Journal of Otology & RhinologyISSN: 2324-8785

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Case Report, J Otol Rhinol Vol: 3 Issue: 6

Recurrent Thyroid Abscess Secondary to Pyriform Sinus Fistula: An Underlying Pathology for an Uncommon Disease

Hashem M. Al-Momani1, Ayman Mismar2*, Raed N. Al-Taher1,Tareq Mahafzah3, Khalid Al-Zaben4 and Mahmoud Mustafa4
1Department of Surgery, Division of Pediatric Surgery, The University of Jordan Hospital, Faculty of Medicine, Amman, Jordan
2Department of Surgery, Division of Endocrine Surgery, The University of Jordan, Faculty of Medicine, Amman, Jordan
3Department of Special Surgery, Division of Otorhinolaryngology, The University of Jordan Hospital, Faculty of Medicine, Amman, Jordan
4Department of Anesthesia, The University of Jordan, Faculty of Medicine, Amman, Jordan
*Corresponding author : Ayman Mismar
P.O. Box: 13764, Amman 11942, Jordan
Tel: +962 799060822;
E-mail: [email protected]
Received: June 11, 2014 Accepted: August 25, 2014 Published: August 27, 2014
Citation: Al-Momani HM, Mismar A, Al-Taher RN, Mahafzah T, Al-Zaben K, et al. (2014) Recurrent Thyroid Abscess Secondary to Pyriform Sinus Fistula: An Underlying Pathology for an Uncommon Disease. J Otol Rhinol 3:6. doi:10.4172/2324-8785.1000188

Abstract

Secondary to Pyriform Sinus Fistula: An Underlying Pathology for an Uncommon

Introduction: Thyroid gland is very resistant to microbial infection, and whenever this happens, the presence of underlying pathology should be raised, especially with recurrent thyroid abscess. Presentation of Case: We report a clinical case of a 10 year old male child presented with recurrent thyroid abscess and diagnosed as a pyriform sinus fistula on the clinical grounds confirmed by esophagography and Computed tomography. The patient was treated by excision of the fistulous tract along with the involved thyroid tissue after an inflammation-free period. Surgery resulted in complete resolution and cure. Discussion: Although rare, there are a few reported cases in the literature discussing the entity of pyriform sinus fistula as a cause of recurrent thyroid abscess or suppurative thyroiditis. Strong clinical suspicion is the key to the diagnosis. Fistulogram could be confirmative especially when performed in an inflammatory-free period, and helps the surgeon to direct the plan of management. Surgical excision of the fistulous tract with hemithyroidectomy or lobectomy is the management of choice. Conclusion: Pyriform sinus fistula should be kept in mind as an underlying cause of n cases of recurrent thyroid abscess or suppurative thyroiditis.

Keywords: Thyroid abscess; Pyriform sinus fistula; Branchial cleft anomalies

Keywords

Thyroid abscess; Pyriform sinus fistula; Branchial cleft anomalies

Introduction

The thyroid gland is particularly resistant to bacterial infection. A pyriform sinus fistula should be suspected in the unusual cases of left sided, recurrent, neck abscesses or acute suppurative thyroiditis in children [1]. Pyriform sinus fistula is a congenital fistula that originates from the pyriform sinus, penetrates the cricothyroid muscle, and usually ends in the left lobe of the thyroid gland or the surrounding perithyroid tissues. This rare developmental anomaly that arises from the third and fourth pharyngeal pouches is notorious for causing recurrent thyroid suppuration.
Surgical neck exploration with removal of the entire fistulous tract along with the involved portion of the thyroid lobe during an inflammation-free period is the treatment of choice.
We present a case of 10 year old child with recurrent left thyroid lobe abscess secondary to pyriform sinus fistula.

