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  Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 56  |  Issue : 4  |  Page : 325-329
 

Unusual loco-regional presentation in papillary carcinoma of thyroid: A case series


1 Department of Head and Neck Surgery, Tata Medical Center, Kolkata, West Bengal, India
2 Department of Plastic and Reconstructive Surgery, Tata Medical Center, Kolkata, West Bengal, India
3 Department of Nuclear Medicine, Tata Medical Center, Kolkata, West Bengal, India

Date of Web Publication11-Oct-2019

Correspondence Address:
Pattatheyil Arun
Department of Head and Neck Surgery, Tata Medical Center, Kolkata, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijc.IJC_368_18

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 » Abstract 


BACKGROUND: Papillary carcinoma of thyroid (PTC) is usually indolent with good prognosis and excellent long-term survival. However, PTC sometimes presents itself in unusual situations, posing diagnostic and therapeutic challenges. Owing to paucity of data, there is lack of consensus as to what treatment should be prescribed in patients with loco-regional spread other than the usual sites.
MATERIALS AND METHODS: Six patients of PTC presenting with involvement of the aero-digestive tract, retropharyngeal, and para-pharyngeal lymph nodes and great vessels of the neck are included in this case series.
RESULTS AND CONCLUSION: Though rare, unusual loco-regional presentation of PTC poses challenges in diagnosis and treatment. A keen clinical sense is paramount in effectively diagnosing these cases. Aggressive surgical resection and reconstruction results in good functional and aesthetic outcomes. Further studies are required for establishing specific guidelines on the approach to the treatment of these cases.


Keywords: Papillary carcinoma thyroid, retropharyngeal node, tracheal infiltration


How to cite this article:
Jain PV, Sharan R, Manikantan K, Biswas G, Ray S, Arun P. Unusual loco-regional presentation in papillary carcinoma of thyroid: A case series. Indian J Cancer 2019;56:325-9

How to cite this URL:
Jain PV, Sharan R, Manikantan K, Biswas G, Ray S, Arun P. Unusual loco-regional presentation in papillary carcinoma of thyroid: A case series. Indian J Cancer [serial online] 2019 [cited 2019 Nov 18];56:325-9. Available from: http://www.indianjcancer.com/text.asp?2019/56/4/0/267599





 » Introduction Top


Papillary thyroid carcinoma (PTC) comprises 70–80% of thyroid carcinomas.[1] They are usually confined to the thyroid gland at presentation. Widely invasive thyroid cancer invading the aero-digestive tract including larynx, trachea, hypopharynx, and esophagus, is seen in 1–8% of patients.[2] Regional lymph node metastasis is evident in 40–50% cases and is most commonly seen in the nodes along the internal jugular vein and recurrent laryngeal nerve.[1] Distant metastases are seen in 4–15% cases.[3]

The 10-year survival for PTC is between 85% and 95%.[4] This number falls to 50% in the presence of distant metastasis.[4] Presence of laryngo-tracheal invasion is also associated with decreased survival and higher local recurrence rates.[5] Owing to paucity of data, there is lack of consensus about the treatment to be prescribed in patients with loco-regional spread other than the usual sites. We are presenting a case series of six patients of PTC who presented with unusual loco-regional involvement.


 » Case Reports Top


This case series includes six patients of unusual presentation of PTC, the details of which are depicted in [Table 1].
Table 1: Patient details

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Case 1: Retropharyngeal Nodal Metastases

A 32 year old woman presented with complaints of progressive neck swelling on the right side since 3 years, clinically suggestive of a lymph node mass. The node showed metastatic papillary carcinoma from the thyroid. Thyromegaly was not apparent on clinical examination. There was a bulge in the right lateral wall of the nasopharynx with intact mucosa. Computed tomography (CT) scan showed a hypodense lesion to the right of the nasopharynx, measuring 3.9 × 2.11 cm with radiologic features of a metastatic lymph node in the para-nasopharyngeal space abutting the skull base [Figure 1].
Figure 1: Case 1: CT Neck showing retronasopharyngeal node on right side. (Arrow)

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She underwent total thyroidectomy with central and bilateral selective neck dissection and excision of nasopharyngeal node through a mandibulotomy access.[6] Postoperative histopathology showed that two out of three para-nasopharyngeal lymph nodes showed metastatic papillary carcinoma. She received 205 mCi of radioiodine-131 postoperatively. Patient is currently 26 months out of treatment and is disease free.

