Low-Fat Plexiform Spindle Cell Lipoma with Prominent Myxoid
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Brief Clinical Studies The Journal of Craniofacial Surgery Volume 28, Number 2, March 2017 In conclusion, we suggest that even the large ACC defects could 20. Ross DA, Laurie SWS, Coombs CJ, et al. Aplasia cutis congenita: failed be managed conservatively with proper care. With the proper conservative treatment. Plast Reconstr Surg 1995;95:124–129 dressing, infection control with no serious complications and 21. Kim CS, Tatum SA, Rodziewicz G. Scalp aplasia cutis congenita complete healing of the defect could be achieved. We also suggest presenting with sagittal sinus hemorrhage. Arch Otolaryngol Head that topical application of bFGF which has positive effects over Neck Surg 2001;127:71–74 wound healing decreases the time of hospitalization with superior 22. Tsuboi R, Rifkin DB. Recombinant basic fibroblast growth factor stimulates wound healing in healing-impaired db/db mice. J Exp wound healing compared to treatment without such application and Med 1990;172:245–251 should be used in order to provide more data for a better treatment 23. Akita S, Akino K, Imaizumi T, et al. The quality of pediatric burn scars modality of the condition. Individual assessment of the patients with is improved by early administration of basic fibroblast growth factor. J an experienced team of good communication including pedia- Burn Care Res 2006;27:333–338 tricians, plastic surgeons, and neurosurgeons in order to provide 24. Hayashida K, Akita S. Quality of pediatric second-degree burn wound the best treatment should be made. Recognizing the complications scars following the application of basic fibroblast growth factor: results due to the mortality associated and knowledge of prevention is of a randomized, controlled pilot study. Ostomy Wound Manage essential for an ideal management. 2012;58:32–36 25. Akita S, Akino K, Imaizumi T, et al. Basic fibroblast growth factor accelerates and improves second-degree burn wound healing. Wound Repair Regen 2008;16:635–641 REFERENCES 26. Maeda T, Oyama A, Funayama E, et al. Successful conservative 1. Browning JA. Aplasia cutis congenital: approach to evaluation and management of a large congenital scalp and skull defect. J management. Dermatol Ther 2013;26:439–444 Craniofac Surg 2015;26:275–277 2. Komuro Y, Yanai A, Seno H, et al. Surgical treatment of aplasia cutis 27. Jung WK, Koo HC, Kim KW, et al. Antibacterial activity and congenita of the scalp associated with bilateral coronal synostosis. mechanism of action of the silver ion in Staphylococcus aureus J Craniofac Surg 2002;13:513–519 and Escherichia coli. Appl Environ Microbiol 2008;74:2171–2178 3. Bernbeck B, Schwabe J, Groninger A, et al. Aplasia cutis congenita of the scalp: how much therapy is necessary in large defects? Acta Paediatr 2005;94:758–760 4. Demmel U. Clinical aspects of congenital skin defects. I. Congenital skin defects on the head of the newborn. Eur J Pediatr 1975;121:21–50 5. Stephan MJ, Smith DW, Ponzi JW, et al. Origin of scalp vertex aplasia cutis. J Pediatr 1982;101:850–853 Low-Fat Plexiform Spindle Cell 6. Winston KR, Ketch LL. Aplasia cutis congenita of the scalp, composite type: the criticality and inseparability of neurosurgical and plastic Lipoma With Prominent Myxoid surgical management. Pediatr Neurosurg 2016;51:111–120 7. Fro¨jd V, Maltese G, Ko¨lby L, et al. Conservative healing of an 11 Â 9- Stroma: An Unusual Oral cm aplasia cutis congenita of the scalp with bone defect. J Neurol Surg Rep 2014;75:220–223 Presentation and 8. Rocha D, Rodrigues J, Marques JS, et al. Aplasia cutis congenita: a conservative approach of a case with large, extensive skin, and Immunohistochemical Analysis underlying skull defect. Clin Case Rep 2015;3:841–844 Ã 9. Basterzi Y, Bagdatoglu C, Sari A, et al. Aplasia cutis congenita of Jessica Luana dos Santos, DDS, MSc, the scalp and calvarium: conservative wound management with Eduardo Akira Ocamoto, DDS,y novel wound dressing materials. J Craniofac Surg 2007;18: Luciana Yamamoto Almeida, DDS, PhD,z 427–429 Lucas Ribeiro Teixeira, DDS,§ Alfredo Ribeiro-Silva, MD, PhD,Ã 10. Dutra LB, Pereira M, Kreniski TM, et al. Aplasia cutis congenita: § management of a large skull defect with acrania. J Craniofac Surg and Jorge Esquiche Leo´n, DDS, PhD 2009;20:1288–1292 11. Gospodarowicz D. Biological activities of fibroblast growth factors. Ann Abstract: Spindle cell lipoma (SCL) and pleomorphic lipoma N Y Acad Sci 1991;638:1–8 constitute a spectrum of lipomatous lesions with distinctive clin- 12. Hayek A, Culler FL, Beattie GM, et al. An in vivo model for study of the icopathological features. Multiple variants of SCL have been angiogenic effects of basic fibroblast growth factor. Biochem Biophys Res Commun 1987;147:876–880 reported including fibrous, plexiform, vascular, pseudoangioma- 13. Puvabanditsin S, February M, Garrow E, et al. Our experience with a tous, low-fat/fat-free, and myxoid changes. This paper describes an severe case of aplasia cutis congenita with a large skull defect. Int J Dermatol 