Indian Journal of Dermatology
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   Table of Contents - Current issue
November-December 2020
Volume 65 | Issue 6
Page Nos. 441-572

Online since Friday, October 23, 2020

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Time for next transformation Highly accessed article p. 441
Dwijendra Nath Gangopadhyay
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Chemical vitiligo: A subset of vitiligo Highly accessed article p. 443
Sanjay Ghosh
Chemical leucoderma, an under-diagnosed common condition often mimicking idiopathic vitiligo, represents an acquired depigmentation induced by repeated exposure to specific chemical compounds in subjects with genetic susceptibility to vitiligo. This has been increasing rapidly in incidence in recent decades in developing countries like India. The term 'chemical vitiligo' was first coined by us to indicate the possible relationship between chemical leucoderma and vitiligo, which has been supported recently by other authors to designate the term 'chemical-induced vitiligo'. The largest case series showed that household chemical exposure was the major etiological factor. Causative chemicals are mostly phenolic and catecholic derivatives. Vitiligo pathogenesis is induced by genetic and environmental factors like many other autoimmune diseases. Innate immunity acts as a bridge between cellular stress and adaptive immunity. Multiple patches are commonly seen; children below 12 years are also affected in good numbers. The most common presence of confetti macules indicates these as characteristic, although not pathognomonic, of chemical leucoderma. Chemical leucoderma has been broadened into 'chemical leucoderma syndrome' with proper staging. The clinical criteria for diagnosis of chemical leucoderma have been specifically outlined. Same pathomechanism of chemical leucoderma might elucidate trigger factors and reasons for progression and chronicity in idiopathic vitiligo. Depigmentation in chemical vitiligo spreads to distant sites, in the same way as generalized idiopathic vitiligo. The study showed that chemical triggering factors played a very significant role in the induction and progression of vitiligo. Thus it should be rational to consider chemical vitiligo not as a separate entity but as a major subset of vitiligo spectrum.
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Post kala-azar dermal leishmaniasis p. 450
Nilay Kanti Das
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Immune responses in post kala-azar dermal leishmaniasis p. 452
Mitali Chatterjee, Ritika Sengupta, Debanjan Mukhopadhyay, Shibabrata Mukherjee, Aishwarya Dighal, Srija Moulik, Shilpa Sengupta
Kala-azar, commonly known as visceral leishmaniasis (VL), is a neglected tropical disease that has been targeted in South Asia for elimination by 2020. Presently, the Kala-azar Elimination Programme is aimed at identifying new low-endemic foci by active case detection, consolidating vector control measures, and decreasing potential reservoirs, of which Post Kala-azar Dermal Leishmaniasis (PKDL) is considered as the most important. PKDL is a skin condition that occurs after apparently successful treatment of VL and is characterized by hypopigmented patches (macular) or a mixture of papules, nodules, and/or macules (polymorphic). To achieve this goal of elimination, it is important to delineate the pathophysiology so that informed decisions can be made regarding the most appropriate and cost-effective approach. We reviewed the literature with regard to PKDL in Asia and Africa and interpreted the findings in establishing a potential correlation between the immune responses and pathophysiology. The overall histopathology indicated the presence of a dense, inflammatory cellular infiltrate, characterized by increased expression of alternatively activated CD68+ macrophages, CD8+ T cells showing features of exhaustion, CD20+ B cells, along with decreased CD1a+ dendritic cells. Accordingly, this review is an update on the overall immunopathology of PKDL, so as to provide a better understanding of host-parasite interactions and the immune responses generated which could translate into availability of markers that can be harnessed for assessment of disease progression and improvement of existing treatment modalities.
