Recurrent suppurative thyroiditis due to pyriform sinus fistula: a case report

Acute suppurative thyroiditis is a rare disease, particularly in childhood. We present a case with recurrent acute suppurative thyroiditis due to a pyriform sinus fistula originating from the fourth branchial pouch. The typical symptoms of a piriform sinus fistula are recurrent left-sided pain and swelling of the neck with signs of acute bacterial inflammation. Diagnosis should be made by high resolution ultrasound, barium meal studies and endoscopic examination. During acute exacerbations treatment with antibiotics is indicated, but permanent cure can only be attained by complete fistulectomy.     

Pyriform sinus fistula

Pyriform sinus fistulae/sinuses are rare causes of recurrent cervical abscess, especially on the left side. They can also present as acute thyroiditis. Treatment in the form of simple incision and drainage is invariably unsuccessful, and the entity may be confused with the residual tract of a second branchial arch anomaly. We report a case of pyriform sinus fistula, and believe that this is only the second case report in India. We feel that greater awareness can lead to proper and appropriate diagnosis of this anomaly.     

Pyriform sinus fistula

Pyriform sinus fistulae/sinuses are rare causes of recurrent cervical abscess, especially on the left side. They can also present as acute thyroiditis. Treatment in the form of simple incision and drainage is invariably unsuccessful, and the entity may be confused with the residual tract of a second branchial arch anomaly. We report a case of pyriform sinus fistula, and believe that this is only the second case report in India. We feel that greater awareness can lead to proper and appropriate diagnosis of this anomaly.     

新生儿梨状窝瘘的诊治特点

目的 阐述新生儿梨状窝瘘这一少见疾病的诊治特点.方法 收集我院2001年7月至2010年1月收治的9例新生儿梨状窝瘘病例,分析其发病年龄、临床表现、辅助检查及临床治疗经过,总结其诊治特点和预后.结果 9例梨状窝瘘均为左侧.均以颈部囊性肿块就诊.8例为新生儿时期出现症状,1例为产前发现.5例颈部增强CT提示肿块中含有气体.新生儿时期手术治疗7例.随访时间6个月至9年,患儿临床均无反复感染或复发迹象.结论 新生儿的梨状窝瘘临床表现不同于儿童,常因颈部无痛性、囊性肿块就诊,缺少反复脓肿切开引流史.超声或CT检查中常可发现囊肿中含有气体影.新生儿早期手术瘘管结扎率高,部分需要胃镜辅助.治疗安全性可靠,术后复发率低且预后良好.     

新生儿梨状窝瘘的诊治特点

目的 阐述新生儿梨状窝瘘这一少见疾病的诊治特点.方法 收集我院2001年7月至2010年1月收治的9例新生儿梨状窝瘘病例,分析其发病年龄、临床表现、辅助检查及临床治疗经过,总结其诊治特点和预后.结果 9例梨状窝瘘均为左侧.均以颈部囊性肿块就诊.8例为新生儿时期出现症状,1例为产前发现.5例颈部增强CT提示肿块中含有气体.新生儿时期手术治疗7例.随访时间6个月至9年,患儿临床均无反复感染或复发迹象.结论 新生儿的梨状窝瘘临床表现不同于儿童,常因颈部无痛性、囊性肿块就诊,缺少反复脓肿切开引流史.超声或CT检查中常可发现囊肿中含有气体影.新生儿早期手术瘘管结扎率高,部分需要胃镜辅助.治疗安全性可靠,术后复发率低且预后良好.     

Suspicion of prenatal pyriform sinus cyst and fistula: a case report

There has been no report describing suspected prenatal pyriform sinus (PS) cyst and fistula. We report a case suspected by prenatal ultrasonography and fetal MRI. A large cystic mass was found in the left neck of the fetus. After the baby was born, preoperative laryngoscopic catheterization of the fistula tract was used to confirm the diagnosis and greatly facilitated the identification and excision of the PS cyst and fistula.     

儿童梨状窝瘘反复发作的原因与胃镜辅助手术

目的分析儿童梨状窝瘘反复发作的原因，介绍胃镜辅助手术方法及其优越性。方法12例患儿。男8例，女4例，年龄2．5～9岁；曾行左颈部瘘管或囊肿切除术8例，左颈部反复感染者4例；吞钡造影、B超、CT、同位素扫描等检查诊断为梨状窝瘘。12例均行胃镜辅助瘘管切除术。取此前未经胃镜辅助而直接手术的8例临床资料作对照。结果经胃镜辅助切除瘘管，平均手术时间1h 35min，术后恢复顺利，随访无复发。对照组手术时间长，2例复发。结论缺乏对梨状窝瘘胚胎发生和病理过程的了解是治疗失误的主要原因，胃镜辅助手术是一种简便、有效的治疗手段。     

