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Pediatric earlobe keloids: Outcomes and patterns of recurrence
YJPSU-59275; No of Pages 4 Journal of Pediatric Surgery xxx (xxxx) xxx
Contents lists available at ScienceDirect
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Pediatric earlobe keloids: Outcomes and patterns of recurrence☆ Faraz A. Khan ⁎, Natalie A. Drucker, Shawn D. Larson, Janice A. Taylor, Saleem Islam Division of Pediatric Surgery, Department of Surgery, University of Florida College of Medicine, 1600 SW Archer Rd. P.O. Box 10019, Gainesville, FL, USA
a r t i c l e
i n f o
Article history: Received 10 September 2018 Received in revised form 23 June 2019 Accepted 15 July 2019 Available online xxxx Key words: Earlobe Keloids Pediatric Recurrence
a b s t r a c t Purpose: Keloids occur as a result of abnormal wound healing and as many as 70% of keloids and hypertrophic scars affect the pediatric population. Earlobe keloids, similar to lesions elsewhere in the body, remain a challenging problem given the high rates of recurrence and lack of consensus regarding treatment strategy. This study aims to evaluate the outcomes and patterns of recurrences following treatment of earlobe keloids in a large cohort of pediatric patients to facilitate identification of the optimal treatment strategy. Methods: All pediatric patients who underwent surgical therapy for earlobe keloids over a 10-year period (2004–2015) were identified and demographic, clinical and outcomes data were collected. A follow-up phone survey was administered to assess rates of long-term recurrence and overall satisfaction with the treatment strategy. Data analysis was performed using Student's t-test and Fisher's exact as appropriate. Results: A total of 94 patients with 135 keloids were identified. Mean age was 14 years with slight preponderance of females (52%) and a majority (75%) of the patients were African American (AA). Mean keloid size was 2.4 cm (0.25–11 cm) with ear piercing being the most common inciting etiology. Excision with steroid injection (59%) was the most frequent treatment approach followed by excision alone (25%). During a median follow up of 2 years, keloid recurrence occurred in 27 (28.7%) patients, 19 (70.4%) underwent additional therapy with 10 (52.6%) re-recurrences noted. Median time to recurrence was 23 months, with a slightly higher recurrence rate in AA patients (32%; p = 0.26) and in those age b 10 years (50%; p = 0.11). Conclusion: This represents the largest series of earlobe keloids treatment in children. Our data suggest that recurrence rates are unaffected by the age at excision and race of the patient. Moreover, adjuncts to excisional therapy such as steroid injection, compression therapy and radiation also did not appear to influence recurrence. Given the pattern of recurrences studies with longer term follow-up are needed to assess the efficacy of treatment strategies. Type of study: Case series. Level of evidence: IV © 2019 Elsevier Inc. All rights reserved.
Any injury to the deep dermis irrespective of the mechanism is followed by wound healing which clinically manifests with the formation of a scar. [1] While scars are typically classified based on morphological appearance or histopathology, in essence clinically the appearance and characteristics of a patient's scar are simply the endpoint of genetic and environmental factors affecting the wound healing process. [2] Globally, approximately 100 million individuals per year acquire a scar following surgical/traumatic insult, and approximately 15 million are subsequently complicated by keloids and hypertrophic scars which are because of excess collagen deposition. Almost 70% of keloids or hypertrophic scars are noted to occur in children. [2] ☆ Funding Disclosure and Conflicts of Interest:No external funding was secured for this study. The authors have no financial relationships or any other conflicts of interest to disclose. ⁎ Corresponding author. Tel.: +1 352 265 8800. E-mail addresses: [email protected] (F.A. Khan), [email protected] (N.A. Drucker), [email protected]fl.edu (S.D. Larson), [email protected]fl.edu (J.A. Taylor), [email protected]fl.edu (S. Islam).
Hypertrophic scars by definition remain within the boundaries of the original injury, whereas keloids extend beyond and frequently have a delayed onset as compared to hypertrophic scars. Keloids disproportionately occur in dark skinned individuals, are associated with a familial history, and are related to hormonal changes associated with puberty. [2,4,5] These lesions are frequently symptomatic and can be associated with pruritus, pain, and tenderness. [13] Moreover, there is significant social and psychological impact on the affected patient. [6] Certain anatomic areas of the body such as the chest, shoulders, neck and ears appear to be more predisposed to the development of keloids and have important cosmetic significance. [7,12] Given the frequency of earlobe piercing associated with an approximately 2.5% risk of keloid development, earlobes are one of the most frequent locations these lesions are seen in pediatric patients. [8,9] Earlobe keloids, similar to lesions elsewhere in the body, remain a challenging problem to manage given the high rates of recurrence and lack of consensus regarding treatment strategy. Numerous treatment strategies have been proposed including surgical excision, intralesional
https://doi.org/10.1016/j.jpedsurg.2019.07.006 0022-3468/© 2019 Elsevier Inc. All rights reserved.