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Histopathology of post kala-azar dermal leishmaniasis p. 461
V Ramesh, M Ramam
The various lesions seen in the clinical presentation of post kala-azar dermal leishmaniasis (PKDL) are reflected in the histopathology of the type of lesion biopsied. The cells that form the dermal infiltrate include lymphocytes, histiocytes, and plasma cells in varying proportions. The infiltrate, which is mild and confined to the superficial dermis in macular lesion becomes denser with the increasing severity of the lesion. Leishman–Donovan bodies (LDB) in general are rarely demonstrable in macules and somewhat infrequently in the rest, though at times they may be numerous; mucosal lesions offer a greater chance of visualizing LDB than biopsies from the skin. A characteristic histomorphology in nodules is prominent follicular plugging with a dense plasma cell-rich lymphohistiocytic dermal infiltrate that shows an abrupt cut-off in the lower dermis, an appearance highly suggestive of PKDL even in the absence of LDB. Russell bodies within plasma cells, vascular changes, and xanthoma-like hue have been seen in plaques from chronic PKDL. The histopathologic picture in some may also mimic that seen in tuberculoid and lepromatous leprosy, and other granulomatous dermatoses. In contrast to Indian PKDL, epithelioid cell granulomas with giant cells are more common in African PKDL, and vascular changes are rare though neuritis showing LDB has been described.
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Advancement in molecular diagnosis of post kala-azar dermal leishmaniasis p. 465
Keerti Kaumudee Dixit, Ruchi Singh, Poonam Salotra
Post kala-azar dermal leishmaniasis (PKDL), a clinical sequela of visceral leishmaniasis (VL), plays a critical role in the anthroponotic transmission of VL, particularly in the Indian subcontinent (ISC). The early, accurate, and feasible diagnosis of PKDL is essential for the attainment and sustenance of VL elimination goal in ISC. PKDL poses a stumbling block for this goal, considering the heterogeneity presented with regard to time after cure of VL and onset of PKDL, chronicity, and clinical variations. In most of the endemic regions the diagnosis is based on clinical examination, previous history of VL, by ruling out other disorders, and by the response to treatment. The conventional microscopic examination involving the demonstration of Leishman–Donovan bodies (LDB) in macrophages is pathognomonic, however, the method faces constraints in terms of being invasive, less sensitive, technically demanding, and difficult to be applied in field conditions. Serological evidences are of limited use because antileishmanial antibodies remain positive for years after VL treatment. Molecular tools such as PCR, nested-PCR, Q-PCR overcome these constraints and have become increasingly popular due to their high sensitivity and specificity along with their applicability in diverse clinical samples. Molecular methods not only play a key role in early detection but also provide quantification and monitoring of treatment effectiveness. NCBI PubMed search tool was used for locating, selecting, and extracting research articles pertinent for this review article by using various related terminologies on the molecular diagnosis of leishmaniasis.
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A nationwide, multicentric case–control study on vitiligo (MEDEC-V) to elicit the magnitude and correlates p. 473
Nilendu Sarma, Sayantani Chakraborty, Shital Poojary, B M Shashi Kumar, Lalit Kumar Gupta, Leelavathy Budamakuntla, Leishiwon Kumrah, Suchibrata Das, Ajay Govindrao Ovhal, Nirmal Kumar Mandal, Shuvankar Mukherjee, TV Anoop, Binod Kumar Thakur, L Eswari, Joan Felicita Samson, Krina Bharat Patel, Rajesh Rajagopalan, Sanjeev Gupta, Tejinder Kaur
Background: Vitiligo is an acquired, idiopathic, and common depigmentation disorder. The values of various epidemiologic parameters are often doubtful due to the methodological weaknesses of the studies. Aims: To elicit the magnitude of various epidemiological parameters and important correlates of vitiligo. Materials and Methods: Every vitiligo patient attending the outpatient department of medical colleges spread over most of the Indian states were examined over a period of 1 year. Various epidemiological and clinical variables were examined and compared with age and sex-matched controls (registered in the Clinical Trial Registry of India CTRI/2017/06/008854). Results: A total of 4,43,275 patients were assessed in 30 medical colleges from 21 Indian states. Institutional prevalence of vitiligo was 0.89% (0.86% in males and 0.93% in females,P <0.001). The mean age at presentation and mean age at onset were 30.12 ± 17.97 years and 25.14 ± 7.48 years, respectively. Head–neck was the most common primary site (n = 1648, 41.6%) and most commonly affected site (n = 2186, 55.17%). Most cases had nonsegmental vitiligo (n = 2690, 67.89%). The disease started before 20 years of age in more than 46% of cases. About 77% of all cases had signs of instability during the last 1 year. The family history, consanguinity, hypothyroid disorders, and depressed mood were significantly (P < 0.001) higher among the cases. First, second, and third-degree family members were affected in 269 (60.04%), 111 (24.78%), and 68 (15.18%) cases, respectively. Work-related exposure to chemicals was significantly higher among cases (P < 0.008). Obesity was less common among vitiligo cases [P < 0.001, odds ratio (OR) 0.78, 95% confidence interval (CI): 0.71–0.86]. Conclusion: This is one of the largest studies done on vitiligo in India. The prevalence of vitiligo was found to be 0.89% among hospital attendees. Prevalence of vitiligo was higher among females than in males and prevalence of family history, consanguinity, hypothyroid disorders were higher in vitiligo than among controls.
