International Journal of Current Research and Review
ISSN: 2231-2196 (Print)ISSN: 0975-5241 (Online)
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IJCRR - 4(13), July, 2012

Pages: 74-78

Date of Publication: 18-Jul-2012


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SYRINGOCYSTADENOMA PAPILLIFERUM IN NEVUS SEBACEOUS AT UNUSUAL SITE

Author: Dayananda S. Biligi, Deepa S. Anand, Yogeeshwarappa E.N., D.C. Prabhu

Category: Healthcare

Abstract:Nevus sebaceous is a benign, congenital hamartoma of the skin, with a predilection for the scalp and less commonly occurs on the face, around the ears, neck or on the trunk. In late childhood and early adulthood, there is a significant risk of developing secondary tumors, the most common of which are syringocystadenoma papilliferum and basal cell carcinoma. A case of syringocystadenoma papilliferum associated with nevus sebaceous in a 26 year old female patient in the nipple areolar region. A rare site of occurance of syringocystadenoma papilliferum arising in nevus sebaceous is described. To the best of our knowledge, this appears to be the first case reported in the nipple areolar complex.

Keywords: Nevus sebaceous, Syringocystadenoma papilliferum, nipple areola

Full Text:

INTRODUCTION Nevus sebaceous is a congenital skin lesion commonly seen by pediatricians, dermatologists, and plastic surgeons.[1] It has a predilection for the scalp and less commonly occurs on the face, around the ears, neck, or on the trunk. The most common benign and malignant neoplasms arising in this disorder include trichilemmoma, syringocystadenoma papilliferum, keratoacanthoma, apocrine cystadenoma and basal cell carcinoma. Syringocystadenoma papilliferum is a benign adnexal tumor of the skin,[2] which can occur either de novo or on some organoid nevus such as nevus sebaceous. This tumor presents generally over the scalp and forehead. The clinical presentation of nevus sebaceous and syringocystadenoma papilleferum varies widely but the histological appearance is uniform and characteristic, forming the basis of diagnosis.[3] CASE REPORT A 26 year old female patient presented with history of pigmented papules in bilateral nipple and areola region, since birth. Lesion formed an irregular nodule during puberty and progressed to a verrucous plaque, on right side, since one month. On examination, her general condition was good. Local examination showed a pigmented, non tender, verrucous nodule in right nipple areolar complex, measuring 3.5x2.5x2cm. (Fig. 1) Excision biopsy of the lesion was done on the right side and sent for histopathological examination. Grossly, the specimen was a single, irregular, warty mass measuring 3.5x2.5x2cm. Cut surface was grey white with tiny cysts. Microscopy showed dilated, keratin filled infundibulum surrounded by numerous mature and immature sebaceous glands. (Fig.2) There were cystic invaginations from the surface, lined by stratified sqaumous epithelium transforming into a double layered epithelium. (Fig.3) Outer layer of epithelium was lined by cuboidal cells and the inner layer by columnar cells, a few of which showed decapitation secretion. Papillary projections into the cystic invagination were evident. Dense mononuclear inflammatory infiltrate, predominantly plasma cells were seen in the papillary core and around the cyst wall. (Fig.4) A good number of apocrine cysts were also seen. These classical histological features favored a diagnosis of Syringocystadenoma Papilliferum associated with Nevus Sebaceous. DISCUSSION Nevus sebaceous is a congenital hamartoma of the skin, first described by Jadassohn. It is defined by hyperplasia of the epidermis, hair follicles, sebaceous and apocrine glands. Nevus sebaceous often presents at birth, is quiescent in childhood and grows during puberty, probably due to increased levels of serum androgens present in neonatal period and at puberty.[4] It is sporadic and occurs with equal frequency in males and females of all races.[1] In a study by Rosen et al,[1] out of 631 cases, most common locations of nevus sebaceous were scalp, face or neck [Scalp (62.8%), Forehead (10.3%), Cheek ( 9.8%), Neck (3.7%), Eyebrow (1.7%), Back (1.5%), Eyelid (0.8%), Chest (0.5%), Abdomen (0.2%) and Breast ( 0.2%)]. In late childhood and early adulthood, there is a significant risk of developing secondary tumors, mainly syringocystadenoma papilliferum and basal cell carcinoma.[4] Syringocystadenoma papilliferum, formerly known as nevus syringocystadenomatous papilliferus, is a proliferating tumor with mostly apocrine differentiation but eccrine differentiation can also be seen. The histogenesis of this rare neoplasm is still unclear. It is theorized that syringocystadenoma papilliferum arises from the pluripotent cells of apocrine lineage.[5] In about 75% of cases, syringocystadenoma papilliferum arises within a preexistent naevus sebaceous of Jadassohn according to a large series of 100 cases by Helwig and Hackney.[6] In their series,[6] 55 lesions were on the scalp, 11 on the forehead and temple, 5 on the face and 3 on the upper lip, but none was reported on the nipple areolar complex. Less frequently reported sites are chest, abdomen, arm, eyelid and thigh.[2][7][8] Clinically, the differential diagnoses of these lesions include linear epidermal nevus, nevus comedonicus, basaloid follicular hamartoma, cylindroma and eccrine nevus.[9] However, in our case, histopathological features were unmistakable and had no differential diagnosis. The only treatment for syringocystadenoma papilliferum is excisional biopsy, which also confirms the diagnosis. CO2 laser excision of syringocystadenoma papilliferum of the head and neck is a clinical treatment option in anatomic areas unfavorable to excision and grafting.[10] CONCLUSION Syringocystadenoma papilliferum in nevus sebaceous is well described, but a relatively rare tumor, diagnosed only by histopathological examination. We report a case of syringocystadenoma papilliferum associated with nevus sebaceous in the right nipple areolar region, an exceptional site of presentation.