Acute suppurative thyroiditis in children secondary to pyriform sinus fistula

Acute suppurative thyroiditis (AST), a potential complication of pyriform sinus fistula (PSF), is a rare clinical condition as the thyroid gland is remarkably resistant to infections. Lack of awareness of the entity contributes to the rarity and frustrating recurrences. We performed a retrospective review of all cases of AST due to PSF treated at our institution over a 10-year period. The clinical data, investigations, operative findings and procedures, microbial culture reports and follow-up were recorded and analyzed. Between January 1997 and September 2006, 12 cases (8 males and 4 females) of AST due to PSF were treated. Nine patients (75%) underwent successful complete excision, seven of whom had initial incision and drainage procedures. In three patients (25%) with recurrence, one underwent complete excision at a later procedure, one patient had multiple recurrences with six incision and drainage procedures and two failed attempts of excision of PSF before final successful complete excision. The third patient is awaiting re-excision of the PSF tract. All patients, except the one awaiting re-excision, are well with no further recurrences during the follow-up period that ranged from 18 to 96 months (median, 46.5 months). AST due to PSF is a challenging entity in terms of diagnosis and management as recurrences are common despite meticulous dissection. High index of suspicion and radiological investigations such as barium studies and computed tomography scan aid in the delineation and excision of the fistulous tract.     

Imaging studies of pyriform sinus fistula

BACKGROUND: Pyriform sinus fistula (PSF) refers to a persistent embryologic third or fourth pharyngeal pouch, which typically presents as a congenital sinus tract that originates from the pyriform sinus. The sinus tract is often diagnosed by a barium study or direct endoscopic inspection. Utilization of advanced imaging studies, including ultrasound (US), computed tomography (CT), and magnetic resonance imaging (MRI), may aid in the diagnosis of this disease entity. OBJECTIVES: To review the imaging findings of PSF and demonstrate the value of various cross-sectional imaging (US, CT, and MRI) in the diagnosis of PSF. MATERIALS AND METHODS: PSF in five children was verified surgically. Preoperative barium esophagography, US, CT, and MRI were performed selectively in these patients. The clinical and imaging findings are reviewed retrospectively. RESULTS: Barium studies demonstrated the sinus tract in all five patients. US, CT, and MRI demonstrated an associated inflammatory process. By utilizing the trumpet maneuver, the presence of sinus tract was evident in two patients during US. The sinus tract is also demonstrated by CT in another patient. CONCLUSIONS: Although barium esophagography is advantageous in demonstration of the sinus tract in PSF, US and CT are also capable of showing the sinus tract. The extent of inflammatory process related to PSF is better delineated by US, CT, and MRI.     

Successful endoscopic ablation of a pyriform sinus fistula in a child: case report and literature review

Recurrent thyroid infections are rare in children. When present, patients should be evaluated for anatomic anomalies such as pyriform sinus fistulae. We describe a 12-year-old girl with history of recurrent thyroid abscesses secondary to a pyriform sinus fistula and managed with concurrent endoscopic ablation and incision and drainage.     

儿童梨状窝瘘41例

目的提高对儿童梨状窝瘘的认识，介绍相关诊断与治疗进展。方法报告41例梨状窝瘘病例，男16例，女25例，年龄9个月至12岁3个月；左侧40例，右侧1例；33例行颈部超声显像，20例行CT柃查，29例行甲状腺核素显像，39例行食管吞钡造影。28例手术治疗，其中20例应用胃镜辅助，并对37例进行随访，随访时间18个月至17年6个月。结果超声检查显示：32例病变侧甲状腺实质不均质占位或软组织实质不均质占位，与甲状腺关系密切；CT检查提示：病变侧颈部炎性肿块或合并甲状腺内炎性占位性病变，其中5例直接诊断为梨状窝瘘；核素显像提示：26例甲状腺左叶、特别是上极放射性稀疏，2例呈“冷结节”；食管吞钡检查显示：左侧或右侧梨状窝底部有垂直下行的细小瘘管。10例单纯切除术中8例治愈，2例复发，经胃镜辅助治愈；20例经胃镜辅助切除病例中，2例复发；13例未手术，其中9例获随访，5例自愈或未发作，4例仍反复发作。结论儿童梨状窝瘘炎症消退后食道吞钡检查可明确诊断，CT、B超及同位素检查在梨状窝瘘的诊断中也起着非常重要的作用，手术切除瘘管是主要的治疗方法，关键在于完整切除或消灭瘘管；胃镜辅助检查有助于瘘管的寻找，从而保证瘘管的完整切除，是一种简便、有效的治疗手段。     

Acute suppurative thyroiditis in young children

We report two children with acute suppurative thyroiditis (AST). They presented with typical features of AST, which include fever, painful goiter and biochemical euthyroidism. An anatomical defect predisposed to thyroid infection, pyriform sinus fistula, was identified in one patient. Both patients responded well to surgical pus drainage and antibiotic treatment. Anatomical defects must be sought in all children with AST to perform specific surgical treatment and prevent recurrent infection.     

