otolaryngologia polska 68 (2014) 46–49
Available online at www.sciencedirect.com
journal homepage: www.elsevier.com/locate/otpol
Case report/Kazuistyka
Intralaryngeal ectopic thyroid Tarczyca ektopowa w lokalizacji wewna˛ trzkrtaniowej Tomasz Zaton´ ski 1,*, Marek Bolanowski 2, Diana Je˛drzejuk 2, Katarzyna Zaton´ ska 3, Tomasz Kre˛cicki 1 Department of Otolaryngology, Head & Neck Surgery, Medical University in Wrocław, Head: Tomasz Kre˛cicki, Poland Department of Endocrinology, Diabetology and Isotope Therapy, Medical University in Wrocław, Head: Andrzej Milewicz, Poland 3 Department of Social Medicine, Medical University in Wroclaw, Head: Katarzyna Zaton´ ska, Poland 1 2
article info
abstract
Article history:
Lingual thyroid is the most common presentation of ectopic thyroid tissue. In contrast, to
Received: 05.06.2012
that laryngeal location is extremely rare. We report a case of 59 years old woman with
Accepted: 26.06.2012
a history of progressive dyspnea and nodular thyroid goiter. Endoscopic examination
Available online: 04.07.2012
revealed subglottic smooth tumor of the right side of the larynx. CT scans revealed mass localized in infraglottic part of the larynx, causing infraglottic stenosis. The biopsy of the
Keywords: Ectopic thyroid Laryngeal location Surgical treatment
tumor revealed: Struma nodosa. Reviewing the literature we found only seven cases described. We present development of the thyroid gland and origins causing ectopy. © 2012 Polish Otorhinolaryngology - Head and Neck Surgery Society. Published by Elsevier Urban & Partner Sp. z o.o. All rights reserved.
Słowa kluczowe: tarczyca ektopowa lokalizacja krtaniowa leczenie chirurgiczne
Introduction Lingual ectopy is the most common location of ectopic thyroid gland. In contrast, to that presence of ectopic tissue in the larynx is extremely rare. We have performed review of the literature using following databases: U.S. National Institutes of Health’s National Library of Medicine (NIH/NLM): http://www. ncbi.nlm.nih.gov, PMC: http://www.ncbi.nlm.nih.gov/pmc,
Pubmed: http://www.ncbi.nlm.nih.gov/pubmed and Google Scholar: http://scholar.google.pl. As a result we found seven cases of the laryngeal ectopic thyroid described.
Case report A 59-year-old woman was referred to the Otolaryngology Department of the University Hospital in Wroclaw, with
* Corresponding author at: Department of Otolaryngology, Head and Neck Surgery, Medical University of Wrocław, Borowska 213, 50-556 Wrocław, Poland. Tel.: +48 605414963. E-mail address: [email protected] (T. Zaton´ski). 0030-6657/$ – see front matter © 2012 Polish Otorhinolaryngology - Head and Neck Surgery Society. Published by Elsevier Urban & Partner Sp. z o.o. All rights reserved.
