Association of Recurrence of Infected Congenital Preauricular Cysts Following Incision and Drainage vs Fine-Needle Aspiration or Antibiotic Treatment: A Retrospective Review of Treatment Options

Key Points

Question  Is incision and drainage of infected congenital preauricular cysts associated with increased rate of recurrence when compared with fine-needle aspiration (FNA) or antibiotic treatment?

Finding  In a cohort study of children with a history of preoperative infection, an increase in recurrence was found in patients undergoing preoperative incision and drainage when compared with patients treated with FNA or antibiotics alone.

Meaning  These results guide the practitioner toward more conservative treatment modalities, such as oral antibiotics or FNA, and avoidance of incision and drainage for initial management.

Importance  Treatment modalities for preauricular sinus tract infections vary.  Effort should be taken to decrease methods that lead to increased recurrence after surgical excision.

Objective  To determine whether incision and drainage (I & D) of infected congenital preauricular cysts is associated with increased rate of recurrence when compared with fine-needle aspiration or antibiotic treatment.

Design, Setting, and Participants  This was a 9-year (2006-2014) retrospective cohort study undertaken at a tertiary care pediatric hospital. Children treated for preauricular sinus tract infections were identified using the procedure code for excision of preauricular pit, cyst, or sinus tract.

Main Outcomes and Measures  Postexcision recurrence.

Results  Sixty-nine children ranging in age from 4 months to 17 years (mean age, 5.9 years) underwent excision of a preauricular cyst. Thirty-seven of 69 patients (54%) were female. Fifty-seven of 69 (83%) had a preoperative history of infection; the remainder had chronic drainage. Of children with preoperative infection, 27 were initially treated with incision and drainage (I & D), 12 were treated with fine-needle aspiration only, and 18 received antibiotic therapy alone. Overall, the recurrence rate was 8 in 69 (11.6%). Among the 27 patients with a preoperative history of infection treated with I & D, 5 lesions (18.5%) recurred, and among those who only received preoperative antibiotic therapy or fine-needle aspiration 1 in 30 lesions (3.3%) recurred (absolute difference of 15.2%; 95% CI, −1.7% to 33.6%).

Conclusions and Relevance  Among infants and children undergoing excision of preauricular cysts, a history of infection was not associated with a higher recurrence rate. There was, however, evidence to suggest that a higher rate of recurrence exists among children who had a preoperative history of infection treated with I & D. Our results suggest a more conservative treatment of infected preauricular pit and/or sinus.

Preauricular pits (PAPs) are commonly encountered congenital malformations that are often seen on examination but may not have clinical significance. Preauricular pits can arise sporadically or are inherited in an incomplete autosomal dominant fashion, with reduced penetrance and variable expressivity.¹ The incidence of PAPs varies in the literature, depending on the patient population, and has been reported to be 0.23% in studies in New York, 0.47% in studies in Hungary,² and 1.9% in studies in South Korea.^2,3 Preauricular pits are more common among people of African and Asian descent.⁴ They are theorized to be the result of an incomplete embryonic fusion of the 6 Hillocks of His.

Preauricular pit sinus tracts typically present as indolent pinpoint depressions, located along the anterior margin of the ascending limb of the helix.¹ Preauricular pits may also extend superior to the auricle, along the cymba concha, at the lobule, or posterior to the auricle.⁵ Occasionally, PAPs can become infected, manifesting as facial cellulitis or a localized abscess, and require antibiotic therapy and, at times, incision and drainage (I & D).

Surgical treatment ideally consists of complete excision of the PAP sinus tract, preferably when the tissue is not actively infected. Surgical methods and PAP recurrence rates following sinus tract excision have been documented, but the impact of recurrence after I & D remains unclear.^5,6 Incision and drainage procedures are typically avoided in acute infections because I & D may increase risk of recurrence after definitive tract removal. Results from studies investigating whether preexcision I & D is associated with increased postexcision recurrence rates, however, have been mixed.^5,6 Hence, further investigation is warranted.

The purpose of this study is to investigate whether I & D performed prior to a staged excision is associated with increased rate of recurrence of PAPs. Our hypothesis is that I & D of an infected preauricular cyst is not associated with an increased rate of recurrence, compared with fine-needle aspiration or antibiotic treatment alone.

After obtaining approval from the Children’s National Medical Center’s institutional review board, we undertook a 9-year retrospective review (January 1, 2006, to December 31, 2014) of all children with PAPs. Study patients were identified using the Current Procedural Terminology (CPT) procedure code for excision of PAP, cyst, or sinus tract. Each medical record was individually reviewed. The data extracted included sex, age at surgery, preoperative antibiotic treatment, preoperative fine-needle aspiration, preoperative I & D, time from surgery to recurrence, size of specimen, intraoperative rupture, size of excised tract, and final pathology reports. All cysts were excised using the same technique: lacrimal probes guided a wide excision of surrounding soft tissue along with a small section of cartilage. Recurrence was defined as a persistent preauricular mass or drainage from the area. We excluded children who did not have a cyst confirmed by pathology reports. Statistical analysis was completed using the 2-sided Fisher exact test to compare the rates of recurrence with or without preoperative infection and with or without I & D, and a Kaplan-Meier curve examining percentages of recurrence-free patients. Results were deemed to be significant if P < .05.

