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Laryngeal amyloidosis: An uncommon cause of dysphonia
V. J.. Villagómez-Ortiza, M.. Villegas-Gonzáleza, J. L.. Treviño-Gonzáleza, R.. Santos-Lartiguea, B.. González-Andradea, N.. Montemayor-Peñaa
a Otolaryngology and Head and Neck Surgery Departament, “Dr. José Eleuterio González” University Hospital, Universidad Autónoma de Nuevo León, Monterrey, N.L., Mexico
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    "textoCompleto" => "<p class="elsevierStylePara"><span class="elsevierStyleBold">Introduction </span></p><p class="elsevierStylePara"> Amyloidosis is used to describe a range of disorders defined by extracellular deposition of abnormal protein fibrils&#46; Amyloid deposits were first described by Von Rokitansky in 1842&#44; and the term amyloid was first used by Virchow in 1851 to describe the reaction of this tissue to iodine and sulfate&#46;<span class="elsevierStyleSup">1 </span>Borrow and Neuman &#40;1873&#41; were the first to report laryngeal amyloidosis&#46; Over 300 cases have been reported since then&#46;<span class="elsevierStyleSup">2</span> Current classifications of amyloidosis are based on the biochemical nature of the protein subunit &#40;Chastonay&#44; Hurliman in 1986 and Lewis et al&#46; in 1992&#41;&#46; AL amyloidosis &#40;derived from light immunoglobulin chains&#41; is associated with systemic amyloidosis&#46;</p><p class="elsevierStylePara"> Fibrillar proteins have a characteristic pattern when observed under an electronic microscope after Congo red staining&#44; displaying apple-green birefringence when examined with polarized light&#46;<span class="elsevierStyleSup">3</span></p><p class="elsevierStylePara"> The larynx is the most common site of localized amyloidosis in the head and neck region and constitutes less than 1&#37; of benign laryngeal lesions&#46;<span class="elsevierStyleSup">4</span> Hoarseness is the most common symptom&#46; The male-female ratio is 3&#58;1&#44; with an age range between 8 and 80 years old&#44; and a peak incidence occurring in the 5<span class="elsevierStyleSup">th</span> decade of life&#46;<span class="elsevierStyleSup">5-7</span></p><p class="elsevierStylePara"> There are different treatments and resection methods for these lesions&#44; though a few stand out&#44; such as resection with a CO<span class="elsevierStyleInf">2</span> laser and cold technique&#46; Radiotherapy has recently been described in cases where the lesions cannot be completely resected&#46; Follow up is important in the long term&#44; because these lesions have a high recurrence rate&#46;<span class="elsevierStyleSup">3&#44;4&#44;8&#44;9 </span></p><p class="elsevierStylePara"> Methods</p><p class="elsevierStylePara"> Presented are 4 cases of patients with localized laryngeal amyloidosis who were treated at the Otolaryngology and Head and Neck Surgery Service at the <span class="elsevierStyleItalic">&#8220;Dr&#46; Jos&#233; Eleuterio Gonz&#225;lez&#8221; </span>University Hospital in Monterrey&#44; Mexico&#46;</p><p class="elsevierStylePara"> A flexible nasal fiber laryngoscopy was performed on all of the patients&#46; Lab studies were ordered to rule out the presence of a systemic disease&#58; a complete blood count &#40;CBC&#41; and basic metabolic panel&#44; urinalysis&#44; hepatic function test&#44; thoracic X-rays&#44; and a full physical examination was performed&#44; all of which were reported to be within normal parameters&#46; Rectal or bone marrow biopsies were not performed&#46; Maximum phonation time&#44; voice handicap index&#44; reflux symptoms &#40;described by Belafsky et al&#46;&#41;&#44; and glottic function and features in all patients were measured&#46;</p><p class="elsevierStylePara"> A resection by microlaryngoscopy with cold technique on all the lesions was performed&#46; Congo red stains were applied to all surgical specimens&#46;</p><p class="elsevierStylePara"> The approval of the ethics Committee of the <span class="elsevierStyleItalic">&#8220;Dr&#46; Jos&#233; Eleuterio Gonz&#225;lez&#8221;</span> University Hospital was obtained&#44; as well as signed informed consents from all the patients&#46;</p><p class="elsevierStylePara"><span class="elsevierStyleBold">Cases presentation </span></p><p class="elsevierStylePara"> A laryngeal amyloidosis diagnosis was performed on 3 men and one woman&#44; whose ages ranged from 14 to 67 years&#46; The most common symptoms are&#58; hoarseness&#44; pharyngodynia&#44; vocal pauses&#44; and vocal fatigue&#46; The duration of the symptoms before diagnosis was