References:

1. Rosen H, Schmidt B, Lam H.P, Meara J.G, Labow B.I. Management of Nevus Sebaceous and the Risk of Basal Cell Carcinoma: An 18-Year Review. Pediatric Dermatology Vol. 26 No. 6 676–681, 2009

2. Rao VA, Kamath GG, Kumar A. An unusual case of syringocystadenoma papilliferum of the eyelid. Indian J Ophthalmol 1996;44:168- 9

3. Karg E, Korom I, Varga E et al. Congenital syringocystadenoma papilliferum. Pediatr Dermatol 2008;25:132–133

4. Hamilton K.S, Johnson S, Smoller B.R. The Role of Androgen Receptors in the Clinical Course of Nevus Sebaceus of Jadassohn. Mod Pathol 2001;14(6):539–542

5. Stokes JH. A clinico-pathologic study of an unusual cutaneous neoplasm combining nevus syringadenomatosus papilliferus and a granuloma. J Cutan Dis. 1917; 35: 411-9.

6. Helwig EB, Hackney VC. Syringoadenoma papilliferum. Arch Dermatol 1995; 71: 361- 372.

7. Premalatha S, Rao NR, Yesudian P, Razack A, Zahra A. Syringocystadenoma Papilliferum in an unusual location. Int J Dermaatol 1985;24:520-1

8. Malhotra P, Singh A, Ramesh V. Syringocystadenoma papilliferum on the thigh: An unusual location. Indian J Dermatol Veneral Leprol 2009;75:170-2

9. Philipone E, Chen S. Unique case: syringocystadenoma papilliferum associated with an eccrine nevus. Am J Dermatopathol. 2009; 31(8): 806-7.

10. Fz Elfatoiki, K Khadir, A Ouakadi, S Azzouzi, N Bahechar, H Benchikhi. Syringocystadenoma papilliferum:Unusual location.Dermatology Online Journal 2011; 17(6):7