Branchial fistula: CT manifestations

A patient with a recurrent left neck abscess was found to have a branchial fistula from the pyriform sinus. This uncommon anomaly was demonstrated by CT and barium swallow. The embryology, pathology and clinical features of this lesion are discussed and contrasted with those of the branchial cyst.     

"Massive hemoptysis" and shock after fever and cough北大核心CSCD

11岁男性患儿,因间断发热15 d,咳嗽10 d,“咯血”7 d入院。患儿15 d前以发热咳嗽起病,伴左侧颈部疼痛,抗生素治疗有效。病程中突然出现大“咯血”致休克,后经纤维支气管镜检查发现左侧梨状窝内瘘口并持续渗血,结合颈部及血管影像学检查考虑为先天性梨状窝瘘感染并颈部脓肿致颈内静脉受损血栓形成,通过低温等离子消融梨状窝瘘封闭术治疗后好转,随访1年半未反复。检索文献未发现梨状窝瘘感染引起大出血休克报道,该文总结此例患儿的临床特点及诊治经过,为早期诊断该类疾病及其并发症防治提供参考依据。     

Cervical infection secondary to pyriform sinus fistula of branchial origin

Complete third branchial arch anomalies are rare and have been described only in case reports, affecting mainly children and typically presenting as a cervical inflammatory process. Anomalies of the third and fourth branchial apparatus, though rare, usually present as sinuses/incomplete fistulas of pyriform sinus or recurrent suppurative thyroiditis. A 6‐year‐old girl presented with a small opening on the left side of her anterior neck, which had been present since birth and was associated with recurrent infection. She had no history of incision and drainage of swelling. Computed tomography with contrast injection into the cervical opening revealed a fistulous tract extending from the cervical neck skin to the pyriform fossa. Complete excision of the fistulous tract and left hemithyroidectomy were performed. There was no recurrence at 22 months of follow‐up.     

Pyriform sinus fistula appearing as a neck tumor in the neonatal period: a case report

: Pyriform sinus cyst and fistula is a relatively rare tumor of the neck, even less is the neonatal period. We experienced a case of this one, and preoperative diagnosis was made by using endoscopy and cine-esophagography. A dyeing method that confirmed the fistula tract of cyst lead us to successful resection. Accepted: 1 October 1998     

儿童颈部梨状窝瘘诊治进展

梨状窝瘘多以小儿反复发作的单侧（左侧为主）颈部炎性脓肿或急性化脓性甲状腺炎等症状就诊,以往在治疗上多为反复颈部脓肿切开引流及联合多种抗生素对症缓解症状或是开放性常规分离瘘管手术寻求根治,但复发率较高。该文通过总结近年梨状窝瘘相关诊断和治疗进展,旨在强调对于疑似患儿需及时行超声、CT、上消化道钡餐等检查,必要时做内镜检查,治疗上在非急性期以内镜辅助下导管置入或亚甲蓝内口注射行梨状窝瘘切除术可明显减少复发率。     

支气管镜在儿童气管食管瘘全程手术管理中的应用研究

目的 探讨支气管镜在儿童气管食管瘘术前、术中和术后的临床应用价值.方法 对14例疑似气管食管瘘患儿(其中1例支气管食管瘘)尝试应用支气管镜进行手术全程管理.结果 术前对4例患儿应用支气管镜检查明确诊断为气管食管瘘;运用相同方法确诊1例患儿为气管窦道,排除了气管食管瘘;利用支气管镜引导下气管插管技术,成功封堵1例因误食烧碱造成左主支气管-食管瘘.在术中应用"支气管镜透射照明"法和"导丝环"法精确定位3例气管食管瘘,减少了手术创伤和探查瘘管的时间.在术后应用支气管镜发现2例气管食管瘘复发和2例存在气管软化;支气管镜还充当了一次"超细胃镜",将导丝引入食道,然后进行食管狭窄的球囊扩张.结论 支气管镜在儿童气管食管瘘诊治中有很大的临床应用价值,可应用于气管食管瘘的全程管理.     

Recurrent pseudothyroiditis and cervical abscess. The fourth branchial pouch's role. Apropos of 16 cases

Among the causes of inflammatory swelling of the neck in children, the "cysts" and ducts joining in the hypopharynx deserve being individualized. This unrecognized pathology is indeed responsible for suppurative pseudothyroiditis or cervical abscesses relapsing in spite of adequate antibiotic treatment and incision-drainage. X-ray films may help as they often show the fistula. Diagnosis relies on hypopharyngoscopy. This investigation only may assess the origin of this "internal fistula" by showing the mucous opening of the bottom of the pyriform sinus, from which sometimes springs some pus when pressing the neck. The clear left predominance of this canal and its junction between the superior and inferior laryngeal nerves suggest that its origin could be the 4th branchial pouch. Its treatment consists of complete surgical excision, which avoids relapses.