between 2 and 12 years&#46;</p><p class="elsevierStylePara"> Diagnosis and treatment were implemented to rule out systemic involvement&#59; the results were negative in all patients&#46;</p><p class="elsevierStylePara"> Resections of the lesions by microlaryngoscopy with cold technique were executed&#44; and samples were sent out for histopathology analysis&#46; Congo red staining was used&#44; where we were able to observe green birefringence through the use of the electronic microscope&#46;</p><p class="elsevierStylePara"> The results of the questionnaires conducted before and after the surgical procedure were documented&#46; With the use of this technique we were able to observe favorable results in the subjective symptoms as well as in the maximum phonation time&#46;</p><p class="elsevierStylePara"> Case 1</p><p class="elsevierStylePara"> A 67-year-old male&#44; came to the Voice Clinic for the first time presenting hoarseness with an evolution of 10 years&#44; progressive&#44; vocal fatigue&#44; glottic leak and inability to reach high notes&#46; He denies any systemic diseases or smoking&#46; He scored a glottic closure force index of 9&#44; reflux index of 8 and quality of life index of 3&#46;</p><p class="elsevierStylePara"> At the physical examination&#58; Maximum phonation time was 17 seconds&#44; S&#58;Z ratio of 25&#58;27&#46; The videolaryngoscopy displayed an elevated lump towards the right false vocal cord and anterior commissure &#40;Fig&#46; 1&#41;&#46;</p><p class="elsevierStylePara"><img alt="Figure 1 Patient 1&#44; first visit&#46; An increase in submucus volume can be observed in the photograph&#44; altering the anatomy of the false vocal cords at the supraglottic level&#46;" src="304v16n64-90367590fig1.jpg"></img></p><p class="elsevierStylePara"><span class="elsevierStyleBold">Figure 1 </span>Patient 1&#44; first visit&#46; An increase in submucus volume can be observed in the photograph&#44; altering the anatomy of the false vocal cords at the supraglottic level&#46;</p><p class="elsevierStylePara"> Case 2</p><p class="elsevierStylePara"> A 52-year-old female&#44; teacher&#44; came to the Voice Clinic as a result of hoarseness with a 3-year evolution&#44; accompanied by vocal fatigue&#44; vocal pauses and inability to reach high notes&#46; She did not improve by resting her voice&#46; She denies smoking or ethilism&#46; She previously received steroids and antibiotics without improvement&#46;</p><p class="elsevierStylePara"> Her glottic closure force index was 12&#44; reflux index of 17 and quality of life index of 7&#46; Her maximum phonation time was 7 seconds&#44; S&#58;Z ratio of 5&#58;3&#46; We were able to observe a major glottis edema with the videolaryngoscopy &#40;Fig&#46; 2&#41;&#46; A phonomicrosurgery with cold technique was performed&#44; and the sample was sent for histopathological analysis with an amyloidosis report&#46;</p><p class="elsevierStylePara"><img alt="Figure 2 Patient 2&#44; first visit&#46; An increase in volume is observed in the left lower lip of the true vocal cord&#44; creating premature contact upon glottal closure&#46;" src="304v16n64-90367590fig2.jpg"></img></p><p class="elsevierStylePara"><span class="elsevierStyleBold">Figure 2 </span>Patient 2&#44; first visit&#46; An increase in volume is observed in the left lower lip of the true vocal cord&#44; creating premature contact upon glottal closure&#46;</p><p class="elsevierStylePara"> Post-surgically&#44; she showed an improvement in her maximum phonation time to 12 seconds&#44; a glottic force closure rate index to 4&#44; reflux index to 4&#44; and a quality of life index of 3 &#40;Fig&#46; 3&#41;&#46;</p><p class="elsevierStylePara"><img alt="Figure 3 Patient 2&#44; one month post-surgery&#46; A lessening of the increase in volume can be observed at the true vocal cords&#46; There is now no premature contact upon glottal closure&#46;" src="304v16n64-90367590fig4.jpg"></img></p><p class="elsevierStylePara"><span class="elsevierStyleBold">Figure 3 </span>Patient 2&#44; one month post-surgery&#46; A lessening of the increase in volume can be observed at the true vocal cords&#46; There is now no premature contact upon glottal closure&#46;</p><p class="elsevierStylePara"> Case 3</p><p class="elsevierStylePara"> A 33-year-old male&#44; presenting hoarseness with an evolution of 3 years&#44; accompanied by vocal fatigue&#44; vocal pauses and inability to reach high notes&#44; occasional ethilism and denied smoking&#46;</p><p class="elsevierStylePara"> At physical examination&#58; Glottic leak&#44; a glottic