oc000064 10.3205/oc000064 urn:nbn:de:0183-oc0000647 Case Report Karaci Karaci Selman S MD

Department of Plastic and Reconstructive Surgery, Kanuni Eĝitim ve Araştirma Hastanesi, Kasüstül, Kaşüstü, Trabzon, Turkey, Phone: +90 536 4177518, Fax: +90 462 3447777Department of Plastic and Reconstructive Surgery, Kanuni Eĝitim ve Araştirma Hastanesi, Kasüstü, Kaşüstü, Trabzon, Turkey

selmankaraci@gmail.com author Livaoğlu Livaoğlu Ayten A

Department of Pathology, Kanuni Eğitim ve Araştırma Hastanesi, Kaşüstü, Trabzon, Turkey

author Köse Köse Rüştü R

Department of Plastic Surgery, Recep Tayyip Erdoğan University School of Medicine, Merkez, Rize, Turkey

author Kömür Kömür Barış B

Department of Ophthalmology, Kanuni Eğitim ve Araştırma Hastanesi, Kaşüstü, Trabzon, Turkey

author German Medical Science GMS Publishing House

Düsseldorf

610 20170519 engl This is an Open Access article distributed under the terms of the Creative Commons Attribution 4.0 License. Dieser Artikel ist ein Open-Access-Artikel und steht unter den Lizenzbedingungen der Creative Commons Attribution 4.0 License (Namensnennung). 2193-1496 7 GMS Ophthalmology Cases GMS Ophthalmol Cases 13 A 74-year-old man was examined for a mass in the left upper eyelid. It was inconspicuous in inspection. There was no visual restriction, however a feeling of friction on the superior orbit during lid movement. Through eyelid crease lid incision, a solitary lesion appeared in the preaponeurotic space that orginated from the upper tarsal plate. Excisional biopsy revealed adipose proliferation with patch form vessels. This case represents the second reported angiolipoma of the eyelid in English literature. IntroductionAngiolipomas are common variants of lipomatous tumors. They most commonly occur on the extremities and trunk of children and adolescents. It is rarely encountered in the orbital region. We present a rare case of angiolipoma within the eyelid with associated friction in the glide zone. Case descriptionA 74-year-old man was referred to the opthalmology clinic with a history of a mass in the left upper eyelid that had been present for four years. Visual acuity was 7/10 for the right eye and 6/10 for the left eye, anterior segment examination revealed grade 2 nuclear cataract for the right eye and grade 3 nuclear cataract for the left eye. Motility was bilateral normal with no diplopia. Levator functions were bilateral normal, except there was a minimal mechanical ptosis for the left eye. There was also a subcutaneous mass on the left forearm. Systemic check-up relevead no other illness or malignancy. On clinical examination, there was a subcutaneous, firm and round mass located on the central left upper eyelid, which was non-tender to palpation. It was inconspicuous on inspection. There was no eyelid movement restriction but a feeling of friction on the superior orbit during lid movement. Ultrasonography showed a homogeneous echogenic and well defined mass in the anterior orbit. No internal vascularity was seen. An excisional biopsy was taken through the crease of the upper eyelid under local anesthesia. The lesion was 9x7x7 mm in diameter and located 15 mm superior to the eyelid margine and located under the orbicularis muscle. During the removal operation, it was noted that the lesion was stuck to the tarsal border from its base. The mass was removed completely and its location was cauterized while preserving the underlying tarsal plate (Figure 1 ). Histologically, this nodular lesion, which was composed of mature adipose lobules with a patch of proliferating vascular components and well-circumscribed by fibrous connective tissue, was classified as angiolipoma (Figure 2 ). Postoperatively, visual acuity was 7/10 for the right eye and 6/10 for the left eye, with no change in the anterior segment examination. The patient did not have any motility restrictions and there was no recurrence during follow-up for the next 12 months. DiscussionAngiolipoma accounts for 6–17% of all lipomas. It rarely occurs in the head and neck region . Eyelid angiolipoma has been reported in only one patient while orbital angiolipoma has been shown in two two cases .Several hypotheses have been proposed for the cause of this lesion: embryonic sequestration of multipotential cells, fatty metamorphosis of a central hemangioma, and vascular proliferation of congenital lipoma. Mast cell-derived vascular endothelial growth factor (VEGF) was suggested as a possible reason for increased vascularity in this particular tumor . Angiolipoma characteristically occurs as an encapsulated mass of mature adipose tissue containing clusters of vascular network that was first described by Bowen in 1912. These tumors are