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   2018| January-March  | Volume 19 | Issue 1  
    Online since December 28, 2017

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Stevens-Johnson syndrome and toxic epidermal necrolysis in children
Sudip Das, Ramkumar Ramamoorthy
January-March 2018, 19(1):9-14
SJS and TEN are diseases characterised by epidermal detachment and necrolysis predominantly of drug induced etiology. SJS/TEN begins with a prodrome of fever, malaise, anorexia, pharyngitis, and headache lasting for 2-3 days, at times, extending to 10-11 days. Mucosal lesions usually precede skin lesion. Usually, two mucosal membranes are involved, most commonly conjunctiva and oral mucosa. Oral mucosal involvement is seen in 90% of SJS and almost all patients diagnosed with TEN. In SJS, mucosal involvement is widespread and confl uent in contrast to erythema multiforme where it is focal and seen in only 25%-60% of cases. The exact pathogenesis of SJS and TEN is not fully elucidated. In view of the paucity of T-cell infiltrate,keratinocyte apoptosis could be the result of autocrine or paracrine interaction between Fas, a death receptoron keratinocyte and Fas Ligand (FasL) produced by the keratinocytes along with the substantial contributionof soluble FasL from peripheral mononuclear cells. FasL upregulation in keratinocytes is nitric oxide-dependent anddriven by T-cell derived tumor necrosis factor (TNF) alpha and Interferon-gamma.The management revplves around immediate stoppage of drug and.,supportive care .IVIG,corticosteroids and cyclosporine are all effective drugs but no RCT is available for any of them
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Tinea capitis in children: A report of four cases trichoscopic with trichoscopic features
Ebtisam Elghblawi
January-March 2018, 19(1):51-56
Tinea capitis (TC) is the most prevalent pediatric superficial dermatophyte infection. The culprit species differ across the continents; for instance, Microsporum canis prevails in Europe, whereas Trichophyton tonsurans dominates in North America. Oral medication is always* the main required remedy as TC does not respond well to topical therapy unaided. However, in some countries, griseofulvin is no longer available. Fungal culture should be included in the study of persistent, scalp lesions and trichoscopy can offer a speedy diagnosis by its characteristic findings. Scalp dermoscopy or “trichoscopy” represents a valuable, noninvasive technique for the evaluation of patients with hair loss due to TC. It is simple, quick, and easy to perform.
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Vascular birthmarks: A hidden world behind a word
Carlo Gelmetti
January-March 2018, 19(1):1-8
A confusing nomenclature concerning the subject of vascular birthmark and angiomas lead (and leads!) to the consequence that parents with children affected by this kind of disorders become medical nomads, and therefore, the best treatment, when available, is frequently postponed. The concept of maternal imprint that began to crumble in the XVIII century is, unfortunately, still widespread. While a maternal responsibility as it has been conceived in the past has been definitively excluded, the pivotal classification proposed by John Mulliken that divides vascular anomalies in vascular tumors and vascular malformations is practically very useful. The rapid progress of genetic studies has explained to medical community a huge number of genotype-phenotype correlations, and it will individuate new forms in the next future. Understanding the biology of vascular birthmarks is a fundamental step forward to implement effective and specific drugs for specific forms. Discoveries by serendipity occur but are rare.
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Unusual presentations and associations of hyper IgE Syndrome: Retrospective analysis of ten cases at tertiary care institute – With review of indian published reports
Nayan H Patel, Jigna K Padhiyar, Yogesh B Shah, Trusha P Gajjar, Mansi D Buch
January-March 2018, 19(1):31-36
Background: Job syndrome also known as hyper IgE syndrome (HIES) is primary immunodeficiency syndrome. Autosomal dominant variant caused by mutation in signal transducer and activator of transcription-3 gene is characterized by recurrent staphylococcal skin infections, sinopulmonary infections, eczema, recurrent bone fractures, and coarse facial features. Autosomal recessive (AR) variant is because of mutation in DOCK8 gene which lacks the skeletal and dental involvement but manifest with severe viral infection and develop neurological complications. Aims: This study aims to evaluate variable presentations and associations of job syndrome. Methods: Analysis of ten cases, presented at our department between October 2015 and September 2016, with suspected HIES was done retrospectively. All cases were studied for their presentations and associations and were investigated accordingly for the same. Score for HIES was counted as per National Institutes of Health (NIH) scoring system. Indian published reports were found by internet search engine and were reviewed for unusual presentations. Results: NIH scoring was indeterminate in two patients and was highly likely for autosomal dominant-HIES in five patients. Three patients were diagnosed as AR-HIES. Two of our patients had multiple endocrinopathy, one had pyoderma gangrenosum and two patients had autoimmune disorders. Limitations: Genetic analysis was not done because of nonavailability of testing in our state and poor financial conditions of patients. It is a retrospective study. Conclusion: Our patients had unusual association in the form of multiple endocrinopathy, pyoderma gangrenosum, dermatomyositis, and all were diagnosed very much late in life. Hence, a vigilant eye for suspicion at a younger age might benefit patients.