force closure rate index of 19&#44; reflux index of 25&#44; and a quality of life index of 18&#44; maximum phonation time was 9 seconds&#44; S&#58;Z ratio of 32&#58;8&#46; The videolaryngoscopy displayed an elevated lump towards the right false vocal cord which impedes glottic closure&#44; with a wide glottal gap &#40;Fig&#46; 4&#41;&#46; A resection using the cold technique was performed&#44; and we confirmed a diagnosis of amyloidosis&#46; Post-surgical data&#58; Glottic force closure rate index of 10&#44; reflux index of 5&#44; quality of life index of 9&#44; and maximum phonation time of 16 seconds &#40;Fig&#46; 5&#41;&#46;</p><p class="elsevierStylePara"><img alt="Figure 4 Patient 3&#44; first visit&#46; Increased exophytic volume can be seen&#44; at the right level of the false vocal cords&#44; which does not obstruct the glottal light&#46; And a plain deformity of the right true vocal cord with premature contact and alteration of the chordal wave vibration&#46;" src="304v16n64-90367590fig3.jpg"></img></p><p class="elsevierStylePara"><span class="elsevierStyleBold">Figure 4 </span>Patient 3&#44; first visit&#46; Increased exophytic volume can be seen&#44; at the right level of the false vocal cords&#44; which does not obstruct the glottal light&#46; And a plain deformity of the right true vocal cord with premature contact and alteration of the chordal wave vibration&#46;</p><p class="elsevierStylePara"><img alt="Figure 5 Patient 3&#44; one month post-surgery&#46; There is no evidence of supraglottic lesions&#44; or obstruction of glottic light&#46;" src="304v16n64-90367590fig5.jpg"></img></p><p class="elsevierStylePara"><span class="elsevierStyleBold">Figure 5 </span>Patient 3&#44; one month post-surgery&#46; There is no evidence of supraglottic lesions&#44; or obstruction of glottic light&#46;</p><p class="elsevierStylePara"> Case 4</p><p class="elsevierStylePara"> A 14-year-old male with hoarseness&#44; with a 2-year evolution&#44; progressive&#44; with vocal fatigue and vocal pauses during conversation&#46;</p><p class="elsevierStylePara"> A videolaryngoscopy was performed&#44; where we were able to observe a lump which deformed both false and true vocal cords&#44; thus conditioning a premature glottic closure &#40;Fig&#46; 6&#41;&#46;</p><p class="elsevierStylePara"><img alt="Figure 6 Patient 4&#44; first visit&#46; Submucous lesions at the left false vocal cord level can be observed&#46;" src="304v16n64-90367590fig6.jpg"></img></p><p class="elsevierStylePara"><span class="elsevierStyleBold">Figure 6 </span>Patient 4&#44; first visit&#46; Submucous lesions at the left false vocal cord level can be observed&#46;</p><p class="elsevierStylePara"> Glottic force closure rate index of 13&#44; reflux index of 10&#44; and a quality of life index of 6&#44; maximum phonation time was 12 seconds&#44; S&#58;Z ratio of 17&#58;12 seconds&#46;</p><p class="elsevierStylePara"> The same surgical procedure was executed&#44; and we sent a sample for histopathological analysis&#46; Post-surgical data&#58; Maximum phonation time of 21 seconds&#44; glottic force closure rate index of 4&#44; reflux index of 2&#44; quality of life index of 5 &#40;Fig&#46; 7&#41;&#46;</p><p class="elsevierStylePara"><img alt="Figure 7 Patient 4&#44; post-surgery&#46; No evidence of lesions at the supraglotic or glotic level&#46;" src="304v16n64-90367590fig7.jpg"></img></p><p class="elsevierStylePara"><span class="elsevierStyleBold">Figure 7 </span>Patient 4&#44; post-surgery&#46; No evidence of lesions at the supraglotic or glotic level&#46;</p><p class="elsevierStylePara"><span class="elsevierStyleBold">Discussion </span></p><p class="elsevierStylePara"> The larynx is the most common site for primary amyloidosis in the head and neck region&#46; The male-female ratio in our patients was the same as the one reported in medical literature&#44; with a variation in age range&#46;</p><p class="elsevierStylePara"> In order of frequency&#44; lesions were most commonly found in the ventricle&#44; vestibular folds&#44; vocal folds&#44; epiglottis&#44; and aryepiglottic folds&#46; In our patients the most common was the involvement of false vocal cords&#46;</p><p class="elsevierStylePara"> In general&#44; the symptoms of this pathology are specific and do not lead to diagnosis&#44; thus requiring a high level of suspicion&#44; because they will appear depending on the size and site of the lesion&#46; The most common reported symptom in our patients was hoarseness and vocal fatigue&#46;</p><p class="elsevierStylePara"> It is important to rule out the