Please cite this article as: F.A. Khan, N.A. Drucker, S.D. Larson, et al., Pediatric earlobe keloids: Outcomes and patterns of recurrence, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.07.006
2
F.A. Khan et al. / Journal of Pediatric Surgery xxx (xxxx) xxx
steroid injection, radiotherapy, cryotherapy and cryosurgery. [6–8] Unfortunately, majority of these treatment strategies are known to be associated with frequent recurrences. [10] While numerous management options are available as discussed above there are no specific guidelines for the pediatric and adolescent population and evidence supporting or refuting each approach is either extrapolated from adult studies or relies on very small pediatric studies. [2,11] Given the paucity of data in pediatric/adolescent population, our study aims to review the outcomes of various treatment strategies, factors associated with recurrence and options for management of recurrences. 1. Methods 1.1. Patient selection Following approval by the institutional review board at the University of Florida, divisional, hospital and institutional databases were searched for pediatric patients having undergone surgical interventions for earlobe keloids over a 10-year period. The International Classification of Diseases, Ninth Revision (ICD-9) and Current Procedural Terminology (CPT) codes were used to identify all the cases treated by any service (Pediatric surgery, plastic surgery, or dermatology) 1.2. Definitions and data Patients between the ages of 1 and 19 years were identified using Current Procedural Terminology (CPT) codes 11,440–11,446 and International Classification of Diseases-9 (ICD-9) code 701.4 treated between 2004 and 2015. Patient demographics, presurgical clinical characteristics of the lesion (location, laterality and size), details of treatment strategy and postoperative follow-up data were collected. We also attempted to reach each patient by telephone and mail and administered a questionnaire (supplement 1) with an aim to evaluate keloid recurrence, additional surgical procedures and overall satisfaction with the treatment strategy. This survey instrument was not independently validated; however, focus groups within the department were used to test it prior to phone call. Each subject was sent a letter two weeks prior to attempted contact for informational purposes, and four separate attempts to call the patient were made. The rationale for the phone follow up was to increase the follow-up duration, as well as ascertain patients who may have been lost to our center, and also see the satisfaction with the results. Majority of the patients (96%) had routine postoperative follow-up (median follow-up duration of 8 months) in addition to the phone survey. 1.3. Statistical analysis Data analysis was performed using Minitab 17 Statistical Software (2010); State College, PA: Minitab, Inc. (www.minitab.com). Continuous variables were analyzed using Student's t-test, or the Mann– Whitney U test for nonparametric data. Categorical variables were analyzed using proportional statistics (Fisher's exact test). Significance was inferred at p b 0.05. 2. Results
Table 1 Patient demographics/ lesion characteristics (n = 94). Age (years)
Gender Race
Etiology Architecture
Size (cm)
Mean
14.0
Range
2–19
Male Female African American Caucasian Hispanic Asian Earrings Surgery/Trauma Nodular Pedunculated Mixed Mean Range
42 (45%) 52 (55%) 75 (80%) 12 (12%) 5 (5%) 2 (2%) 84 (89%) 10 (11%) 72 (76%) 13 (14%) 9 (9%) 2.4 0.25–11
2.2. Treatment strategy Treatment strategies utilized were (i) excision alone (n = 24), (ii) excision with intralesional injection of triamcinolone acetonide (Kenalog, Bristol-Myers Squibb, New York NY) n = 56, (iii) excision with intralesional injection of triamcinolone acetonide and compression earrings (n = 5) and (iv) excision with postoperative radiation (n = 9). (Table 2) In all cases, the technical aspects of the procedure including the choice of suture were made by the attending surgeon, but absorbable sutures were universally used. Most common was polyglactin acid (braided) suture (VICRYL R; Ethicon, Somerville, NJ) for the deep layer, while monofilament absorbable suture (MONOCRYL R; Ethicon, Somerville, NJ) was used in the superficial layer. Most patients (70%) reported being satisfied or very satisfied with the eventual cosmetic outcome based on a 5-point Likert scale (Very Satisfied, Satisfied, Neutral, Dissatisfied, Very Dissatisfied) irrespective of the treatment strategy employed. This satisfaction included those with mild recurrences as well. 2.3. Risk factors for recurrence Overall rate of recurrences in the entire cohort was n = 27 (28.7%). No statistically significant effect was noted on the rate of recurrences related to the patient's age (p = .08), gender (p = .05), race (p = 0.08), size (p = 0.09), type (nodular vs peduncular) of the lesion (p = 1.00) or treatment strategy employed (p = 0.53). When patients were stratified by age in two groups (less than or more than 10 years of age at the time of initial treatment) no differences were noted in the rate of recurrence (p = 0.11). Similarly, no differences in the rate of recurrences were noted when stratified by gender (p = 0.17) or by race (p = 0.26). 2.4. Re-recurrence Among the 27 patients who had a recurrence 19 (70.4%) underwent additional therapy for the recurrence. The following treatment strategies were used, and 10 (52.6%) re-recurrences were observed: (1) Excision alone in 2 patients (1 re-recurrence), (2) Steroids alone in 2 patients (1 re-recurrence), (3) Excision and steroids in 12 patients (6 re-recurrences) and (4) Excision with postoperative radiation in 3
2.1. Demographic data A total of 94 patients with 135 keloids were identified. Mean age was 14 (2–19) years with slight preponderance of female (52%) patients and a majority (75%) of the patients were African American (AA). Mean keloid size was 2.4 cm (0.25–11 cm), with nodular morphology being the most common and ear lobe piercing was the most common inciting etiology. (Table 1) The phone survey was attempted on all patients, with an overall response rate of 54%.
