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

Pepper, Jon Paul; Kupfer, Robbi A.; McHugh, Jonathan B.; Hogikyan, Norman D.

doi:10.1001/archoto.2011.119

Cited by 2 publications

(2 citation statements)

References 12 publications

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“…This finding suggests that factors other than pulmonary fibrosis are involved in the tracheal abnormalities that occur in SSc patients. Consistent with this observation, several studies have shown impairment of anatomic structures above the trachea in scleroderma patients; laryngeal dysfunction is relatively common, with pathologic findings demonstrating fibrinoid degeneration and an increase in collagen fibers [ 30 , 31 ]. Considering the promising advent of user-friendly software, evaluation of the trachea in patients with scleroderma may be an interesting approach both in clinical practice and in trials.…”

Section: Discussionmentioning

confidence: 62%

Computed tomography trachea volumetry in patients with scleroderma: Association with clinical and functional findings

et al. 2018

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BackgroundIn scleroderma, excessive collagen production can alter tracheal geometry, and computed tomography (CT) volumetry of this structure may aid in detecting possible abnormalities. The objectives of this study were to quantify the morphological abnormalities in the tracheas of patients with scleroderma and to correlate these findings with data on clinical and pulmonary function.MethodsThis was a cross-sectional study in which 28 adults with scleroderma and 27 controls matched by age, gender and body mass index underwent chest CT with posterior segmentation and skeletonization of the images. In addition, all participants underwent pulmonary function tests and clinical evaluation, including the modified Rodnan skin score (mRSS).ResultsMost patients (71.4%) had interstitial lung disease on CT. Compared to controls, patients with scleroderma showed higher values in the parameters measured by CT trachea volumetry, including area, eccentricity, major diameter, minor diameter, and tortuosity. The tracheal area and equivalent diameter were negatively correlated with the ratio between forced expiratory flow and forced inspiratory flow at 50% of forced vital capacity (FEF50%/FIF50%) (r = -0.44, p = 0.03 and r = -0.46, p = 0.02, respectively). The tracheal tortuosity was negatively correlated with peak expiratory flow (r = -0.51, p = 0.008). The mRSS showed a positive correlation with eccentricity (r = 0.62, p < 0.001) and tracheal tortuosity (r = 0.51, p = 0.007), while the presence of anti-topoisomerase I antibody (ATA) showed a positive correlation with tracheal tortuosity (r = 0.45, p = 0.03).ConclusionsIn a sample composed predominantly of scleroderma patients with associated interstitial lung disease, there were abnormalities in tracheal geometry, including greater eccentricity, diameter and tortuosity. In these patients, abnormalities in the geometry of the trachea were associated with functional markers of obstruction. In addition, tracheal tortuosity was correlated with cutaneous involvement and the presence of ATA.

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Section: Discussionmentioning

confidence: 62%

Computed tomography trachea volumetry in patients with scleroderma: Association with clinical and functional findings

et al. 2018

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“…Facial skin thickening and muscular atrophy contribute to bone resorption 3. In turn, laryngeal inflammation has been attributed to gastro-oesophageal reflux, vocal fold thickening, nodularity or fibrosis,4 5 while vocal cord paralysis has been related to cricoarytenoid ankyloses and nerve injury 6 7. According to literature, mandibular resorption in SSc has a variable prevalence raising from 6.6% to 46.7%,3 and it correlates with longer disease duration.…”

Section: Introductionmentioning

confidence: 99%

Mandibular resorption and vocal cord paralysis: a catastrophic form of systemic sclerosis

Mendonça

Taulaigo²,

Caetano

et al. 2019

BMJ Case Rep

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Sudden respiratory distress in association with severe weight loss are unusual features of systemic sclerosis (SSc). We report the case of a 56-year-old Caucasian woman with a 9-year history of a diffuse form of SSc who presented with acute stridor due to vocal cord paralysis and required an emergency tracheostomy. She had sought medical attention only after 4 years of disease onset, presenting with a mask-like face, diffuse skin thickening, acro-osteolysis and severe interstitial lung disease. Even though skin tightness improved after immunosuppressive treatment, several spontaneous facial fractures and episodes of dysphagia and choking occurred in the years that followed. At the time of stridor, she was severely malnourished and a percutaneous endoscopic gastrostomy was required for feeding. Permanent vocal cord damage in combination with severe loco-regional bone resorption resulted in severe disability and impaired nutrition. We hereby highlight the features of SSc for which therapy remains challenging.

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

Cited by 2 publications

References 12 publications

Computed tomography trachea volumetry in patients with scleroderma: Association with clinical and functional findings

Computed tomography trachea volumetry in patients with scleroderma: Association with clinical and functional findings

Mandibular resorption and vocal cord paralysis: a catastrophic form of systemic sclerosis

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