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Critical Care |

Diffuse Alveolar Hemorrhage in Severe Obstructive Sleep Apnea in a Patient With Antiphospholipid Antibody Syndrome

Sadaf Mir, MD; Silviana Marineci, MD; Mary O'Sullivan, MD
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St. Luke's Roosevelt Hospital Center, New York, NY


Chest. 2014;145(3_MeetingAbstracts):165A. doi:10.1378/chest.1834682
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Abstract

SESSION TITLE: Critical Care Case Report Posters

SESSION TYPE: Case Report Poster

PRESENTED ON: Sunday, March 23, 2014 at 01:15 PM - 02:15 PM

INTRODUCTION: Diffuse alveolar hemorrhage (DAH) is associated with negative pressure pulmonary edema secondary to acute upper airway obstruction. There have been no associations with the upper airway obstruction of obstructive sleep apnea. Negative intrathoracic pressure leads to an increase in pulmonary vascular volume and pulmonary capillary transmural pressure, creating a risk of disruption of the alveolar-capillary membrane. We present a case of DAH in a patient on anticoagulation with positive anti-cardiolipin antibody and low level anti-dsDNA triggered by severe obstructive sleep apnea.

CASE PRESENTATION: A fifty year old obese African American male presented with shortness of breath, 5 months of intermittent hemoptysis and fluctuating patchy ground glass opacities on serial chest computed tomographies (CT). Past history included nephrectomy for papillary renal cancer, hypercoagulable state with chronic deep venous thrombosis on anticoagulation with therapeutic INR and severe obstructive sleep apnea (OSA). He was noncompliant with continuous positive airway pressure (CPAP) machine. Physical examination was remarkable for android obesity (BMI=32). Platelet count fluctuated between 90 and 140k/ul. Negative ANCA and myeloperoxidase Ab. Normal V/Q scan. Echocardiogram revealed moderate pulmonary hypertension. Pulmonary function test revealed restriction with moderate reduction of diffusion capacity. The ground glass CT opacities fluctuated in location and size consistent with DAH. Bronchoscopy, off anticoagulation, while asleep, revealed markedly redundant false vocal cords collapsing circumferentially into his airway during extreme respiratory effort with marked facial discoloration. BAL was increasingly pink over three instillations yielding hemosiderin laden macrophages. ENT exam sitting and awake revealed only moderate circumferential collapse. A surgical biopsy confirmed DAH with no evidence of vasculitis or malignancy and mild emphysema. The patient was advised that the severity of his sleep apnea was contributing to his hemoptysis. He become compliant with his CPAP and the hemorrhage ceased and the pulmonary infiltrates cleared.

DISCUSSION: Pulmonary hemorrhage is a dynamic process that often features multiple contributing factors. We present a case of DAH precipitated by severe OSA in a patient on anticoagulation with underlying antiphospholipid antibody syndrome and presumably some degree of vasculopathy without actual vasculitis. Although our diagnosis is presumptive, we hypothesize that severe negative intrathoracic force against a completely closed airway in this patient triggered DAH which resolved with CPAP compliance. Also, bronchoscopic exam, supine and asleep reveals a more accurate assessment of the severity of the dynamic forces involved in OSA.

CONCLUSIONS: Physicians should be made aware of such complication of DAH in patients with severe obstructive sleep apnea.

Reference #1: Case Rep Pulmonol. 2012;2012:836017 Diffuse alveolar hemorrhage: a rare life-threatening condition in systemic lupus erythematosus. Virdi RP, Bashir A, Shahzad G, Iqbal J, Mejia JO.

Reference #2: Nihon Kokyuki Gakkai Zasshi. 2011 Oct;49(10):733-8. A case of negative pressure pulmonary hemorrhage associated with upper-airway obstruction due to a giant vocal cord nodule. Hoshika Y, Horie Y, Yamamoto Y, Haga T, Mizoo A.

Reference #3: Semin Arthritis Rheum. 2005 Dec;35(3):154-65. Antiphospholipid antibodies as a cause of pulmonary capillaritis and diffuse alveolar hemorrhage: a case series and literature review. Deane KD, West SG.

DISCLOSURE: The following authors have nothing to disclose: Sadaf Mir, Silviana Marineci, Mary O'Sullivan

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