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Clinico-dermoscopic features and treatment responsiveness in pediatric alopecia – Experience from a tertiary care pediatric dermatology clinic Highly accessed article p. 483
Rahul Mahajan, Manju Daroach, Dipankar De, Sanjeev Handa
Background: Data on clinical and epidemiologic profile on pediatric alopecia is relatively scarce. Aims and Objectives: We aimed to study the clinical, epidemiological, and dermoscopic profile of children presenting with alopecia, and assess the responsiveness to different treatment modalities in a real-life setting. Materials and Methods: This cohort study involved analyzing children presenting with hair loss during the study period. After a detailed history, clinical, and trichoscopic examination, treatment offered to patients and follow-up response to treatment along with relapse of symptoms were noted. Results: Around 119 children were included. Nearly 90% were of acquired etiology. The most common cause of alopecia was alopecia areata (AA) in 85 (71%) patients followed by tinea capitis 9 (7.5%), lichen planopilaris 4 (3.3%), and other less common causes. In patients of AA, dermoscopy showed the presence of black dots in 68% cases, exclamation mark hair in 54% of patients followed by off-white dots, yellow dots, and vellus hair. Patients with an acute course and black dots on dermoscopy responded better to treatment. Relapse was common in patients with early age of onset and longer disease duration. Conclusions: Hair loss is frequently seen in pediatric dermatology clinics. Dermoscopy of pediatric AA shows scarce yellow dots while off-white dots are more frequent; the presence of black dots is a good prognostic indicator.
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Correlation of venous clinical severity score and venous disability score with dermatology life quality index in chronic venous insufficiency p. 489
Subhadeep Mallick, Tanusree Sarkar, Tirthankar Gayen, Biswanath Naskar, Adrija Datta, Somenath Sarkar
Background: Chronic venous insufficiency (CVI) is an underestimated public health problem involving the lower limbs. It exerts a significant impact on patient's quality of life (QoL). The severity of the disease was measured by venous clinical severity score (VCSS) and venous disability score (VDS). Aims: The aim of the study was to evaluate VCSS, VDS, and dermatology life quality index (DLQI) among the patients of CVI and to evaluate the correlation among DLQI with VCSS, VDS, and leg ulcer. Materials and Methods: In this institution-based cross-sectional study, clinically and sonographically confirmed cases of CVI were included. Clinical severity of the disease and disability were assessed by using VCSS and VDS, respectively. QoL was assessed by a validated DLQI questionnaire. Correlation between DLQI with VCSS and VDS was analyzed. The association between DLQI with different characteristics of the ulcer was also evaluated. Results: Mean VCSS, VDS, and DLQI in the study population were 11 ± 4.96, 1.47 ± 0.67, and 6.94 ± 3.87, respectively. Both VCSS and VDS had a strong positive correlation with DLQI. The number of active ulcers, size of the ulcer, and duration of the ulcer had a strong positive correlation, whereas the age of onset of the disease had a negative correlation and duration of the disease had poor correlation with DLQI. Pain (P = 0.03) and edema (P = 0.04) had significant association with VDS. Conclusion: VCSS and VDS are important tools for measuring severity and disability in CVI, respectively. CVI had a strong impact on patients QoL more than it was thought hitherto.