Case 2: Parapharyngeal Nodal Metastases

A 38 year old man, who had undergone a total thyroidectomy, central compartment clearance and left modified radical neck dissection, followed by ablation with 157mCi, presented to our center 5 months later for follow up. His iodine scan was normal but thyroglobulin level was raised (935 mIU/mL) with normal thyroglobulin antibody levels. Positron emission tomography scan showed metabolically active enhancing left parapharyngeal mass lesion [Figure 2].
Figure 2: Case 2: MRI Neck showing Left parapharyngeal node (Arrow)

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He underwent transoral robot-assisted excision of the parapharyngeal mass, which on histopathology showed papillary carcinoma thyroid of 3.5 × 2 × 1.5 cm. He received 200 mCi of radioiodine postoperatively. He is loco-regionally free of disease, 14 months post-treatment.

Case 3: Jugular Venous Thrombosis in Papillary Carcinoma Thyroid

A 44-year old woman presented for follow up after total thyroidectomy, central compartment clearance, and left-level IV nodal dissection. She received 153 mCi of radioiodine treatment. On post-therapy scan, an elongated neck uptake of radioiodine was evident [Figure 3]. SPECT CT used to localize the uptake identified a thrombus in the left internal jugular vein. CT angiography showed the embolus in the left internal jugular vein with lower extent just short of superior vena cava. An embolectomy was planned but she defaulted on the procedure.
Figure 3: Case 3: Iodine scan showing uptake in left internal jugular vein (Arrow)

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Case 4: Tracheal Infiltration

A 52-year-old woman patient presented after total thyroidectomy followed by radioiodine ablation. Post-treatment scan showed a nodular infiltrating hypoechoic mass with vascularity in the upper part of the left thyroid bed extending up to the midline, with involvement of strap muscles and invasion of the anterior part of the cricoid ring and three rings of tracheal cartilage [Figure 4]. A subglottic infiltrating mass was visible on fiber-optic laryngoscopy [Figure 5]. She was diagnosed to have recurrent papillary thyroid carcinoma and underwent surgical resection as a two-stage procedure. In the first stage, two segments of the seventh rib were harvested and placed in a subcutaneous pocket in the radial artery forearm (RAFF) territory for prefabrication. In the second stage, 3 weeks following the first stage, the lesion was removed along with the involved part of cricoid ring and tracheal rings. The surgical defect was then closed with the prefabricated radial artery forearm free flap. Following surgery, she received 195 mCi radio-iodine as adjuvant therapy. She is disease-free 27 months after her treatment. She is without tracheostomy tube and has intelligible speech.
Figure 4: Case 4: MRI Neck showing infiltrating tracheal mass (Arrow)

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Figure 5: Case 4: Fibre-optic laryngoscopy showing subglottic infiltrating mass

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Case 5: Tracheal Infiltration

A 59 year old woman presented with a thyroid nodule and intermittent hemoptysis for 2 years. Examination revealed a 4 × 4 cm nodule, fixed to underlying structures. Bronchoscopy showed tracheal infiltration. CT scan showed a 4.4 × 2.4 × 4.4 cm right thyroid mass with tracheal infiltration [Figure 6]. Bronchoscopy showed infiltration of the tracheal mucosa [Figure 7]. She underwent total thyroidectomy with tracheal resection and bilateral selective neck dissection. The tracheal ends were anastomosed primarily after resection. She received radioiodine treatment (100 mCi) after surgery. She has intelligible speech and is disease-free at present, 16 months post-treatment.
Figure 6: Case 5: CT Neck showing thyroid mass infiltrating the trachea

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Figure 7: Case 5: Tracheal infiltration visible on excised specimen of total thyroidectomy

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Case 6: Tracheal Infiltration

A 14 year old girl presented with raised thyroglobulin levels, after total thyroidectomy, bilateral neck dissection and radioiodine ablation 6 years back. On examination of the neck, the neck had no palpable mass and the left vocal cord was paralysed. She had a left vocal cord palsy. CT scan of the neck showed a recurrent lesion in the left trachea-esophageal groove with suspicious involvement of the trachea and oesophagus. [Figure 8]. Bronchoscopy and upper gastrointestinal endoscopy excluded mucosal involvement. She was planned for the excision of the mass. In view of anticipated tracheal excision, prefabrication of the left radial artery forearm free flap with rib graft was done. As a second-stage surgery, the mass could be shaved off the trachea with removal of single tracheal ring. Mucosa was intact after the procedure and no reconstruction was required. She is disease-free 9 months post treatment.
Figure 8: Case 6: Recurrent thyroid mass in left tracheo-esophageal groove