Table 2 Recurrence by treatment groups. Treatment Group
N
Recurrence N (%)
Overall Median FUP (months)
Excision alone Excision + Steroids Excision + Steroids + Compression Excision + Radiation
24 56 5 9
7 (24%) 13 (23%) 3 (60%) 4 (44%)
22 22 27 70
Please cite this article as: F.A. Khan, N.A. Drucker, S.D. Larson, et al., Pediatric earlobe keloids: Outcomes and patterns of recurrence, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.07.006
F.A. Khan et al. / Journal of Pediatric Surgery xxx (xxxx) xxx
patients (2 re-recurrences). No differences in the rate of re-recurrences were noted with the treatment strategy chosen. (Fisher's exact p = 1.0). These recurrences were reported over the duration of the study with a majority occurring within 2 years (median 23 months). No statistically significant effect was noted on the rate of re-recurrences related to the patient's age (p = 0.38), gender (p = 1.0), race (p = 1.0), or size of the lesion (p = 0.14). 3. Discussion This study, to our knowledge, is the largest series evaluating outcomes and treatment of ear lobe keloids in the pediatric population. With a median follow-up of 2-years, a higher incidence of recurrences was noted in our cohort as compared to those previously reported in pediatric patients. [11] A possible explanation for this observed difference may be the length of follow-up; our data set suggests that the median time to recurrence postexcision is six months, and therefore longer follow-ups are necessary to accurately quantify the burden of recurrence. In addition, we have likely captured patients who had secondary procedures with other providers owing to the telephone-based survey. There remains a paucity of evidence-based guidelines to optimally manage earlobe keloids in pediatric patients. Our data set demonstrates that while there was a high rate of recurrence, it was unaffected by the treatment strategy employed and patient characteristics, including race. This is a key finding, as while AAs have the highest burden of keloids, we may be selecting a subset of patients across race, gender and age that are susceptible to further keloid formation, which is suggested by the even higher rate of recurrence after a second keloid developed. An additional interesting finding of this study was that ear lobe keloids affected both genders equally, as females are more more likely to have ear lobe piercings due to a societal bias, it can be hypothesized that males may have a higher likelihood of developing ear lobe keloids. Intralesional corticosteroids are considered a main stay of therapy of keloids owing to their ease of use and high degree of tolerability. The efficacy of corticosteroids is thought to be secondary to their ability to suppress inflammation, promotion of collagen degeneration and inhibition of further collagen production. [2] The majority of data evaluating use of corticosteroids is several decades old and is limited to adult cases with the exception of one pediatric study. [11,14,15] The response rate in these studies for corticosteroids is reported as 50%–100% with a 9%–50% recurrence rate. [16] Potential for local cutaneous atrophy, hypopigmentation and pain associated with injections are other obstacles limiting repeated injections. In summary while there may be a potential benefit for adjuvant injection of corticosteroids the magnitude of benefit and optimal strategy of use are yet to be adequately defined. Our data set noted no appreciable effect in reduction of recurrence rates or cosmetic outcomes related to the use of corticosteroids. Fortunately, there were no complications related to use of intralesional corticosteroids; therefore, they may be considered as a safe adjunct to surgical therapy. There are some small series that report the use of radiation in trying to reduce the rate of recurrence [17]. Radiation theoretically works by decreasing cellular proliferation. However, the therapy may not be available at all institutions and certainly leads to an increase in the overall expense of therapy. There were 9 cases in our report where this modality was utilized. We did not see any effect on the rate of recurrences; however, given the small number in this subset no definitive conclusions can be drawn. As this cohort was selected on the basis of having undergone a surgical excision of the keloid, all had an operation performed. While surgical removal is likely to be the most common management approach especially for larger lesions, without a denominator of all ear lobe keloids presenting to our institution we cannot appropriately comment on the efficacy of non-surgical approaches. Choice of technical aspects of the procedure (selection of suture type, use of cautery, etc.) is also an interesting variable and may contribute to the risk of recurrence.
