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6.DS2008-Upper eyelid myocutaneous flap

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					Large Cutaneous Epithelioid Angiomatous Nodule on the
Glabella Repaired Using a Medially Based Upper Eyelid
Myocutaneous Flap
CHIEN-HSUN CHEN, MD, YI-LUN TSAI, MD, PO-YU SHIH, MD, AND CHENG-SHENG CHIU, MD, MPHÃ


The authors have indicated no significant interest with commercial supporters.




C     utaneous epithelioid angiomatous nodule
      (CEAN) is a relatively new entity. Eighteen
cases have been reported in the literature. We report
                                                                  orbitale. The levator complex was not disturbed. In a
                                                                  lateral to medial fashion, the myocutaneous flap was
                                                                  carefully dissected from the orbital septum and el-
a 67-year-old man with a large CEAN on the                        evated with the aid of double hooks, with the flap
glabella area that was repaired using a medially
based bilateral upper eyelid myocutaneous flap after
local wide excision.



Case Report

A 67-year-old man with end-stage renal disease re-
ceiving hemodialysis developed an exophytic nodule
on the glabella that grew over 2 weeks (Figure 1).
Although it bled easily, it was otherwise asymptom-
atic. On examination, the lesion was a 2.5-cm-
diameter rounded nodule with a peripheral bluish
hue, focal ulceration, and eschar formation. It was
excised with a 2-mm margin under the clinical im-
pression of a benign vascular tumor, then repaired
using a medially based upper eyelid myocutaneous
flap.

Under local anesthesia, the skin islands were marked
on both upper eyelids, with the total dimension
equal in size to the defect. The lower margin of the
flap was at the palpebral sulcus, 8 to 10 mm above
the eyelid margin (Figure 2A). An incision was made
                                                                  Figure 1. A 67-year-old man with a protruding nodule mea-
through the skin and the preseptal and orbicularis                suring 2.5 cm in diameter on the glabella. The nodule had
oculi muscles down to the level above the septum                  eschar formation 2 weeks later, as shown in Figure 2.

ÃAll authors are affiliated with the Department of Dermatology, Chang Gung Memorial Hospital, Chang Gung
University College of Medicine, Taipei, Taiwan

& 2008 by the American Society for Dermatologic Surgery, Inc.  Published by Wiley Periodicals, Inc. 
ISSN: 1076-0512  Dermatol Surg 2008;34:1–5  DOI: 10.1111/j.1524-4725.2008.34398.x

                                                                                                                              1
    GLABELLAR CEAN




    Figure 2. The glabellar defect was reconstructed using a medially based upper eyelid myocutaneous flap. (A) Design of the
    myocutaneous flap. (B) Bilateral upper eyelid myocutaneous flap after elevation. (C) Immediate postoperative appearance
    after Penrose drain insertion.




    remaining in continuity with the medial fat pocket.           infiltrates surrounded the nodule (Figure 3A and B).
    Dog ears were corrected with sharp scissors, and              Mitotic figures were seen, but no atypia or
    with adequate dissection, a motile myocutanous flap           invasive growth was found (Figure 3C). The
    was created for ensuing rotation into the defect              surgical margins were free. Based on the clinical
    (Figure 2B). During these processes, care was taken           and histopathological findings, CEAN was
    to avoid damage to the peripheral arterial arcade or          diagnosed.
    the lacrimal drainage system. The flaps reached the
    skin defect on the glabella with little tension and no        The patient has been free of disease for 30 months
    sign of vascular compromise. Donor site defects were          since excision. A minor postoperative lagophthalmos
    closed primarily, but because prominent vascularity           was seen, which resolved spontaneously after several
    was noted on the left side, a Penrose drain was               weeks. At 30-month follow-up, the flap had a highly
    placed there to avoid hematoma formation (Figure              satisfactory cosmetic result. The eyebrows remained
    2C). Cold compression was applied postoperatively             symmetrical, and no ectropion was noted (Figure 4A
    for the same purpose. The postoperative course was            and B).
    devoid of complications.