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A study by Dorothy Ebere Adimora et al. entitled \"Remediation for Effects of Domestic Violence on Psychological well-being, Depression and Suicide among Women During COVID-19 Pandemic: A Cross-cultural Study of Nigeria and Spain\" is awarded Best Article of Vol 14 issue 23
A study by Muhas C. et al. entitled \"Study on Knowledge & Awareness About Pharmacovigilance Among Pharmacists in South India\" is awarded Best article for Vol 14 issue 22
A study by Saurabh Suvidha entitled \"A Case of Mucoid Degeneration of Uterine Fibroid with Hydrosalphinx and Ovarian Cyst\" is awarded Best article of Vol 14 issue 21
A study by Alice Alice entitled \"Strengthening of Human Milk Banking across South Asian Countries: A Next Step Forward\" is awarded Best article of Vol 14 issue 20
A study by Sathyanarayanan AR et al. entitled \"The on-task Attention of Individuals with Autism Spectrum Disorder-An Eye Tracker Study Using Auticare\" is awarded Best article of Vol 14 issue 19
A study by Gupta P. et al. entitled \"A Short Review on \"A Novel Approach in Fast Dissolving Film & their Evaluation Studies\" is awarded Best Article of Vol 14 issue 18.
A study by Shafaque M. et al. entitled \"A Case-Control Study Performed in Karachi on Inflammatory Markers by Ciprofloxacin and CoAmoxicillin in Patients with Chronic Suppurative Otitis Media\" is awarded Best Article of Vol 14 issue 17
A study by Ali Nawaz et al. entitled \"A Comparative Study of Tubeless versus Standard Percutaneous Nephrolithotomy (PCNL) \? A Randomized Controlled Study\" is awarded Best Article for Vol 14 issue 16.
A study by Singh R. et al. entitled \"A Prospective Study to Find the Association of Astigmatism in Patients of Vernal Keratoconjunctivitis (VKC) in a Tertiary Health Care Centre in India (Vindhya Region MP)\" is awarded Best Article for Vol 14 issue 15
A Study by Humaira Tahir et al. entitled "Comparison of First Analgesic Demand after Major Surgeries of Obstetrics and Gynecology between Pre-Emptive Versus Intra-Operative Groups by Using Intravenous Paracetamol: A Cross-Sectional Study" is awarded Best Article for Vol 14 issue 14
A Study by Monica K. entitled "Risk Predictors for Lymphoma Development in Sjogren Syndrome - A Systematic Review" is awarded Best Article for Vol 14 issue 13
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A Study by Vinita S & Ayushi S entitled "Role of Colour Doppler and Transvaginal Sonography for diagnosis of endometrial pathology in women presenting with Abnormal Uterine Bleeding" is awarded Best Article for Vol 14 issue 08
A Study by Prabhu A et al. entitled "Awareness of Common Eye Conditions among the ASHA (Accredited Social Health Activist) Workers in the Rural Communities of Udupi District- A Pilot Study" is awarded Best Article for Vol 14 issue 07
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A Study by Ajai KG & Athira KN entitled "Patients’ Gratification Towards Service Delivery Among Government Hospitals with Particular Orientation Towards Primary Health Centres" is awarded Best Article for Vol 14 issue 04
A Study by Mbungu Mulaila AP et al. entitled "Ovarian Pregnancy in Kindu City, D.R. Congo - A Case Report" is awarded Best Article for Vol 14 issue 03
A Study by Maryam MJ et al. entitled "Evaluation Serum Chemerin and Visfatin Levels with Rheumatoid Arthritis: Possible Diagnostic Biomarkers" is awarded Best Article for Vol 14 issue 02
A Study by Shanthan KR et al. entitled "Comparison of Ultrasound Guided Versus Nerve Stimulator Guided Technique of Supraclavicular Brachial Plexus Block in Patients Undergoing Upper Limb Surgeries" is awarded Best Article for Vol 14 issue 01
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A Study by Manoj KP et al. entitled "A Randomized Comparative Clinical Trial to Know the Efficacy of Ultrasound-Guided Transversus Abdominis Plane Block Against Multimodal Analgesia for Postoperative Analgesia Following Caesarean Section" is awarded Best Article Award of Vol 13 issue 23
A Study by Karimova II et al. entitled "Changes in the Activity of Intestinal Carbohydrases in Alloxan-Induced Diabetic Rats and Their Correction with Prenalon" is awarded Best Article of Vol 13 issue 22
A Study by Ashish B Roge et al. entitled "Development, Validation of RP-HPLC Method and GC MS Analysis of Desloratadine HCL and It’s Degradation Products" is awarded Best Article of Vol 13 issue 21
