Sleep Disorders: Student/Resident Case Report Poster - Sleep Disorders |

A Forgotten Problem: Sleep Disordered Breathing in Amyloidosis FREE TO VIEW

Brendon Colaco, MBBS; Clinton Colaco, MBBS; Melissa Lipford, MD
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Mayo Clinic, Rochester, MN

Copyright 2016, American College of Chest Physicians. All Rights Reserved.

Chest. 2016;150(4_S):1293A. doi:10.1016/j.chest.2016.08.1407
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SESSION TITLE: Student/Resident Case Report Poster - Sleep Disorders

SESSION TYPE: Student/Resident Case Report Poster

PRESENTED ON: Tuesday, October 25, 2016 at 01:30 PM - 02:30 PM

INTRODUCTION: Amyloidosis is a systemic disease involving the deposition of amyloid protein in various body organs and structures. The literature describing the relationship between amyloidosis and sleep disordered breathing is sparse. We present a case of a lady with severe obstructive sleep apnea (OSA) secondary to macroglossia. Although her symptoms were managed with continuous positive airway pressure therapy (CPAP), there were significant challenges associated with continuance of therapy.

CASE PRESENTATION: A 48 year old Hispanic lady was diagnosed with amyloidosis at the age of 33 following symptoms of bilateral lower extremity pain, swelling in submandibular gland and tongue, dysarthria, dysphagia to solids, peripheral edema and initial 30 pound weight loss. Patient was noted to have audible breathing at night, abnormal overnight oximetry and daytime sleepiness. Patient had documented amyloid involvement of the heart, peripheral nerve and tongue. On physical exam, patient’s weight was 69 kg. He was noted to have a large tongue and a Friedman class 4 oropharynx. EKG revealed first degree AV block and echocardiogram showed increased ventricular septum thickness with small pericardial effusion. Chest X-ray revealed a small pleural effusion. Overnight oximetry was suggestive of severe sleep disordered breathing. PSG showed an apnea hypopnea index of 121 per hour of sleep which was non-positional with an oxyhemoglobin nadir saturation of 74%. The events were predominantly obstructive with a few mixed events. Patient was subsequently tried on CPAP with control of sleep disordered breathing at 17 cm of water pressure and was issued a prescription.

DISCUSSION: Amyloidosis can precipitate or worsen existing sleep disordered breathing through several pathways. Amyloid deposition in the tongue, submandibular and sublingual glands as well as soft tissues around the neck, can contribute to obstructive sleep apnea. Cardiac and cerebral amyloid deposition can mediate central sleep apnea and amyloid involvement of nerves and diaphragm can result in hypoventilation. Careful history, physical exam and awareness of manifestations of the disease when reading polysomnograms can be of great help in reaching the correct diagnosis and treatment. Other treatment options include surgical tongue reduction and laser tongue reduction.

CONCLUSIONS: Our patient had severe OSA secondary to primary systemic amyloidosis. She was well controlled on CPAP at 17 cm H2O pressure. She did not have significant central sleep apnea or hypoventilation, despite cardiac and nervous system involvement of amyloid.

Reference #1: Carbone JE, Barker D, Stauffer JL. Sleep apnea in amyloidosis. Chest 1985 Mar;87(3):401-3.

Reference #2: Gasparini G, Saltarel A,Carboni A, Maggiulli F, Becelli R. Surgical management of macroglossia: Discussion of 7 cases. Oral Surg Oral med Pathol Oral Radio Endod 2002; 94:566-71.

DISCLOSURE: The following authors have nothing to disclose: Brendon Colaco, Clinton Colaco, Melissa Lipford

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