2, and 6 months after initiation of treatment. Degree of clinical improvement in overall appearance (including color and size) was rated as follows: worse or stable (0), slight (<25%), moderate (25-50%), good (50-75%), or excellent (>75%). A second assessment was done using a 100-mm visual analog scale to rate improvement at 6 months. Pre and post-treatment imaging was available for several patients. Twelve pairs of infants with IH were analyzed. At 1 month, clinical improvement in the propranolol group was moderate to good in all patients. In the prednisone group, only one patient had moderate improvement, with others showing slight (7/12) or no improvement or stabilization (3/12) from baseline and one case worsening. At 6 months, the propranolol group showed good to excellent response in all cases, whereas nine in the prednisone group showed slight to moderate response. Doppler ultrasound and magnetic resonance imaging correlated with the clinical improvement in the cases in which it was performed. No major side effects were observed in either group. Propranolol appears superior to oral prednisone in inducing more-rapid and greater clinical improvement in this study. A larger prospective study comparing these two treatment modalities is warranted.
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http://dx.doi.org/10.1111/j.1525-1470.2011.01551.xDOI Listing
March 2012

Childhood linear IgA bullous disease induced by trimethoprim-sulfamethoxazole.

J Dermatol Case Rep 2010 Dec;4(3):33-5

Division of Dermatology, CHU Sainte-Justine, University of Montreal, Montreal, Quebec, Canada.

Background: Linear IgA bullous disease (LABD) is a rare mucocutaneous autoimmune subepidermal blistering disease that can affect children mostly of pre-school age. As many as two-thirds of LABD are related to drug ingestion, particularly certain antibiotics, non-steroidal anti-inflammatory drugs and diuretics.

Main Observation: We describe a 3-year-old boy who presented a CMV infection followed by LABD induced by trimtheporim-sulfametoxazole. To our knowledge, this is the first reported case of trimethoprim-sulfamethoxazole that was confirmed by a rechallenge.

Conclusions: Most cases of drug-induced LABD are patients being treated with multiple systemic drugs that could induce the LABD. In the lack of suitable alternative treatment, the identification of the causative drug can be achieved by a rechallenge under close medical surveillance.
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http://dx.doi.org/10.3315/jdcr.2010.1053DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3157814PMC
December 2010

Targetoid hemosiderotic hemangiomas (hobnail hemangiomas) are vascular lymphatic malformations: a study of 12 pediatric cases.

J Am Acad Dermatol 2012 Jan 27;66(1):116-20. Epub 2011 Jul 27.

Division of Dermatology, CHU Sainte Justine, University of Montreal, Montreal, Quebec, Canada.

Background: Targetoid hemosiderotic hemangioma (THH), also called hobnail hemangioma, is a benign vascular lesion and thought to be of lymphatic origin.

Objective: We sought to perform a clinicopathologic analysis of cases diagnosed as THH in a tertiary care children's hospital.

Methods: Clinical and histopathologic data were obtained from a chart review of 12 confirmed pediatric cases of THH. To determine the presence or absence of lymphatic vessels in lesional biopsy specimens, we evaluated the expression of the lymphatic endothelial cell marker podoplanin using the D2-40 antibody. Wilms tumor-1 gene immunostaining and Ki-67 proliferation index were also performed to evaluate the proliferative nature of these lesions.

Results: Three children had a lesion since birth and 4 had a history of trauma before appearance of the THH. D2-40 immunostaining was positive in every case. Wilms tumor-1 gene immunostaining was negative in 9 cases, focally positive in two cases, and not performed in one case. The Ki-67 proliferation index was very low in all cases studied.

Limitations: The small number of cases and restriction to a pediatric population were limitations.

Conclusion: Our findings suggest that THH should be classified as a lymphatic vascular malformation.
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http://dx.doi.org/10.1016/j.jaad.2011.05.025DOI Listing
January 2012

Treatment of periocular infantile hemangiomas with propranolol: case series of 18 children.

Ophthalmology 2011 Jun 3;118(6):1184-8. Epub 2011 Feb 3.

Division of Dermatology, CHU Sainte Justine, University of Montreal, Montreal, Quebec, Canada.

Purpose: To study the efficacy of propranolol in the treatment of periocular infantile hemangiomas (IHs).

Design: Retrospective interventional case series.

Participants: Eighteen children presenting periocular IH with occlusion of the pupil, anisometropic astigmatism, proliferating eyelid IH, or cosmetically disfiguring periocular IH.

Methods: All patients received treatment with propranolol started at 0.5 mg/kg/day with an incremental increase by 0.5 mg/kg/day every 4 days, up to a maximum of 2 to 3 mg/kg/day. Complete eye examinations and serial photographs were obtained before, during, and after treatment. Doppler ultrasound and magnetic resonance imaging performed pre- and post-treatment were compared when available.

