Long-Term Outcomes and Quality of Life (QOL) in Survivors of Pediatric and Adolescent Osteosarcoma



Fig. 20.1
QOL in survivors of pediatric osteosarcoma; various aspects and factors are involved in the QOL of long-term survivors of osteosarcoma. For QOL assessment, individual factors must be examined



In this chapter, we introduce long-term outcomes (education, employment, marriage, fertility, multiple primary cancers, and psychosocial outcomes) and QOL in survivors of pediatric and adolescent osteosarcoma based on our study results [28].



20.2 Education and Employment



20.2.1 Our Report on Education [2]


In 41 survivors of pediatric and adolescent osteosarcoma who were treated in our hospital between 1976 and 1995, a questionnaire survey regarding education (returning to school, educational background) was conducted. In addition, they were divided into two groups based on affected limb conditions at the time of the survey: amputation (including rotationplasty) and limb-sparing groups. The results were compared between the two groups.

Of the 41 subjects, responses were obtained from 27 (response rate, 65.9 %). These consisted of 11 males and 16 females. The mean age at the initial presentation was 13.6 years. That at the time of the survey was 34 years. The amputation group consisted of 18 subjects, and the limb-sparing group consisted of 9. The mean interval from the completion of treatment was 218 months.

Of the 27 subjects, 19 could return to school, whereas 7 could not return. There was no description in 1. In 73.1 % (19/26), it was possible to return to the former school. The educational background was senior high school in 12 subjects and university in 13. There was no description in 2. Of these subjects, 52 % (13/25) graduated from a university, being similar to the proportion of Japanese who graduate from a university (45 %). In the limb-sparing group, the proportion of survivors who graduated from university was significantly higher, suggesting that affected limb conditions influence education (Table 20.1, published from Reference [2] based on approval).


Table 20.1
Status of education




























Final education level

Amputation group (18 patients)

Limb-sparing group (9 patients)

University

5

5

College

1

2

High school

11

1

No mention

1

1


Reference [2]


20.2.2 Other Reports on Education


Nagarajan et al. reported that the education level was lower in an amputation group [9]. Novakovic et al. indicated that there was no difference in the education level between survivors and their siblings [10]. These results were similar to those of our study.


20.2.3 Our Report on Employment [2]


We investigated employment (occupation, annual income) in the above 27 survivors of pediatric and adolescent osteosarcoma. There were 18 clerical workers, four technicians, one sales worker, and two housewives. There was no description in 2. Of these, 72 % (18/25) had clerical jobs. The mean annual income was 4,010,000 yen, being similar to that of Japanese businessmen (4,280,000 yen). There was no difference in the mean annual income between the amputation and limb-sparing groups. In Japan, laws for the handicapped have been implemented, and there were few problems regarding survivors’ employment.


20.2.4 Other Reports on Employment


Nagarajan et al. reported that the employment rates were higher in survivors who had received high-level education and male survivors, and that there was no difference in the employment rate between amputation and limb-sparing groups [9]. Hays et al. indicated that there were few financial problems in pediatric cancer patients [11]. In our study, there were also few problems regarding survivors’ employment. Nicholson et al. reported that there was no difference in the employment rate between survivors and their siblings [12]. On the other hand, Novakovic et al. emphasized that the employment rate in survivors was significantly lower [10].


20.3 Marriage and Fertility



20.3.1 Our Report on Marriage [3, 4]


In 62 survivors of pediatric and adolescent osteosarcoma who were treated in our hospital between 1976 and 2002, a questionnaire survey regarding marriage was conducted. Of these, responses were obtained from 46 (response rate, 74.2 %). We investigated the marital rate (number of married survivors/total number of survivors) in the 46 survivors. In addition, they were divided into two groups: amputation and limb-sparing groups. The marital rate was examined in each group. As controls, it was investigated in 52 siblings.

Overall, the marital rate was 63.0 % (29/46). In the siblings, it was 63.5 % (33/52) (Table 20.2, Reference [4]). The marital rates were slightly lower in male survivors and in the amputation group, although there were no significant differences.


Table 20.2
Summary of marital rate





















































































 
Marital rate

p-Value

Total

 Patients

63.0 % (29/46)
 

 Siblings

63.5 % (33/52)

>0.999

Patients

 Males

55.0 % (11/20)
 

 Females

69.2 % (18/26)

0.369

 Amputation group

60.0 % (15/25)
 

 Limb-sparing group

66.7 % (14/21)

0.762

 Males in amputation group

45.5 % (5/11)
 

 Females in amputation group

71.4 % (10/14)

0.241

 Males in limb-sparing group

66.7 % (6/9)
 

 Females in limb-sparing group

66.7 % (8/12)

>0.999

Siblings

 Males

63.0 % (17/27)
 