http://dx.doi.org/10.1016/j.otpol.2012.06.026
otolaryngologia polska 68 (2014) 46–49
47
Fig. 1 – The tumor
Fig. 3 – Post biopsy endoscopy of the larynx
subglottic laryngeal tumor for an evaluation and treatment. She had a 2–3 year history of progressive dyspnea, nodular thyroid goiter (not treated), arterial hypertension, hospitalized because of depression and anxiety disorders. Never smoked, not relevant family history. Laryngological examination revealed septal deviation, without significant pathology within the ear and throat. Laryngeal endoscopy demonstrated normal epiglottis and symmetric movement of the vocal folds. Subglottic smooth tumor of the right side of the larynx, on the level of cricoid cartilage. Surface of the tumor had intense vascular pattern. Diameter of the larynx was reduced by the tumor mass to 33% (Fig. 1). Autofluorescence endoscopy did not show a loss of green fluorescence (which is present in malignant lesions) (Fig. 2). The ultrasound examination of the neck revealed enlarged, heterogeneous, thyroid gland with presence of numerous normoechoic focal lesion with calcifications and fluid parts in both lobes with diameter till 1.6 cm in the right and 0.9 cm in the left lobe. Increased vascular flow pattern on Doppler examination was observed. The ultrasonography
demonstrated multinodular goiter. Along the large neck vessels on the right side and in submandibular region on the left side hypoechoic lymph nodes with hyperechoic hilum (1.2–1.6 cm) were present. CT was performed: in supraglottic and glottic part of the larynx no evidence of pathology was observed. CT scans revealed mass localized in infraglottic part of the larynx on the right wall with dimensions 2.2 cm long and 1 cm wide, causing infraglottic stenosis. Tumor had well-defined borders. Heterogeneous pathological enhancement was observed after intravenous administration of contrast. Furthermore not enlarged thyroid gland with numerous hypoechoic nodules, fibrous strands and calcifications were described. Along the course of the large cervical vessels single lymph nodes with maximal diameter 1.3 cm were observed. The decision of surgical biopsy was undertaken. In the first step of surgery patient underwent tracheotomy and further biopsy in direct laryngoscopy (in Kleinsasser suspension laryngoscope) was proceeded. Cystic tumor filled by mucosal yellow liquid was removed. The patient had no complications during the postoperative period (Fig. 3). Tracheostomic tube was removed on the second day after surgery. Patient went back home. The biopsy of the tumor revealed: Struma nodosa [M71624]. Scintigraphy of thyroid gland showed: bilobal, enlarged, proper located thyroid gland. In external lower part of the right lobe cold area without uptake of isotope. Uptake of Iodium isotope after 24 h on the level of 10.5% (Fig. 4). The patient was consulted in the Department of Endocrinology with conclusion: normal thyroid function, no need of endocrinological treatment. Two weeks later the patient was admitted again to the ENT department with progressive hoarseness and mixed (inspiratory–expiratory) dyspnea. She was qualified for surgical removal of the tumor. First condition of thyroid gland and trachea were evaluated. Frontal wall of trachea was controlled and revealed normal cartilaginous structure of the trachea without any pathological findings. Afterwards by the laryngofissure approach the tumor mass was found on the level of the cricoid cartilage and totally removed (Fig. 5). There were no complications in postoperative period. Endoscopy performed
Fig. 2 – Autofluorescence endoscopy of the tumor
48
otolaryngologia polska 68 (2014) 46–49
Fig. 4 – Scintigraphy of thyroid gland
otolaryngologia polska 68 (2014) 46–49
49
parts were completely separated. As in another location surgical excision is the mainstay of the treatment.
Authors’ contributions/Wkład autoro´w According to order.
Conflict of interest/Konflikt interesu None declared.
Financial support/Finansowanie Fig. 5 – Postoperative endoscopy of the larynx
None declared.
Ethics/Etyka on the follow up visit four weeks after operation showed good movement of the vocal folds and no presence of any subglottic lesion.
Discussion The thyroid gland develops from endoderm, arises from the base of tongue (leaving vestigal depression on the dorsal part of the tongue, the foramen cecum) and migrates downwards to the location inferior to the larynx where is located in postnatal life. In the time of development migration of the thyroid along the thyroglossal duct can cause ectopy. The most often localization of ectopic thyroid is base of tongue (lingual ectopy) with frequency 1/100,000 patients [1]. There are numerous reports of the lingual ectopic thyroid. Such cases were operated several times in the Otolaryngology Department of the University Hospital in Wroclaw with good results. To avoid removing of the only thyroid tissue scintigraphy is obligatory to determine the presence of the main thyroid gland. The literature concerning laryngeal location of the ectopic thyroid is extremely rare. We found 7 cases described [2–8]. Heinemann and Pult in their opinion first and the only authors in 1982 described a case of intralaryngeal ectopic thyroid gland [3]. In contrast to our case thyroid tissue was connected with external goiter through a hole in the cartilage disc just below the left vocal fold. We expected such an anomaly. This has been verified during the second surgery where we found intact wall of the larynx and trachea. In their case it is difficult to determine how this anatomical anomaly could develop. Hypothetically, the tissue of the thyroid gland had to migrate to the larynx before the final formation of laryngeal cartilage was done (between 10 and 13 weeks of fetal life). In our case, the continuity of thyroid tissue has been broken and both
The work described in this article have been carried out in accordance with The Code of Ethics of the World Medical Association (Declaration of Helsinki) for experiments involving humans; EU Directive 2010/63/EU for animal experiments; Uniform Requirements for manuscripts submitted to Biomedical journals. The own research were conducted according to the Good Clinical Practice guidelines and accepted by local Bioethics Committee, all patients agreed in writing to participation and these researches.
