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CLINICAL FORENSIC MEDICINE Journal of Clinical Forensic Medicine 13 (2006) 349–352 www.elsevier.com/locate/jcfm
Case report
Fatal asphyxia by a thyroglossal duct cyst in an adult Anny Sauvageau MD, MSc (Forensic Pathologist) *, E´milie P. Belley-Coˆte´ (Medical Student), Ste´phanie Racette BSc (Research Assistant) Laboratoire de Sciences Judiciaires et de Me´decine Le´gale, E´difice Wilfrid-Derome, 1701 Parthenais Street, 12th Floor, Montreal, Que., Canada. H2K 3S7 Available online 5 October 2006
Abstract Thyroglossal duct cysts arise from remnants of embryonic thyroglossal duct that connects the foramen cecum at the base of the tongue to the thyroid gland. The remnants enlarge secondary to secretions from the epithelial lining. Usually, thyroglossal cysts present as non-tender masses. However, they may also become infected, produce fistulas or give hoarseness and dysphagia. Rarely, especially if the mass is located at the base of the tongue, airway obstruction and dyspnea can ensue. This unusual presentation has been mainly seen in very young children and has caused death in about half of these cases. Nevertheless, in the adult population, very few cases of airway obstruction by thyroglossal duct cysts have been reported, only one being fatal. We present the case of a 55-year-old man who died from fatal asphyxia caused by a thyroglossal cyst. 2006 Elsevier Ltd and AFP. All rights reserved. Keywords: Thyroglossal cyst; Asphyxia; Autopsy; Forensic
1. Introduction Thyroglossal duct cysts result from remnants of embryonic thyroglossal duct. During embryonic development, the thyroid gland arises form a diverticulum at the base of the tongue and migrates down the neck through a canal called the thyroglossal duct. This duct normally degenerates once the thyroid reaches its final position in the neck. If degeneration does not occur, remnants enlarge secondary to secretions form epithelium lining.1,2 Such thyroglossal duct cysts may develop anywhere along the path of the persistent thyroglossal duct: 60% are located between the hyoid bone and the thyroid gland, 24% suprahyoidally, 13% suprasternally and 1% intralingually.3 Grossly, the thyroglossal cyst usually contains a tangrey mucoid fluid. Microscopically, epithelium lining may be ciliated, cuboidal, squamous, columnar or transitional epithelium. Thyroid follicles are sometimes present in the cyst wall, as well as lymphocytic infiltrate.1,2
Thyroglossal duct cysts are the most common congenital neck mass, accounting for about 40% of cases.3 Although they generally manifest clinically in children, the lesion can also manifest in adults, even in elderly persons.4–6 Usual symptom consists of a painless anterior neck mass. Cysts infection, with pain, redness and swelling of the neck is a usual complication, as well as fistulas and spontaneous rupture.7,8 Less encountered complications have been reported: modification of the voice,9–13 dysphagia,10,14–16 dyspnea14,15,17 and snoring.9,18 Although there are some reports of infant death from fatal airway obstruction by thyroglossal duct cyst, there is however only one old report from such a fatal case in an adult.19 It is the case of a healthy 35-year-old man who died from sudden fatal asphyxia from rupture of an unsuspected thyroglossal cyst with secondary inflammatory swelling of the epiglottis and tongue. We here report a case of fatal asphyxia from respiratory tract compression by a thyroglossal duct cyst. 2. Case report
*
Corresponding author. Tel.: +1 514 873 3300; fax: +1 514 873 4847. E-mail address:
[email protected] (A. Sauvageau).
A 55-year-old man was found dead in his bed, lying supine. Having no particular medical history, the autopsy
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was done at the forensic institute to exclude a violent death. The man was a three-pack-a-day smoker. Family members reported a recent history of shortness of breath, which the victim attributed to his smoking habits and did not consult for. Autopsy revealed a morbid obese white man of 1.68 m (5 0 600 ) and 109.09 kg (240 lb) (BMI of 38.6), with a short thick neck (Fig. 1). A 2.5 · 2.3 · 1.5 cm thyroglossal cyst was found tied to the mid portion of the hyoid bone (Fig. 2). Extending above the latter towards the base of the tongue, the cyst was pressing on the anterior aspect of the lower portion of the epiglottis. On cut section, a tan-grey mucoid fluid filled the cyst (Fig. 3). Microscopically, the cyst wall was lined by pseudostratified ciliated epithelium (Fig. 4). Apart from the thryroglossal cyst, autopsic findings compatible with asphyxia were noted: presence of spuma in the lower respiratory tract as well as heavy and congested lungs (right lung of 760 g and left lung of 740 g). The rest of the autopsy was negative, except for the presence of very mild
Fig. 2. (a) A 2.5 cm cyst tied to the mid portion of the hyoid bone and extending above. (b) Lower view.
