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Juan Bass, MD, FRCSC; S. Muirhead, MD, FRCPC
From the Departments of Surgery (Dr Bass) and Pediatrics (Dr Muirhead), Children's Hospital of Eastern Ontario, Ottawa.

Arch Pediatr Adolesc Med. 2000;154:523-524.

A PREVIOUSLY healthy 15-year-old boy with a 7-day history of intermittent fever with temperatures up to 38.5°C, anorexia, nausea, irritability, persistent migraine headaches, and a 9.5-kg weight loss was seen in the emergency department complaining of left lateral neck pain. No discrete masses were palpable, and the thyroid and overlying skin felt normal. On day 9 symptoms persisted, and the left thyroid lobe was enlarged (4-cm long) and firm. A clinical diagnosis of subacute thyroiditis was made, and nonsteroidal anti-inflammatory drugs (NSAIDs) were prescribed pending results of thyroid function tests. Two days later he returned with a further 2.7-kg weight loss, dysphagia, positional dyspnea, and marked fatigue. The left thyroid lobe was hard, tender, and 6.5-cm long. The isthmus and the right lobe felt normal, and there was no cervical lymphadenopathy. Tracheal compression was present on chest radiograph, and ultrasonography was performed (Figure 1). An inadequate sample was obtained for fine-needle aspiration; no purulent material was aspirated. Serum thyroxine levels from day 7 were markedly elevated at 97.9 pmol/L (reference range, 10-25 pmol/L), and TSH was incompletely suppressed at 0.03 mIU/L. Antimicrosomal antibodies were present at a titer of 1:1600 and thyroglobulin antibodies at 155.6 pmol/L (reference range, 0-32 pmol/L). Failure of response to NSAIDs led to the administration of a tapering course of prednisone. A barium esophagram was performed and demonstrated the cause of the problem (Figure 2).

Antibiotics were administered with normalization of thyroid function test findings and clinical status during 4 weeks; however, a unilateral goiter persisted. An ultrasonogram of the thyroid showed a 2.9 x 5.5 x 2.9-cm homogenous mass within the left thyroid lobe. No cystic components were present, and some hyperemia and bilateral cervical chain lymphadenopathy were noted. After administrating antibiotics, the patient underwent direct laryngoscopy. An opening of a fistula from the left pyriform sinus was identified, and a left hemithyroidectomy was performed. During the dissection of the upper pole, laryngoscopy was repeated, and light was applied directly to the pyriform sinus with its transillumination identified in the operative field. The sinus tract was identified and transected, and a probe was introduced through the sinus and visualized with the laryngoscope (Figure 3). The fistulous tract was completely resected. On histopathologic examination there was fibrosis within the left lobe of the thyroid and the fistulous tract was lined with squamous epithelium.

Figure 1.

Figure 2.

Figure 3.

Denouncement and Discussion: Pyriform Sinus Fistula to the Left Lobe of the Thyroid

Figure 1. Ultrasound of the left thyroid lobe.

Figure 2. Barium esophagram. Arrow indicates a left-sided pyriform sinus fistula.

Figure 3. Operative view. Surgical exposure (left); arrow demonstrates probe introduced into pyriform sinus. Endoscopic view (right); probe coming through pyriform sinus.

Pyriform sinus fistula is a rare condition. There is usually a history of repeated upper respiratory tract infection, pain, and tenderness of the thyroid.¹ Although the entire thyroid may become firm and tender, the focus of inflammation is usually located in the left lobe.¹ If suppuration occurs, the overlying skin becomes erythematous and warm.¹ Considerable airway compression is uncommon, but hoarseness and odynophagia is seen.¹

Fistulae arise from the apex of the pyriform sinus of the hypopharynx and end in or adjacent to the upper pole of the thyroid lobe^2-3; thus, these cases can present as acute thyroiditis or as an anterior cervical abscess. The exact origins of the fistula are controversial,^2-3 but suggested origins include the third pharyngeal pouch,^4-7 fourth branchial arch or pouch,^8-10 or the ultimobranchial body.¹¹ Growth of the ultimobranchial body is often restricted or absent on the right side in the lower vertebrates, including reptiles.^11-12 These findings might elucidate the left-sided predominance of the fistula. Findings from barium esophagram may identify the fistulous tract^{5, 13-17}; however, it may fail to do so during the acute inflammatory phase, and studies should be repeated 6 to 8 weeks later.¹

A pyriform sinus fistula should be considered in the presence of unilateral (mostly left-sided) thyroiditis (not necessarily suppurative) and recurrent left anterior cervical abscesses. Recurrence is the rule unless complete excision of the fistulous tract is performed.

AUTHOR INFORMATION
Accepted for publication October 20, 1998.

Reprints: Juan Bass, MD, FRCSC, Department of Surgery, Children's Hospital of Eastern Ontario, 401 Smyth Rd, Ottawa, Ontario K1H 8L1, Canada (e-mail: bass{at}cheo.on.ca).

REFERENCES
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SECTION EDITOR: BEVERLY P. WOOD, MD

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