r e f e r e n c e s / p i s´ m i e n n i c t w o
[1] Gue´rin N, Urtasun A, Chauveau E, Julien M, Lebreton M, Dumon M. Lingual thyroid and intra-lingual thyroglossal cyst. Apropos of 2 cases. Revue de Laryngologie-OtologieRhinologie (Bord) 1997;118(3):183–188. [2] Bone RC, Biller HF, Irwin TM. Intralaryngotracheal thyroid. Annals of Otology Rhinology and Laryngology 1972; 81(3):424–428. [3] Heinemann M, Pult P. Intralaryngeal struma. Laryngologie Rhinologie Otologie (Stuttg) 1982;61(9):531–533. [4] Jime´nez Oliver V, Ruiz Rico R, Da´vila Morillo A, Ferna´ndez Ruiz E, Ruiz del Portal JM, Pe´rez Arcos JA, et al. Intralaryngeal ectopic thyroid tissue. Report of one case and review of the literature. Acta Otorrinolaringologica Espanola 2002;53(1):54–59. [5] Myers EN, Pantangco Jr IP. Intratracheal thyroid. Laryngoscope 1975;85(11 pt 1):1833–1840. [6] Riabina VP. Rare location of accessory lobe of thyroid gland in larynx. Vestnik Otorinolaringologii 1968;30(6): 102–103. [7] Tapia Acuna R. Aberrant thyroid gland in the larynx. Gaceta Me´dica de Me´xico 1958;88(12):899–903. [8] Waggoner LG, Intralaryngeal intratracheal thyroid. Annals of Otology Rhinology and Laryngolo 1958;67(1):61–71.
Available online at www.sciencedirect.com
journal homepage: www.elsevier.com/locate/otpol
Case report/Kazuistyka
Intralaryngeal ectopic thyroid Tarczyca ektopowa w lokalizacji wewna˛ trzkrtaniowej Tomasz Zaton´ ski 1,*, Marek Bolanowski 2, Diana Je˛drzejuk 2, Katarzyna Zaton´ ska 3, Tomasz Kre˛cicki 1 Department of Otolaryngology, Head & Neck Surgery, Medical University in Wrocław, Head: Tomasz Kre˛cicki, Poland Department of Endocrinology, Diabetology and Isotope Therapy, Medical University in Wrocław, Head: Andrzej Milewicz, Poland 3 Department of Social Medicine, Medical University in Wroclaw, Head: Katarzyna Zaton´ ska, Poland 1 2
article info
abstract
Article history:
Lingual thyroid is the most common presentation of ectopic thyroid tissue. In contrast, to
Received: 05.06.2012
that laryngeal location is extremely rare. We report a case of 59 years old woman with
Accepted: 26.06.2012
a history of progressive dyspnea and nodular thyroid goiter. Endoscopic examination
Available online: 04.07.2012
revealed subglottic smooth tumor of the right side of the larynx. CT scans revealed mass localized in infraglottic part of the larynx, causing infraglottic stenosis. The biopsy of the
Keywords: Ectopic thyroid Laryngeal location Surgical treatment
tumor revealed: Struma nodosa. Reviewing the literature we found only seven cases described. We present development of the thyroid gland and origins causing ectopy. © 2012 Polish Otorhinolaryngology - Head and Neck Surgery Society. Published by Elsevier Urban & Partner Sp. z o.o. All rights reserved.