3
These aspects of the surgical care, while interesting variables, however can only be adequately controlled in the setting of a prospective study. In all cases, the choice of suture was made by the attending surgeon, but absorbable sutures were universally used. Most common was polyglactin acid (braided) suture for the deep layer, while monofilament absorbable was used in the superficial layer. Similarly, the postoperative course including the use of postoperative steroids, compression earrings and radiation therapy was chosen by the patient's physician. Given the retrospective nature of this data set it is difficult to adequately control for these variables. As previously discussed, given no differences in the complications when used as adjunct, there may be a role of outpatient intralesional steroids for smaller lesions in reducing the need for surgery. Further work will be needed to assess the feasibility of this approach. Our preferred approach to pediatric ear lobe keloids is excision with closure using absorbable sutures and steroid injection, which our data set suggests at least is not inferior to any other treatment plan and is relatively simple without requiring radiation therapy. We contend this data set suggests that pediatric ear lobe keloids are a challenging condition to treat with a high rate of recurrence regardless of the treatment modality chosen. With the evidence currently available to us decision of approach should be tailored to the individual patient at the discretion of treating physician until prospectively collected data are available. There are some key limitations of this study that bear additional discussion. There are inherent biases and limitations from the retrospective nature of the study itself. The subsets of the patients in various treatment strategies were not controlled for baseline characteristics; similarly, the decisions involving the strategy employed were not controlled. Additionally, the limited number of patients in the adjuvant radiation therapy and compression device strategy limits any meaningful conclusion to be drawn pertaining to these strategies. The use of compression earrings has also not been systemically studied for children. We had limited use in our series mainly owing to patient interest and compliance; therefore, we cannot comment on its usefulness. However, despite these limitations, the strength of this report includes the duration of follow up, large number of cases in the cohorts, and evaluation of multiple recurrences with telephone follow up to include patients who sought care elsewhere subsequently. 4. Conclusion Treatment of ear lobe keloids is a challenging problem among pediatric patients given the high rate of recurrence. Our study adds to the known literature in children and notes the lack of difference with concomitant steroid use. Surgical management is safe, but patients should be made aware of the recurrence risk. Data from this study suggest that there are no marked differences in outcomes related to the surgical strategy employed; plans for patients should be tailored individually by the treating physician. Future studies will investigate the role of compression and steroids as primary therapy. While prospective studies are needed to better define the risk factors associated with recurrence, it would be reasonable because of this to consider deferring surgical intervention until the child is older, particularly among African American patients. Appendix A. Supplementary data Supplementary data to this article can be found online at https://doi. org/10.1016/j.jpedsurg.2019.07.006. References [1] English RS, Shenefelt PD. Keloids and hypertrophic scars. Dermatol Surg 1999 Aug 1; 25(8):631–8. [2] Krakowski AC, Totri CR, Donelan MB, et al. Scar management in the pediatric and adolescent populations. Pediatrics 2016 Feb 1;137(2):e20142065.
Please cite this article as: F.A. Khan, N.A. Drucker, S.D. Larson, et al., Pediatric earlobe keloids: Outcomes and patterns of recurrence, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.07.006
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[4] Marneros AG, Norris JE, Olsen BR, et al. Clinical genetics of familial keloids. Arch Dermatol 2001 Nov 1;137(11):1429–34. [5] Bayat A, Arscott G, Ollier WE, et al. Keloid disease: clinical relevance of single versus multiple site scars. Br J Plast Surg 2005 Jan 31;58(1):28–37. [6] Kelly AP. Medical and surgical therapies for keloids. Dermatol Ther 2004 Jun 1;17 (2):212–8. [7] Litrowski N, Boullie MC, Dehesdin D, et al. Treatment of earlobe keloids by surgical excision and cryosurgery. J Eur Acad Dermatol Venereol 2014 Oct 1;28(10): 1324–31. [8] Stahl S, Barnea Y, Weiss J, Amir A, Zaretski A, Leshem D, Miller E, Shafir R, Ben-Yosef R, Gur E. Treatment of earlobe keloids by extralesional excision combined with preoperative and postoperative “sandwich” radiotherapy. Plast Reconstr Surg 2010 Jan 1;125(1):135–41. [9] Crockett DJ. Regional keloid susceptibility. Br J Plast Surg 1964 Jan 1;17:245–53. [10] Ogawa R, Huang C, Akaishi S, Dohi T, Sugimoto A, Kuribayashi S, Miyashita T, Hyakusoku H. Analysis of surgical treatments for earlobe keloids: analysis of 174 lesions in 145 patients. Plast Reconstr Surg 2013 Nov 1;132(5):818e-25e.
[11] Hamrick M, Boswell W, Carney D. Successful treatment of earlobe keloids in the pediatric population. J Pediatr Surg 2009 Jan 31;44(1):286–8. [12] Bayat A, Arscott G, Ollier WE, et al. Description of site-specific morphology of keloid phenotypes in an Afrocaribbean population. Br J Plast Surg 2004 Mar 31;57(2): 122–33. [13] Lee SS, Yosipovitch G, Chan YH, et al. Pruritus, pain, and small nerve fiber function in keloids: a controlled study. J Am Acad Dermatol 2004 Dec 31;51(6):1002–6. [14] Griffith BH. The treatment of keloids with triamcinolone acetonide. Plast Reconstr Surg 1966 Sep 1;38(3):202–8. [15] Ketchum LD, Robinson DW, Masters FW. Follow-up on treatment of hypertrophic scars and keloids with triamcinolone. Plast Reconstr Surg 1971 Sep 1;48(3):256–9. [16] Robles DT, Berg D. Abnormal wound healing: keloids. Clin Dermatol 2007 Feb 28;25 (1):26–32. [17] Shen J, Lian X, Sun Y, et al. Hypofractionated electron-beam radiation therapy for keloids: retrospective study of 568 cases with 834 lesions. J Radiat Res 2015 Jul 28;56 (5):811–7.
