Case Report
An Unusual Cause of Hoarseness - Hamartoma Larynx.
Authors
Mona Sarawgi, Resident,
Pritikanta Sahu, Resident,
Binayak Baruah, Associate Specialist,
Alok Kumar, Senior Registrar,
KP Dubey, Senior Specialist and Head of Department,
Ajay Gupta, Senior Specialist,
Department Of ENT, Tata Main Hospital, Bistupur, Jamshedpur-831001, Jharkhand, India.
Address for Correspondence
Dr Mona Sarawgi,
Department Of ENT
Tata Main Hospital
Bistupur, Jamshedpur-831001
Jharkhand, India.
E-mail:
mona_sarawgi@yahoo.co.in
Citation
Sarawgi M, Sahu P, Baruah B, Kumar A, Dubey KP, Gupta A. An Unusual Cause of Hoarseness - Hamartoma Larynx. Online J Health Allied Scs.
2015;14(2):5. Available at URL:
http://www.ojhas.org/issue54/2015-2-5.html
Open Access Archives
http://cogprints.org/view/subjects/OJHAS.html
http://openmed.nic.in/view/subjects/ojhas.html
Submitted: Apr 22,
2015; Accepted: Jun 27, 2015; Published: July 15, 2015 |
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Abstract: A hamartoma is a benign, focal malformation that resembles a neoplasm in the tissue of its origin. It is a rare finding in the head and neck area and usually occurs as an age-related tumour in the lung. Herein, we report a case of 52 years old male presented with hoarseness and non productive cough since 2 months. On flexible direct laryngoscopy a smooth, sessile, hyperaemic mass arising from the right vocal cord was seen. Under video-endoscopy, excision of the mass was done using radiofrequency probe and the mass revealed hamartoma on histopathology. Laryngeal hamartoma should be included in the differential diagnosis of benign laryngeal lesions. Complete surgical removal is the method of choice but should be limited in order to preserve laryngeal function, as the prognosis is excellent.
Key Words:
Larynx, Hamartoma, Radiofrequency |
Introduction:
A hamartoma is characterized by the formation of a tumour like mass, composed of disorganized, but mature specialized cells or tissue elements that are indigenous to the location where it is found.(1) Albrecht introduced the term in 1901 and distinguished between true neoplasms and tumour like lesions. It may arise from any of germ layers and it does not metastasize.(2) Hamartomas may occur anywhere in the body but are encountered most frequently in the lungs, kidney, and liver. They are seldom seen in ENT practice.(3) Hamartoma of the larynx is a very rare, non-neoplastic developmental anomaly that may present clinically with symptoms of upper respiratory tract obstruction, dysphonia, choking, hoarseness and progressive, persistent stridor.(4) Pathological findings may be confused with other benign tumours of larynx including choristomas, chondromas, fibromas, chondrometaplastic nodules and angiolipomas. Since the lesion is benign and has a good prognosis unless it shows a wide expansion throughout the larynx, an adequate conservative excision must be path of management.(5)
We report a case of an adult male of laryngeal hamartoma with change in voice and non productive cough. The clinicopathological features and treatment are discussed along with review of relevant literature.
Case Report
A 52 years old male came to our outpatient department with change in voice since 2 months and non productive cough since 15 days. There was no history of throat pain, fever, difficulty in breathing or swallowing or any swelling in the neck. Patient underwent thorough clinical examination and on indirect laryngoscopic examination there was smooth, sessile, hyperemic polypoidal mass over anterior 2/3 of right vocal cord extending till anterior commissure.
Direct flexible laryngoscopy revealed smooth, sessile, hyperaemic mass that originated from the right true cord with no involvement of subglottis region (FIG 1). Computed tomography scan was done to confirm the extent of mass. Under general anaesthesia using videoendoscopy, local surgical excision of the mass was done using radiofrequency micro-larynx probe in both cut and coagulate mode, with the preservation of laryngeal function.
Fig-1 (above) Direct flexible laryngoscopy showing smooth, sessile, hyperaemic mass originating from the right true cord with no involvement of subglottis region
Fig-2 (right) Section showing squamous epithelium with fibromixoid stroma with enlarged vascular spaces (H&E *100) |
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Histopathological examination showed fragments of non-keratinizing squamous epithelium lined tissue, fibromixoid edematous lamina propria showing enlarged dilated blood filled vascular space, with no evidence of malignancy(FIG 2& 3), which was suggestive of hamartomatous vascular polyp.
At first post-operative visit, 1 week after surgery, patient’s symptoms had resolved. The surgical site had healed. There has been no evidence of recurrent lesion till last follow up of 1 year.
Discussion
Although hamartoma is a benign lesion, may become large enough to cause trouble according to size and location. It rarely becomes malignant.(3) They grow concurrently with the host forming a mass of recognizable, but unorganized tissues that contains the structure derived from any of the three germinal layers. It is very rarely described in the head and neck, with few cases reported in the larynx and pharynx.(4)
The first report of laryngeal hamartoma was published by Climie et al in 1963.(6) Zapf et al in 1981, described a supraglottic polypoidal lesion in an infant that caused stridor.(7) In adults sign and symptoms are more diverse. They may be asymptomatic detected incidentally or cause dysphagia, hoarseness of voice and stridor when large in size. Any laryngeal tumour has to be investigated by videolaryngoscopy and computed tomography scan to assess the extent of the tumour.(8) In our case, it presented as a well localized, smooth, hyperaemic, polypoidal mass causing hoarseness with nonproductive cough. Flexible laryngoscopy was suggestive of a vascular lesion of larynx. Subglottic region was clear which was also confirmed by computed tomography scan. Simple microlaryngeal excision of the polyp with radiofrequency probe reduced bleeding and postoperative pain and resulted in well preserved laryngeal function. Treatment modalities include local surgical excision with preservation of laryngeal function or debulking/resection with CO2 laser. Partial or total laryngectomy is reserved for larger lesions.
In conclusion, laryngeal hamartomas are extremely rare. They can be locally destructive and can cause airway obstruction. It should be included in the differential diagnosis of benign laryngeal lesions. Complete surgical removal is the method of choice but should be limited in order to preserve laryngeal function, as the prognosis is excellent.
References
- Buckmire RA, Kwon TK. Bilateral obstructing laryngeal epithelial adenomatous hamartomas. Arch Otolaryngol Head Neck Surg. 2005 Mar;131:259-61.
- Albrecht EU. Hamartome. Verh Dtsch Ges Pathol 1904;7:153-57
- Millas-Gomez T, Benito-Orejas JI, Morais-Pérez D, Fernandez-Salazar L, Alvarez-Quinones SM. Hamartomatous polyp of the hypopharynx. Acta Otorrinolaringol Esp. 2011;62:472-4
- Fine ED, Dahmas B, Arnold JE. Laryngeal hamartoma: A rare congenital abnormality. Ann Otol Rhinol Laryngol. 1995;104:87-9
- Archer SM, Crockett DM, McGill TJI. Hamartoma of the larynx: report of two cases and review of the literature. Int J Ped Otorhinolaryngol 1988;16(3):237
- Climie ARW, Waggoner LG, Krabbenhoft KL .Lymphoid hamartoma of the larynx. Laryngoscope. 1963; 74(10):1381-8.
- Zapf B, Lehmann WB, Snyder GG 3rd. Hamartoma of the larynx: an unusual cause for stridor in an infant. Otolaryngol Head Neck Surg 1981;89(5):797
- Windfuhr JP. Laryngeal hamartoma. Acta Otolaryngol 2004;124:301-8.
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