International Journal of Pediatric Otorhinolaryngology 78 (2014) 1953–1957
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Pediatric recurrent acute suppurative thyroiditis of third branchial arch origin—Our experience in 17 cases Pradipta Kumar Parida *, Surianarayanan Gopalakrishnan, Sunil Kumar Saxena Department of Otorhinolaryngology and Head-Neck Surgery, JIPMER, Puducherry, India
A R T I C L E I N F O
A B S T R A C T
Article history: Received 10 May 2014 Received in revised form 29 July 2014 Accepted 24 August 2014 Available online 1 September 2014
Objective: To describe clinical presentations, management and treatment outcomes of 17 cases of congenital pyriform sinus fistula (PSF) of third branchial arch origin presenting as left recurrent acute suppurative thyroiditis with cervical abscess. Method: Medical record of these 17 cases (5-males, 12-females) presented during 2009–2013 were reviewed. Results: Average age was 9.6 years (range 3–15 years). Fistulous opening in neck was present in 10 cases (58.8%). Average number of episode of infection from first presentation to definitive diagnosis was 3 (range 2–5). All patient had history of incision and drainage (ID) of abscess (average 2, range 1–3). All cases had barium swallow and CT scan. Sixteen cases had telescopic hypopharyngoscopy. Barium swallow and telescopic hypopharyngoscopy detected PSF in 88.23% (15/17) and 100% (16/16) cases respectively. Fourteen cases were treated by transcervical excision (TE) (fistulectomy with left hemithyroidectomy), two cases were treated by endoscopic chemical cauterization (ECC) of internal opening at pyriform sinus using silver nitrate and only ID of abscess was done in one case. Success rate of TE and ECC was 93% and 100% respectively. Recurrence in one case initially treated by TE was managed successfully by ECC. Conclusion: Presence of congenital PFS should be suspected when left-sided intra-thyroidal abscess formation occurs as gland is resistant to infection. Strong clinical suspicion, barium swallow study, telescopic pharyngoscopy and CT scan are the key to diagnosis. Both TE and ECC has comparable success rate. ECC may prove a useful and equally effective method of treatment for congenital PFS in future. ß 2014 Elsevier Ireland Ltd. All rights reserved.
Keywords: Branchial arch Fistula Thyroidectomy Pyriform sinus Thyroiditis
1. Introduction Congenital piriform sinus fistula (PSF) of third branchial arch origin comes out by penetrating the cricothyroid muscle and terminates in capsule of thyroid. When it gets infected, it manifests as recurrent acute thyroid and/or neck abscess [1]. This condition is higher on left side and usually manifest in first decade of life. Presence of PSF should be suspected if a child presents with recurrent left-sided neck or thyroid abscesses with or without discharging cervical fistula. The initial treatment of thyroid abscess is broad spectrum antibiotics with incision and drainage (ID) of abscess. Excision of the entire fistulous tract (FT) together with hemi-thyroidectomy during an inflammation-free interval is the treatment of choice [2].
* Corresponding author at: Department of Otorhinolaryngology and Head-Neck Surgery, JIPMER, Puducherry 605006, India. Tel.: +91 9488820160. E-mail address: [email protected] (P.K. Parida). http://dx.doi.org/10.1016/j.ijporl.2014.08.034 0165-5876/ß 2014 Elsevier Ireland Ltd. All rights reserved.
Endoscopic chemical cauterization (ECC) internal opening of fistula at piriform sinus (PS) has also been recommended in recent years [3–6]. Here, we present 17 pediatrics cases of PS fistulae of third branchial arch presented with left sided, recurrent acute suppurative thyroiditis (AST) and describe the clinical features, investigations and treatment modalities and their outcomes of this rare condition. 2. Methods We undertook a retrospective medical record review of 17 pediatric cases of recurrent AST secondary to third branchial arch PSF treated in the ENT department of Jawaharlal Institute of Postgraduate Medical Education and Research, Pondicherry, India, between 2009 and 2013. Data regarding the patients’ demographics, presenting symptoms, time from presentation to definitive diagnosis, diagnostic studies, treatment modalities, intra-operative findings and recurrence were collected and analyzed by using descriptive statistics.