                                                                  Discussion
    Microscopic examination revealed a circumscribed
    nodule composed of lobules of vessels, plump                  Brenn and Fletcher first described and named cuta-
    epithelioid endothelial cells with occasional intra-          neous epithelioid angiomatous nodule.1 It usually
    cytoplasmic vacuoles, and diffuse hemorrhage in               presents as a rapidly growing, small, solitary nodule
    the dermis. Fibrosis and lymphoplasmacytic                    with erythematous to bluish discoloration and has a




2   D E R M AT O L O G I C S U R G E RY
                                                                                                               CHEN ET AL




Figure 3. (A) A large subcutaneous tumor nodule comprising lobules of vessels with plump epithelioid endothelial cells.
Diffuse hemorrhage with fibrosis and lymphoplasmacytic infiltrates can also be seen (hematoxylin and eosin (H&E); original
magnification, Â 20). (B) Intralesional vascular channels and sheet-like proliferation of plump endothelial cells (H&E; orig-
inal magnification, Â 100). (C) Large epithelioid cells with eosinophilic cytoplasm and occasional intracytoplasmic vacuoles.
Mitotic figures can been seen, but no cellular atypia is observed (H&E; origininal magnification, Â 400).




predilection for the trunk and limbs in adults. There           cell atypia is absent.1 Our patient had a unique
is no definitive association with infection, immuno-            clinical presentation and bizarre histopathologic
suppression, or previous trauma.1–4 The clinical                findings. Clinically, there have been no reports of
nature of cutaneous epithelioid angiomatous nodule              this lesion presenting as an unusually large facial
is benign, and no recurrence or metastasis has been             nodule located in the glabellar area.1–4 Histopatho-
observed. Histopathologically, lobular, well-demar-             logically, the lobulated tumor mass, vacuolated
cated proliferation of large epithelioid endothelial            epithelioid cells, and obvious mitotic figures
cells located within the dermis or submucosa                    raise concern as to the possibility of vascular
characterize the tumor. These epithelioid endothelial           malignancy.
cells contain abundant eosinophilic and occasionally
vacuolated cytoplasm and vesicular nuclei with                  The differential diagnosis thus comprises a hetero-
prominent nucleoli. Mitotic figures may be seen, but            geneous spectrum of disorders including epithelioid




Figure 4. (A) Two weeks after surgery. (B) At 30-month follow-up. No ectropion was noted, and the patient was highly
satisfied with the cosmetic result.




                                                                                                                 34:**:2008     3
    GLABELLAR CEAN




    hemangioma, bacillary angiomatosis, epithelioid an-     forehead flaps,8 the cosmetic results of those flaps
    giosarcoma, and epithelioid hemangioendothelioma.       are not superior to a medially based upper eyelid
                                                            myocutaneous flap. Also, the supratrochlear
    In our case, the tumor was solitary and confined to     artery may be damaged in a midline forehead flap
    the dermis, with few eosinophils, and its short clin-   because of the central location and the large size of
    ical course without recurrence, did not favor the       the tumor.
    diagnosis of epithelioid hemangioma. Furthermore,
    because there were no acute inflammatory infiltrates    Possible complications of the medially based upper
    or neutrophils, and because our patient was not         eyelid myocutaneous flap include lagophthalmos,
    immunosuppressed, bacillary angiomatosis could be       distal flap necrosis, and trauma to the lacrimal
    reasonably excluded. Because the lesion was well        drainage system. Lagophthalmos is usually
    circumscribed and lacked any cytologic atypia or        temporary and resolves quickly. A careful preoper-
    pleomorphism, it could be further differentiated        ative external eye examination is critical to prevent
    from epithelioid angiosarcoma. It also lacked the       ectropion or permanent lagophthalmos. In a review
    endothelial multilayering or dissection of dermal       of previous reports, distal flap necrosis occurred in
    collagen by vascular channels seen in cutaneous an-     three of 63 patients.7 Although the potential for
    giosarcoma. Epithelioid hemangioendothelioma            trauma to the lacrimal drainage system has never
    typically presents as a well-circumscribed nodule       been specifically reported, some authors have
    with an infiltrative growth pattern of ovoid, cubo-     emphasized it, and thus diligent care must be taken
    idal, or short spindle cells arranged in cords and      during dissection. In properly selected patients,
    strands and embedded in a distinctive hyalinized or     complications are rare and mild, with highly
    mucoid stroma but no obvious vascular channels.         satisfactory functional and aesthetic results.