A review of CPT codes yielded 69 patients who underwent excision during the study period, with pathology report–confirmed anomalies. The average age of the patients was 5.9 years (range, 4 months to 17 years), and 37 of 69 of patients (54%) were female. Fifty-seven of the patients (83%) had a history of infection prior to the scheduled surgical excision, and 12 patients had chronic drainage as the indication for surgery (17%). Of the 57 patients with preoperative infection, 27 were treated with I & D, 12 underwent fine-needle aspiration, and 18 were treated medically with antibiotics. See Figure 1.

The recurrence rate in our study was found to be 8 in 69 (11.6%). In patients with a preoperative history of infection treated with I & D, 5 of 27 lesions (18.5%) recurred, and among those who only received preoperative antibiotic therapy or fine-needle aspiration 1 of 30 lesions (3.3%) recurred (absolute difference of 15.2%; 95% CI, −1.7% to 33.6%). There was a 15.2% difference in recurrence between patients treated with I & D and those who received preoperative antibiotic therapy or fine-needle aspiration, and this difference could be as great as 34.0%. The data are also compatible with a slight (2%) decrease in recurrence among those treatment without I & D. A multivariate analysis comparing age, sex, presence of preoperative infection, and size of the specimen did not yield any other significant parameters influencing recurrence. Kaplan-Meier analysis displays a fairly steep curve showing rapid recurrence among those who had I & D compared with those who had no I & D (Figure 2).

In our study, we retrospectively compared postoperative pit recurrence rates in patients undergoing preoperative I & D with those undergoing antibiotic treatment or fine-needle aspiration. In patients who had a preoperative history of infection that required I & D prior to definitive removal, 5 of 27 (18.5%) lesions recurred. The data are inconclusive regarding the relationship between type of treatment for preoperative infection and recurrence but do suggest that recurrence is greater for those who undergo I & D.

This finding is supported by Tang et al,⁴ who compiled a retrospective analysis of 71 patients over 5 years at a single tertiary care facility. They found an overall recurrence rate of 14.1%. Among the 12 patients who underwent drainage for an abscess before the definitive surgery, 4 (33.3%) experienced recurrence. Among the 39 patients who did not have an abscess, only 6 (15.3%) experienced recurrence, with an absolute difference of 18% (95% CI, −9% to 50%). Furthermore, among the 13 sinuses that were actively infected at the time of definitive surgery, 4 lesions (30.7%) recurred, while among the 38 noninfected sinus tracts, only 6 recurred (15.7%), for an absolute difference of 15% (95% CI, −11% to 47%). Tang et al⁴ surmised that complete excision of the sinus tract is more difficult owing to the fibrotic and edematous changes associated with postinfection inflammation and scarring.⁵ Similar findings were reported in a study by Gur et al,⁶ who examined 165 patients over the course of 14 years. In patients who underwent surgical drainage of an abscess before sinus tract excision, 16.7% recurred, whereas in the patients who did not have incision prior to the procedure only 8.16% recurred (P = .25; odds ratio, 2.25). The result of Gur et al,⁶ however, failed to reach statistical significance; the authors attributed the statistical insignificance to small sample size. As in the study by Tang et al,⁴ the article hypothesizes that changes from I & D distort the normal tissue structure and make definitive sinus tract surgery more complex, leading to higher recurrence rates.⁶

Indeed, histological studies show that there is a branching pattern to the preauricular cyst. In 2009, Dunham et al⁷ examined the relationship between the epithelial tract of excised PAPs and the adjacent auricular cartilage. They⁷ stated that the epithelial tracts commonly arborize with narrow, tortuous paths, and their findings suggest they are found less than 5 mm from the cartilage in 50% of the specimens.

Similarly, in 2012, a study by Gan et al⁸ demonstrated that PAP epithelialization does not merely occur along a single tract. Gan et al⁸ divided PAPs into 2 excision groups: 114 participants were placed into an excision under microscope group and 94 participants into a methylene blue dye and probe group. Surgical excision with microscope guidance had significantly lower recurrence rate (0.9%) compared with surgical excision with methylene blue dye and probe guidance (4.3%), with an odds ratio of 28.4 (95% CI, 1.22-659.99). The lower recurrence rate was attributed to clear identification of the PAP and magnified surgical planes.⁹

Finally, Lam et al⁹ also found that preauricular cysts are multibranched. They compared wide local excision of PAPs with simple fistulectomy. Finding that wide local excision leads to lower recurrence rates, the authors stated that preauricular sinuses are multibranched and ramifying within the soft tissue and can be difficult to identify as a result of the inflammation from previous abscess formation.