P.K. Parida et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1953–1957
1954
16 (100%) cases (Fig. 4). Fourteen cases were treated by transcervical excision (TE) of entire FT with left hemithyroidectomy (Fig. 5), two cases were treated by trans-oral rigid ECC of fistulous opening in PS using silver nitrate (Fig. 6) and only ID of abscess was done for one case. The FT originating from the piriform sinus and coming into the left thyroid lobe could be identified in all 14 cases who underwent TE. The histopathologic examination of the specimen confirmed the presence of FT. The lumen FT was lined by pseudostratified, ciliated epithelium with moderate inflammatory infiltration and fibrosis in tract walls. The post-operative period was uneventful in all cases with no incidence of recurrent laryngeal nerve paralysis. The success rate of TE and ECC were 93% and 100% respectively. There was no recurrence except one case (case no 15) over 9 months to 4 years of regular follow up. The fistula recurred in case 15, three months after initial treatment by TE and the recurrence was managed successfully by ECC with no evidence of recurrence over 2 years follow up period. This study was approved by our institutional review committee. Fig. 1. Clinical photograph showing fistulous opening along with scar mark on left side of neck (case 15).
3. Results Out of 17 children, five were males and twelve were females. The mean age was 9.6 years (range 3–15 years). The fistulous opening in neck was present in 10 cases (58.8%) (Fig. 1; Table 1). The average number of episode of infection from first presentation to the definitive diagnosis was 3 (range 2–5 episodes). All patient had history of ID of abscess (average 2, range 1–3). All cases had barium swallow study and Contrast enhanced CT scan of but telescopic hypopharyngoscopy was done in 16 cases. In 15 cases (88.23%), the fistula was detected preoperatively by barium swallow study (Fig. 2a and b). CT scan was taken immediately after barium swallow study. CT scan revealed the presence of inflammation and suppuration surrounding the left thyroid lobe extending up to the level of PS (Fig. 3a). CT scans also demonstrated presence of air in posterior aspect of gland suggestive of FT (Fig. 3b). Per-operative telescopic hypopharyngoscopy using 08 Hopkins Rod endoscope detected PSF in all
4. Discussion The thyroid gland exhibits a high resistance to the bacterial infection because of its high blood supply, rich lymphatics, iodide content, and presence of a tough capsule. Hence, recurrent AST in children would suggest an underlying congenital PSF of branchial arch origin [1,2]. Most cases of recurrent AST in infants and children involve the left lobe of thyroid [1,7]. In our patients, recurrent AST was the most common mode of presentation. PSF of third branchial arch originates anterior to the internal laryngeal nerve fold and pierces the thyro-hyoid membrane cranial to the superior laryngeal nerve and passes over the hypoglossal nerve and behind the internal carotid artery before passing through the left thyroid lobe [8]. Barium swallow study performed four to six week after the resolution of acute inflammation and telescopic examination of PS can demonstrate PSF. So these two investigations should be considered in cases of AST [1,9,10]. Barium oesophagogram could identify the sinus or FT in 50% (8/16) of patients in a study conducted by Park et al. [6]. CT scan performed immediately after
Table 1 Demography, presentations, radiology and surgical modalities used. Age in years/sex
Presentation
No. of episodes of RTA
Time period between 1st episode and definitive treatment in years
PSF on barium swallow study
PSF on pharyngo-scopy
Treatment modalities
Recurrence
9/M 6/F 13/F 15/M 10/F 11/M 3/F 15/F
RTA + NF RTA RTA + NF RTA + NF RTA RTA + NF RTA RTA + NF
3 2 3 5 3 4 3 5
4 2.5 5 7 3.2 4 1 Definitive surgery not done
+ + + + + + _ +
Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT I&D
Nil Nil Nil Nil Nil Nil Nil Nil
9 10 11 12 13 14 15
5/F 9/F 7/F 10/F 7/F 8/M 12/F
RTA + NF RTA RTA + NF RTA + NF RTA RTA + NF RTA + NF
2 3 2 2 3 3 3
1 2.5 1.5 2.5 2 2.6 2.7
+ _ + + + + +
+ + + + + + + Pharyngo-scopy not done + + + + + + +
Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT
16 17
12/F 11/M
RTA RTA
4 2
2.5 3.25
+ +
+ +
ECC ECC
Nil Nil Nil Nil Nil Nil Recurred 3 months after surgerya Nil Nil
Sl. no. 1 2 3 4 5 6 7 8
M = male, F = female, PSF = piriform sinus fistula, RTA = recurrent thyroid abscess, NF = neck fistula, + = positive for PSF, ECC = endoscopic chemical cauterization. a Successfully treated by ECC.
= negative for PSF, HT = hemithyroidectomy,
P.K. Parida et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1953–1957
1955
Fig. 2. Barium swallow study showing left piriform sinus fistula. (a) Antero-posterior vies, (b) lateral view (case 15).