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 » Discussion Top


Thyroid cancer comprises 1–5% of all cancers worldwide.[7] The most common initial presentation of PTC is thyromegaly. Incidental diagnosis of thyroid nodule in otherwise asymptomatic patients is also on the rise, due to newer diagnostic modalities, such as high-resolution ultrasonography.[8]

Presence of nodal metastasis at the time of diagnosis is evident in 40–50% of cases of PTC.[1] The nodes are usually present in the internal jugular and recurrent laryngeal chain on the side of the lesion.[9] Only 39 cases of histopathologically proven retropharyngeal or parapharyngeal nodal metastasis in PTC have been reported in the last two decades.[10] Desuter et al. reported that only 0.43% (3/696) cases of PTC had parapharyngeal nodal metastasis.[11] Kainuma et al. suggested that prior neck dissection and/or metastasis to cervical nodes may result in a retrograde flow leading to retropharyngeal nodal deposits.[10] In our report, one of the patients had a history of prior neck dissection while the other patient had lateral cervical nodes, thereby is in accordance with the theory suggested by Kainuma et al. It is reported that there are two lymphatic pathways that connected with a retro-pharyngeal metastasis from PTC, one is retrograde channel from jugular chain lymphatics and the other is direct through the superior thyroid pole by way of posterior lymphatic trunks, reported to be present in 20% of cases.[12]

Lymphatic spread is more common in PTC than hematogenous spread. Local recurrence due to microscopic vascular invasion can be seen in PTC after surgery. But invasion into the great cervical veins or an artery from the local thyroid carcinoma recurrence is rare and is associated with high mortality rate.[13] According to Graham, Kauffman described the first case of PTC thrombi.[14] An autopsy identified thyroid carcinoma with massive invasion into the superior vena cava, the right auricle, and into the internal jugular, subclavian, and innominate vein on both sides. In our case, CT angiography showed the embolus in left internal jugular vein with lower extent just short of superior vena cava. Neck ultrasound, MRI, and CT scan can be performed for the diagnosis. Radiologists should attempt to detect the signs of tumor thrombi in all patients with thyroid masses. CT can be useful to show a dilated vein with an intraluminal filling defect and adjacent mass. Absence of normal flow void on MR images is diagnostic of venous thrombosis. Intraluminal extension is not a contraindication to aggressive surgical treatment in PTC, owing to relatively good prognosis as well as decreased risk of superior vena cava obstruction, sudden death from airway occlusion, tumor embolization, or fatal right atrial obstruction.[15] The reported survival of patients with internal jugular vein invasion or other great cervical veins by thyroid cancer is 2–5 years.[16]

Laryngo-tracheal invasion occurs in 1–8% of cases of PTC, with trachea being the most common site seen in 35–60% of cases.[2] Laryngo-tracheal invasion in PTC represents a gradual mechanism of infiltration of the tracheal wall layers from the outside into the lumen of the trachea.[2] Description of inter-cartilage PTC infiltration into the trachea was first provided by Shin et al. in 1993.[17] The most effective treatment in such cases include complete surgical resection of the tumor. The surgical options include shave procedure, partial laryngectomy, and total laryngectomy. However, laryngectomy results in substantial morbidity, including dysphonia, dysgeusia, and dysosmia. A function preserving surgical method of resection and reconstruction for PTC invading into the cricotracheal region has not yet been established. Enomoto et al. in 2015 reported a surgical technique, viz., Windmill resection and Tetris reconstruction, which offered improved comfort, more aesthetic results, and good postoperative recovery.[18] In our series, we used the radial artery forearm free flap prefabricated with ribs for reconstruction of cricotracheal defect. This technique has been used for reconstruction of ear but its use for cricotracheal reconstruction has not been reported so far.[19] In our experience, the technique provided good functional and aesthetic recovery with minimal donor-site morbidity. Postoperative recovery of the patients was uneventful. The only disadvantage of this procedure was that it was a two-staged procedure, but the indolent nature of PTC allows for the two-staged procedure. This helped us avoid a laryngectomy in this patient. Tracheal invasion was treated with mobilization of the mediastinal part of the trachea and primary closure.