Please cite this article as: F.A. Khan, N.A. Drucker, S.D. Larson, et al., Pediatric earlobe keloids: Outcomes and patterns of recurrence, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.07.006
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Pediatric earlobe keloids: Outcomes and patterns of recurrence☆ Faraz A. Khan ⁎, Natalie A. Drucker, Shawn D. Larson, Janice A. Taylor, Saleem Islam Division of Pediatric Surgery, Department of Surgery, University of Florida College of Medicine, 1600 SW Archer Rd. P.O. Box 10019, Gainesville, FL, USA
a r t i c l e
i n f o
Article history: Received 10 September 2018 Received in revised form 23 June 2019 Accepted 15 July 2019 Available online xxxx Key words: Earlobe Keloids Pediatric Recurrence
a b s t r a c t Purpose: Keloids occur as a result of abnormal wound healing and as many as 70% of keloids and hypertrophic scars affect the pediatric population. Earlobe keloids, similar to lesions elsewhere in the body, remain a challenging problem given the high rates of recurrence and lack of consensus regarding treatment strategy. This study aims to evaluate the outcomes and patterns of recurrences following treatment of earlobe keloids in a large cohort of pediatric patients to facilitate identification of the optimal treatment strategy. Methods: All pediatric patients who underwent surgical therapy for earlobe keloids over a 10-year period (2004–2015) were identified and demographic, clinical and outcomes data were collected. A follow-up phone survey was administered to assess rates of long-term recurrence and overall satisfaction with the treatment strategy. Data analysis was performed using Student's t-test and Fisher's exact as appropriate. Results: A total of 94 patients with 135 keloids were identified. Mean age was 14 years with slight preponderance of females (52%) and a majority (75%) of the patients were African American (AA). Mean keloid size was 2.4 cm (0.25–11 cm) with ear piercing being the most common inciting etiology. Excision with steroid injection (59%) was the most frequent treatment approach followed by excision alone (25%). During a median follow up of 2 years, keloid recurrence occurred in 27 (28.7%) patients, 19 (70.4%) underwent additional therapy with 10 (52.6%) re-recurrences noted. Median time to recurrence was 23 months, with a slightly higher recurrence rate in AA patients (32%; p = 0.26) and in those age b 10 years (50%; p = 0.11). Conclusion: This represents the largest series of earlobe keloids treatment in children. Our data suggest that recurrence rates are unaffected by the age at excision and race of the patient. Moreover, adjuncts to excisional therapy such as steroid injection, compression therapy and radiation also did not appear to influence recurrence. Given the pattern of recurrences studies with longer term follow-up are needed to assess the efficacy of treatment strategies. Type of study: Case series. Level of evidence: IV © 2019 Elsevier Inc. All rights reserved.
Any injury to the deep dermis irrespective of the mechanism is followed by wound healing which clinically manifests with the formation of a scar. [1] While scars are typically classified based on morphological appearance or histopathology, in essence clinically the appearance and characteristics of a patient's scar are simply the endpoint of genetic and environmental factors affecting the wound healing process. [2] Globally, approximately 100 million individuals per year acquire a scar following surgical/traumatic insult, and approximately 15 million are subsequently complicated by keloids and hypertrophic scars which are because of excess collagen deposition. Almost 70% of keloids or hypertrophic scars are noted to occur in children. [2] ☆ Funding Disclosure and Conflicts of Interest:No external funding was secured for this study. The authors have no financial relationships or any other conflicts of interest to disclose. ⁎ Corresponding author. Tel.: +1 352 265 8800. E-mail addresses: [email protected] (F.A. Khan), [email protected] (N.A. Drucker), [email protected]fl.edu (S.D. Larson), [email protected]fl.edu (J.A. Taylor), [email protected]fl.edu (S. Islam).