Fig. 3. (a) Contrast enhanced computerized tomography of neck, axial section showing the abscess in left lobe of thyroid and neck (case 7). (b) Computerized tomography of neck, axial section showing inflammation on left side of neck and presence of air pocket (shown by a blue arrow) suggestive of fistulous tract (case 7). (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.)
the barium swallow or a direct contrast injection through the skin opening will be more useful in delineating the entire FT. CT scan is also useful in excluding other lateral cervical pathology and to guide the plane and direction of dissection. Park et al. [6] could able to identify the FT in all cases (14/14) on CT scan. Telescopic pharyngoscopy under general anesthesia is useful for
Fig. 4. Telescopic hypopharyngoscopy showing the internal opening of fistula at left piriform sinus shown by a blue arrow and the green arrow indicates the aryepiglottic fold. (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.)
Fig. 5. Per-operative photograph showing left hemithyroidectomy along with excision of the fistulous tract. The fistulous tract was ligated at the apex of left piriform sinus.
1956
P.K. Parida et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1953–1957
Fig. 6. Telescopic hypopharyngoscopy showing endoscopic chemical cauterization of the internal opening of fistula at left piriform sinus and the green arrow indicates the ary-epiglottic fold.
diagnosis and it could identify the fistulous opening in all cases in our series. Telescopic hypopharyngoscopy could able to identify the fistulous opening in PS in all cases in other reported case series [3,4,6]. Though empirical administration of broad spectrum antibiotics reduces inflammation, suppurative thyroiditis often requires ID which makes the definitive surgery more difficult [1]. Complete excision of the FT with a hemithyroidectomy, during a quiescent period, is the treatment of choice. Excision of FT up to the level of thyrohyoid membrane may be sufficient. Wide exposure may be achieved by retracting or excising a vertical strip of the posterior border of the thyroid ala. Endoscopic cannulation of internal opening at PS, with injection of 1 percent methylene blue dye may help in localizing the FT [11]. Incomplete excision carries a high risk of recurrence. Sai Prasad et al. [12] reported 12 cases of AST in children secondary to PSF treated by surgical excision, nine patients (75%) underwent successful complete excision and three patients (25%) had recurrence. Mali et al. [8] reported successful treatment of 3 cases of PSF by surgical excision. Minimal invasive procedure like endoscopic obliteration of internal opening at PS during an infection free period with silver nitrate, trichloroacetic acid, fibrin or an insulated electro-cautery probe is a safe and effective treatment option with similar success rate as of TE [1,3–6,13,14]. The endoscopic procedure carries a very low morbidity and reduces hospital stay. The reported success rate for ECC varies from 78% to 100% [3–6,14]. Fifteen out of 17 patients who underwent chemo-cauterization of sinus or fistulous tract in Park et al. [6] series had good surgical outcome. Cigliano et al. [14] reported successful endoscopic management of congenital PSF by injection of fibrin adhesive in one case. Fourth branchial cleft sinus tracts can be managed by endoscopic cauterization. Seven out of 10 cases of 4th branchial arch fistula treated with endoscopic cauterization had no recurrence with an average follow up of 3 years [5]. Watson et al. [15] reported 5 cases of fourth branchial arch fistulae obliterated using electro-cautery in 1 patient, CO2 laser/silver nitrate chemo-cautery in 2 patients and Silver Nitrate chemo-cautery in a further 2 patients. There were no complications and no recurrences over a mean follow-up period of 25 months (range 11–41 months). Chen et al. [4] reported the success rate and recurrence rate of endoscopic electro-cauterization of PSF 78% (7/9) and 22% (2/9) respectively. They also suggested that endoscopic electro-cauterization of PSF tract as definitive treatment reserving open excision
