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.     

Thyroiditis. Differentiation of acute suppurative and subacute. Case report and review of the literature

Acute suppurative thyroiditis is rarely seen during childhood. The classic clinical features of this illness (fever, neck pain, and a swollen, tender mass over the thyroid gland) can differentiate acute thyroiditis from the more common subacute thyroiditis. In less typical cases, however, this distinction can be difficult. An adolescent male presented with a swollen, tender thyroid gland. Atypical laboratory findings and the lack of fever and toxicity delayed the diagnosis and treatment of acute suppurative thyroiditis. A review of the pediatric literature summarizes clinical and diagnostic features valuable in the differential between acute suppurative thyroiditis and subacute thyroiditis in childhood. The important contribution of fistulae between the piriform sinus and thyroid gland to the pathogenesis and acute suppurative thyroiditis is emphasized. Such a fistula should be sought in every patient in whom this entity is diagnosed.     

Acute suppurative thyroiditis in children

BACKGROUND: Acute suppurative thyroiditis in children is rare and is often related to a pyriform sinus fistula or thyroglossal duct remnant, especially when it is recurrent. METHODS: From January, 1985, through December, 2000, 15 children with acute suppurative thyroiditis were treated. Their clinical, laboratory and radiologic findings were reviewed and analyzed. RESULTS: There were 8 girls and 7 boys, with a mean age at diagnosis of 6.1+/-2.9 years (range, 1.5 to 9.8). A thyroid mass was present on the left in 13 and on the right in 2 (P < 0.05). Fever, neck pain and swelling were the most common symptoms and signs. Seven patients (46.7%) had recurrent disease. Needle aspiration for Gram stain and bacterial cultures were done, and pathogenic organisms were identified on culture in 8 patients but were found only on Gram stain in 2 patients. In one-half of the patients with positive cultures, mixed pathogens were found. The most common organisms isolated were streptococcal species (50%). Barium esophagography was performed in all patients, and 5 (33.3%) had a pyriform sinus fistula on the left. Only 1 of the recurrent patients had a fistula. Thyroid scans were performed in 13 patients, of whom 12 (92.3%) had decreased radioactive uptake. Thyroid function tests were normal in all 15. CONCLUSIONS: Acute suppurative thyroiditis is usually caused by oropharyngeal flora, resulting in mixed pathogens on culture. Broad spectrum antibiotics should be given once cultures have been obtained. Imaging studies might be helpful in the diagnosis of acute suppurative thyroiditis.     

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.     

Acute thyroiditis caused by Eikenella corrodens and abnormality of the left pyriform sinus

A case of suppurative thyroiditis is reported. The initial course was insidious and mimicked De Quervain subacute thyroiditis. The abscess was surgically drained. It contained numerous Eikenella corrodens bacilli. Fibroscopy of pharyngo-laryngeal region showed that the left pyriform sinus was abnormal. Surgical removal of a fistula of the fourth branchial pouch was performed in order to prevent recurrence of the thyroiditis.     

Light guided procedure for congenital pyriform sinus fistula; new and simple procedure for impalpable fistula

Pyriform sinus fistula causes acute suppurative thyroiditis, and there is a risk of recurrence if the sinus tract is not excised completely. The tract should be dissected as high as possible toward the pyriform fossa. We report our devised technique “the light guided procedure” for the impalpable fistula. A bronchoscope was inserted into the pyriform sinus fistula with the help of a gastrofiberscope positioned at the larynx. The tract of the fistula was identified with the help of the light from the bronchoscope. We were able to reach the proximal end of the fistula, directly. This procedure is easy, safe, and minimally invasive.     

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。     

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

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.     

Subacute thyroiditis in children: patient report and review of the literature

Subacute thyroiditis is extremely rare during childhood. We treated a 10 year-old prepubertal girl who presented with typical clinical features of subacute thyroiditis. Considering this case and previous reports in children, we characterized the clinical features of this disorder as it occurs in childhood. In contrast to the predominance of subacute thyroiditis in adults, the incidence of subacute thyroiditis is lower than that of acute suppurative thyroiditis in children. This may cause difficulty with differential diagnosis, particularly when leukocytosis is present or the lesion is localized to the left lobe. Ultrasonography can be helpful in differentiation.     