Hypertrophic scars by definition remain within the boundaries of the original injury, whereas keloids extend beyond and frequently have a delayed onset as compared to hypertrophic scars. Keloids disproportionately occur in dark skinned individuals, are associated with a familial history, and are related to hormonal changes associated with puberty. [2,4,5] These lesions are frequently symptomatic and can be associated with pruritus, pain, and tenderness. [13] Moreover, there is significant social and psychological impact on the affected patient. [6] Certain anatomic areas of the body such as the chest, shoulders, neck and ears appear to be more predisposed to the development of keloids and have important cosmetic significance. [7,12] Given the frequency of earlobe piercing associated with an approximately 2.5% risk of keloid development, earlobes are one of the most frequent locations these lesions are seen in pediatric patients. [8,9] Earlobe keloids, similar to lesions elsewhere in the body, remain a challenging problem to manage given the high rates of recurrence and lack of consensus regarding treatment strategy. Numerous treatment strategies have been proposed including surgical excision, intralesional
https://doi.org/10.1016/j.jpedsurg.2019.07.006 0022-3468/© 2019 Elsevier Inc. All rights reserved.
Please cite this article as: F.A. Khan, N.A. Drucker, S.D. Larson, et al., Pediatric earlobe keloids: Outcomes and patterns of recurrence, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.07.006
2
F.A. Khan et al. / Journal of Pediatric Surgery xxx (xxxx) xxx
steroid injection, radiotherapy, cryotherapy and cryosurgery. [6–8] Unfortunately, majority of these treatment strategies are known to be associated with frequent recurrences. [10] While numerous management options are available as discussed above there are no specific guidelines for the pediatric and adolescent population and evidence supporting or refuting each approach is either extrapolated from adult studies or relies on very small pediatric studies. [2,11] Given the paucity of data in pediatric/adolescent population, our study aims to review the outcomes of various treatment strategies, factors associated with recurrence and options for management of recurrences. 1. Methods 1.1. Patient selection Following approval by the institutional review board at the University of Florida, divisional, hospital and institutional databases were searched for pediatric patients having undergone surgical interventions for earlobe keloids over a 10-year period. The International Classification of Diseases, Ninth Revision (ICD-9) and Current Procedural Terminology (CPT) codes were used to identify all the cases treated by any service (Pediatric surgery, plastic surgery, or dermatology) 1.2. Definitions and data Patients between the ages of 1 and 19 years were identified using Current Procedural Terminology (CPT) codes 11,440–11,446 and International Classification of Diseases-9 (ICD-9) code 701.4 treated between 2004 and 2015. Patient demographics, presurgical clinical characteristics of the lesion (location, laterality and size), details of treatment strategy and postoperative follow-up data were collected. We also attempted to reach each patient by telephone and mail and administered a questionnaire (supplement 1) with an aim to evaluate keloid recurrence, additional surgical procedures and overall satisfaction with the treatment strategy. This survey instrument was not independently validated; however, focus groups within the department were used to test it prior to phone call. Each subject was sent a letter two weeks prior to attempted contact for informational purposes, and four separate attempts to call the patient were made. The rationale for the phone follow up was to increase the follow-up duration, as well as ascertain patients who may have been lost to our center, and also see the satisfaction with the results. Majority of the patients (96%) had routine postoperative follow-up (median follow-up duration of 8 months) in addition to the phone survey. 1.3. Statistical analysis Data analysis was performed using Minitab 17 Statistical Software (2010); State College, PA: Minitab, Inc. (www.minitab.com). Continuous variables were analyzed using Student's t-test, or the Mann– Whitney U test for nonparametric data. Categorical variables were analyzed using proportional statistics (Fisher's exact test). Significance was inferred at p b 0.05. 2. Results
Table 1 Patient demographics/ lesion characteristics (n = 94). Age (years)
Gender Race
Etiology Architecture
Size (cm)
Mean
14.0
Range
2–19
Male Female African American Caucasian Hispanic Asian Earrings Surgery/Trauma Nodular Pedunculated Mixed Mean Range
42 (45%) 52 (55%) 75 (80%) 12 (12%) 5 (5%) 2 (2%) 84 (89%) 10 (11%) 72 (76%) 13 (14%) 9 (9%) 2.4 0.25–11
2.2. Treatment strategy Treatment strategies utilized were (i) excision alone (n = 24), (ii) excision with intralesional injection of triamcinolone acetonide (Kenalog, Bristol-Myers Squibb, New York NY) n = 56, (iii) excision with intralesional injection of triamcinolone acetonide and compression earrings (n = 5) and (iv) excision with postoperative radiation (n = 9). (Table 2) In all cases, the technical aspects of the procedure including the choice of suture were made by the attending surgeon, but absorbable sutures were universally used. Most common was polyglactin acid (braided) suture (VICRYL R; Ethicon, Somerville, NJ) for the deep layer, while monofilament absorbable suture (MONOCRYL R; Ethicon, Somerville, NJ) was used in the superficial layer. Most patients (70%) reported being satisfied or very satisfied with the eventual cosmetic outcome based on a 5-point Likert scale (Very Satisfied, Satisfied, Neutral, Dissatisfied, Very Dissatisfied) irrespective of the treatment strategy employed. This satisfaction included those with mild recurrences as well. 2.3. Risk factors for recurrence Overall rate of recurrences in the entire cohort was n = 27 (28.7%). No statistically significant effect was noted on the rate of recurrences related to the patient's age (p = .08), gender (p = .05), race (p = 0.08), size (p = 0.09), type (nodular vs peduncular) of the lesion (p = 1.00) or treatment strategy employed (p = 0.53). When patients were stratified by age in two groups (less than or more than 10 years of age at the time of initial treatment) no differences were noted in the rate of recurrence (p = 0.11). Similarly, no differences in the rate of recurrences were noted when stratified by gender (p = 0.17) or by race (p = 0.26). 2.4. Re-recurrence Among the 27 patients who had a recurrence 19 (70.4%) underwent additional therapy for the recurrence. The following treatment strategies were used, and 10 (52.6%) re-recurrences were observed: (1) Excision alone in 2 patients (1 re-recurrence), (2) Steroids alone in 2 patients (1 re-recurrence), (3) Excision and steroids in 12 patients (6 re-recurrences) and (4) Excision with postoperative radiation in 3
2.1. Demographic data A total of 94 patients with 135 keloids were identified. Mean age was 14 (2–19) years with slight preponderance of female (52%) patients and a majority (75%) of the patients were African American (AA). Mean keloid size was 2.4 cm (0.25–11 cm), with nodular morphology being the most common and ear lobe piercing was the most common inciting etiology. (Table 1) The phone survey was attempted on all patients, with an overall response rate of 54%.