with or without thyroid lobectomy for failures. The recurrent cases underwent excision of sinus tract with left thyroid lobectomy. Smith et al. [3] reported 5 cases of congenital PSF treated successfully without recurrence. Three were managed with complete excision of their tract and 2 with cauterization of the internal opening with silver nitrate. Sun et al. [16] reported 23 cases of third and fourth branchial arch PSF who underwent endoscopic electro-cauterization with an overall success rate of 91% on the first attempt (21 of 23 patients). The 2 cases with recurrence experienced the symptoms within 1 month of cauterization and were treated with either open excision or re-cauterization. Endoscopic cauterization was able to definitely treat 9 patients whose treatment with incision and drainage (7 cases) or open excision (2 cases) had failed [16]. Sanchis Blanco et al. [17] reported 9 cases of PSF treated by fistulectomy (4 cases), endoscopic sclerosis with diathermy (4 cases) and the ninth case received both treatments performing electro-cauterization after a surgical recurrence. Three of the patients who underwent surgery relapsed; none treated by endoscopic sclerosis with diathermy did or had complications. Initial ECC of PSF should be considered, with open excision reserved for endoscopic failures [4]. In our series the case who had recurrence after open excision well responded to ECC. So, ECC may be recommended for the recurrent cases who have not undergone ECC previously. Roll of ECC for recurrence following either initial ECC or open excision has to be studied. In view of rarity of this condition, multi-institutional studies are required to verify the effectiveness of ECC, repeated ECC and open surgical procedure. Post-operative complications following open surgery include temporary vocal fold paralysis, salivary fistula and wound infection. These are more common in children younger than eight years [11]. Recurrence is possible many years after surgery, usually following inadequate excision or excision during an acute episode of suppurative thyroiditis. Hence, long-term follow up is necessary. 5. Conclusion The presence of congenital PFS of should be suspected when left-sided intra-thyroidal abscess formation occurs as the gland is resistant to infection. Strong clinical suspicion, barium swallow study, telescopic pharyngoscopy and CT scan are the key to diagnose this rare entity. Though the both open surgical excision and ECC has good success rate, ECC of internal opening at PS is a minimally invasive and safe. ECC may prove a useful and equally effective method of treatment for congenital PSF in future. References [1] N. Seki, T. Himi, Retrospective review of 13 cases of pyriform sinus fistula, Am. J. Otolaryngol. 28 (2007) 55–58. [2] A.T. Ahuja, J.F. Griffiths, D.J. Roebuck, W.K. Loftus, K.Y. Lau, C.K. Yeung, et al., Ultrasound and oesophagography in the management of acute suppurative thyroiditis in children associated with congenital pyriform fossa sinus, Clin. Radiol. 53 (1998) 209–211. [3] S.L. Smith, K.D. Pereira, Suppurative thyroiditis in children: a management algorithm, Pediatr. Emerg. Care 24 (2008) 764–767. [4] E.Y. Chen, A.F. Inglis, H. Ou, J.A. Perkin, K.C. Sie, J. Chiara, et al., Endoscopic electrocauterisation of piriform fossa sinus tracts as definitive treatment, Int. J. Pediatr. Otorhinolaryngol. 73 (2009) 1151–1156. [5] D.J. Verret, J. McClay, A. Murray, M. Biavati, O. Brown, Endoscopic cauterization of fourth branchial cleft sinus tracts, Arch. Otolaryngol. Head Neck Surg. 130 (4) (2004) 465–468, Apr. [6] S.W. Park, M.H. Han, M.H. Sung, I.O. Kim, K.H. Kim, K.H. Chang, et al., Neck infection associated with pyriform sinus fistula: imaging findings, Am. J. Neuroradiol. 21 (5) (2000) 817–822, May. [7] M. Kubota, S. Suita, T. Kamimura, Y. Zaizen, Surgical strategy for the treatment of pyriform sinus fistula, J. Pediatr. Surg. 32 (1997) 34–37. [8] V.P. Mali, K. Prabhakaran, Recurrent acute thyroid swellings because of pyriform sinus fistula, J. Pediatr. Surg. 43 (2008) 27–30. [9] H. Tucker, M. Skolnick, Fourth branchial cleft (pharyngeal pouch) remnant, Trans. Am. Acad. Ophthalmol. Otol. 77 (1973) 368–371. [10] S. Takai, A. Miyauchi, F. Matsuzuka, K. Kuma, G. Kosaki, Internal fistula as a route of infection in acute suppurative thyroiditis, Lancet (1) (1979) 751–752.
P.K. Parida et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1953–1957 [11] A. James, C. Stewart, P. Warrick, C. Tzifa, V. Forte, Branchial sinus of the piriform fossa: reappraisal of the third and fourth branchial anomalies, Laryngoscope 117 (2007) 1920–1924. [12] T.R. Sai Prasad, C.L. Chong, A. Mani, C.H. Chui, C.E. Tan, W.S. Tee, et al., Acute suppurative thyroiditis in children secondary to pyriform sinus fistula, Pediatr. Surg. Int. 23 (2007) 779–783. [13] R. Garrel, E. Jouzdani, Q. Gardiner, M. Makeieff, M. Mondain, P. Hagen, et al., Fourth branchial pouch sinus: from diagnosis to treatment, Otolaryngol. Head Neck Surg. 134 (2006) 157–163. [14] B. Cigliano, L. Cipolletta, N. Baltogiannis, C. Esposito, A. Settimi, Endoscopic fibrin sealing of congenital pyriform sinus fistula, Surg. Endosc. 18 (March (3)) (2004) 554–556.