Presentation of Case

A 10 year old male child presented to the emergency department with painful neck swelling of three days duration. Upon examination, the patient was febrile, has an anterior neck swelling and redness just to the left side of the midline. He had leukocytosis with neutrophilia. Ultrasound showed left thyroid lobe abscess. Computed tomography scan was performed and confirmed the presence of a well-defined cystic lesion with thick enhancing wall occupying most of the left thyroid lobe measuring 22mm×18mm, associated with few enlarged bilateral cervical lymph nodes (Figure 1).
Figure 1: CT scan of the neck: cystic lesion with thick enhancing wall occupying most of the left thyroid lobe measuring 22mm x 18mm, associated with few enlarged bilateral cervical lymph nodes.
IV antibiotic therapy and surgical drainage was done. Two weeks later, the patient came back to the clinic with good general status, healed wound, and no more inflammatory signs. Esophagogram was obtained and showed a fistulous tract arising from the pyriform sinus ending with a cystic lesion to the left of the midline (Figure 2).
Figure 2: Esophagogram: a fistulous tract arising from the pyriform sinus ending with a cystic lesion to the left of the midline.
The patient was lost to follow up for ten months, and then the child reappeared with anterior neck swelling at the same site. The clinical examination and CT findings (Figure 3) confirmed a recurrent thyroid abscess. Surgical drainage was done and he was given antibiotics.
Figure 3: CT-scan of the neck: recurrent thyroid abscess.
One month after resolution of the inflammatory signs, thyroid scan was obtained and showed homogenous uptake throughout the thyroid gland except for marked reduction at the upper half of left thyroid lobe.
After three months of resolution of the inflammation, the patient underwent surgical exploration. Under general anesthesia, direct laryngoscopy with trial to canulate the fistula was attempted. Neck collar skin incision was made. Exploration of left thyroid lobe showed a well defined cystic lesion with a well formed fibrous wall, around 1.5cm in diameter located at the upper pole. Fistulous tract was identified, resected and ligated at its most proximal point in conjunction with left partial thyroid lobectomy (Figure 4).
Figure 4: Operative photograph showing the fistula after excising the upper pole of the thyroid gland; a probe has been passed through the fistula tract (right).
The post-operative period was uneventful. Follow up eight months later showed complete resolution with no complications.