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Clinico-etiological evaluation of chronic leg ulcer in a tertiary care center of eastern India p. 495
Falguni Nag, Gobinda Chatterjee, Arghyaprasun Ghosh, Abhishek De
Background: A chronic leg ulcer (CLU) is a significant public health problem. It has various etiologies. Racial, familial, occupational, and social factors may also have an impact on the prevalence of different causes of leg ulcers. Though there are western data on the epidemiology of leg ulcer, similar data are largely unavailable from our part of the world. Aims: We undertook a study in a tertiary care center in eastern India to determine the clinical and etiological pattern of patients with CLU. Materials and Methods: Hundred consecutive patients presenting with CLU, fulfilling the criteria, were included after informed consent. Patients were subjected to proper history taking, clinical examination, routine blood test, and pus for culture and sensitivity test (where needed) along with Ankle Brachial Index (ABI). Results: Among the 100 patients, venous ulcer (34%) was predominant followed by arterial ulcer (14%), mixed arterial and venous ulcer (11%). History of smoking (56%) and obesity (BMI >25) (32%) were the common risk factors in leg ulcer patients. Fifty nine percent of the total CLU were infected and out of this, 86.4% showed growth of microorganisms.Staphylococcus aureus (39%) was the most commonly isolated organism, followed by Pseudomonas aeruginosa (15%). Eleven (24.44%) clinically diagnosed venous ulcer patients showed significantly lower ABI (<0.9) and were diagnosed as mixed ulcer (a venous ulcer with a peripheral arterial disease). Conclusion: Venous ulcer and mixed ulcer are the most common type of CLU.
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Podoplanin as a predictive marker for identification of high-risk mycosis fungoides patients: An immunohistochemical study p. 500
Amal A El-Ashmawy, Maha M Shamloula, Nashwa N Elfar
Background: Podoplanin, an important protein, has been implicated in various cellular processes, including lymphangiogenesis. Podoplanin is a mucin-type transmembrane glycoprotein that is accepted as a novel marker of lymphatic endothelial cells. Objectives: To study the immunohistochemical expression of podoplanin in the different stages of mycosis fungoides (MF) in comparison to control and to correlate their expression with disease severity and progression. Materials and Methods: The study included 50 patients of MF, clinically diagnosed and assessed by World Health Organization/European Organization for Research And Treatment Of Cancer Consensus and 20 normal persons as control. Skin biopsy specimens were taken from all and examined for expression of podoplanin immunohistochemically. Results: Significant upregulation of podoplanin expression was detected in all studied patients of MF in comparison to control group. Podoplanin expression in malignant lymphocytes and also lymph vessel density showed significant upregulation in the aggressive clinical presentations as well as the highest stages regarding TNMB staging of MF. Conclusions: Evaluation of podoplanin expression may be taken into consideration in the future as a useful tool to identify high-risk MF patients. Furthermore, it may open new therapeutic options for the clinical management of those patients.
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Expression of programmed death-ligand 1 in cutaneous squamous cell carcinoma arising in sun-exposed and nonsun-exposed skin p. 506
Hiroyuki Goto, Kazunari Sugita, Osamu Yamamoto
Background: A recent clinical trial has shown the efficacy of an anti-programmed death-1 (PD-1) antibody against advanced squamous cell carcinoma (SCC). The expression of PD-ligand 1 (PD-L1) in tumor cells correlates with a favorable response to anti-PD-1 therapy in various malignancies. In recent studies, it has been shown that SCC frequently expresses PD-L1. However, there has been no previous study focusing on the difference in PD-L1 expression between SCC in sun-exposed skin and that in nonsun-exposed areas. Aims: The purpose of this study was to investigate the relationship between sun-exposure status and PD-L1 expression in patients with SCC. Materials and Methods: We investigated 80 patients with SCC (40 patients with SCC in sun-exposed skin and 40 patients with SCC in nonsun-exposed skin) by immunohistochemical staining for PD-L1. Fisher's exact test was used for statistical analyses of the differences between the two groups. Results: Patients with SCC in sun-exposed skin showed a significantly higher expression level of PD-L1 in tumor cells than did patients with SCC in nonsun-exposed skin (P = 0.0133). Conclusions: We found that the expression level of PD-L1 in patients with SCC in sun-exposed skin was higher than in patients with SCC in nonsun-exposed skin. Practical data are needed for appropriate applications of new therapeutic options for SCC.