Differences in the characteristics and management of pyriform sinus fistula between neonates and young children

Purpose  

Pyriform sinus fistula (PSF), which originates from the third or fourth branchial pouch remnant, is relatively rare. In young children and adults, it is characterized by lateral neck infections and suppurative thyroiditis, while neonatal PSF presents as a large cervical cystic mass and causes respiratory distress. We hypothesized that the characteristics and management of PSF may differ between neonates and young children.     

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.     

Acute suppurative thyroiditis as a rare complication of aggressive chemotherapy in children with acute myelogeneous leukemia

Acute suppurative thyroiditis (AST) is quite rare, even in immunocompromised patients. The authors describe 2 cases of AST during aggressive chemotherapy for acute myelogeneous leukemia (AML). They were treated with aggressive combination chemotherapy and achieved complete remission. After several courses of chemotherapy, they developed fever and pain in the region of the thyroid gland. Laboratory tests showed hyperthyroidism and elevated levels of thyroglobulin and C-reactive protein. Ultrasonography revealed hypoechoic areas in the thyroid gland. A diagnosis of AST was made. Bacterial infections were suspected because they were successfully treated with antibiotics. After a month, the patients' thyroid function and thyroglobulin levels returned to normal without a period of transient hypothyroidism. A pyriformsinus fistula was not demonstrated. The results suggest that neutropenia and preceding cellulitis around the thyroid gland, which might be subsequent to oral mucosal damage induced by anticancer drugs, may play a role in the development of AST. AST should be considered a potential complication of aggressive chemotherapy for leukemia.     

Atypical presentation of subacute thyroiditis.

Subacute thyroiditis is extremely rare during the first decade of life. We describe a case of subacute thyroiditis in a 2 year old child whose initial clinical, sonographic, and radioisotopic features were indistinguishable from acute suppurative thyroiditis. The diagnosis was established by a raised antibody titre against adenovirus and typical increased thyroid function tests.     

Pyriform sinus fistulae

Pyriform sinus fistulae (PSF) are rare branchial pouch anomalies. In most previously described cases the anomaly is located on the left side. PSF should be suspected in cervical inflammatory processes (cervical abscesses and types of suppurative thyroiditis). We report two cases of acute thyroiditis and deep cervical abscesses secondary to PSF, which were diagnosed and treated in our hospital in the last 2 years. Both presented inflammatory cervical masses associated with painful swallowing, high fever and laboratory findings compatible with acute infection. In both cases the diagnosis of PSF was confirmed by barium esophagogram. Cervical ultrasonography and computed tomography were also performed. The treatment of choice consists of broad-spectrum antibiotic therapy during acute exacerbation and subsequent fistulectomy. Definitive surgical treatment prevents recurrences.     

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.     

Papillary carcinoma of the thyroid gland in a child of thyrotoxicosis patient receiving radioactive iodine therapy: report of a case

Herein we present the case of a 9-year-old girl who had an enlarged right lobe of the thyroid gland and sub-clinical hypothyroidism (thyroid stimulating hormone at 9.24 mIU/L). The patient had a history of unintentional exposure to radiation while her mother was receiving radionuclide therapy for diffuse toxic goiter. Ultrasonography of the young girl showed right lobe enlargement with diffuse coarse heterogenous echogenicity, compatible with a microcalcification pattern identified in both lobes of the thyroid gland. Histopathology of the tissue from a thyroidectomy revealed papillary thyroid carcinoma in the right lobe and chronic lymphocytic thyroiditis in the remaining tissue. Molecular pathology demonstrated an RET/PTC1 rearrangement in both tumor and non-tumorous tissue harboring thyroiditis. Considering the history of exposure and the characteristics of the thyroid pathology together, the PTC in this patient was likely a secondary-to-genetic alteration induced by external radiation. This case emphasizes the importance of stringent restrictions when giving radioactive iodine therapy to a patient with small children.     

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。     

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.