A Study by Isha Gaurav et al. entitled "Association of ABO Blood Group with Oral Cancer and Precancer – A Case-control Study" is awarded Best Article for Vol 13 issue 20
A Study by Amr Y. Zakaria et al. entitled "Single Nucleotide Polymorphisms of ATP-Binding Cassette Gene(ABCC3 rs4793665) affect High Dose Methotrexate-Induced Nephrotoxicity in Children with Osteosarcoma" is awarded Best Article for Vol 13 issue 19
A Study by Kholis Ernawati et al. entitled "The Utilization of Mobile-Based Information Technology in the Management of Dengue Fever in the Community Year 2019-2020: Systematic Review" is awarded Best Article for Vol 13 issue 18
A Study by Bhat Asifa et al. entitled "Efficacy of Modified Carbapenem Inactivation Method for Carbapenemase Detection and Comparative Evaluation with Polymerase Chain Reaction for the Identification of Carbapenemase Producing Klebsiella pneumonia Isolates" is awarded Best Article for Vol 13 issue 17
A Study by Gupta R. et al. entitled "A Clinical Study of Paediatric Tracheostomy: Our Experience in a Tertiary Care Hospital in North India" is awarded Best Article for Vol 13 issue 16
A Study by Chandran Anand et al. entitled "A Prospective Study on Assessment of Quality of Life of Patients Receiving Sorafenib for Hepatocellular Carcinoma" is awarded Best article for Vol 13 issue 15
A Study by Rosa PS et al. entitled "Emotional State Due to the Covid – 19 Pandemic in People Residing in a Vulnerable Area in North Lima" is awarded Best Article for Vol 13 issue 14
A Study by Suvarna Sunder J et al. entitled "Endodontic Revascularization of Necrotic Permanent Anterior Tooth with Platelet Rich Fibrin, Platelet Rich Plasma, and Blood Clot - A Comparative Study" is awarded Best Article for Vol 13 issue 13
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A Study by Kumar S. et al. entitled "A Study on Clinical Spectrum, Laboratory Profile, Complications and Outcome of Pediatric Scrub Typhus Patients Admitted to an Intensive Care Unit from a Tertiary Care Hospital from Eastern India" is awarded Best Article for Vol 13 issue 09
A Study by Mardhiah Kamaruddin et al. entitled "The Pattern of Creatinine Clearance in Gestational and Chronic Hypertension Women from the Third Trimester to 12 Weeks Postpartum" is awarded Best Article for Vol 13 issue 08
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A Study by Anu et al. entitled "Effectiveness of Cytological Scoring Systems for Evaluation of Breast Lesion Cytology with its Histopathological Correlation" is awarded Best Article of Vol 13 issue 04
A Study by Sharipov R. Kh. et al. entitled "Interaction of Correction of Lipid Peroxidation Disorders with Oxibral" is awarded Best Article of Vol 13 issue 03
A Study by Tarek Elwakil et al. entitled "Led Light Photobiomodulation Effect on Wound Healing Combined with Phenytoin in Mice Model" is awarded Best Article of Vol 13 issue 02
A Study by Mohita Ray et al. entitled "Accuracy of Intra-Operative Frozen Section Consultation of Gastrointestinal Biopsy Samples in Correlation with the Final Histopathological Diagnosis" is awarded Best Article for Vol 13 issue 01
A Study by Badritdinova MN et al. entitled "Peculiarities of a Pain in Patients with Ischemic Heart Disease in the Presence of Individual Combines of the Metabolic Syndrome" is awarded Best Article for Vol 12 issue 24
A Study by Sindhu Priya E S et al. entitled "Neuroprotective activity of Pyrazolone Derivatives Against Paraquat-induced Oxidative Stress and Locomotor Impairment in Drosophila melanogaster" is awarded Best Article for Vol 12 issue 23
A Study by Habiba Suhail et al. entitled "Effect of Majoon Murmakki in Dysmenorrhoea (Usre Tams): A Standard Controlled Clinical Study" is awarded Best Article for Vol 12 issue 22
A Study by Ghaffar UB et al. entitled "Correlation between Height and Foot Length in Saudi Population in Majmaah, Saudi Arabia" is awarded Best Article for Vol 12 issue 21
A Study by Siti Sarah Binti Maidin entitled "Sleep Well: Mobile Application to Address Sleeping Problems" is awarded Best Article for Vol 12 issue 20
A Study by Avijit Singh"Comparison of Post Operative Clinical Outcomes Between “Made in India” TTK Chitra Mechanical Heart Valve Versus St Jude Mechanical Heart Valve in Valve Replacement Surgery" is awarded Best Article for Vol 12 issue 19
