Sorafenib treatment for papillary thyroid carcinoma with diffuse lung metastases in a child with autism spectrum disorder: a case report

Abstract

BackgroundPediatric papillary thyroid carcinoma frequently presents with lymph node involvement and distant metastases. Sorafenib, an oral multikinase inhibitor, has been used to treat radioactive iodine (RAI) therapy-refractory thyroid carcinoma in adults; however, pediatric experience is limited. Medical procedures and hospitalization for children with autism spectrum disorder may be challenging.Case presentationAn 11-year-old boy with autism spectrum disorder and moderate intellectual impairment presented with dyspnea on exertion with thyroid carcinoma and diffuses lung metastases. Total thyroidectomy and adjuvant RAI therapy is the standard treatment; however, the latter therapy was impractical because of his respiratory status and challenging behaviors. He was therefore started on sorafenib 200 mg/day (150 mg/m2/day) and this dosage was increased to 400 mg/day (300 mg/m2/day). The adverse effects were mild and tolerable. After administration of medication, his dyspnea improved and surgery was performed. We attempted to administer RAI therapy after surgery; however, we abandoned it because he had difficulty taking care of himself according to isolation room rules. Thyrotropin suppression therapy was therefore started and sorafenib treatment (400 mg/day) resumed. Follow-up imaging showed regression of pulmonary metastases. The metastases have remained stable for over 24 months on continuous sorafenib treatment without serious adverse events.ConclusionWe inevitably used sorafenib as an alternative to standard therapy because of the patient’s specific circumstances. Individualized strategies for pediatric cancer patients with autism spectrum disorder are needed.

Thyroid Cancer in the Pediatric Population

“…In the setting of adult late-stage metastatic and progressive radioactive iodine-refractory differentiated thyroid carcinoma, sorafenib and lenvatinib have both shown improved progression free survival compared to placebo, 10.8 months vs. 5.8 months (hazard ratio 0.59) and 18.3 months vs. 3.6 months (hazard ratio 0.21) respectively [116,119,120]. In the pediatric population, response to sorafenib has been reported both in progressive RAI refractory PTC, PTC with diffuse metastatic disease not amenable to upfront RAI, and as gap therapy in a patient who could not receive RAI in a timely fashion (one case each) [121,122,123], while stable disease has been reported in three pediatric patients with extensive bilateral metastatic pulmonary disease treated with lenvatinib (including one previously treated with sorafenib) [124]. Clinical trials evaluating the efficacy of lenvatinib in the treatment of children with refractory or relapsed solid malignancies (including thyroid cancer) are ongoing.…”

New insights into the management of differentiated thyroid carcinoma in children and adolescents (Review)

“…Targeted therapy. Despite the excellent prognosis of DTC in paediatric patients, a small subset of this population may show progressive and RAI refractory disease (13)(14)(15)(16)(17). In such a case, systemic therapy should be considered (13).…”

“…A North American population study conducted between 1992 and 2014 found 20-year survival rates of 99.7 and 96.3% for PTC and FTC, respectively, regardless of the disease stage (12). Notwithstanding, a subgroup of patients presents an aggressive clinical course, with increased morbidity and mortality (13)(14)(15)(16)(17). Currently, a significant challenge in the management of DTC is identifying patients at high risk of unfavourable outcomes.…”

Radioiodine (131I) therapy in a child with autism spectrum disorder: A complex and demanding task

“…Total thyroidectomy and adjuvant radioiodine ( 131 I) (RAI) therapy is the accepted standard treatment of patients with thyroid carcinoma but is challenging for children with behavioral issues. In fact, in a recent reported case of a child with autism spectrum disorder, RAI treatment was abandoned due to behavior-related issues and the patient was treated with sorafenib [1] as a second-line option. For our 11.5-year-old child, it became clear, from the first consultation session, that behavioral obstacles related to the child's autism, intellectual disability, and family environment needed to be overcome.…”