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Clinical Note |

Histopathologic Findings and Clinical Manifestations in a Patient With Dysphonia and Vocal Fold Involvement by Systemic Sclerosis

Jon-Paul Pepper, MD; Robbi A. Kupfer, MD; Jonathan B. McHugh, MD; Norman D. Hogikyan, MD
Arch Otolaryngol Head Neck Surg. 2011;137(8):816-819. doi:10.1001/archoto.2011.119.
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Systemic sclerosis (SSc), or systemic scleroderma, is an autoimmune disease characterized by excessive interstitial fibrosis throughout the body.1 The skin is most frequently affected, resulting in diffuse thickening known as scleroderma (“hard skin”). Common otolaryngologic manifestations include trismus, gastroesophageal reflux disease (GERD), and ankyloglossia, in addition to the aforementioned skin changes. One study reported that approximately 9% of patients with scleroderma have at least intermittent dysphonia.2 Laryngoscopic findings in patients with scleroderma may include a wide range of nonspecific changes associated with reflux laryngitis due to a patulous lower esophageal sphincter and distal third of the esophagus.3 More rarely, SSc has been associated with vocal fold paresis and cricoarytenoid joint fixation.47 This report describes a patient with SSc presenting with moderate to severe dysphonia due to bilateral submucosal vocal fold lesions. The histopathologic characteristics of these lesions demonstrate changes that are characteristic of SSc and that to date have not been well described for the vocal folds. Case data, surgical approach, and videostroboscopic findings are discussed.

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Figure 1. Videostroboscopy on initial evaluation. Findings during inspiration (A) and phonation (B) demonstrated multiple yellowish submucosal masses at the central vocal folds bilaterally. Mucosal irregularities were apparent at the free edge of the bilateral vocal folds.

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Figure 2. Elevation of mucosal flaps performed intraoperatively. Findings revealed yellow masses within the submucosal plane of the left (A) and right (B) vocal folds. The vocal ligament remained intact after removal of abnormal-appearing tissue (C). The mucosal flaps were redraped to approximate the cordotomy incision (D).

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Figure 3. Biopsy findings of the vocal cord submucosal masses show extensive sclerosis effacing the submucosa with residual entrapped epithelium (arrow). In addition, perivascular lymphoplasmacytic inflammation and degenerating collagen (arrowhead and inset) characterized by less fibrillar and deeper pink (eosinophilic) collagen are present. These features are consistent with scleroderma. Hematoxylin-eosin, original magnification ×100 (inset, ×400).

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Figure 4. Videostroboscopy 8 weeks after surgery demonstrated normal vocal fold contour and absence of submucosal nodularity.

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