Słowa kluczowe: tarczyca ektopowa lokalizacja krtaniowa leczenie chirurgiczne
Introduction Lingual ectopy is the most common location of ectopic thyroid gland. In contrast, to that presence of ectopic tissue in the larynx is extremely rare. We have performed review of the literature using following databases: U.S. National Institutes of Health’s National Library of Medicine (NIH/NLM): http://www. ncbi.nlm.nih.gov, PMC: http://www.ncbi.nlm.nih.gov/pmc,
Pubmed: http://www.ncbi.nlm.nih.gov/pubmed and Google Scholar: http://scholar.google.pl. As a result we found seven cases of the laryngeal ectopic thyroid described.
Case report A 59-year-old woman was referred to the Otolaryngology Department of the University Hospital in Wroclaw, with
* Corresponding author at: Department of Otolaryngology, Head and Neck Surgery, Medical University of Wrocław, Borowska 213, 50-556 Wrocław, Poland. Tel.: +48 605414963. E-mail address: [email protected] (T. Zaton´ski). 0030-6657/$ – see front matter © 2012 Polish Otorhinolaryngology - Head and Neck Surgery Society. Published by Elsevier Urban & Partner Sp. z o.o. All rights reserved.
http://dx.doi.org/10.1016/j.otpol.2012.06.026
otolaryngologia polska 68 (2014) 46–49
47
Fig. 1 – The tumor
Fig. 3 – Post biopsy endoscopy of the larynx
subglottic laryngeal tumor for an evaluation and treatment. She had a 2–3 year history of progressive dyspnea, nodular thyroid goiter (not treated), arterial hypertension, hospitalized because of depression and anxiety disorders. Never smoked, not relevant family history. Laryngological examination revealed septal deviation, without significant pathology within the ear and throat. Laryngeal endoscopy demonstrated normal epiglottis and symmetric movement of the vocal folds. Subglottic smooth tumor of the right side of the larynx, on the level of cricoid cartilage. Surface of the tumor had intense vascular pattern. Diameter of the larynx was reduced by the tumor mass to 33% (Fig. 1). Autofluorescence endoscopy did not show a loss of green fluorescence (which is present in malignant lesions) (Fig. 2). The ultrasound examination of the neck revealed enlarged, heterogeneous, thyroid gland with presence of numerous normoechoic focal lesion with calcifications and fluid parts in both lobes with diameter till 1.6 cm in the right and 0.9 cm in the left lobe. Increased vascular flow pattern on Doppler examination was observed. The ultrasonography
demonstrated multinodular goiter. Along the large neck vessels on the right side and in submandibular region on the left side hypoechoic lymph nodes with hyperechoic hilum (1.2–1.6 cm) were present. CT was performed: in supraglottic and glottic part of the larynx no evidence of pathology was observed. CT scans revealed mass localized in infraglottic part of the larynx on the right wall with dimensions 2.2 cm long and 1 cm wide, causing infraglottic stenosis. Tumor had well-defined borders. Heterogeneous pathological enhancement was observed after intravenous administration of contrast. Furthermore not enlarged thyroid gland with numerous hypoechoic nodules, fibrous strands and calcifications were described. Along the course of the large cervical vessels single lymph nodes with maximal diameter 1.3 cm were observed. The decision of surgical biopsy was undertaken. In the first step of surgery patient underwent tracheotomy and further biopsy in direct laryngoscopy (in Kleinsasser suspension laryngoscope) was proceeded. Cystic tumor filled by mucosal yellow liquid was removed. The patient had no complications during the postoperative period (Fig. 3). Tracheostomic tube was removed on the second day after surgery. Patient went back home. The biopsy of the tumor revealed: Struma nodosa [M71624]. Scintigraphy of thyroid gland showed: bilobal, enlarged, proper located thyroid gland. In external lower part of the right lobe cold area without uptake of isotope. Uptake of Iodium isotope after 24 h on the level of 10.5% (Fig. 4). The patient was consulted in the Department of Endocrinology with conclusion: normal thyroid function, no need of endocrinological treatment. Two weeks later the patient was admitted again to the ENT department with progressive hoarseness and mixed (inspiratory–expiratory) dyspnea. She was qualified for surgical removal of the tumor. First condition of thyroid gland and trachea were evaluated. Frontal wall of trachea was controlled and revealed normal cartilaginous structure of the trachea without any pathological findings. Afterwards by the laryngofissure approach the tumor mass was found on the level of the cricoid cartilage and totally removed (Fig. 5). There were no complications in postoperative period. Endoscopy performed
Fig. 2 – Autofluorescence endoscopy of the tumor
48
otolaryngologia polska 68 (2014) 46–49
Fig. 4 – Scintigraphy of thyroid gland
otolaryngologia polska 68 (2014) 46–49
49
parts were completely separated. As in another location surgical excision is the mainstay of the treatment.
