Laryngeal Obstruction and Hoarseness Associated With Rosai-Dorfman Disease

Abstract

Hypoxic lung injury during cancer therapy poses a serious problem. Multiple agents can be implicated. In this patient, vinorelbine most likely was the causative agent of the pulmonary findings. While filgrastim has been associated with hypoxemia during neutrophil recovery, 1,2 the lack of temporal association, leukocytosis, or positive rechallenge during subsequent chemotherapy makes it an unlikely offender in this case. No other causes could be elucidated after extensive work-up, though remote history of radiotherapy to the chest wall may have been a sensitizing factor. In a pooled analysis of three large multicenter trials conducted in North America with vinorelbine (n ϭ 327), dyspnea was reported in 5% of patients; in 3% it was severe. 3 This complication may occur after either the first or subsequent treatments. In fact, it may manifest as late as on the tenth cycle of treatment. 4,5 Potential risk factors include concomitant use of mitomycin-C and history of dyspnea from a previous infusion. 5,6 Two types of respiratory distress associated with vinorelbine have been reported: acute and subacute. The acute form, typically associated with acute dyspnea, bronchospasm, fever, hypotension, and alveolar infiltrates, usually occurs within minutes after vinorelbine infusion. 3,6 The subacute form, occurring hours to days after infusion, is marked by progressive dyspnea, diffuse interstitial infiltrates, and in rare cases, acute respiratory distress syndrome. 7 Vinorelbine, a semisynthetic vinca alkaloid, interacts with tubulin. It causes imbalance between polymerization and depolymerization and leads to a disruption of microtubule assembly-an essential component of vascular permeability regulation. 8 Microtubule inhibitors cause a dysfunctional actomyosin cytoskeleton, leading to endothelial cell barrier dysfunction, which is similar to the pathogenesis of many conditions such as sepsis and acute lung injury. 8 After infusion, vinorelbine achieves much higher concentration in heart and lung tissues than in serum. 9 In fact, rare cases of myocardial infarction have also been reported in association with vinorelbine. 10,11 Treatment of acute lung injury associated with vinorelbine or other suspected chemotherapeu-tic agents is largely supportive, and most reports recommend the use of corticosteroids. Typically, respiratory distress associated with single-agent vinorelbine is followed by full recovery. This syndrome, as with other vinca alkaloids such as vinblastine, may result in chronic lung disease, especially when mitomycin-C was concurrently administered. 12 After one episode of respiratory distress, re-treatment with vinorelbine may lead to a more severe reaction, though some investigators re-treated patients, noting only a mild reaction, using premedication with corticosteroids. 3,6 Vinorelbine most likely was the causative agent in this patient, and while resulting in a critical situation, was associated with a full recovery. As the role for vinorelbine in solid tumors, including breast cancer, continue...

Doença de Rosai-Dorfman como diagnóstico diferencial de lesão ulcerada de prega vocal

“…1 It is a rare benign lymphoproliferative disorder featuring painless neck lymphadenopathy, fever, neutrophilia, an elevated erythrocyte sedimentation rate (ESR), and polyclonal hypergamaglobulinemia. 2 Published cases describe laryngeal involvement associated with lymphadenopathy or disease in other sites; 3 , 5 however, there are no descriptions of glottic involvement only, as in the present case. The etiology of RDD remains unclear.…”

“…2 Published cases describe laryngeal involvement associated with lymphadenopathy or disease in other sites; 3,5 however, there are no descriptions of glottic involvement only, as in the present case. The etiology of RDD remains unclear.…”

“…3 The etiology is unclear; an association with immune diseases has been documented. 4 The clinical picture is not always present.…”

Radiotherapy for Steroid-Resistant Laryngeal Rosai–Dorfman Disease

“…Extranodal rdd of the larynx has been documented in the literature [8][9][10][11][12][13][14][15][16][17][18] . In these cases, treatment was initiated because of symptoms such as stridor, dysphagia, dysphonia, and dyspnea because of a laryngeal lesion.…”

Extranodal Rosai–Dorfman disease involving bilateral ovaries in a patient with a ventriculoperitoneal shunt

“…The mean age at onset is reported to be 20.6 years with a male : female incidence ratio of 1.4:1 2 . Extranodal involvement is present in approximately 43% of cases, and the commonly involved sites are the skin, upper respiratory tract, bone, orbit, paranasal sinuses, the central nervous system (CNS) etc 3 . Involvement of the female genital tract is extremely rare, with only one reported case of uterine cervical involvement 4 .…”