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Unilateral Proptosis as the Presenting Feature of Papillary Thyroid Carcinoma.

INTRODUCTION

The most common malignancy involving the thyroid gland is papillary thyroid carcinoma (PTC; 75%), with a rare distant metastasis (1%-2%) (1). Orbital metastasis from thyroid malignancy is observed in 3%-6% cases, mostly from follicular or medullary carcinoma (1). A recent review has documented four cases of orbital metastasis from PTC (2). Thus far, only one case of PTC involving muscles in the orbit has been published (3). Here, we describe a patient with orbital metastasis involving muscles as the primary clinical manifestation of PTC.

CASE REPORT

A 69-year-old-female with unilateral proptosis in her right eye for 1 year presented at the ophthalmology clinic (Figure 1). She had a history of degenerative myopia and low vision for several years. Upon examination, her best-corrected visual acuity was light perception in the right eye and 20/200 in the left eye; a fixed dilated pupil was noted in the right eye. Measurements with the Hertel exophthalmometer showed values of 30 mm for the right eye and 27 mm for the left eye (base 102 mm). A dilated fundus examination revealed posterior staphylomas and other degenerative myopic changes in both eyes and optic disc pallor in the right eye. Orbital computed tomography and magnetic resonance imaging scans showed an extra-axial, well-defined, 30x30x45-mm mass located in the middle cranial fossa, next to the temporal pole, with its borders indistinguishable from the leptomeningeal and osseous tissues, associated with sphenoidal bone lysis and invading the retrobulbar fat tissue (Figure 2). She was submitted to surgery by the neurosurgery department with pterional craniotomy. Gross anatomic finding was noted in the surgery as an elastic, purple-colored, hemorrhagic mass originating from the muscle cone. At this stage, the presumptive diagnosis was rhabdomyosarcoma. The mass was completely resected.

A histopathological examination revealed an infiltrative neoplastic epithelial lesion, neoplastic cells having a medium-sized eosinophilic cytoplasm, and mildly pleomorphic round vesicular nuclei. Some nuclei had a ground glass appearance and nuclear overlapping. Neoplastic cells mostly had a follicular appearance, with some papillary projections. There were few colloid-like materials in certain follicular neoplastic areas. The neoplastic cells infiltrated muscular fibers and bone trabeculae. An immunohistochemical analysis showed pancytokeratin, TTF-1, thyroglobulin, cytokeratin 19, galectin, and epithelial membrane antigen positivity and the absence of staining against estrogen and progesterone receptors, mesothelin, and S-100. These findings were consistent with the follicular variant of PTC (Figure 3).

The patient was referred to the surgical department of our hospital. She was clinically and biochemically euthyroid. The thyroidstimulating hormone level was 2.13 pUI/mL, free T3 level was 2.55 pg/mL, and free T4 level was 1.28 ng/dL. At this time, the serum thyroglobulin level was 290 ng/mL (immunometric assay) and antithyroglobulin antibody level was 8.07 U/mL (quimioluminescence assay). A thyroid ultrasonography revealed an increased left lobe with multiple nodules, with the largest nodule sized 2.5x3 cm in diameter having peripheral calcifications and internal microcalcifications. She also had presumed images of metastatic lymph nodes in the paravascular region in zones 3-4 beside the internal jugular vein, with the biggest node sized 15x11 mm in diameter. The fine-needle aspiration biopsy of nodules revealed benign lesions. The patient was submitted for bilateral total thyroidectomy with left modified radical neck dissection. The histopathological examination confirmed the diagnosis of PTC without any invasion to the extrathyroidal soft tissue. She was transferred to the nuclear medicine department for further radioiodine therapy.

DISCUSSION

Orbital metastasis from all types of cancer is rare. The presenting signs of patients treated for orbital metastasis of any origin by frequency are diplopia, proptosis, pain, decreased vision, and ptosis.(4) The most common symptom of orbital metastasis from various thyroid cancers is proptosis.(2) Our patient had a history of low vision and pseudoproptosis due to high myopia and axial length, which might have led to her delay in the recognition of symptoms.