1957
[15] G.J. Watson, J.R. Nichani, M.P. Rothera, I.A. Bruce, Case series: endoscopic management of fourth branchial arch anomalies, Int. J. Pediatr. Otorhinolaryngol. 77 (2013) 766–769. [16] J.Y. Sun, E.E. Berg, J.E. McClay, Endoscopic cauterization of congenital pyriform fossa sinus tracts: an 18-year experience, JAMA Otolaryngol. Head Neck Surg. 140 (2014) 112–117. [17] G. Sanchis Bianco, C. Gutierrez San Roman, M. Bordallo Vazquez, J. Cartes Saez, J.E. Barrias Fontoba, J. Liuna Gonzalez, et al., Endoscopic sclerosis with pneumatic distension for pyriform sinus fistula treatment, Cir. Pediatr. 27 (2014) 1–5.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Pediatric recurrent acute suppurative thyroiditis of third branchial arch origin—Our experience in 17 cases Pradipta Kumar Parida *, Surianarayanan Gopalakrishnan, Sunil Kumar Saxena Department of Otorhinolaryngology and Head-Neck Surgery, JIPMER, Puducherry, India
A R T I C L E I N F O
A B S T R A C T
Article history: Received 10 May 2014 Received in revised form 29 July 2014 Accepted 24 August 2014 Available online 1 September 2014
Objective: To describe clinical presentations, management and treatment outcomes of 17 cases of congenital pyriform sinus fistula (PSF) of third branchial arch origin presenting as left recurrent acute suppurative thyroiditis with cervical abscess. Method: Medical record of these 17 cases (5-males, 12-females) presented during 2009–2013 were reviewed. Results: Average age was 9.6 years (range 3–15 years). Fistulous opening in neck was present in 10 cases (58.8%). Average number of episode of infection from first presentation to definitive diagnosis was 3 (range 2–5). All patient had history of incision and drainage (ID) of abscess (average 2, range 1–3). All cases had barium swallow and CT scan. Sixteen cases had telescopic hypopharyngoscopy. Barium swallow and telescopic hypopharyngoscopy detected PSF in 88.23% (15/17) and 100% (16/16) cases respectively. Fourteen cases were treated by transcervical excision (TE) (fistulectomy with left hemithyroidectomy), two cases were treated by endoscopic chemical cauterization (ECC) of internal opening at pyriform sinus using silver nitrate and only ID of abscess was done in one case. Success rate of TE and ECC was 93% and 100% respectively. Recurrence in one case initially treated by TE was managed successfully by ECC. Conclusion: Presence of congenital PFS should be suspected when left-sided intra-thyroidal abscess formation occurs as gland is resistant to infection. Strong clinical suspicion, barium swallow study, telescopic pharyngoscopy and CT scan are the key to diagnosis. Both TE and ECC has comparable success rate. ECC may prove a useful and equally effective method of treatment for congenital PFS in future. ß 2014 Elsevier Ireland Ltd. All rights reserved.
Keywords: Branchial arch Fistula Thyroidectomy Pyriform sinus Thyroiditis
1. Introduction Congenital piriform sinus fistula (PSF) of third branchial arch origin comes out by penetrating the cricothyroid muscle and terminates in capsule of thyroid. When it gets infected, it manifests as recurrent acute thyroid and/or neck abscess [1]. This condition is higher on left side and usually manifest in first decade of life. Presence of PSF should be suspected if a child presents with recurrent left-sided neck or thyroid abscesses with or without discharging cervical fistula. The initial treatment of thyroid abscess is broad spectrum antibiotics with incision and drainage (ID) of abscess. Excision of the entire fistulous tract (FT) together with hemi-thyroidectomy during an inflammation-free interval is the treatment of choice [2].
* Corresponding author at: Department of Otorhinolaryngology and Head-Neck Surgery, JIPMER, Puducherry 605006, India. Tel.: +91 9488820160. E-mail address: [email protected] (P.K. Parida). http://dx.doi.org/10.1016/j.ijporl.2014.08.034 0165-5876/ß 2014 Elsevier Ireland Ltd. All rights reserved.