2016;55:1151–1153 From the ÃDepartment of Pathology and Forensic Medicine, Ribeira˜o Preto 14. Rhee ST, Colville C, Buchman SR, et al. Complete osseous regeneration Medical School (FMRP/USP), University of Sa˜o Paulo (USP), Ribeira˜o of a large skull defect in a patient with cutis aplasia: a conservative Preto; yPublic Health Service, Jaboticabal; zHematology Division, approach. J Craniofac Surg 2002;13:497–500 Department of Clinical Medicine, Ribeira˜o Preto Medical School 15. de Oliveira RS, Juca´ CEB, Lins-Neto AL, et al. Aplasia cutis congenita (FMRP/USP), University of Sa˜o Paulo (USP), Ribeira˜o Preto; and §Oral of the scalp: is there a better treatment strategy? Childs Nerv Syst Pathology, Department of Stomatology, Public Oral Health, and Foren- 2006;22:1072–1079 sic Dentistry, University of Sa˜o Paulo, School of Dentistry of Ribeira˜o 16. Ingall NW. Congenital defects of the scalp; studies in pathology of Preto (FORP/USP), University of Sa˜o Paulo, Ribeira˜o Preto, Brazil. development. Am J Obstet Gynecol 1933;25:861–873 Received August 16, 2016. 17. Rudolph RI, Schwartz W, Leyden II. Bitemporal aplasia cutis congenita. Accepted for publication October 7, 2016. Occurrence with other cutaneous abnormalities. Arch Dermatol Address correspondence and reprint requests to Jessica Luana dos Santos, 1974;110:615–618 DDS, MSc, Universidade de Sa˜o Paulo, Avenida Bandeirantes, S/N, 18. Burkhead A, Poindexter G, Morrell DS. A case of extensive aplasia cutis 14040-904, Ribeira˜o Preto, Brazil; congenita with underlying skull defect and central nervous system E-mail: [email protected] malformation: discussion of large skin defects, complications, treatment The authors report no conflicts of interest. and outcome. J Perinatol 2009;29:582–584 Copyright # 2016 by Mutaz B. Habal, MD 19. Frieden IJ. Aplasia cutis congenita: a clinical review and proposal for ISSN: 1049-2275 classification. J Am Acad Dermatol 1986;14:646–660 DOI: 10.1097/SCS.0000000000003348 e158 # 2017 Mutaz B. Habal, MD Copyright © 2017 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited. The Journal of Craniofacial Surgery Volume 28, Number 2, March 2017 Brief Clinical Studies unusual patient with a 1-cm submucosal nodular lesion excised from the buccal mucosa of a 55-year-old woman with classic histopathological and immunohistochemical features of ‘‘low- fat’’ plexiform SCL with prominent myxoid stroma, which initially suggested a soft-tissue myxomatous lesion other than SCL. The current lesion exhibited microscopically few adipocytes supported by network-like myxoid proliferations with retraction artifacts from the surrounding stromal connective tissue. Immunohistochemistry was positive for vimentin, CD34, CD10, and S100, the latter only on adipocytes. The Ki-67 was <1%. Pan-cytokeratin (AE1/AE3), desmin, alpha-SMA, EMA, bcl-2, p53, and remarkably retinoblas- toma protein (pRb) were negative. ‘‘Low-fat’’ plexiform SCL bear no significant prognostic significance, but this lesion may challenge the diagnosis even experienced pathologists. Key Words: Immunohistochemistry, low-fat lipoma, oral cavity, plexiform variant, spindle cell lipoma FIGURE 2. (A) Microscopic view showing a lobulated mass, containing net-like pindle-cell lipoma (SCL) is a distinct histological variant of proliferations (H&E staining, Â2.5). (B) Microscopic view showing a myxoid stroma and scarce adipocytes. Inset shows cellular details of mast cells (H&E S lipoma which is derived from prelipoblastic mesenchymal staining, Â20). (C) Microscopic view exhibiting network-like myxoid 1 cells. Spindle cell lipoma is a circumscribed subcutaneous lesion, proliferations presenting retraction artifacts (Ã) from the surrounding stromal typically occurring on the neck and upper back of male individuals, fibrosis (arrow) (H&E staining, Â10). (D) Presence of mast cells by toluidine blue and it is composed of an admixture of well-aligned spindle cells, staining, Â40. Inset shows cellular details of mast cells. mature adipocytes, dense collagen bundles, myxoid interstitial matrix, mast cells, and blood vessels.2,3 Mitoses are extremely rare. While the presence of myxoid matrix is not uncommon, the Following surgical excision, the specimen was fixed in 10% relative proportions of spindle cells and adipocytic elements vary buffered formalin, routinely processed, and embedded in paraffin from SCL patient to patient and it can be designated as ‘‘low-fat’’ or wax. The 3-mm thick sections were stained with hematoxylin and ‘‘free-fat’’ if has a very small amount of adipocytes (<5%) or is eosin. Additional sections were cut for immunohistochemical pro- absent, respectively.4–6 Moreover, 6 patients with dermal SCL have cedures. Immunohistochemical studies were performed using the been reported, often affecting middle-aged females, which micro- following antibodies: Pan-cytokeratin (AE1/AE3), EMA, vimentin, scopically exhibited a moderately circumscribed plexiform pattern, S100, alpha-SMA, desmin, CD10, CD30, CD34, bcl-2, p53, Ki-67, with net-like proliferations and retraction artifacts against the and retinoblastoma protein (pRb).