 » Conclusion Top


Unusual loco-regional presentation of PTC pose challenges in diagnosis and treatment. A keen clinical sense is paramount in effectively diagnosing these cases. Aggressive surgical resection and reconstruction results in good functional and aesthetic outcomes. Further studies are required for establishing specific guidelines on the approach to the treatment of these cases.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 » References Top

1.
Mazzaferri EL, Jhiang SM. Long-term impact of initial surgical and medical therapy on papillary and follicular thyroid cancer. Am J Med 1994;97:418-28.  Back to cited text no. 1
    
2.
Brauckhoff M, Dralle H. Extrathyroidal thyroid cancer: Results of tracheal shaving and tracheal resection. Chirurg 2011;82:134-40.  Back to cited text no. 2
    
3.
Song HJ, Xue YL, Xu YH, Qiu ZL, Luo QY. Rare metastases of differentiated thyroid carcinoma: pictorial review. Endocr Relat Cancer 2011;18:11-68.  Back to cited text no. 3
    
4.
Madani A, Jozaghi Y, Tabah R, How J, Mitmaker E. Rare metastases of well-differentiated thyroid cancers: A systematic review. Ann Surg Oncol 2015;22:460-6.  Back to cited text no. 4
    
5.
McCaffrey JC. Aerodigestive tract invasion by well-differentiated thyroid carcinoma: Diagnosis, management, prognosis, and biology. Laryngoscope 2006;116:1-11.  Back to cited text no. 5
    
6.
Chu F, Tagliabue M, Giugliano G, Calabrese L, Preda L, Ansarin M. From transmandibular to transoral robotic approach for parapharyngeal space tumors. Am J Otolaryngol 2017;38:375-9.  Back to cited text no. 6
    
7.
Kilfoy BA, Zheng T, Holford TR, Han X, Ward MH, Sjodin A et al. International patterns and trends in thyroid cancer incidence, 1973-2002. Cancer Causes Control 2009;20:525-31.  Back to cited text no. 7
    
8.
Enewold L, Zhu K, Ron E, Marrogi AJ, Stojadinovic A, Peoples GE et al. Rising thyroid cancer incidence in the United States by demographic and tumor characteristics, 1980-2005. Cancer Epidemiol Biomarkers Prev 2009;18:784-91.  Back to cited text no. 8
    
9.
Scheumann GF, Gimm O, Wegener G, Hundeshagen H, Dralle H. Prognostic significance and surgical management of locoregional lymph node metastases in papillary thyroid cancer. World J Surg 1994;18:559-67.  Back to cited text no. 9
    
10.
Kainuma K, Kitoh R, Yoshimura H, Usami S. The first report of bilateral retropharyngeal lymph node metastasis from papillary thyroid carcinoma and review of the literature. Acta Otolaryngol 2011;131:1341-8.  Back to cited text no. 10
    
11.
Desuter G, Lonneux M, Plouin-Gaudon I, Jamar F, Coche E, Weynand B et al. Parapharyngeal metastases from thyroid cancer. Eur J Surg Oncol 2004;30:80-4.  Back to cited text no. 11
    
12.
Rouvière H. Anatomy of human lymphatic system: English Edition. Ann Arbor, Michigan: Edwards Brothers; 1939. p. 5-65.  Back to cited text no. 12
    
13.
Niederle B, Hausmaninger C, Kretschmer G, Polterauer P, Neuhold N, Mirza DF. Intraatrial extension of thyroid cancer: Technique and results of a radical surgical approach. Surgery 1990;108:951-6.  Back to cited text no. 13
    
14.
Graham A. Malignant tumors of the thyroid: Epithelial types. Ann Surg 1925;82:30-44.  Back to cited text no. 14
    
15.
Gross M, Mintz Y, Maly B, Pinchas R, Muggia-Sullam M. Internal jugular vein tumor thrombus associated with thyroid carcinoma. Ann Otol Rhinol Laryngol 2004;113:738-40.  Back to cited text no. 15
    
16.
Boedeker CC, Ridder GJ, Weerda N, Maier W, Klenzner T, Schipper J. Etiology and therapy of the internal jugular vein thrombosis. Laryngorhinootologie 2004;83:743-9.  Back to cited text no. 16
    
17.
Shin DH, Mark EJ, Suen HC, Grillo HC. Pathologic staging of papillary carcinoma of the thyroid with airway invasion based on the anatomic manner of extension to the trachea: A clinicopathologic study based on 22 patients who underwent thyroidectomy and airway resection. Hum Pathol 1993;24:866-70.  Back to cited text no. 17
    
18.
Enomoto K, Uchino S, Noguchi H, Enomoto Y, Noguchi S. A novel surgical technique for thyroid cancer with intra-cricotracheal invasion: Windmill resection and tetris reconstruction. Indian J Surg 2015;77:319-26.  Back to cited text no. 18
    
19.
Akin S. Burned ear reconstruction using a prefabricated free radial forearm flap. J Reconstr Microsurg 2001;17:233-6.  Back to cited text no. 19
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]
 
 
    Tables

  [Table 1]



 

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