Yeo et al¹⁰ studied factors contributing to recurrence after surgery among 191 patients with preauricular sinus tract infections in South Korea. The overall recurrence rate was 4.9%. In contrast to our findings and proposed hypothesis, they found an absolute difference in recurrence rate of only 5% (95% CI, −2.5% to 12%) between patients with a history of drainage compared with those who did not have drainage. They found that recurrence increased only if the procedure was performed under local anesthesia, which was attributed to the tortuous course and pattern of arborizations frequently associated with PAP tracts and the difficulty of effective dissection on conscious patients.¹⁰ Another article, by Huang et al,¹¹ stratified PAP operative procedures into 3 groups based on degree of inflammation. In group 1, PAPs with mild inflammation, lesions were excised with a simple sinusotomy; group 2, lesions with moderate inflammation were excised with a local wide excision; and in group 3 they was removed in a “figure 8” method. Their overall recurrence rate was 7.34% (8 of 109). Of the 34 sinuses drained for an abscess, 5 (14.7%) had recurrence, while 3 of the 65 (4.6%) that did not have previous I & D recurred for an absolute difference of 10.1% (95% CI, −3% to 28%). This rate is similar to ours. They found presence of preoperative I & D was not significant. This could be related to their method of a figure 8 excision, which was composed of 2 wedge resections down to the temporalis fascia for an extended fistulectomy. This widened approach could capture fragmented tracts after I & D.

Similar to PAPs, other congenital malformations have been associated with postoperative recurrence. The relationship of malformation recurrence in the setting of preoperative I & D has also been investigated in these other lesions. In a study by Simon and Magit,¹² no significant relationship between preoperative I & D of thyroglossal duct cysts and cyst recurrence was found. Their study,¹² however, found that the rate of recurrence among the 49 patients with infection was 20%, the rate of recurrence among the 71 without infection was 4% for an absolute difference of 16% (95% CI, 3%-31%). The difference between these findings and our findings in PAPs could be attributed to the multibranched, arborized nature of the PAP in contrast to the thyroglossal duct cyst, which forms along a single tract.¹²

Unlike other studies, our study included patients who underwent preoperative fine-needle aspiration in our analysis. No difference in recurrence rate was noted between patients undergoing fine-needle aspiration and I & D or between patients undergoing fine-needle aspiration and those treated with antibiotics alone. It is likely, however, that this lack of relationship is due to small population size, so further investigation is warranted. In our analysis we compared patients undergoing I & D with patients undergoing either fine-needle aspiration or antibiotic therapy. The decision to combine fine-needle aspiration and antibiotic groups into 1 group is justified because fine-needle aspiration is less likely to disrupt the sinus tract when compared with I & D.

The genetic pattern of dominant inheritance may often prompt the parent or guardian to avoid interventions that may not seem to be urgent or necessary. In addition, the progression of the infection in the PAP may be very rapid and treatment can become complex if the patient is in pain and or has extensive swelling, abscess formation, and expansion of the fluid collection on the anterior aspect of the face.

Thus, this study has the following limitations: because patients were not randomized, the decision to proceed with I & D, fine-needle aspiration, or antibiotic therapy may create a source of bias. The retrospective nature of the study limits the variables available for analysis. In addition, this study has a small sample size, which could reduce the likelihood that a statistically significant result reflects a true effect. Severity of infection is one confounding variable that might require drainage prior to definitive excision. Most treatment modalities prior to operative incision and drainage in our review were managed by primary care and emergency medicine physicians. At this time, treatment guidelines are not available to influence clinical decision making. It is the aim of future endeavors to establish such treatment protocols. As demonstrated in our study, attempts should be made to treat the PAP with antibiotics and fine-needle aspiration.

Preoperative I & D of the infected PAP or sinus, is associated with increased incidence of recurrence after surgical excision. The absence of a history of infection enhances the chances that the excision of the PAP will be complete and a long-term success. Our results guide the practitioner toward more conservative treatment modalities, such as oral antibiotics or fine-needle aspiration, and avoidance of I & D treatments.

Corresponding Author: Holly Rataiczak, DO, Walter Reed National Military Medical Center, 8901 Wisconsin Ave, Bethesda, MD 20889 (rataichm@gmail.com).

Accepted for Publication: August 7, 2016.

Published Online: October 27, 2016. doi:10.1001/jamaoto.2016.2988

Author Contributions: Dr Rataiczak had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.

Concept and design: Reilly.

Acquisition, analysis, or interpretation of data: Rataiczak, Lavin, Levy, Preciado, Reilly.

Drafting of the manuscript: Rataiczak, Lavin.

Critical revision of the manuscript for important intellectual content: Rataiczak, Lavin, Levy, Preciado, Reilly.

Statistical analysis: Rataiczak, Lavin, Levy, Preciado.

Administrative, technical, or material support: Rataiczak, Reilly.

Study supervision: Lavin, Preciado, Reilly.

Conflict of Interest Disclosures: All authors have completed and submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest, and none were reported.

Previous Presentation: This study was presented at Combined Otalaryngology Spring Meetings/American Society of Pediatric Otolaryngology; April 24, 2015; Boston, Massachusetts.