frequently tender and occur on the trunk, extremities, and especially forearm of young men. Angiolipomas are classified as infiltrating and non-infiltrating based on the presence of a complete capsule. The infiltrating type is usually poorly encapsulated and has a high recurrence rate after surgical removal . Unlike most adipose tissue tumors, angiolipoma do not have chromosomal alterations. For this reason, it has been proposed that they may represent hamartomas rather than true neoplasm .There have been a few reports of lipomatous variants involving the eyelids: dermolipomas, myolipomas, spindle cell lipomas, and others. The nasopalpebral lipoma-coloboma syndrome associated eye abnormalities and palpebral lipoma are rare congenital abnormalities . The upper eyelid consists of two layers; the anterior skin/orbicularis oculi layer and posterior layers of the levator aponeurosis, Müller’s muscle, and the tarsal plate. The glide zone is in the middle of the two layers consisting of the preaponeurotic fat and all potential space. When the levator contracts, the tarsal plate glides up under the skin and orbicularis, therefore the glide zone acts like a friction-free layer .Our patient was experiencing discomfort when looking straight ahead from downgaze. The mass expanded up to the tarsal plate so it was touching the superior orbit. The case presented here is the second reported angiolipoma that appears as an eyelid mass. A histologic variant of lipoma should be considered in diagnosis, if it occurs in unusual locations. NotesCompeting interestsThe authors declare that they have no competing interests. Arenaz Búa J Luáces R Lorenzo Franco F García-Rozado A Crespo Escudero JL Fonseca Capdevila E López-Cedrún JL Angiolipoma in head and neck: report of two cases and review of the literature 2010 Int J Oral Maxillofac Surg 610-5 Arenaz Búa J, Luáces R, Lorenzo Franco F, García-Rozado A, Crespo Escudero JL, Fonseca Capdevila E, López-Cedrún JL. Angiolipoma in head and neck: report of two cases and review of the literature. Int J Oral Maxillofac Surg. 2010 Jun;39(6):610-5. DOI: 10.1016/j.ijom.2009.12.021 http://dx.doi.org/10.1016/j.ijom.2009.12.021 Deprez M Uffer S Clinicopathological features of eyelid skin tumors. A retrospective study of 5504 cases and review of literature 2009 Am J Dermatopathol 256-62 Deprez M, Uffer S. Clinicopathological features of eyelid skin tumors. A retrospective study of 5504 cases and review of literature. Am J Dermatopathol. 2009 May;31(3):256-62. DOI: 10.1097/DAD.0b013e3181961861 http://dx.doi.org/10.1097/DAD.0b013e3181961861 Carruth BP Meyer DR Angiolipoma of the Orbit: A Rare Tumor in an Unusual Location 2015 Ophthal Plast Reconstr Surg e142-5 Carruth BP, Meyer DR. Angiolipoma of the Orbit: A Rare Tumor in an Unusual Location. Ophthal Plast Reconstr Surg. 2015 Nov-Dec;31(6):e142-5. DOI: 10.1097/IOP.0000000000000180 http://dx.doi.org/10.1097/IOP.0000000000000180 Sciot R Akerman M Dal Cin P De Wever I Fletcher CD Mandahl N Mertens F Mitelman F Rosai J Rydholm A Tallini G Van den Berghe H Vanni R Willen H Cytogenetic analysis of subcutaneous angiolipoma: further evidence supporting its difference from ordinary pure lipomas: a report of the CHAMP Study Group 1997 Am J Surg Pathol 441-4 Sciot R, Akerman M, Dal Cin P, De Wever I, Fletcher CD, Mandahl N, Mertens F, Mitelman F, Rosai J, Rydholm A, Tallini G, Van den Berghe H, Vanni R, Willen H. Cytogenetic analysis of subcutaneous angiolipoma: further evidence supporting its difference from ordinary pure lipomas: a report of the CHAMP Study Group. Am J Surg Pathol. 1997 Apr;21(4):441-4. DOI: 10.1097/00000478-199704000-00010 http://dx.doi.org/10.1097/00000478-199704000-00010 Oliphant H Rajak S Selva D Simple lipoma of the eyelid: a rare entity 2016 Clin Exp Ophthalmol Oliphant H, Rajak S, Selva D. Simple lipoma of the eyelid: a rare entity. Clin Exp Ophthalmol. 2016 Nov 6. DOI: 10.1111/ceo.12870 http://dx.doi.org/10.1111/ceo.12870 Chen WP The concept of a glide zone as it relates to upper lid crease, lid fold, and application in upper blepharoplasty 2007 Plast Reconstr Surg 379-86 Chen WP. The concept of a glide zone as it relates to upper lid crease, lid fold, and application in upper blepharoplasty. Plast Reconstr Surg. 2007 Jan;119(1):379-86. DOI: 10.1097/01.prs.0000244908.04694.32 http://dx.doi.org/10.1097/01.prs.0000244908.04694.32 0

1 1 Figure 1: An intraoperative photograph of the lesion showing a mass on the upper eyelid crease, which was adherent to the tarsal plate

2 2 Figure 2: Microphotography of the specimen showing encapsulated tumor mass and mature adipocytes separated by a vascular patch (H & E stain; 10X)

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