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NX:	A Severe Case of Steroid-Induced Cheek Lipoatrophy
Reconstructed via Deepithlized Submental Flap

Masaaki Kosaka, M.D., Ph.D.


Department of Plastic Surgery, 
International University of Health and Welfare Hospital
Iguchi 537-3, Nasu-shiobara city, Tochigi 329-2763, Japan
E-Mail:  HYPERLINK "mailto:kosaka@iuhw.ac.jp" kosaka@iuhw.ac.jp
Disclosure
The authors declared no potential conflict of interest with respect to the research, authorship, and publication of this article.

Funding
The authors received no financial support for the research, authorship, and publication of this article.
0Abstract0
Subcutaneous atrophy developed in a 69-year-old woman at the site of repetitive triamcinolone acetonide injection for cheek lymphocytoma (pseudo-lymphoma). Subcutaneous atrophy following local injection of corticosteroids is generally reversible and spontaneously improves within several years after the injection; however, improvement was not observed and subsequent filler or autologous fat injection had no effect. In this case, a deepithelized submental island flap was utilized for contour surgery; moreover, a satisfactory result was obtained. The deepithelized submental flap is an excellent option for cheek reconstruction due to reliability of flap circulation, versatility and easy application in elderly patients.

[Key words]
Steroids, Side effect, Lipoatrophy, Submental flap, Reconstruction
Introduction
Pseudolymphoma, which is reactive extranodal lymphocytosis, displays minimally atypical lymphocytes that are basically benign.1 The optimal treatment for pseudolymphoma remains uncertain: many practitioners recommend frequent observation, whereas some clinicians have applied corticosteroids as an ointment or injection therapy. However, atrophy of the skin and subcutaneous tissue, an adverse effect of corticosteroid injection, at the administration site is well documented.2 Such atrophy is known to improve spontaneously over time; in contrast, cases in which spontaneous improvement fails to occur require appropriate intervention including surgery. 3, 4
     Reconstructive surgery involving deepithelized submental island flap transfer was performed in the current case of steroid-induced cheek lipoatrophy.  Satisfactory results were obtained.

Case Report
A 69-year-old Japanese woman presented to our outpatient clinic with a ten-year history of cheek lesion. Ten years earlier, a dome-shaped nodule had arisen on her right cheek. The patient was diagnosed with pseudolymphoma of skin on the basis of biopsy findings.  The patient received repetitive local injection of Triamcinolone Acetonide by dermatologist (more than 20 injections). Gradually, her cheek became atrophic and demonstrated concavity. In order to correct the atrophy, hyaluronic acid injection and autologous fat injection were conducted as an aesthetic surgery; however, the fillers completely disappeared within several years. Consequently, she was referred to our clinic.
Upon physical examination, the patient exhibited a 5 x 4 cm concave area in the right buccal region (Figure 1). The skin overlying the lesion was atrophic and hypopigmented. CT imaging revealed severely atrophied buccal fat tissue (Figure 2). The submental area demonstrated adiposity and drooping; as a result, the submental island flap was selected for reconstruction.
Under general anesthesia, a 11 x 4 cm submental flap, supplied by both submental vessels, was raised. The contralateral left submental artery was ligated and divided. Dissection was carried down to the origin of the facial artery. Following complete de-epithelization of the flap epidermis, the flap was transferred to the concave area via a subcutaneous tunnel; moreover, the surgical defect was sutured primarily without excessive skin tension. The post-operative course was uneventful (Figure 3). Six months later, a partial revision to achieve the desired contour was performed under local anesthesia. The patient was fully satisfied with the final results of reconstruction and inconspicuous donor scarring (Figure 4). 