involvement of a systemic disease in patients with this pathology&#46; It is worth noting the fact that in order to obtain our diagnosis&#44; we did not perform any invasive procedures in our patients &#40;i&#46; E&#46; Rectal or bone marrow biopsies&#41;&#46;</p><p class="elsevierStylePara"> The treatment for these patients is surgical&#44; and a complete resection is essential to avoid recurrence&#59; however&#44; there are few comparative studies between lesion resection with cold technique and laser to estimate their recurrence&#46; Studies have suggested the use of radiotherapy combined with surgery in those lesions where complete resection is not possible&#46;<span class="elsevierStyleSup">7 </span></p><p class="elsevierStylePara"><span class="elsevierStyleBold">Conclusions </span></p><p class="elsevierStylePara"> Laryngeal amyloidosis is a rare disease with a rate of 8 in 1&#44;000&#44;000 and a higher incidence during the 5<span class="elsevierStyleSup">th</span> decade of life&#46; Diagnosis is confirmed by Congo red staining observing an apple-green birefringence under an electronic microscope&#46; Resection with cold technique is an adequate method to remove lesions with positive subjective and objective results&#46; It is important to rule out a systemic involvement in these patients&#46;</p><p class="elsevierStylePara"><span class="elsevierStyleBold">Conflicts of interest </span></p><p class="elsevierStylePara"> The authors have no conflicts of interest to declare&#46;</p><p class="elsevierStylePara"><span class="elsevierStyleBold">Funding </span></p><p class="elsevierStylePara"> No financial support was provided&#46;</p><hr></hr><p class="elsevierStylePara"> Received&#58; November 2013&#59; <br></br> Accepted&#58; January 2014</p><p class="elsevierStylePara"> &#42; Corresponding author&#58; <br></br> Otolaryngology and Head and Neck Surgery Departament&#44; <br></br><span class="elsevierStyleItalic">&#8220;Dr&#46; Jos&#233; Eleuterio Gonz&#225;lez&#8221;</span> University Hospital&#46; <br></br> Francisco I&#46; Madero and Gonzalitos Avenue&#44; <br></br> Mitras Centro&#44; Monterrey&#44; N&#46;L&#46;&#44; Mexico&#46; <br></br> Telephone&#58; &#40;81&#41; 8333 2917&#46; <br></br><span class="elsevierStyleItalic">E-mail address</span>&#58; <a href="mailto&#58;vicentevilla&#64;yahoo&#46;com" class="elsevierStyleCrossRefs">vicentevilla&#64;yahoo&#46;com</a> &#40;V&#46; J&#46; Villag&#243;mez-Ortiz&#41;&#46;</p>"
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        "resumen" => "<p class="elsevierStylePara"> <span class="elsevierStyleItalic">Introduction</span>&#58; Amyloidosis is used to describe a range of disorders defined by extracellular deposition of abnormal protein fibrils&#46; The larynx is the most common site of localized amyloidosis in the head and neck region and constitutes less than 1&#37; of benign laryngeal lesions&#46; Hoarseness is the most common symptom&#46;</p> <p class="elsevierStylePara"> <span class="elsevierStyleItalic">Objective</span>&#58; Prospective clinical evaluation of patients with localized laryngeal amyloidosis&#46;</p> <p class="elsevierStylePara"> <span class="elsevierStyleItalic">Clinical cases</span>&#58; Presented are 4 cases of patients with localized laryngeal amyloidosis who were treated at the Otolaryngology and Head and Neck Surgery Department at the <span class="elsevierStyleItalic">&#8220;Dr&#46; Jos&#233; Eleuterio Gonz&#225;lez&#8221;</span> University Hospital in Monterrey&#44; Mexico&#46; Three patients underwent phonomicrosurgery by direct microlaryngoscopy with the removal of the amyloid implantation using a cold knife excision with great results&#46; In each patient the major site of involvement was the supraglottis with a small focus on the false vocal cord&#46; A medical work-up&#44; including a complete blood count &#40;CBC&#41;&#44; a basic metabolic panel&#44; urinalysis&#44; liver function test&#44; chest X-ray and physical examination were performed to rule out the presence of systemic disease&#59; no amyloidosis or signs of systemic disease were found&#46; Congo red staining confirms the diagnosis of amyloidosis in all surgical specimens&#46;</p> <p class="elsevierStylePara"> <span class="elsevierStyleItalic">Conclusions</span>&#58; In laryngeal amyloidosis&#44; the treatment should be directed toward the improvement of the voice and the maintenance of the airway&#46;</p>"
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Article information
ISSN: 16655796
Original language: English
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