There are four cases in literature documenting the orbital metastasis of PTC.(1, 3, 5, 6) Three of these cases had distant organ metastasis other than orbit at the time of diagnosis, and only one case similar to the present case showed no other site of distant metastasis other than orbit. In their case presented by Boughattas et al. (6), the patient had a history of the diagnosis of PTC and sites of metastasis in the lung and on the supraorbital ridge were detected on postablative imaging and was confirmed via radiological imaging. Rocha Filho et al. (5) have also documented a case with orbital metastasis of PTC on the frontal bone as the initial clinical manifestation of the tumor and treated with radiotherapy and chemotherapy. The case also had diffuse bone metastasis. Repanos et al. (3) have demonstrated a case of orbital metastasis of PTC to the medial rectus muscle, similar to our case, in which muscles were involved. Surgical exenteration was the treatment option for the case because of debilitating diplopia and visual acuity showing an unlikely improvement with any other treatment option. Shyla et al. (1) have reported a case of a well-encapsulated mass involving the orbit and extending to ethmoids, sphenoid sinus, and nasal cavity. Both histopathological examination of the incisional biopsy and total surgical of the mass revealed acinic cell carcinoma of the minor salivary gland; however, they associated the lesion with orbital metastasis of PTC after the histopathological analysis showed the diagnosis of PTC from thyroidectomy specimen.

CONCLUSION

This case demonstrates a very rare presentation of PTC with distant orbital metastasis. Our case shows that special care should be taken while examining patients with a long history of low vision because the most common symptoms of orbital tumors, such as diplopia, loss of vision, and proptosis, may remain unnoticed by these patients in the absence of a specific evaluation. Moreover, although very rare for orbital masses, thyroid should also be considered while assessing the primary cause for the most common suspected sites.

Informed Consent: Written informed consent was obtained from the patient who participated in this study.

Peer-review: Externally peer-reviewed.

Author Contributions: Conceived and designed the experiments or case: DGS., MU., OK., AS. Performed the experiments or case: DGS., OK., AS., Analyzed the data: DGS., MU., OK. Wrote the paper: DGS., MU. All authors have read and approved the final manuscript.

Conflict of Interest: No conflict of interest was declared by the authors.

Financial Disclosure: The authors declared that this study has received no financial support.

REFERENCES

(1.) Shyla PR, Nair RM, Somanathan T. Rare case of orbital tumor. Indian J Otolaryngol Head Neck Surg 2007; 59(2): 174-5. [CrossRef]

(2.) Besic N, Luznik Z. Choroidal and orbital metastases from thyroid cancer. Thyroid 2013; 23(5): 543-51. [CrossRef]

(3.) Repanos C, Ho YM, Bird K, Coman S, Coman WB, Wallwork B. Metastatic papillary thyroid carcinoma involving orbit: a case report and review. ANZ J Surg 2011; 81(5): 375-6. [CrossRef]

(4.) Char DH, Miller T, Kroll S. Orbital metastases: diagnosis and course. Br J Ophthalmol 1997; 81(5): 386-90. [CrossRef]

(5.) Rocha Filho FD, Lima GG, Ferreira FV, Lima MG, Hissa MN. Orbital metastasis as primary clinical manifestation of thyroid carcinoma --case report and literature review. Arq Bras Endocrinol Metabol 2008; 52(9): 1497-500. [CrossRef]

(5.) Rocha Filho FD, Lima GG, Ferreira FV, Lima MG, Hissa MN. Orbital metastasis as primary clinical manifestation of thyroid carcinoma --case report and literature review. Arq Bras Endocrinol Metabol 2008; 52(9): 1497-500. [CrossRef]

Duygu Gulmez Sevim (1) [iD], Metin Unlu (1) [iD], Olgun Kontas (2) [iD], Ahmet Selcuklu (3) [iD]

(1) Department of Ophthalmology, Erciyes University Faculty of Medicine, Kayseri, Turkey

(2) Department of Medical Pathology, Erciyes University Faculty Faculty of Medicine, Kayseri, Turkey

(3) Department of Neurosurgery, Erciyes University Faculty of Medicine, Kayseri, Turkey

Submitted 25.09.2017

Accepted 11.12.2017

Correspondence Duygu Gulmez Sevim, Department of Ophthalmology, Erciyes University Faculty of Medicine, Kayseri, Turkey Phone: +90352 207 66 66 e.mail: duygugsevim@gmail.com

DOI: 10.5152/etd.2018.17111

Cite this article as: Gulmez Sevim D, Unlu M, Kontas O, Selcuklu A. Unilateral proptosis as the presenting feature of papillary thyroid carcinoma. Erciyes Med J 2018; 40(1): 50-3.
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Title Annotation:CASE REPORT
Author:Sevim, Duygu Gulmez; Unlu, Metin; Kontas, Olgun; Selcuklu, Ahmet
Publication:Erciyes Medical Journal
Article Type:Report
Date:Mar 1, 2018
Words:1349
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