Fig. 3. Grossly, on cut section, cyst contained a tan-grey mucoid fluid.
Fig. 1. (a,b) The victim, with morbid obesity and a short thick neck, found dead in his bed, lying supine.
cardiac atherosclerosis (460 g heart with less than 25% coronary stenosis). Despite the heart weight, there were no autopsy signs of heart failure: absence of heart failure cells (i.e., haemosiderin filled macrophages) in the lungs and of centrilobular necrosis in the liver. Heart microscopic exam did not reveal any evidence of recent or old infarct. There was no significant sign of chronic obstructive pulmonary disease. Toxicological analyses were negative.
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Table 1 Cases of asphyxia by thyroglossal cyst in infants reported in the literature Ref.
Author
Number of cases
Age at presentation
Deaths
[20] [21] [22] [23] [24] [25]
Lewison and Lim Paez, Warren and Srouji Solomon and Rangecroft Hanzlick Byard, Bourne and Silver Samuel, Freeman and Sajwany Kuint et al. Weldon and Krafcik Mahboudi and Gheyi
2 1 1 1 2 2
6 and 17 weeks 6 months 1 day 4 weeks 3 months 10 weeks and 3 months At birth 39 days ? (<21 months)
1 – – 1 2 –
[26] [27] [28]
Fig. 4. Under the microscope, epithelium lining was of the pseudostratified ciliated type: (a) 250· and (b) 400·.
Cause of death was attributed to asphyxia by airway compression secondary to a thyroglossal cyst in a morbid obese man. 3. Discussion Upon neck dissection, it was clearly demonstrated that the cyst was tied to the mid portion of the hyoid bone and extending above it, therefore, pressing just at the base of the epiglottis. This particular position was thought to have created a valve effect on the epiglottis (i.e., closure of the trachea by the pressed epiglottis), with secondary asphyxia by choking. The victim’s recent complaints of shortness of breath further supports this conclusion. In infants, airway obstruction by thyroglossal cyst is a well-known phenomenon,20–28 with a lot of those cases being fatal20,23,24,26 (Table 1). In adults however, thyroglossal duct cyst is generally a benign condition that does not cause serious health problems. Nevertheless, on rare occasions, these cysts may, by virtue of their position and size, produce significant obstruction of the airway even in the adult population.9 Perez-Calderon et al. described in 2003 the case of a 55year-old woman who presented with a 2-year history of 5–
2 1 2
1 – –
10 min length recurrent episodes of cough, dyspnea and stridor. Cough was related to some body positions, such as hyperextension of the neck. Physical examination revealed an obese woman with a short thick neck, without any evident mass on neck palpation. Pulmonary function tests (spirometry with flow-volume loops) showed a cutoff of the inspiratory peak in conjunction with normal expiratory measurements. The symptoms were found to be caused by a 4 cm thyroglossal duct cyst which caused substantial obstruction of the airway at the level of the epiglottis.17 Olumide reported the case of a 23-year-old man with a 1-year history of swelling in the anterior aspect of the neck, associated with hoarseness of the voice, marked snoring during sleep and shortness of breath at rest or on moderate exertion. Symptoms disappeared after surgical excision of a 6 cm thyroglossal duct cysts located over the thyroid cartilage.9 Finally, there is also the case of a 68-year-old man with a huge anterior neck swelling from a thyroglossal duct cyst causing difficulty in breathing, particularly on exertion, along with dysphagia. The man’s symptoms disappeared after surgical excision.14 In the literature, there is only one case of fatal asphyxia by a thyroglossal duct cyst in an adult. It is the case of a healthy 35-year-old man who suddenly presented throat pain and inability to swallow. Even though a doctor was called, the cause of the symptoms could not be discovered. Shortly afterwards, the patient developed breathing difficulty and died from asphyxia within 1 h after the initial symptoms. Autopsy revealed a 1.5 cm ruptured thyroglossal duct cyst with secondary swelling of the epiglottis and tongue.19 Our case is thus the second reported case of fatal asphyxia by a thyroglossal cyst in an adult. Acknowledgments The authors thank the Laboratoire de sciences judiciaires et de me´dicine le´gale for their support, especially M. Yves Bob Dufour, director of the laboratory, as well as Mme Gabrielle Tre´panier, chief of the legal medicine section.
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