Main Outcome Measures: Evolution of the treated IH was evaluated with respect to astigmatism, amblyopia, and size of the lesion.

Results: The IH size decreased in 17 of 18 patients. We noted a greater reduction when treatment was administered during the proliferative phase of growth of IHs. At the conclusion of treatment, none of our patients had amblyopia. The mean value of amblyogenic astigmatism (n = 7) decreased from 2.71 diopters (D) pretreatment to 1.03 D post-treatment. On radiology, 8 patients had significant regression of the lesion size of their IH and 1 patient had a limited progression. Propranolol had to be temporarily discontinued in only 1 patient because of symptomatic hypotension.

Conclusions: Propranolol seems to be an effective modality of treatment for periocular IH. It seems to be most efficacious when initiated in the proliferative phase of IH but may be beneficial even in the later stage.

Financial Disclosure(s): The author(s) have no proprietary or commercial interest in any materials discussed in this article.
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http://dx.doi.org/10.1016/j.ophtha.2010.10.031DOI Listing
June 2011

Large congenital melanocytic nevi and neurocutaneous melanocytosis: one pediatric center's experience.

J Am Acad Dermatol 2009 Nov 18;61(5):766-74. Epub 2009 Sep 18.

Dermatology Service, Department of Pediatrics, Sainte-Justine Hospital Center, Montreal, Quebec, Canada.

Background: Large congenital melanocytic nevi (LCMN) predispose to neurocutaneous melanocytosis (NCM), which is associated with significant morbidity and mortality.

Objective: To identify risk factors for NCM in patients with LCMN and suggest guidelines for their management.

Methods: Medical records of patients with LCMN were reviewed at Sainte-Justine Hospital between 1980 and 2006. Presence of multiple satellite nevi and posterior midline location were evaluated as risk factors for NCM using chi-square test. Magnetic resonance imaging scans were reviewed by a neuroradiologist.

Results: Twenty-six of 52 patients underwent radiologic investigation. Six of 26 (23%) had NCM. Patients with this condition are more likely to have multiple satellite nevi (100% vs 50%, P = .03) and have a trend to posterior midline location of their LCMN (100% vs 60%, P = .08). Patients with NCM are more likely to have both multiple satellite nevi and posterior midline location (100% vs 25%, P = .002). Radiologic findings are also presented.

Limitations: This was a retrospective case series with imprecise chart data in 38% of cases.

Conclusion: The presence of multiple satellite nevi alone or with associated posterior midline location of LCMN is associated with a higher risk of NCM. We recommend magnetic resonance imaging testing before 4 months of age in patients with these features.
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http://dx.doi.org/10.1016/j.jaad.2008.11.022DOI Listing
November 2009

A prospective randomized trial comparing the efficacy and adverse effects of four recognized treatments of molluscum contagiosum in children.

Pediatr Dermatol 2006 Nov-Dec;23(6):574-9

CHU Sherbrooke, University of Montreal, Montreal, Quebec, Canada.

Molluscum contagiosum is a common viral disease of childhood presenting as small, firm, dome-shaped umbilicated papules. Although benign and generally self-limited, this condition is contagious and can lead to complications such as inflammation, pruritus, dermatitis, bacterial superinfection, and scars. No consensus has been established concerning the management of this condition. We conducted a prospective randomized study comparing four common treatments for molluscum contagiosum in 124 children aged 1 to 18 years. One group was treated with curettage, a second with cantharidin, a third with a combination of salicylic acid and lactic acid, and a fourth with imiquimod. Patients needing, respectively, one, two, or three visits for treatment of their mollusca were: 80.6%, 16.1%, and 3.2% for curettage, 36.7%, 43.3%, and 20.0% for cantharidin, 53.6%, 46.4%, and 0% for salicylic acid and glycolic acid, and 55.2%, 41.4%, and 3.4% for imiquimod. The rate of side effects was 4.7% for group 1, 18.6% for group 2, 53.5% for group 3, and 23.3% for group 4. Curettage was found to be the most efficacious treatment and had the lowest rate of side effects. It must be performed with adequate anesthesia and is a time-consuming procedure. Cantharidin is a useful bloodless alternative particularly in the office setting, but has moderate complications due to blisters and necessitated more visits in our experience. The topical keratolytic used was too irritating for children. Topical imiquimod holds promise but the optimum treatment schedule has yet to be determined. Finally, we believe that the ideal treatment for mollusca depends on the individual patient preference, fear, and financial status, distance from the office, and whether they have dermatitis or blood-borne infections.
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http://dx.doi.org/10.1111/j.1525-1470.2006.00313.xDOI Listing
October 2007
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