    The difficulties in management lay not only in the      In summary, we present a patient with a CEAN with
    diagnosis, but also in the repair of such a large       a peculiar clinicopathologic presentation in the
    glabellar defect. This defect was unsuitable for        glabellar region and demonstrate the usefulness of a
    second-intention healing or primary closure because     medially based upper eyelid myocutaneous flap for
    of its large size. Skin grafting, although simple and   reconstruction of this large skin defect.
    straightforward, provides poor color and texture
    matching. It also usually leaves a conspicuous de-
                                                            Acknowledgments The authors would like to
    pression due to the lack of subcutaneous tissue after
                                                            thank Dr. Fu-Chan Wei, who kindly reviewed the
    grafting. Considering tissue match, as well as the
                                                            manuscript.
    possible morbidity associated with repair at close
    proximity to eyelids and eyebrows, local flaps were
    the favored reconstructive option for this
    cosmetically important location.

                                                            References
    Preoperative assessment revealed deep rhytides and
                                                            1. Brenn T, Fletcher CD. Cutaneous epithelioid angiomatous nodule:
    much skin laxity within the glabella and upper eye-
                                                               a distinct lesion in the morphologic spectrum of epithelioid vascular
    lids. After thorough consideration of our patient’s        tumors. Am J Dermatopathol 2004;26:14–21.
    status, we chose the medially based upper eyelid        2. Kantrow S, Martin JD, Vnencak-Jones CL, Boyd AS. Cutaneous
    myocutaneous flap, most commonly used for re-              epithelioid angiomatous nodule: report of a case and absence of
                                                               microsatellite instability. J Cutan Pathol 2007;34:515–6.
    construction of lower eyelid and inner and outer
                                                            3. Fernandez-Flores A, Montero MG, Renedo G. Cutaneous epithe-
    canthal defects.5–7 Although other possible choices        lioid angiomatous nodule of the external ear. Am J Dermatopathol
    for reconstruction include retroangular and midline        2005;27:175–6.




4   D E R M AT O L O G I C S U R G E RY
                                                                                                                            CHEN ET AL




4. Zamecnik M. Relationship between cutaneous epithelioid an-          8. Iida N, Ohsumi N, Tsutsumi Y. Use of bilateral retroangular flaps
   giomatous nodule and epithelioid hemangioma. Am J Dermatopa-           for reconstruction of the glabella and nose. Br J Plast Surg 2001;
   thol 2004;26:351–2.                                                    54:451–4.

5. Jelks GW, Glat PM, Jelks EB, Longaker MT. Medial canthal re-
   construction using a medially based upper eyelid myocutaneous
   flap. Plast Reconstr Surg 2002;110:1636–43.

6. Maniglia AJ, Megerian CA. The medially based myocutaneous
   upper eyelid flap for lateral nasal defect reconstruction. Am J     Address correspondence and reprint requests to: Cheng-
   Otolaryngol 1996;17:118–21.                                         Sheng Chiu, MD, MPH, Department of Dermatology,
7. Porfiris E, Kalokerinos D, Christopoulos A, et al. Upper eyelid     Chang Gung Memorial Hospital, Chang Gung University
   island orbicularis oculi myocutaneous flap for periorbital recon-   College of Medicine, No. 199, Tung Hwa North Road,
   struction. Ophthal Plast Reconstr Surg 2000;16:42–4.                Taipei 105, Taiwan, or e-mail: cchiu1229@yahoo.com.tw




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