Table 2 Recurrence by treatment groups. Treatment Group
N
Recurrence N (%)
Overall Median FUP (months)
Excision alone Excision + Steroids Excision + Steroids + Compression Excision + Radiation
24 56 5 9
7 (24%) 13 (23%) 3 (60%) 4 (44%)
22 22 27 70
Please cite this article as: F.A. Khan, N.A. Drucker, S.D. Larson, et al., Pediatric earlobe keloids: Outcomes and patterns of recurrence, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.07.006
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patients (2 re-recurrences). No differences in the rate of re-recurrences were noted with the treatment strategy chosen. (Fisher's exact p = 1.0). These recurrences were reported over the duration of the study with a majority occurring within 2 years (median 23 months). No statistically significant effect was noted on the rate of re-recurrences related to the patient's age (p = 0.38), gender (p = 1.0), race (p = 1.0), or size of the lesion (p = 0.14). 3. Discussion This study, to our knowledge, is the largest series evaluating outcomes and treatment of ear lobe keloids in the pediatric population. With a median follow-up of 2-years, a higher incidence of recurrences was noted in our cohort as compared to those previously reported in pediatric patients. [11] A possible explanation for this observed difference may be the length of follow-up; our data set suggests that the median time to recurrence postexcision is six months, and therefore longer follow-ups are necessary to accurately quantify the burden of recurrence. In addition, we have likely captured patients who had secondary procedures with other providers owing to the telephone-based survey. There remains a paucity of evidence-based guidelines to optimally manage earlobe keloids in pediatric patients. Our data set demonstrates that while there was a high rate of recurrence, it was unaffected by the treatment strategy employed and patient characteristics, including race. This is a key finding, as while AAs have the highest burden of keloids, we may be selecting a subset of patients across race, gender and age that are susceptible to further keloid formation, which is suggested by the even higher rate of recurrence after a second keloid developed. An additional interesting finding of this study was that ear lobe keloids affected both genders equally, as females are more more likely to have ear lobe piercings due to a societal bias, it can be hypothesized that males may have a higher likelihood of developing ear lobe keloids. Intralesional corticosteroids are considered a main stay of therapy of keloids owing to their ease of use and high degree of tolerability. The efficacy of corticosteroids is thought to be secondary to their ability to suppress inflammation, promotion of collagen degeneration and inhibition of further collagen production. [2] The majority of data evaluating use of corticosteroids is several decades old and is limited to adult cases with the exception of one pediatric study. [11,14,15] The response rate in these studies for corticosteroids is reported as 50%–100% with a 9%–50% recurrence rate. [16] Potential for local cutaneous atrophy, hypopigmentation and pain associated with injections are other obstacles limiting repeated injections. In summary while there may be a potential benefit for adjuvant injection of corticosteroids the magnitude of benefit and optimal strategy of use are yet to be adequately defined. Our data set noted no appreciable effect in reduction of recurrence rates or cosmetic outcomes related to the use of corticosteroids. Fortunately, there were no complications related to use of intralesional corticosteroids; therefore, they may be considered as a safe adjunct to surgical therapy. There are some small series that report the use of radiation in trying to reduce the rate of recurrence [17]. Radiation theoretically works by decreasing cellular proliferation. However, the therapy may not be available at all institutions and certainly leads to an increase in the overall expense of therapy. There were 9 cases in our report where this modality was utilized. We did not see any effect on the rate of recurrences; however, given the small number in this subset no definitive conclusions can be drawn. As this cohort was selected on the basis of having undergone a surgical excision of the keloid, all had an operation performed. While surgical removal is likely to be the most common management approach especially for larger lesions, without a denominator of all ear lobe keloids presenting to our institution we cannot appropriately comment on the efficacy of non-surgical approaches. Choice of technical aspects of the procedure (selection of suture type, use of cautery, etc.) is also an interesting variable and may contribute to the risk of recurrence.