Authors’ contributions/Wkład autoro´w According to order.
Conflict of interest/Konflikt interesu None declared.
Financial support/Finansowanie Fig. 5 – Postoperative endoscopy of the larynx
None declared.
Ethics/Etyka on the follow up visit four weeks after operation showed good movement of the vocal folds and no presence of any subglottic lesion.
Discussion The thyroid gland develops from endoderm, arises from the base of tongue (leaving vestigal depression on the dorsal part of the tongue, the foramen cecum) and migrates downwards to the location inferior to the larynx where is located in postnatal life. In the time of development migration of the thyroid along the thyroglossal duct can cause ectopy. The most often localization of ectopic thyroid is base of tongue (lingual ectopy) with frequency 1/100,000 patients [1]. There are numerous reports of the lingual ectopic thyroid. Such cases were operated several times in the Otolaryngology Department of the University Hospital in Wroclaw with good results. To avoid removing of the only thyroid tissue scintigraphy is obligatory to determine the presence of the main thyroid gland. The literature concerning laryngeal location of the ectopic thyroid is extremely rare. We found 7 cases described [2–8]. Heinemann and Pult in their opinion first and the only authors in 1982 described a case of intralaryngeal ectopic thyroid gland [3]. In contrast to our case thyroid tissue was connected with external goiter through a hole in the cartilage disc just below the left vocal fold. We expected such an anomaly. This has been verified during the second surgery where we found intact wall of the larynx and trachea. In their case it is difficult to determine how this anatomical anomaly could develop. Hypothetically, the tissue of the thyroid gland had to migrate to the larynx before the final formation of laryngeal cartilage was done (between 10 and 13 weeks of fetal life). In our case, the continuity of thyroid tissue has been broken and both
The work described in this article have been carried out in accordance with The Code of Ethics of the World Medical Association (Declaration of Helsinki) for experiments involving humans; EU Directive 2010/63/EU for animal experiments; Uniform Requirements for manuscripts submitted to Biomedical journals. The own research were conducted according to the Good Clinical Practice guidelines and accepted by local Bioethics Committee, all patients agreed in writing to participation and these researches.
r e f e r e n c e s / p i s´ m i e n n i c t w o
[1] Gue´rin N, Urtasun A, Chauveau E, Julien M, Lebreton M, Dumon M. Lingual thyroid and intra-lingual thyroglossal cyst. Apropos of 2 cases. Revue de Laryngologie-OtologieRhinologie (Bord) 1997;118(3):183–188. [2] Bone RC, Biller HF, Irwin TM. Intralaryngotracheal thyroid. Annals of Otology Rhinology and Laryngology 1972; 81(3):424–428. [3] Heinemann M, Pult P. Intralaryngeal struma. Laryngologie Rhinologie Otologie (Stuttg) 1982;61(9):531–533. [4] Jime´nez Oliver V, Ruiz Rico R, Da´vila Morillo A, Ferna´ndez Ruiz E, Ruiz del Portal JM, Pe´rez Arcos JA, et al. Intralaryngeal ectopic thyroid tissue. Report of one case and review of the literature. Acta Otorrinolaringologica Espanola 2002;53(1):54–59. [5] Myers EN, Pantangco Jr IP. Intratracheal thyroid. Laryngoscope 1975;85(11 pt 1):1833–1840. [6] Riabina VP. Rare location of accessory lobe of thyroid gland in larynx. Vestnik Otorinolaringologii 1968;30(6): 102–103. [7] Tapia Acuna R. Aberrant thyroid gland in the larynx. Gaceta Me´dica de Me´xico 1958;88(12):899–903. [8] Waggoner LG, Intralaryngeal intratracheal thyroid. Annals of Otology Rhinology and Laryngolo 1958;67(1):61–71.