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Childhood leprosy in the postelimination era: A vision achieved or a concern growing at large
Ashwini Babu, M Ramesh Bhat, Jyothi Jayaraman
January-March 2018, 19(1):26-30
Background: The proportion of childhood leprosy has been gaining importance of late as a performance indicator of the National Leprosy Eradication Program. A sizable proportion of the newly detected cases comprises children. The prevalence rate has been used as a measure of existence of infection though leprosy has attained elimination levels in the country. Materials and Methods: A retrospective descriptive study was conducted. Case records of children newly diagnosed with leprosy aged <15 years between April 2005 and October 2015 were studied and included in the study. Findings such as patches, nerve thickness, spectrum of leprosy, grade of deformity, smear status, presence of reaction, histopathological diagnosis, and treatment history were noted. Results: The percentage of childhood leprosy among the newly diagnosed cases in our study was found to be 11.2%. A solitary skin lesion was the most frequent manifestation in 64.4% children. The second most common finding seen in these patients included the presence of thickened nerves in 46.6% children. Smear showed acid-fast bacilli in 8.8% cases. Conclusion: There is an urgent need for improving clinical acumen in diagnosing childhood leprosy. The importance of appropriate and complete treatment along with meticulous surveillance in endemic areas and looking for household contacts needs to be stressed on.
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Effect of occurrence of vitiligo in children over quality of life of their families: A hospital-based study using family dermatology life quality index
Pratik Gahalaut, Sandhya Chauhan, Amit Shekhar, Madhur Kant Rastogi, Nitin Mishra
January-March 2018, 19(1):21-25
Introduction: Vitiligo in an adult patient has a profound effect on the quality of life (QoL) of that particular patient. Although it is known that vitiligo in adult patient affects QoL in their family, very little information is available regarding QoL in a family having children suffering from vitiligo. Materials and Methods: This is a cross-sectional, hospital-based study to ascertain the effect children suffering from vitiligo have on the QoL of their respective families. Study participants were fifty healthy parent/immediate caregivers of fifty corresponding children suffering from vitiligo. The QoL in the family of these vitiliginous children was assessed with the help of Family Dermatology Life Quality Index (FDLQI). Results: There was significantly more impairment of FDLQI among respondents if vitiliginous child was female compared to male. The total FDLQI showed a significant negative correlation with duration of vitiligo in children. Analysis of individual items in FDLQI revealed emotional distress as the most impaired facet of FDLQI and housework as the least affected item. Conclusion: Presence of vitiligo in children affects the QoL of that particular child and his/her family. This impairment of FDLQI is more if the child suffering from vitiligo is female. Treatment of vitiligo in pediatric age group should include psychological counseling and support for the child as well as their parents/caregivers.
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Legius syndrome
Manoj Kumar Sharma, Ramesh Kumar, Savera Gupta, Suresh Kumar Jain
January-March 2018, 19(1):86-88
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Study of physiological and pathological skin changes in neonates: An east indian perspective
Binodini Behera, Yatra Kavadya, Prasenjit Mohanty, Dipanweeta Routray, Subhra Ghosh, Lina Das
January-March 2018, 19(1):40-47
Background: Numerous dermatological conditions are prevalent in neonatal period, i.e., first 28 days of life with varied presentations ranging from transient self-limiting lesions to serious dermatosis requiring clinical attention. Due to lack of complete knowledge, it is distressing for both parents and treating physician. Objectives: This study was done in eastern part of India to record various dermatosis, and their prevalence compared with other part of country and find association between skin conditions and parameters, namely, sex, parity, gestational age, mode of delivery, and birth weight. Materials and Methods: This descriptive cross-sectional study was done in tertiary care hospital entailed recording of 400 neonates having any kind of dermatosis and statistically evaluated using Chi-square test (SPSS) and significance (P < 0.05). Results: Mongolian spot followed by sebaceous gland hyperplasia and epstein pearls were the common dermatologic findings. Physiological desquamation, acrocyanosis, and sebaceous gland hyperplasia were more prevalent in males and was statistically significant while erythema toxicum neonatorum significantly high in females. Epstein pearls, physiological scaling was also significantly high in term babies. Physiological scaling in cesarean patients and acrocyanosis in vaginal delivery was also significantly associated. Some atypical cases such as phakomatosis pigmentovascularis, plantar keratoderma, collodion baby, and congenital whorled hypermelanosis were also present in our study group. Conclusion: Physiological skin lesions are very common in neonates and appreciation of these lesions are important.