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Truncal varicosities and bilateral port-wine stain with spinal deformities associated with Klippel–Trenaunay–Weber syndrome p. 510
Amr Abdelhamed, Soha Aboeldahab, Mohammed Abu El-Hamd, Hanan Assaf
Klippel–Trenaunay–Weber syndrome (KTWS) is a rare congenital disorder characterized by asymmetric limb hypertrophy, usually of the lower limbs, as well as vascular anomalies and capillary malformations under the skin, termed as port-wine stain. KTWS is prevalent in all parts of the world. It has a high degree of diversity of the associated malformations. In the present case, vascular/lymphatic malformations were evident by the presence of bilateral port-wine stain and lymphangioma. More interestingly, prominent aberrant veins (truncal varicosities) were found in the anterior chest wall, together with the presence of multiple angiolipomatosis. Bone deformities were more than limb hypertrophy and macrodactyly and extended to spinal deformities in the form of scoliotic changes.
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Linear scleroderma and primary sclerosing cholangitis: An undescribed association p. 514
Iñigo Navarro Fernández, Susana Armesto Alonso, Carlos Duran Vian, L Reguero Del Cura, Carmen González-Vela, Marcos González-López
Linear scleroderma (LS) is clinically characterized by the presence of sclerotic areas of skin, which develop in a linear pattern. Primary sclerosing cholangitis is a cholestatic disorder that can lead to end-stage liver disease. We present, for the first time in English literature, the case of a patient suffering from both the diseases. This highlights the fact that, even though LS has conventionally been considered to be a form of localized scleroderma, this does not necessarily imply that it is an exclusively cutaneous disease.
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POEMS syndrome with extensive calciphylaxis: A case report and literature review p. 516
Cheng-Han Liu, Yu-Chen Chen, Tsai-Yun Chen, Hang-Wei Huang, Julia Yu-Yun Lee
POEMS syndrome, a paraneoplastic disorder, caused by plasma cell dyscrasia, is characterized by polyneuropathy, organomegaly, endocrinopathy, M-protein, and skin changes. Calciphylaxis is a vasculopathy of subcutaneous arteries with calcification and skin ulceration, most commonly associated with end-stage renal disease. Calciphylaxis occurring in patients with POEMS syndrome is very rare; only 8 cases have been reported to date. We report the 9th case. A middle-aged woman with chronic inflammatory demyelinating polyneuropathy and monoclonal plasma cell proliferative disorder with IgA gammopathy presented with a 3-month history of multiple painful violaceous plaques with gangrenous necrosis and ulceration on the abdomen and extremities. The skin lesions worsened after surgical debridement and fasciotomy but improved with weekly intravenous bortezomib combined with thalidomide and dexamethasone for POEMS syndrome. The present case illustrates that effective control of the disease activity of POEMS syndrome may improve the surgical outcome of extensive skin necrosis in POEMS syndrome-associated calciphylaxis.
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Restrictive dermopathy – A rare congenital skin disorder p. 519
VR Viraraghavan, Sarita Sanke, Vibhu Mendiratta, Aditi Dewan, Ajay Kumar, Rashi Pangti
Restrictive dermopathy is a rare, autosomal recessive, lethal congenital skin disorder. This congenital genodermatosis could be mistaken for various other similar skin disorders. Diagnosis is a must in the context of genetic counseling for the subsequent pregnancy. We herein report a preterm male neonate with restrictive dermopathy, with additional feature of multiple bone fractures.
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Hand-foot skin reaction with sunitinib: A rare case report with review of literature p. 522
Suchibrata Das, Alok K Roy, Srutee Barman, Aritra Roy
Newer multi-kinase inhibitors (MKI) like sunitinib have changed the therapy of patients of renal cell carcinoma, hepatocellular carcinoma, and gastrointestinal stromal tumor. The use of sunitinib also led to cutaneous toxicity, known as hand-foot skin reaction (HFSR). We report a case of hand-foot skin reaction (HFSR) in an Indian patient being treated with sunitinib. Respective literature on this disorder is also reviewed.