Endoscopic chemical cauterization (ECC) internal opening of fistula at piriform sinus (PS) has also been recommended in recent years [3–6]. Here, we present 17 pediatrics cases of PS fistulae of third branchial arch presented with left sided, recurrent acute suppurative thyroiditis (AST) and describe the clinical features, investigations and treatment modalities and their outcomes of this rare condition. 2. Methods We undertook a retrospective medical record review of 17 pediatric cases of recurrent AST secondary to third branchial arch PSF treated in the ENT department of Jawaharlal Institute of Postgraduate Medical Education and Research, Pondicherry, India, between 2009 and 2013. Data regarding the patients’ demographics, presenting symptoms, time from presentation to definitive diagnosis, diagnostic studies, treatment modalities, intra-operative findings and recurrence were collected and analyzed by using descriptive statistics.
P.K. Parida et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1953–1957
1954
16 (100%) cases (Fig. 4). Fourteen cases were treated by transcervical excision (TE) of entire FT with left hemithyroidectomy (Fig. 5), two cases were treated by trans-oral rigid ECC of fistulous opening in PS using silver nitrate (Fig. 6) and only ID of abscess was done for one case. The FT originating from the piriform sinus and coming into the left thyroid lobe could be identified in all 14 cases who underwent TE. The histopathologic examination of the specimen confirmed the presence of FT. The lumen FT was lined by pseudostratified, ciliated epithelium with moderate inflammatory infiltration and fibrosis in tract walls. The post-operative period was uneventful in all cases with no incidence of recurrent laryngeal nerve paralysis. The success rate of TE and ECC were 93% and 100% respectively. There was no recurrence except one case (case no 15) over 9 months to 4 years of regular follow up. The fistula recurred in case 15, three months after initial treatment by TE and the recurrence was managed successfully by ECC with no evidence of recurrence over 2 years follow up period. This study was approved by our institutional review committee. Fig. 1. Clinical photograph showing fistulous opening along with scar mark on left side of neck (case 15).
3. Results Out of 17 children, five were males and twelve were females. The mean age was 9.6 years (range 3–15 years). The fistulous opening in neck was present in 10 cases (58.8%) (Fig. 1; Table 1). The average number of episode of infection from first presentation to the definitive diagnosis was 3 (range 2–5 episodes). All patient had history of ID of abscess (average 2, range 1–3). All cases had barium swallow study and Contrast enhanced CT scan of but telescopic hypopharyngoscopy was done in 16 cases. In 15 cases (88.23%), the fistula was detected preoperatively by barium swallow study (Fig. 2a and b). CT scan was taken immediately after barium swallow study. CT scan revealed the presence of inflammation and suppuration surrounding the left thyroid lobe extending up to the level of PS (Fig. 3a). CT scans also demonstrated presence of air in posterior aspect of gland suggestive of FT (Fig. 3b). Per-operative telescopic hypopharyngoscopy using 08 Hopkins Rod endoscope detected PSF in all
4. Discussion The thyroid gland exhibits a high resistance to the bacterial infection because of its high blood supply, rich lymphatics, iodide content, and presence of a tough capsule. Hence, recurrent AST in children would suggest an underlying congenital PSF of branchial arch origin [1,2]. Most cases of recurrent AST in infants and children involve the left lobe of thyroid [1,7]. In our patients, recurrent AST was the most common mode of presentation. PSF of third branchial arch originates anterior to the internal laryngeal nerve fold and pierces the thyro-hyoid membrane cranial to the superior laryngeal nerve and passes over the hypoglossal nerve and behind the internal carotid artery before passing through the left thyroid lobe [8]. Barium swallow study performed four to six week after the resolution of acute inflammation and telescopic examination of PS can demonstrate PSF. So these two investigations should be considered in cases of AST [1,9,10]. Barium oesophagogram could identify the sinus or FT in 50% (8/16) of patients in a study conducted by Park et al. [6]. CT scan performed immediately after
Table 1 Demography, presentations, radiology and surgical modalities used. Age in years/sex
Presentation
No. of episodes of RTA
Time period between 1st episode and definitive treatment in years
PSF on barium swallow study
PSF on pharyngo-scopy
Treatment modalities
Recurrence
9/M 6/F 13/F 15/M 10/F 11/M 3/F 15/F
RTA + NF RTA RTA + NF RTA + NF RTA RTA + NF RTA RTA + NF
3 2 3 5 3 4 3 5
4 2.5 5 7 3.2 4 1 Definitive surgery not done
+ + + + + + _ +
Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT I&D
Nil Nil Nil Nil Nil Nil Nil Nil
9 10 11 12 13 14 15
5/F 9/F 7/F 10/F 7/F 8/M 12/F
RTA + NF RTA RTA + NF RTA + NF RTA RTA + NF RTA + NF
2 3 2 2 3 3 3
1 2.5 1.5 2.5 2 2.6 2.7
+ _ + + + + +
+ + + + + + + Pharyngo-scopy not done + + + + + + +
Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT Fistulectomy + HT
16 17
12/F 11/M
RTA RTA
4 2
2.5 3.25
+ +
+ +
ECC ECC
Nil Nil Nil Nil Nil Nil Recurred 3 months after surgerya Nil Nil
Sl. no. 1 2 3 4 5 6 7 8
M = male, F = female, PSF = piriform sinus fistula, RTA = recurrent thyroid abscess, NF = neck fistula, + = positive for PSF, ECC = endoscopic chemical cauterization. a Successfully treated by ECC.