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Hoyeraal–Hreidarsson syndrome: A rare dyskeratosis congenita phenotype
Mansoor C Abdulla
January-March 2018, 19(1):83-84
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Congenital insensitivity to pain: Case report of a rare entity
Swati Dahiya, Kondajji Ramchandra Vijayalakshmi, Mubeen Khan
January-March 2018, 19(1):48-50
Hereditary sensory and autonomic neuropathies (HSANs) are a group of disorders characterized by insensitivity to noxious stimuli and autonomic dysfunction, associated with pathological abnormalities of the peripheral nerves. Five types of HSAN have been reported in literature, out of which Type V known as congenital insensitivity to pain (CIP) is a rare autosomal recessive condition. Self-mutilation is an invariable feature of this disorder, involving the teeth and orofacial structures. This case report describes a case of a 6-year-old girl with CIP brought by her parents for prostheses to replace her self-extracted primary teeth.
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Prevalence of dermatoses among the tribal children studying in residential schools of South Chhattisgarh, India
Yasha Upendra, Sampreeti Sendur, Naveen Keswani, Abhishek Pallava
January-March 2018, 19(1):15-20
Introduction: Dermatoses are common among schoolgoing children and lead to significant morbidity. Objective: The present study was conducted to find the prevalence of various dermatoses in children studying in residential schools of remote tribal areas. Materials and Methods: A cross-sectional study was conducted on 1943 children studying in eight residential government schools over a period of 2 months (August and September 2016). Diagnosis of various dermatoses was made on the basis of detailed clinical examination and necessary investigations. Data analysis done by SPSS version 21.0 and Chi-square test was used to analyze categorical variables. Results: The prevalence of dermatoses was 72.1%. Majority had single skin lesion. Among infections and infestations, scabies (13.6%), pediculosis capitis (9.2%), and pyoderma (7.5%) were most prevalent. Among the noninfective conditions, xerosis (32.63%), pityriasis alba (18.9%), acne (8.6%), and atopic dermatitis were most prevalent. The prevalence of pyoderma, tinea corporis, pityriasis versicolor, scabies, and xerosis was higher in boys while pediculosis capitis and pityriasis alba were more prevalent in girls. The prevalence of xerosis, pityriasis alba, and phrynoderma was higher in primary school students while tinea corporis, scabies, acne, and pediculosis capitis were more prevalent in high school students. Conclusions: The burden of dermatoses in residential school children is very high. Health education of children, teachers, and caregivers regarding signs and symptoms of dermatoses is warranted for early detection and timely intervention. There is urgent need to address dermatoses under school health program.
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Dermatopathia pigmentosa reticularis
Gauri Vats, Rohit Kataria, Durgesh Sonare, Vinod Jain
January-March 2018, 19(1):77-79
Dermatopathia pigmentosa reticularis (DPR) is a rare disorder with characteristic triad of generalized reticulate hyperpigmentation, noncicatricial alopecia, and onychodystrophy. A 12-year-old Indian boy presented with classical features of the triad along with adermatoglyphia, hyperhidrosis, punctate hyperkeratosis of palm, and sole along with keratosis pilaris. Histopathology suggested a diagnosis of DPR with a differential of Naegeli–Franceschetti–Jadassohn syndrome and dyskeratosis congenita.
