Secondary Neoplasms After Retinoblastoma Treatment: Retrospective Cohort Study of 754 Patients in Japan

Araki, Yuko; Matsuyama, Yutaka; Kobayashi, Yasuki; Toyokawa, Satoshi; Inoue, Kazuo; Suzuki, Shigenobu; Makimoto, Atsushi

doi:10.1093/jjco/hyq201

Cited by 26 publications

(23 citation statements)

References 31 publications

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“…Indeed, our previous study showed that the incidence of second malignancies appeared to be largely associated with radiotherapy and that systemic chemotherapy or selective ophthalmic arterial injection therapy did not increase the incidence of second malignancies (36). Another investigation focusing on the risk factors for the development of second malignancies found that focal and systemic chemotherapy was not a significant risk factor (37).…”

Section: Discussionmentioning

confidence: 95%

Second primary osteosarcomas in patients with retinoblastoma

Fujiwara

Numoto

et al. 2015

Jpn. J. Clin. Oncol.

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Objective: Second primary malignancies have become the leading cause of death in retinoblastoma survivors. Although osteosarcoma is the most common second malignancy, little is known about its clinical and therapeutic features. Methods: We retrospectively reviewed a database of patients with retinoblastoma and osteosarcoma occurring as a second malignancy between 1964 and 2010 at the National Cancer Center Hospital of Japan. Results: Among 857 patients with retinoblastoma registered in the database, 10 (1.1%) developed osteosarcoma as a second malignancy. The median age at the onset of retinoblastoma was 3 months, being bilateral in nine patients and unilateral in one. Systemic chemoreduction was performed in three patients and intra-arterial chemotherapy in six; all patients received external beam radiotherapy. The median age at the onset of second primary osteosarcoma was 11.2 years; four were radiation-related and six were located in an extremity. Among five patients treated at our institute, four patients with tumors on an extremity were treated by wide resection with neoadjuvant and adjuvant chemotherapy. Three of these four patients (75%) were good responders to high-dose methotrexate-based multi-agent chemotherapy and survived with no evidence of disease (median follow-up period, 17.3 years). One patient whose temporal bone was affected underwent radiotherapy with chemotherapy but died after local recurrence. Conclusions: The clinical outcomes of second primary osteosarcoma in an extremity occurring in retinoblastoma survivors may be more favorable than those of conventional osteosarcoma. Early diagnosis of radiation-related osteosarcoma arising in the craniofacial region should be made at a stage where complete resection is possible.

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Section: Discussionmentioning

confidence: 95%

Second primary osteosarcomas in patients with retinoblastoma

Fujiwara

Numoto

et al. 2015

Jpn. J. Clin. Oncol.

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“…The occurrence of a secondary neoplasm in patients following treatment for retinoblastoma has been reviewed in several studies [8,9,10,11]. The most frequent second malignancies are leiomyosarcoma, osteosarcoma, and melanoma.…”

Section: Discussionmentioning

confidence: 99%

“…The most frequent second malignancies are leiomyosarcoma, osteosarcoma, and melanoma. UPS, schwannoma, and meningioma have been reported but are relatively rare in the setting of treated retinoblastoma [8,9,10,11,12]. A meta-analysis of 676 secondary tumors in patients with retinoblastoma demonstrated that the median age at diagnosis of all secondary tumors was 13.0 years (range, 0.3-60.4), 13.0 years for sarcoma, and 29.0 years for carcinoma [13].…”

Section: Discussionmentioning

confidence: 99%

Quadruple Neoplasms following Radiation Therapy for Congenital Bilateral Retinoblastoma

et al. 2017

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Purpose: The aim of this study was to describe a 34-year-old male with hereditary bilateral retinoblastoma treated with radiotherapy as a child who developed 4 distinct tumors within the radiation field. Methods: A 34-year-old male with bilateral retinoblastoma status postradiation therapy and recurrence requiring enucleation presented with left-eye visual acuity changes. Magnetic resonance imaging demonstrated a left orbital mass and a right parasellar complex lobulated mass (right sphenoid and right cavernous sinus). Two weeks later, the patient underwent excision of the orbital mass and biopsy of an upper-lid nodule. This was followed by craniotomy for removal of the complex mass. Results: Histology revealed 4 distinct tumors, including an undifferentiated pleomorphic sarcoma (left orbit), a radiation-induced meningioma (right sphenoid), a schwannoma (right cavernous sinus), and a basal-cell carcinoma (left lid). Conclusion: Although occurrence of a second neoplasm is a well-known outcome following radiation treatment in patients with hereditary retinoblastoma, the diagnosis of 4 additional neoplasms is rare. Pleomorphic sarcoma, radiation-induced meningioma, and schwannoma are uncommon tumors and not well represented in the literature describing irradiated retinoblastoma patients. Secondary malignancies are a leading cause of early death in retinoblastoma survivors, and long-term follow-up is crucial for patient care.

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“…Of note, 12 of the 14 eyes (86%) that required further chemoreduction received those additional cycles within 6 months of initial treatment suggesting relatively early identification of tumor persistence and/or recurrence. Given the well-known associated risks of radiation therapy including retardation of orbital bone growth [32,33] and increased secondary malignancies [34,35,36,37,38,39,40], we recommend the use of additional chemoreduction prior to IMRT for tumors refractory to initial short-course 3-cycle chemoreduction.…”

Section: Discussionmentioning

confidence: 99%

Long-Term Outcomes of Group B Eyes in Patients with Retinoblastoma Treated with Short-Course Chemoreduction: Experience from Children's Hospital Los Angeles/University of Southern California

et al. 2015

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Background/Aims: Chemoreduction protocols for retinoblastoma vary widely across institutions. Herein, we compare a 3- versus 6-cycle chemotherapy approach for group B retinoblastoma. Methods: A nonrandomized, retrospective review of patients diagnosed with group B retinoblastoma from 1991-2011 at Children's Hospital Los Angeles was performed. A total of 72 eyes of 63 patients were analyzed. Mean follow-up time was 82 months (range 6-272 months). Main outcome measures were globe salvage and need for external beam radiation. Results: Forty-six patients (55 eyes) were treated upfront with 3 cycles of carboplatin, etoposide, and vincristine; 17 patients (17 eyes) received 6 cycles. Thirty-seven eyes (67%) in the 3-cycle group were cured with initial chemoreduction alone. An additional 10 eyes with persistent or recurrent tumors were rescued with 3 more cycles for a total salvage rate of 85% (47/55 eyes). In the 6-cycle group, 16 of 17 eyes (94%) avoided radiation and enucleation. Conclusion: The initial recurrence rate was higher for the 3-cycle group (p = 0.03). However, eyes failing short-course chemoreduction were rescued with 3 additional cycles and achieved a similar overall event-free survival rate (p = 0.16). In our cohort, this short-course approach spared 63% (29/46) of patients with group B retinoblastoma the extra 3 cycles of systemic chemotherapy.

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Secondary Neoplasms After Retinoblastoma Treatment: Retrospective Cohort Study of 754 Patients in Japan

Cited by 26 publications

References 31 publications

Second primary osteosarcomas in patients with retinoblastoma

Second primary osteosarcomas in patients with retinoblastoma

Quadruple Neoplasms following Radiation Therapy for Congenital Bilateral Retinoblastoma

Long-Term Outcomes of Group B Eyes in Patients with Retinoblastoma Treated with Short-Course Chemoreduction: Experience from Children's Hospital Los Angeles/University of Southern California

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