Discussion

The thyroid gland is particularly resistant to bacterial infection because of its rich blood supply extensive lymphatics, high iodide content, and a capsule that separates it from neighboring sources of contamination [1]. The thyroid abscess is a very rare entity and represents less than 0.1% of surgical diseases of the thyroid gland [2]. This is attributed to the existence of congenital malformations such as thyroglossal duct fistulas and branchial fistulas. Patients with these embryological anomalies may present with recurrent episodes of cervical abscess [3]. In the adult population; although most occur due to blood or lymphatic spread from a remote focus, some cases are related to loco-regional causes as thyroid FNA, thyroglossal cyst, perforation of the esophagus or hypo pharynx secondary to ingestion of a chicken bone, for example, and direct neck trauma [4,5]. Patients with immunosuppression are at higher risk. These conditions include retroviral HIV infection, patients receiving chemotherapy or corticosteroids and transplanted subjects [6]. Bacteriologically, the most offending organisms are Staphylococcus aureus, Streptococcus, and anaerobes. These germs are found in 70% of cases [7]. Other species were isolated: Escherichia coli due to bacteremia of urinary or digestive origin [7], Bacteroides fragilis isolated in women post hysterectomy [8]. In addition, other pathogens were isolated and reported in the literature: Klebsiella, Salmonella typhi, Acinetobacter, Mycobacterium tuberculosis, Pseudomonas,Eikenella corrodens, Clostridium, Fusobacterium Mortiferum, carnii pneumonia, Haemophilus as well as fungi such as Candida albicans, andaspergillosis [6,9,10]. These species have been identified in patients with immunosuppression [11,12]. Differential diagnoses are represented by viral subacute thyroiditis and chronic thyroiditis, bleeding intra cystic, primitive neoplasia or metastasis, and amyloidosis [13].
A pyriform sinus fistula should be suspected in all cases of left sided, recurrent, neck abscesses or acute suppurative thyroiditis in children [14]. Pyriform sinus fistula is a congenital fistula that originates from the pyriform sinus, penetrates the cricothyroid muscle, and ends in the thyroid gland or perithyroid tissues.
In literature, the first case of pyriform sinus fistula was reported as a branchial cleft cyst by Sandborn and Shafer in 1972 [15]. In 1973, Tucker and Skolnick demonstrated a fistulous tract using barium swallow study [16]. A few cases have been reported since 1979 when Takai et al described for the first time the situation of acute suppurative thyroiditis caused by pyriform sinus fistula [17].
Branchial anomalies of third and fourth arch although rare, usually present as sinuses or incomplete fistulae of the pyriform sinus [18]. The fistulous tract originates from the pyriform sinus to pass through the cricothyroid muscle and lobe of the thyroid gland to terminate in the neck [19]. This branchial arch anomaly manifests itself by recurrent neck abscess or acute suppurative thyroiditis and is usually noted during first decade of life, more frequently on the left than the right. Godin et al reported a 93% rate of left-sided patients [20]. The cause of this left predominance is related to the asymmetric development of the fourth branchial arch, whereby it becomes part of the aortic arch on the left side while it forms the right subclavian artery on the right [21]. However, on the basis of the histologic appearance of C cells, Miyauchi et al proposed the theory of fifth pouch (ultimobranchial body) derivation [21].
Generally, the clinical presentation of pyriform sinus fistula includes recurrent acute suppurative thyroiditis, retropharyngeal abscess, perithyroid abscess, respiratory distress, neck pain, fever, dysphagia, local erythema, and so on, with recurrent abscess or inflammation on the left anterior neck being the most pathognomonic and warrants investigation to exclude the existence of an underlying abnormality [19].
The important point in clinical diagnosis is maintaining a high index of suspicion [22]. Barium swallow study is an indispensable examination to demonstrate the fistulous tract, but it should be done about two months after an acute inflammatory phase because the edematous wall of the tract may not allow barium to pass and false-negative results may therefore be obtained [23,24]. However, confirming the internal orifice of the fistula and inserting a tube or injecting pigment such as methylene blue is a helpful method for detecting the tract during surgery. Computed tomography scan is a necessary examination to rule out other lateral cervical diseases and to determine the direction of the fistulectomy [25-27]. Ultrasound and CT are an essential aid in the diagnosis of thyroid abscess, it demonstrates the abscess cavity and its relation to adjacent structures including with major vessels of the neck and airway [3,13]. Despite the details provided by imaging, simple needle aspiration may be necessary to confirm the diagnosis of abscesses, and it provides sample for culture and sensitivity and thus it helps in choosing the most suitable antibiotic for treatment [6]. Empirical antibiotic to cover Staphylococcus aureus, Streptococcus, and anaerobes is the first line of treatment; this choice can be changed depending on the clinical response and the culture and sensitivity results. CTguided percutaneous catheter drainage may be an effective and safe therapeutic option [28]. Incision and drainage is indicated whenever there is a purulent collection that cannot be cleared by percutaineous aspiration and antibiotics. Whenever an underlying pathology is identified it should be mmanaged, otherwise recurrences may occur [29]. Removal of the fistula tract and control of internal opening is the mainstay of treatment [30,31]. Some authors recommend resection of the adjacent thyroid lobe, or at least debridement and excision of necrotic tissue, with resection of fistulous connections if possible [6]. Ablation of the fistula can be achieved less invasively through instillation of chemocauterizing agent which has shown satisfactory results [32,33]. Endoscopic obliteration of the internal opening of the fistula with trichloroacetic acid, fibrin glue, or an insulated electrocautery probe has also been reported [32,34,35].

Conclusion

Strong clinical suspicion is the key to the diagnosis of pyriform sinus fistula as a cause of recurrent neck or intra-thyroid abscesses. CT scan and contrast esophagogram are confirmative especially when performed in an inflammation-free period, and help the surgeon to direct the plan of management. Surgical excision of the fistulous tract along with the involved thyroid tissue is the management of choice.

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