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Cutaneous leishmaniasis in children: A case series
Mrinal Gupta
January-March 2018, 19(1):37-39
Background: Cutaneous leishmaniasis (CL) is a protozoal disease usually caused by Leishmania major and Leishmania tropica and transmitted by the bite of a sandfly. It is usually characterized by a single, polymorphous lesion located in an uncovered area. Aims: This study aimed to evaluate the clinico-epidemiological characteristics of CL among children. Materials and Methods: It was a retro-prospective study carried out over a period of 18 months in our center, in which the clinico-epidemiological features of children presenting with CL were assessed. Results: A total of 10 children (male:female - 7:3) were included in the study with the age range of 1–15 years, with a mean age of 8.8 years. Face was the most commonly affected site (n = 8), followed by neck and hands (1 each), and nodulo-ulcerative was the most common clinical type seen in nine patients. Skin smears for Leishman–Donovan (LD) bodies were positive in five patients while the skin biopsy, which was done in four cases, showed the presence of LD bodies in only one patient. Conclusion: CL is a common presentation among children, especially in the endemic areas, and the clinical features are similar to the adult onset disease.
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Recurrent pilomatricoma of the thigh: An unusual site of presentation
Vanlalhriatpuii Hmar, Nandakishore Thokchom, Sandhyarani Kshetrimayum
January-March 2018, 19(1):65-67
Pilomatricoma is a benign tumor of the hair matrix. It is also known as calcifying epithelioma of Malherbe and is commonly located in head, neck, and upper extremities. A 10-year-old female patient presented with a solitary skin-colored firm nodule on her left mid-thigh which progressed to 1.5 cm diameter within 4 months. Surgical excision had been conducted 1 year ago at the same site. Clinical examination revealed a single, firm, well-circumscribed, non-tender, freely mobile nodule with irregular surface and hyperpigmented borders including surrounding skin. Histopathological examination showed dermis displaying benign adnexal tumor tissue with sheets of ghost cells, clusters of basaloid cells, and extensive areas of dystrophic calcification. Histopathological confirmation prior excision should be ideally performed. Although the tumor is most common in head and neck region, clinicians should be aware that tumors can arise rarely in the lower limbs as well.
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Ecthyma gangrenosum as the presenting clinical feature of autoimmune lymphoproliferative syndrome
Aniruddha Ghosh, Rashmita Das, Partha Pratim Halder, Sandipan Dhar
January-March 2018, 19(1):62-64
Ecthyma gangrenosum (EG) is a severe invasive cutaneous infection caused classically by Pseudomonas aeruginosa typically affecting immunocompromised patients especially those with neutropenia. We report the case of a 2-year-old boy with autoimmune lymphoproliferative syndrome presenting with solitary EG on the forehead. Blood culture, as well as culture from the lesion both, showed growth of P. aeruginosa. Pseudomonal sepsis responded well to piperacillin-tazobactam and amikacin therapy. Prompt diagnosis of the lesion is crucial as failure to start timely appropriate therapy may lead to fatal outcome.
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Monomorphic bullous hemorrhagic varicella in a patient on methotrexate
Swastika Suvirya, Amita Jain, Avinash Agrawal, Parul Jain
January-March 2018, 19(1):59-61
Varicella, a common exanthematous disease of childhood, can have atypical presentation in immunosuppressed patients. We report a case of a 14-year-old female who presented with multiple hemorrhagic tense bullae and a monomorphic presentation that created an initial diagnostic dilemma. Patient's treatment history was remarkable for taking methotrexate for arthritis for the last 2 months. The patient was treated with a prolonged course of intravenous acyclovir that eventually led to her uneventful recovery.
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Cutaneous polyarteritis nodosa: Diagnosis lies skin deep
Raghavendraswami Amoghimath, Rajwanti K Vaswani, Chandrahas T Deshmukh, Chhaya Divecha
January-March 2018, 19(1):71-73
Cutaneous polyarteritis nodosa (CPAN) is an uncommon form of cutaneous vasculitis. Definitive diagnosis is based on skin biopsy. We present a rare case of CPAN in a 2-year-old male child who presented with a history of fever, cutaneous ulcers, and digital gangrene. He was treated with steroids and skin lesions had resolved completely. CPAN needs to be differentiated from systemic polyarteritis nodosa for specific prognosis and management. We report this case to highlight the need of early diagnosis for faster recovery with favorable outcome.