A Study by Sonali Banerjee and Mary Mathews N. entitled "Exploring Quality of Life and Perceived Experiences Among Couples Undergoing Fertility Treatment in Western India: A Mixed Methodology" is awarded Best Article for Vol 12 issue 18
A Study by Jabbar Desai et al. entitled "Prevalence of Obstructive Airway Disease in Patients with Ischemic Heart Disease and Hypertension" is awarded Best Article for Vol 12 issue 17
A Study by Juna Byun et al. entitled "Study on Difference in Coronavirus-19 Related Anxiety between Face-to-face and Non-face-to-face Classes among University Students in South Korea" is awarded Best Article for Vol 12 issue 16
A Study by Sudha Ramachandra & Vinay Chavan entitled "Enhanced-Hybrid-Age Layered Population Structure (E-Hybrid-ALPS): A Genetic Algorithm with Adaptive Crossover for Molecular Docking Studies of Drug Discovery Process" is awarded Best article for Vol 12 issue 15
A Study by Varsha M. Shindhe et al. entitled "A Study on Effect of Smokeless Tobacco on Pulmonary Function Tests in Class IV Workers of USM-KLE (Universiti Sains Malaysia-Karnataka Lingayat Education Society) International Medical Programme, Belagavi" is awarded Best article of Vol 12 issue 14, July 2020
A study by Amruta Choudhary et al. entitled "Family Planning Knowledge, Attitude and Practice Among Women of Reproductive Age from Rural Area of Central India" is awarded Best Article for special issue "Modern Therapeutics Applications"
A study by Raunak Das entitled "Study of Cardiovascular Dysfunctions in Interstitial Lung Diseas epatients by Correlating the Levels of Serum NT PRO BNP and Microalbuminuria (Biomarkers of Cardiovascular Dysfunction) with Echocardiographic, Bronchoscopic and HighResolution Computed Tomography Findings of These ILD Patients" is awarded Best Article of Vol 12 issue 13 
A Study by Kannamani Ramasamy et al. entitled "COVID-19 Situation at Chennai City – Forecasting for the Better Pandemic Management" is awarded best article for  Vol 12 issue 12
A Study by Muhammet Lutfi SELCUK and Fatma entitled "Distinction of Gray and White Matter for Some Histological Staining Methods in New Zealand Rabbit's Brain" is awarded best article for  Vol 12 issue 11
A Study by Anamul Haq et al. entitled "Etiology of Abnormal Uterine Bleeding in Adolescents – Emphasis Upon Polycystic Ovarian Syndrome" is awarded best article for  Vol 12 issue 10
A Study by entitled "Estimation of Reference Interval of Serum Progesterone During Three Trimesters of Normal Pregnancy in a Tertiary Care Hospital of Kolkata" is awarded best article for  Vol 12 issue 09
A Study by Ilona Gracie De Souza & Pavan Kumar G. entitled "Effect of Releasing Myofascial Chain in Patients with Patellofemoral Pain Syndrome - A Randomized Clinical Trial" is awarded best article for  Vol 12 issue 08
A Study by Virendra Atam et. al. entitled "Clinical Profile and Short - Term Mortality Predictors in Acute Stroke with Emphasis on Stress Hyperglycemia and THRIVE Score : An Observational Study" is awarded best article for  Vol 12 issue 07
A Study by K. Krupashree et. al. entitled "Protective Effects of Picrorhizakurroa Against Fumonisin B1 Induced Hepatotoxicity in Mice" is awarded best article for issue Vol 10 issue 20
A study by Mithun K.P. et al "Larvicidal Activity of Crude Solanum Nigrum Leaf and Berries Extract Against Dengue Vector-Aedesaegypti" is awarded Best Article for Vol 10 issue 14 of IJCRR
A study by Asha Menon "Women in Child Care and Early Education: Truly Nontraditional Work" is awarded Best Article for Vol 10 issue 13
A study by Deep J. M. "Prevalence of Molar-Incisor Hypomineralization in 7-13 Years Old Children of Biratnagar, Nepal: A Cross Sectional Study" is awarded Best Article for Vol 10 issue 11 of IJCRR
A review by Chitra et al to analyse relation between Obesity and Type 2 diabetes is awarded 'Best Article' for Vol 10 issue 10 by IJCRR. 
A study by Karanpreet et al "Pregnancy Induced Hypertension: A Study on Its Multisystem Involvement" is given Best Paper Award for Vol 10 issue 09

List of Awardees

A Study by Ese Anibor et al. "Evaluation of Temporomandibular Joint Disorders Among Delta State University Students in Abraka, Nigeria" from Vol 13 issue 16 received Emerging Researcher Award


A Study by Alkhansa Mahmoud et al. entitled "mRNA Expression of Somatostatin Receptors (1-5) in MCF7 and MDA-MB231 Breast Cancer Cells" from Vol 13 issue 06 received Emerging Researcher Award


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