= negative for PSF, HT = hemithyroidectomy,
P.K. Parida et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1953–1957
1955
Fig. 2. Barium swallow study showing left piriform sinus fistula. (a) Antero-posterior vies, (b) lateral view (case 15).
Fig. 3. (a) Contrast enhanced computerized tomography of neck, axial section showing the abscess in left lobe of thyroid and neck (case 7). (b) Computerized tomography of neck, axial section showing inflammation on left side of neck and presence of air pocket (shown by a blue arrow) suggestive of fistulous tract (case 7). (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.)
the barium swallow or a direct contrast injection through the skin opening will be more useful in delineating the entire FT. CT scan is also useful in excluding other lateral cervical pathology and to guide the plane and direction of dissection. Park et al. [6] could able to identify the FT in all cases (14/14) on CT scan. Telescopic pharyngoscopy under general anesthesia is useful for
Fig. 4. Telescopic hypopharyngoscopy showing the internal opening of fistula at left piriform sinus shown by a blue arrow and the green arrow indicates the aryepiglottic fold. (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.)
Fig. 5. Per-operative photograph showing left hemithyroidectomy along with excision of the fistulous tract. The fistulous tract was ligated at the apex of left piriform sinus.
1956
P.K. Parida et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1953–1957
Fig. 6. Telescopic hypopharyngoscopy showing endoscopic chemical cauterization of the internal opening of fistula at left piriform sinus and the green arrow indicates the ary-epiglottic fold.
diagnosis and it could identify the fistulous opening in all cases in our series. Telescopic hypopharyngoscopy could able to identify the fistulous opening in PS in all cases in other reported case series [3,4,6]. Though empirical administration of broad spectrum antibiotics reduces inflammation, suppurative thyroiditis often requires ID which makes the definitive surgery more difficult [1]. Complete excision of the FT with a hemithyroidectomy, during a quiescent period, is the treatment of choice. Excision of FT up to the level of thyrohyoid membrane may be sufficient. Wide exposure may be achieved by retracting or excising a vertical strip of the posterior border of the thyroid ala. Endoscopic cannulation of internal opening at PS, with injection of 1 percent methylene blue dye may help in localizing the FT [11]. Incomplete excision carries a high risk of recurrence. Sai Prasad et al. [12] reported 12 cases of AST in children secondary to PSF treated by surgical excision, nine patients (75%) underwent successful complete excision and three patients (25%) had recurrence. Mali et al. [8] reported successful treatment of 3 cases of PSF by surgical excision. Minimal invasive procedure like endoscopic obliteration of internal opening at PS during an infection free period with silver nitrate, trichloroacetic acid, fibrin or an insulated electro-cautery probe is a safe and effective treatment option with similar success rate as of TE [1,3–6,13,14]. The endoscopic procedure carries a very low morbidity and reduces hospital stay. The reported success rate for ECC varies from 78% to 100% [3–6,14]. Fifteen out of 17 patients who underwent chemo-cauterization of sinus or fistulous tract in Park et al. [6] series had good surgical outcome. Cigliano et al. [14] reported successful endoscopic management of congenital PSF by injection of fibrin adhesive in one case. Fourth branchial cleft sinus tracts can be managed by endoscopic cauterization. Seven out of 10 cases of 4th branchial arch fistula treated with endoscopic cauterization had no recurrence with an average follow up of 3 years [5]. Watson et al. [15] reported 5 cases of fourth branchial arch fistulae obliterated using electro-cautery in 1 patient, CO2 laser/silver nitrate chemo-cautery in 2 patients and Silver Nitrate chemo-cautery in a further 2 patients. There were no complications and no recurrences over a mean follow-up period of 25 months (range 11–41 months). Chen et al. [4] reported the success rate and recurrence rate of endoscopic electro-cauterization of PSF 78% (7/9) and 22% (2/9) respectively. They also suggested that endoscopic electro-cauterization of PSF tract as definitive treatment reserving open excision