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Twin-spot phenomenon: A rare association hypomelanosis of ito with nevoid hypertrichosis
Projna Biswas, Abhishek De , Dayamay Pal, Asad Ansari, Sudip Das
January-March 2018, 19(1):88-90
  831 110 -
Autosomal recessive anhidrotic ectodermal dysplasia: A rare disease
Tasleem Arif, Mohammad Adil, Syed Suhail Amin, Konchok Dorjay
January-March 2018, 19(1):84-86
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A rare case of neurocutaneous disorders of the newborn: Incontinentia pigmenti
Melek Aslan Kayiran, Mehmet Salih Gürel, Burçe Can Kuru, Ilkin Zindancı
January-March 2018, 19(1):68-70
Incontinentia Pigmenti (IP) is a rare X-linked dominant disorder which is mostly lethal for males. It effects hair, teeth, nails, eyes and central nervous system along with skin. A twelve day old female patient was referred to the dermatology departmant due to skin eruptions that began the day before. There was a widespread erythema and many papuls and vesicles were seen on the eryhematous skin in linear sequence. Spongiosis with eosinophilia and necrotic keratinocytes were seen on histopathologic examination. The patient was diagnosed with incontinentia pigmenti. Vesicles and bullous eruption are rarely seen in newborns. The physcians should not avoid histopathologic examination especially in females. Since incontinentia pigmenti is a rare but multisystemic disorder, early diagnosis is crucial.
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Kasabach–Merritt syndrome in an infant successfully treated with a combination of propranolol and methotrexate
Thulasi Weerasinghe, AC Ranasinghe, Jayamini Seneviratne
January-March 2018, 19(1):57-58
Kasabach Merritt syndrome is a rare childhood vascular tumour,which develops bleeding as an acute complication. The underlying tumour is either a Kaposiform haemangioendothelioma or a tufted angioma. Tumour associated platelet trapping, activation and concomitant fibrinolysis leads to bleeding. Herein we report a case with Kasabach Merritt syndrome initially treated with a combination of drugs but subsequently successfully managed with Methotrexate.
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Dego's disease in a female child with Anti-Scl-70 antibody positivity
Jigna Krunal Padhiyar, Nayan H Patel, Trusha P Gajjar, Mansi D Buch
January-March 2018, 19(1):80-82
Degos' disease (DD) also known as malignant atrophic papulosis is considered to be a disorder of abnormal coagulation with obliterative arteritis. Its association has been described with connective tissue disorders, human immunodeficiency virus infection (HIV) and wegener's granulomatosis. Gastrointestinal, neurological, ocular, cardiovascular, and pulmonary involvement can also occur in DD. Benign version of DD with only cutaneous manifestations has been described. We are presenting a case of 7-year-old female with multiple irregular-shaped crusted tender lesions with porcelain white scars of healed lesions mainly over the trunk and upper limb and few lesions involving face and legs for the last 6 months. There was a history of occasional headache. Other systemic examination by experts was within normal limit. On further investigation, antinuclear antibody, and anti-Scl-70 antibody were positive with histopathology suggestive of DD. The patient had reduced peak expiratory flaw rate (PEFR). The patient was given oral prednisolone without much improvement. With oral aspirin for a month, the patient had mild improvement. The patient is presently under follow-up for future systemic involvement and response to therapy. Anticipated mortality in DD is 50%–60%. Earlier diagnosis and proper management at such a younger age might benefit the patient; however, they need to be followed up for complications. DD is very rare and very few cases have been reported in children. Ours is unique with anti-Scl-70 antibody positivity.
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A rare co-existence of langerhans cell histiocytosis and lichen planus
Aarti S Salunke, Anil P Gosavi, Ravindranath B Chavan, Pallavi Prataprao Patil
January-March 2018, 19(1):74-76
Langerhans cell histiocytosis (LCH) is the commonest of the histiocytic disorders. Owing to the relative rarity of the condition, it remains a disease in which the diagnosis is often delayed or missed. Commonly seen in infancy or early childhood, the disorder is characterized by proliferation of abnormal and clonal Langerhans cells in skin, bone, lymph nodes, lungs, liver, spleen, and bone marrow. Lichen planus presents as purple, polygonal, pruritic, papular eruption of unknown etiology affecting skin, mucous membranes and nails. Despite of thourough literature search, we could not find any association of lichen planus with LCH. Hereby, we report a case of five year old child with acute onset of polymorphic eruptions all over the body which on histopathology and Immunohistochemistry showed features of Langerhans cell histiocytosis and lichen planus on bilateral lower limb.
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Erratum: Tinea Capitis in Children: A Report of Four Cases Trichoscopic with Trichoscopic features

January-March 2018, 19(1):91-91
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