1. Operation procedure
Based on our practical experience, the mSis procedure has been optimized in the following details: the resection at the cartilaginous joint of hyoid bone, core out to the foramen cecum, and the resection and ligation techniques of the fistula near the foramen cecum. These detailed modifications realized the complete resection of the lesions while reducing injuries, further lowering the recurrence rate.
1.1 Operation procedures of hyoid bone. In the traditional modified Sistrunk surgery, the central body or middle third of the hyoid bone was transversely cut with the bone cutting forceps on both sides of the hyoid body. The operation procedure adapted in the present study used the needle electrotom to divide the cartilaginous junction between the hyoid body and the greater horn by combining the advantages of the anatomical characteristics in childhood. An overview of the hyoid bone discovered that until middle age, the connection between the body and the greater cornu is fibrous[9]. Therefore, for pediatric TGDC, the needle electrotom can easily accomplish the division of the cartilaginous junction between the body and the greater horn without unnecessary bleeding. A similar conclusion was reported by Ryu et al. after studying 26 pediatric patients[10].
1.2 The tracking and anatomy of foramen cecum. It should be emphasized the importance of maintaining the en bloc principle without trying to identify a sinus tract in the region superior to the hyoid bone[11], and the cuff column of about 1-cm tissue volume was completely separated to the mucosal of lingual foramen cecum level, encompassing mylohyoid muscles and genioglossus. According to Sistrunk, the cuff diameter of tissues around the lingual duct separated along the suprahyoid bone is about 3 mm[8]. However, Horisawa et al. found extensive branches at suprahyoid and infrahyoid areas in the three-dimensional reconstruction of the thyroglossal duct[12]. In 1950, Boyd proposed that the rupture of the thyroglossal duct shortens and thickens the proximal portion, and its epithelium resembles that of the tongue and frequently re-develops a lumen from which an extensive system of branches is grown[13]. Soucy and Penning reviewed 44 TGDC pathological sections and demonstrated that the thyroglossal duct delivers many dendritic branches to the surrounding tissues[14], such that the resection range covered these hidden branch fistulae. Since the separation is deep to the mucosal layer of the foramen cecum, the cold knife should be used for dissection to avoid the possible risk of pharyngeal fistula caused by electrotom resection, affecting healing and facilitating visual observation to confirm the fistula mucosal epithelium in the resected tissues. This procedure does not enter the pharyngeal cavity, avoiding the purse string suture and adopting figure-of-eight suture to consolidate the stumps. Since fully bending backward position would cause glossocoma and tracheal intubation would be close to epiglottis and the root of the tongue, the suture should not be extremely deep.
2. Recurrence and the risk factor
The sample size of previous case-control studies on the application of mSis in TGDC ranged from 24–251, and the recurrence rate was 1.6–16.1%[15, 16]. The present study has the largest sample size involving retrospective data of mSis in children, and the recurrence rate (2.30%, during 15–156-months follow-up) was lower than the average reported by other studies. Statistical analysis revealed that the distinct lesion boundary, surgical history, and lesion diameter were independent risk factors for disease recurrence.
2.1Leision boundary. In this study, the indistinct lesion boundary suggested that infection can lead to local inflammatory or cicatricial adhesions. Moreover, the duration of surgery was 2 weeks after the infection was controlled. However, the above findings indicated that although the skin surface indicated controlled infection, the internal peripheral lesions might still be in the inflammatory recovery stage of the infection. When TGDC is in the infection stage, the tissue is edematous and congested, and the fistula wall and small branches have poor elasticity and toughness and are easy to rupture when separated. Although several studies suggested an association between infection and recurrence[17-19], some reports do not support this association[20, 21]. The present findings demonstrated that the preoperative occult infection stage might affect the complete resection of the small branches and fistulas during the procedure, leading to recurrence.
3.2 Secondary surgery. For patients with surgical history of neck masses, local inflammatory or cicatricial adhesions, unclear anatomical level, and blind operation poses significant challenges for a complete resection of the cysts and fistulas in secondary surgery, leading to residual and recurrent fistulas and the formation of small branches. Kim found that even if experienced doctors used the classic Sistrunk procedure to resect TGDC, the mean recurrence rate was >4%, and re-surgery failed in 33% of recurrent patients[11]. In the present study, the failure rate of re-surgery in the recurrent patients was 5.6% (2/36), further suggesting that in children undergoing non-standard surgeries, such as simple cyst resection and non-dissection of hyoid bone, intraoperative procedures should be detailed and thorough. Perkins et al. demonstrated that if the hyoid bone was not dissected in the first surgery, re-surgery after recurrence would increase the risk of second recurrence[22]. Some studies reported a successful experience of the central neck dissection in the treatment of recurrent TGDC, expanding the resection range of Sistrunk procedure to include the anterior tracheal soft tissues to realize the en bloc resection; the width of the resected tissue volume of the strap muscles and hyoid bone was 3-4 cm and that of the foramen cecum reached 2-3 cm[23]. Moreover, no recurrence or serious complication was observed after the central neck dissection, but risks, such as damage to the internal carotid artery, vagus nerve, and entry into the pharyngeal cavity could not be ignored. These findings provided a reference to improve the efficacy in the treatment of children with recurrent and refractory TGDC after repeated surgeries.
3.3 Leision size. In addition, some researchers proposed a correlation between the cyst size and recurrence[21]. Clinically, the volume of the cyst does not change or changes slowly. If the cyst is infected, the volume increases in a short time but is reduced rapidly when the infection is controlled. The present study found that lesion diameter (>2 cm) is a major risk factor for recurrence. Thus, it is speculated that large cysts are prone to rupture during surgery and leakage into the surrounding tissues, resulting in indistinct boundaries. On the other hand, some children may still be in the recovery stage post-infection.
4. Limitations.
Nevertheless, the present study has several limitations. First, due to the limited sample size, combined with the lower recurrence rate, the sample size of the recurrence and the non-recurrence groups was significantly different, causing bias in the results. Second, the follow-up time was limited. Although the minimum follow-up time of this study (15 months) was significantly longer than the recurrence time (<7 months) of most recurrent children (8/9), there may be potential relapses that may not have been found. Third, the duration for the case included in the present study was prolonged, while the experience of the mSis procedure was accumulated gradually. During continuous unification of surgical details, the surgery-related habits of different surgeons also have a potential impact on the findings. Fourth, for children with recurrence after undergoing mSis procedure, constant attention and investigation are warranted for developing therapeutic regimens, including re-resection, ultrasound-guided local injection of hardening agents, and endoscopic resection of masses at the tongue root (for those with secondary lingual TGDC), to accumulate experience.