Clinically isolated laryngeal sarcoidosis

Abstract

Laryngeal sarcoidosis is rare (0.5% of patients with sarcoidosis), the pathogenesis is unknown and the optimal treatment remains a matter of debate. We undertook this study to elucidate possible pathogenic factors in clinically isolated laryngeal sarcoidosis and to describe results of supraglottoplastic surgery. From 1995 to 2009, we identified six patients with histologically proven sarcoidosis of the larynx treated at Rigshospitalet. All patients were subjected to a panel of blood tests and MR scan of the head and neck. All patients had dyspnoea at admission, and five were subjected to a combination of CO(2)-laser excision of supraglottic tissue and closure of the incision with sutures. All serological tests were negative or normal, including angiotensin 1 converting enzyme. The clinical expression was uniform with pale, smooth swellings of the supraglottic structures. Surgery proved successful to maintain normal breathing. None of the many parameters examined--some previously having been found to be abnormal in sarcoidosis--were abnormal in the present cohort. We are therefore unable to elucidate the pathogenesis. The combined surgical approach re-established normal airway function for all five patients and complete remission without further swellings was seen in two patients.

Head and neck sarcoidosis, from wait and see to tumor necrosis factor alpha therapy: A pilot study

“…In the present patient group facial palsy, sinunasal and laryngeal manifestation, as well as inner ear hearing loss accompanied pulmonary disease and did not occur in isolated extrapulmonary manifestations 14–17. In contrast, Plaschke et al9 reported on 6 cases of isolated laryngeal sarcoidosis. In these patients, chest x‐ray and laboratory parameter (including ACE) did not show abnormalities.…”

“…A total of 30 patients had complex extrapulmonary disease 8. The literature provides only a few case reports that describe the clinical manifestation of head and neck sarcoidosis; comprehensive analysis of treatment modalities referring to specific requirements of head and neck manifestation is unavailable 9–13. We treated 31 patients with head and neck manifestation of sarcoidosis.…”

“…In these patients, chest x‐ray and laboratory parameter (including ACE) did not show abnormalities. The biopsies obtained from the lesional laryngeal mucosa were stained for mycosis and mycobacteria; in 4 cases additional tuberculosis (TB) and bacteriologic cultures were performed 9. However, TB‐spot or polymerase chain reaction (PCR) was not performed to exclude tuberculosis.…”

Sarcoidosis of the Head and Neck

“…This results in reduced symptoms and recurrence of swelling. This mucosal sparing surgery, even when demonstrated on only six patients, shows potential as an additional management tool [15]. Figures 2, 3 show laryngeal sarcoidosis at low and high magnification.…”

“…Isolated laryngeal sarcoidosis is found in only 0.5 % of patients [15]. Plaschke, et al published a recent study outlining a successful treatment option, supraglottoplasty using CO 2 -laser excision of tissue, a technique that removes involved mucosa and allows suturing to promote primary healing of mucosal surfaces.…”