Discussion
Pseudolymphoma, which is reactive extranodal lymphocytosis, displays minimally atypical lymphocytes that are basically benign. These lymphocytes may represent exaggerated reactions to diverse external antigens, e.g., insect bite, tattoo, zoster, trauma, etc. Treatment includes antibiotics, intralesional and systemic corticosteroids, excision, radiotherapy and immunosuppressants; however, the optimal treatment for pseudolymphoma remains uncertain.1 In some cases, the tumor rapidly decreased following �biopsy� for analysis of the clonal gene with no additional intervention.5 Therapy depends on assessment and biologic behavior, which is usually benign. 1
Corticosteroid injections are frequently included as a therapeutic option in a wide variety of diseases: skin diseases, asthma or other allergic diseases, and musculoskeletal disorders.  Unfortunately, subcutaneous atrophy due to local injection of corticosteroid was described as early as the 1950�s.6 Many reports stated that steroid-induced atrophy is not permanent; rather, it is a transient reaction.6,7  These atrophic changes were thought to be attributable to ischemia,8 an inhibitory action of fibroblast proliferation in a dose-related manner.9 A review of the literature revealed major adverse events at 5.8%, ranging from pigmentation and atrophy of the skin to cellulitis; generally speaking, these adverse effects could perhaps be classified as �relatively mild�.2 Local atrophy cannot be regarded as a serious complication of corticosteroid therapy; however, the condition is unsightly and may persist for several years. When this type of deformity occurs on the face, patient quality of life (QOL) may be negatively impacted; thus, appropriate treatment including surgery is indicated.
In terms of reconstruction, a number of methods involving the introduction of artificial materials such as collagen, hyaluronic acid injections, dermal fat graft and fat injection,3,4 other platysma flap types, sternocleidomastoid myocutaneous flap or free flap transfers can be selected for augmentation surgery.  Although fillers and fat injection therapy were implemented in another hospital, they demonstrated little efficacy.  The documented results of fat injection vary and depend on fat harvesting and the specific handling technique as well as the recipient area;3 thus, local circulation within the recipient area, particularly atrophic tissue, may be insufficient to permit the survival of free grafted tissue.  Moreover, this patient displayed special medical status, namely, corticosteroid medication for rheumatoid arthritis and bleeding tendency consequent to autoimmune hemolytic anemia. In view of these points, the following requirements are mandatory in the current case:
(1) Autologous tissue characterized by rich blood flow may be preferable to free fat or dermal-fat graft; 
(2) Pedicle flap without vascular anastomoses is preferable; 
(3) A small target area (e.g., 5 x 4 cm) and tissue thickness of 2 ~3 cm is desirable;
(4) Inconspicuous donor scarring; the submental island flap was the method of choice. 
   The submental flap, which was described initially by Martin10 in 1993, has been widely applied as a reconstructive approach for facial tissue defects. A long flap extending over the corner of the mandible can be elevated readily with no serious complications or conspicuous donor scarring. Pistre et al. 11 reported mean flap size of 11.8 x 5.5 cm; moreover, they believed this flap to possess great clinical potential and to be a meaningful addition to the existing surgical armamentarium.
Additionally, the elderly submandible tends to display sagging skin as in the current case; thus, donor scarring is inconspicuous despite the elevation of a relatively large flap. During the procedure, the distal end of the flap is lifted inward and upward with a traction suture in order to prevent the flap from sagging; however, if the lower half is slightly bulky, then subsequent reduction surgery under local anesthesia is necessitated.  �The flap should be made larger than recipient size� is the basic philosophy of flap surgery due to post-operative tissue atrophy of the flap. However, in the elderly, the overlying skin tension may become insufficient to withstand the flap weight. Tan et al.12 used the submental flap in deepithelialized fashion successfully for augmentation of the face in three patients who presented with hemifacial microsomia (n = 2) and longstanding facial paralysis (n = 1). A second debulking procedure was performed in elderly patient. The authors recommend that the volume of the lower half of the flap should be modest from the perspective of �tissue sagging under gravity� versus �post-operative flap atrophy� in elderly patients. The submental flap is an excellent option for cheek reconstruction in elderly patients due to reliability of circulation, versatility and ease of application.

Acknowledgement
The presented case was experienced in Fukuoka Sanno Hospital author�s former post. The author deeply indebted to Drs. Ayumi Tsukamoto, M.D. and Tadayoshi Sakamoto, M.D. for their assistance of surgery.

References
Gilliam AC, Wood GS (2000) Cutaneous lymphoid hyperplasias. Semin Cutan Med Surg 19:133-141. 
Brinks A, Koes BW, Volkers AC, Verhaar JA, Bierma-Zeinstra SM (2010) Adverse effects of extra-articular corticosteroid injections: a systematic review. BMC Musculoskelet Disord 11:206.
Latoni JD, Marshall DM, Wolfe SA (2000) Overgrowth of fat autotransplanted for correction of localized steroid-induced atrophy. Plast Reconstr Surg 106: 1566-1569.
Imagawa K, Ohkuma S (2010) A case of fat injection for treating subcutaneous atrophy caused by local administration of corticosteroid. Tokai J Exp Clin Med 35: 66-69.
Kakizaki A, Fujimura T, Numata I, Hashimoto A, Aiba S (2012) Pseudolymphomatous folliculitis on the nose. Case Rep Dermatol 4:27-30.
Goldman L, Preston RH (1953) Local tissue reactions to cortisone and hydrocortisone (compound F) in man. VIII. Studies of local therapeutic effect of hydrocortisone in diseases of skin. AMA Arch Derm Syphilol 67:163-165.
Goldman L (1962) Reaction following intralesional and sublesional injection of corticosteroids. JAMA; 182: 613-616.
McKenzie AW (1962) Percutaneous Absorption of Steroids. Arch Dermatol 86: 611-614.
McCoy BJ, Diegelmann RF, Cohen IK (1980) In vitro inhibition of cell growth, collagen synthesis, and prolyl hydroxylase activity by triamcinolone acetonide. Proc Soc Exp Biol Med 163:216-222.
Martin D, Pascal JF, Baudet J, Mondie JM, Farhat JB, et al. (1993) The submental island flap: A new donor site. Anatomy and clinical application as a free or pedicled flap. Plast Reconstr Surg 92: 867-873. 
Pistre V, Pelissier P, Martin D, Lim A, Baudet J (2001) Ten years of experience with the submental flap. Plast Reconstr Surg 108: 1576-1581.
Tan O, Atik B, Parmaksizoglu D (2007) Soft-tissue augmentation of the middle and lower face using the deepithelialized submental flap. Plast Reconstr Surg 119: 873-879.

Figure Legend

Figure 1: Pre-operative frontal view
Soft tissue atrophy of right cheek is apparent.

Figure 2: Pre-operative CT image
Soft tissue atrophy of right cheek is apparent.

Figure 3: Operative findings
(a) A submental flap, 11 x 4 cm in size, was drawn. (b) Flap elevation. (c) Flap rotation. (d) De-epithelized flap inserted into the depressed cheek area.

Figure 4: Post-operative view






	
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