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These aspects of the surgical care, while interesting variables, however can only be adequately controlled in the setting of a prospective study. In all cases, the choice of suture was made by the attending surgeon, but absorbable sutures were universally used. Most common was polyglactin acid (braided) suture for the deep layer, while monofilament absorbable was used in the superficial layer. Similarly, the postoperative course including the use of postoperative steroids, compression earrings and radiation therapy was chosen by the patient's physician. Given the retrospective nature of this data set it is difficult to adequately control for these variables. As previously discussed, given no differences in the complications when used as adjunct, there may be a role of outpatient intralesional steroids for smaller lesions in reducing the need for surgery. Further work will be needed to assess the feasibility of this approach. Our preferred approach to pediatric ear lobe keloids is excision with closure using absorbable sutures and steroid injection, which our data set suggests at least is not inferior to any other treatment plan and is relatively simple without requiring radiation therapy. We contend this data set suggests that pediatric ear lobe keloids are a challenging condition to treat with a high rate of recurrence regardless of the treatment modality chosen. With the evidence currently available to us decision of approach should be tailored to the individual patient at the discretion of treating physician until prospectively collected data are available. There are some key limitations of this study that bear additional discussion. There are inherent biases and limitations from the retrospective nature of the study itself. The subsets of the patients in various treatment strategies were not controlled for baseline characteristics; similarly, the decisions involving the strategy employed were not controlled. Additionally, the limited number of patients in the adjuvant radiation therapy and compression device strategy limits any meaningful conclusion to be drawn pertaining to these strategies. The use of compression earrings has also not been systemically studied for children. We had limited use in our series mainly owing to patient interest and compliance; therefore, we cannot comment on its usefulness. However, despite these limitations, the strength of this report includes the duration of follow up, large number of cases in the cohorts, and evaluation of multiple recurrences with telephone follow up to include patients who sought care elsewhere subsequently. 4. Conclusion Treatment of ear lobe keloids is a challenging problem among pediatric patients given the high rate of recurrence. Our study adds to the known literature in children and notes the lack of difference with concomitant steroid use. Surgical management is safe, but patients should be made aware of the recurrence risk. Data from this study suggest that there are no marked differences in outcomes related to the surgical strategy employed; plans for patients should be tailored individually by the treating physician. Future studies will investigate the role of compression and steroids as primary therapy. While prospective studies are needed to better define the risk factors associated with recurrence, it would be reasonable because of this to consider deferring surgical intervention until the child is older, particularly among African American patients. Appendix A. Supplementary data Supplementary data to this article can be found online at https://doi. org/10.1016/j.jpedsurg.2019.07.006. References [1] English RS, Shenefelt PD. Keloids and hypertrophic scars. Dermatol Surg 1999 Aug 1; 25(8):631–8. [2] Krakowski AC, Totri CR, Donelan MB, et al. Scar management in the pediatric and adolescent populations. Pediatrics 2016 Feb 1;137(2):e20142065.
Please cite this article as: F.A. Khan, N.A. Drucker, S.D. Larson, et al., Pediatric earlobe keloids: Outcomes and patterns of recurrence, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.07.006
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[4] Marneros AG, Norris JE, Olsen BR, et al. Clinical genetics of familial keloids. Arch Dermatol 2001 Nov 1;137(11):1429–34. [5] Bayat A, Arscott G, Ollier WE, et al. Keloid disease: clinical relevance of single versus multiple site scars. Br J Plast Surg 2005 Jan 31;58(1):28–37. [6] Kelly AP. Medical and surgical therapies for keloids. Dermatol Ther 2004 Jun 1;17 (2):212–8. [7] Litrowski N, Boullie MC, Dehesdin D, et al. Treatment of earlobe keloids by surgical excision and cryosurgery. J Eur Acad Dermatol Venereol 2014 Oct 1;28(10): 1324–31. [8] Stahl S, Barnea Y, Weiss J, Amir A, Zaretski A, Leshem D, Miller E, Shafir R, Ben-Yosef R, Gur E. Treatment of earlobe keloids by extralesional excision combined with preoperative and postoperative “sandwich” radiotherapy. Plast Reconstr Surg 2010 Jan 1;125(1):135–41. [9] Crockett DJ. Regional keloid susceptibility. Br J Plast Surg 1964 Jan 1;17:245–53. [10] Ogawa R, Huang C, Akaishi S, Dohi T, Sugimoto A, Kuribayashi S, Miyashita T, Hyakusoku H. Analysis of surgical treatments for earlobe keloids: analysis of 174 lesions in 145 patients. Plast Reconstr Surg 2013 Nov 1;132(5):818e-25e.
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Please cite this article as: F.A. Khan, N.A. Drucker, S.D. Larson, et al., Pediatric earlobe keloids: Outcomes and patterns of recurrence, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.07.006