with or without thyroid lobectomy for failures. The recurrent cases underwent excision of sinus tract with left thyroid lobectomy. Smith et al. [3] reported 5 cases of congenital PSF treated successfully without recurrence. Three were managed with complete excision of their tract and 2 with cauterization of the internal opening with silver nitrate. Sun et al. [16] reported 23 cases of third and fourth branchial arch PSF who underwent endoscopic electro-cauterization with an overall success rate of 91% on the first attempt (21 of 23 patients). The 2 cases with recurrence experienced the symptoms within 1 month of cauterization and were treated with either open excision or re-cauterization. Endoscopic cauterization was able to definitely treat 9 patients whose treatment with incision and drainage (7 cases) or open excision (2 cases) had failed [16]. Sanchis Blanco et al. [17] reported 9 cases of PSF treated by fistulectomy (4 cases), endoscopic sclerosis with diathermy (4 cases) and the ninth case received both treatments performing electro-cauterization after a surgical recurrence. Three of the patients who underwent surgery relapsed; none treated by endoscopic sclerosis with diathermy did or had complications. Initial ECC of PSF should be considered, with open excision reserved for endoscopic failures [4]. In our series the case who had recurrence after open excision well responded to ECC. So, ECC may be recommended for the recurrent cases who have not undergone ECC previously. Roll of ECC for recurrence following either initial ECC or open excision has to be studied. In view of rarity of this condition, multi-institutional studies are required to verify the effectiveness of ECC, repeated ECC and open surgical procedure. Post-operative complications following open surgery include temporary vocal fold paralysis, salivary fistula and wound infection. These are more common in children younger than eight years [11]. Recurrence is possible many years after surgery, usually following inadequate excision or excision during an acute episode of suppurative thyroiditis. Hence, long-term follow up is necessary. 5. Conclusion The presence of congenital PFS of should be suspected when left-sided intra-thyroidal abscess formation occurs as the gland is resistant to infection. Strong clinical suspicion, barium swallow study, telescopic pharyngoscopy and CT scan are the key to diagnose this rare entity. Though the both open surgical excision and ECC has good success rate, ECC of internal opening at PS is a minimally invasive and safe. ECC may prove a useful and equally effective method of treatment for congenital PSF in future. References [1] N. Seki, T. Himi, Retrospective review of 13 cases of pyriform sinus fistula, Am. J. Otolaryngol. 28 (2007) 55–58. [2] A.T. Ahuja, J.F. Griffiths, D.J. Roebuck, W.K. Loftus, K.Y. Lau, C.K. Yeung, et al., Ultrasound and oesophagography in the management of acute suppurative thyroiditis in children associated with congenital pyriform fossa sinus, Clin. Radiol. 53 (1998) 209–211. [3] S.L. Smith, K.D. Pereira, Suppurative thyroiditis in children: a management algorithm, Pediatr. Emerg. Care 24 (2008) 764–767. [4] E.Y. Chen, A.F. Inglis, H. Ou, J.A. Perkin, K.C. Sie, J. Chiara, et al., Endoscopic electrocauterisation of piriform fossa sinus tracts as definitive treatment, Int. J. Pediatr. Otorhinolaryngol. 73 (2009) 1151–1156. [5] D.J. Verret, J. McClay, A. Murray, M. Biavati, O. Brown, Endoscopic cauterization of fourth branchial cleft sinus tracts, Arch. Otolaryngol. Head Neck Surg. 130 (4) (2004) 465–468, Apr. [6] S.W. Park, M.H. Han, M.H. Sung, I.O. Kim, K.H. Kim, K.H. Chang, et al., Neck infection associated with pyriform sinus fistula: imaging findings, Am. J. Neuroradiol. 21 (5) (2000) 817–822, May. [7] M. Kubota, S. Suita, T. Kamimura, Y. Zaizen, Surgical strategy for the treatment of pyriform sinus fistula, J. Pediatr. Surg. 32 (1997) 34–37. [8] V.P. Mali, K. Prabhakaran, Recurrent acute thyroid swellings because of pyriform sinus fistula, J. Pediatr. Surg. 43 (2008) 27–30. [9] H. Tucker, M. Skolnick, Fourth branchial cleft (pharyngeal pouch) remnant, Trans. Am. Acad. Ophthalmol. Otol. 77 (1973) 368–371. [10] S. Takai, A. Miyauchi, F. Matsuzuka, K. Kuma, G. Kosaki, Internal fistula as a route of infection in acute suppurative thyroiditis, Lancet (1) (1979) 751–752.
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