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Superficial CD34-positive fibroblastic tumor: Report of an extremely rare entity p. 526
Ebru Zemheri, Ayşe Serap Karadag, İsmail Yılmaz
Superficial CD34-positive fibroblastic tumor (SCPFT), a newly described neoplasm is a rare mesenchymal neoplasm of intermediate malignancy. A 63-year-old man presented with a painless, slow-growing, skin-colored nodule of 8 × 4 mm in diameter on the right side of the neck. It was completely resected. Histologically, a tumor located in the subcutis with the minimally infiltrative pattern was detected. The tumor was composed of variably enlarged bizarre and pleomorphic spindle to polygonal cells. Tumor cells were stained strongly diffuse positive with CD34 and weak positive with keratin, negative with STAT6, FLI-1, ERG, S100, desmin, and smooth muscle actin. The fluorescence in-situ hybridization (FISH) analysis was negative for
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An unusual etiology for an acral psoriasiform dermatosis p. 530
Dimitra Koumaki, Vasiliki Koumaki, Vrettos Haniotis, Nikolaos Galanakis, Alexandros Katoulis, Sotirios Boumpoucheropoulos, George Evangelou, Maria Stefanidou, Sabine E Krueger-Krasagakis, Konstantinos Krasagakis
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A large multinodular mass on the back p. 533
Ryan Gillihan, Diana Braswell, Kiran Motaparthi
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“Walter Cooper Dendy (1794-1871) and the first book on pediatric dermatology” - Reader's question p. 536
Sarvin Sanaie, Javad Ghazi Sha'rbaf, Reza Mohammadinasab
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Author's Reply p. 537
Amiya K Mukhopadhyay
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Michelin tire baby syndrome with dermal sclerosis: A novel association p. 538
Komal Agarwal, Indrashis Podder, Abhisekh Bandyopadhyay, Satyendra N Chowdhury
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Complete phenotypic expression of hypohidrotic ectodermal dysplasia in a female patient p. 540
Sumit Sen, Somnath Das, Surabhi Sharma
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Flexural circumscribed juvenile pityriasis rubra pilaris: An unusual presentation p. 542
Md Zeeshan, Abhijeet K Jha, Noorussaba Arfeen, RK P Chaudhary
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A case of linear IgA bullous dermatosis successfully treated with omalizumab p. 543
Nayankumar H Patel, Jignaben K Padhiyar, Tejas D Patel, Nishi S Trivedi, Vidhi S Chandibhamar, Ranjan Raval
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A case of laryngeal mucous membrane pemphigoid possibly triggered by radiotherapy p. 546
Giovanni M Palleschi, Michela Di Pietro, Marta Barzacchi, Luca G Locatello, Maria B Ninu
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A case of localized bullous pemphigoid associated with an arteriovenous fistula p. 547
Ju Wang Jang, Chang Hwa Song, You Jin Jung, Tae Lim Kim, Hyun-Min Seo, Young Gyun Kim, Joung Soo Kim
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Rowell's syndrome with lupus hepatitis: A case report from China p. 549
Ming Zhang, Jiejie Lu, Bei Zhang, Zhen Li, Weiwei Wu
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Oral allergy syndrome associated with eggplant particles manifesting as lichenoid reaction– A sequential diagnostic workout and long-term follow up p. 550
Santosh Palla, Abu Raghavan Srinivasan, Krishnan Subramanian, Vandana Shekar
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Disseminated histoplasmosis with oral and cutaneous manifestations in an immunocompetent patient p. 552
Sumit Sen, Arpita Hati, Subhadeep Mallick, Somnath Das
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Palatal ulceration of long evolution p. 554
Tamara Gracia-Cazaña, Ana L Morales-Moya, Lucia Prieto-Torres, Isabel Sanjoaquin-Conde
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Spectrum of dermoscopic pattern in a patient with angiolymphoid hyperplasia with tissue eosinophilia p. 556
Mayuri Kalantri, Uday Khopkar
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Dermatofibroma botryoides on nipple: A report p. 558
Sara Behbahani, Katrice M Karanfilian, Marcus L Elias, Shreya Patel, William C Lambert
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Solitary eccrine syringofibroadenoma of the dorsum of hand p. 560
Peimei Zhou, Fan Li, Hongjie Liu, Lin Wang
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Reviewers, 2020 p. 562
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Author Index, 2020 p. 563
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Title Index, 2020 p. 573
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