 Females

64.0 % (16/25)

>0.999

Males

 Patients

55.0 % (11/20)
 

 Siblings

63.0 % (17/27)

0.765

Females

 Patients

69.2 % (18/26)
 

 Siblings

64.0 % (16/25)

0.771


Reference [4]


20.3.2 Other Reports on Marriage


Nagarajan et al. reported that the marital rate in female survivors was higher than in male survivors, and that the marital rate in survivors was lower than in their siblings. In addition, they indicated that there was no difference in the marital rate between amputation and limb-sparing groups [9]. Our study also showed that the marital rate was lower in male survivors. Novakovic et al. reported that in survivors, the marital rate was lower than in their siblings, and that the number of divorces was greater [10]. On the other hand, Nicholson et al. indicated that there was no difference in the marital rate between survivors and their siblings [12]. In our study, there was also no difference in the marital rate between survivors and their siblings.


20.3.3 Our Report on Fertility [3, 4]


In 29 survivors who were married, we investigated the fertility rate (number of survivors raising a child/number of married survivors). They were divided into two groups: MC group in which moderate-dose chemotherapy was performed between 1976 and 1986 and IC group in which intensive-dose chemotherapy was performed between 1987 and 2002. The fertility rate was examined in each group. As controls, it was investigated in 33 siblings.

Overall, the fertility rate was 58.6 % (17/29). In the siblings, it was 81.8 % (27/33). In male survivors, the fertility rate was slightly lower (Table 20.3, Reference [4]). In particular, the fertility rate in male survivors in the IC group was significantly lower than in male siblings (p = 0.018), suggesting that recent intensified chemotherapy for osteosarcoma affects male survivors’ fertility.


Table 20.3
Summary of fertility rate


























































































































 
Fertility rate

p-Value

Total

 Patients

58.6 % (17/29)
   

 Siblings

81.8 % (27/33)

0.055
 

Patients

 Males

36.4 % (4/11)
   

 Females

72.2 % (13/18)

0.119
 

 MC group

52.9 % (9/17)
   

 IC group

16.7 % (1/6)

0.703
 

Siblings

 Males

76.5 % (13/17)
   

 Females

87.5 % (14/16)

0.656
 

Males

 Patients

36.4 % (4/11)
   

 Siblings

76.5 % (13/17)

0.053
 

Females

 Patients

72.2 % (13/18)
   

 Siblings

87.5 % (14/16)

0.405
 

 Male patients in MC group

60.0 % (3/5)
   

 Male siblings

76.5 % (13/17)

0.585
 

 Female patients in MC group

71.4 % (5/7)
   

 Female siblings

87.5 % (14/16)

0.557
 

 Male patients in IC group

16.7 % (1/6)
   

 Male siblings

76.5 % (13/17)

0.018

*

 Female patients in IC group

72.7 % (8/11)
   

 Female siblings

87.5 % (14/16)

0.371
 


Reference [4]

MC group: moderate-dose chemotherapy group

IC group: intensive-dose chemotherapy group

*Statistically significant (Fisher’s exact probability test)


20.3.4 Other Reports on Fertility


Williams et al. reported that ifosfamide influenced fertility [13]. Longhi et al. indicated that ifosfamide-related infertility was more frequent in males [14]. The influence of chemotherapy on fertility is an important issue for long-term survivors; this should be further reviewed in the future.


20.4 Multiple Primary Cancers



20.4.1 Our Report on Multiple Primary Cancers


We investigated the incidence of multiple primary cancers in 162 patients with osteosarcoma who were treated in our department between 1976 and 2009, with an age of 30 years or younger on initial presentation. Nine patients with second malignant neoplasms following osteosarcoma were assigned to Group A. Four patients with osteosarcoma as a second malignant neoplasm were assigned to Group B. We examined the clinical features of these 13 patients.

In Group A, second malignant neoplasms were diagnosed as breast carcinoma in four patients, acute myelogenous leukemia in two, a malignant phyllodes tumor in one, ovarian carcinoma in one, and small intestine carcinoma in one. Ages at the development of second malignant neoplasms ranged from 14 to 34 years (mean, 25.9 years). Intervals from the development of osteosarcoma until that of second malignant neoplasms ranged from 3 to 16 years (mean, 9.6 years) (Table 20.4).


Table 20.4
Multiple primary cancers in patients with pediatric osteosarcoma
























 
Sex

First malignant neoplasm (FMN)

Interval from FMN to SMN (years)

Second malignant neoplasm (SMN)

Age at final follow-up (years)

Outcome

Age (years)

Diagnosis

Age (years)

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Jun 4, 2017 | Posted by in ORTHOPEDIC | Comments Off on Long-Term Outcomes and Quality of Life (QOL) in Survivors of Pediatric and Adolescent Osteosarcoma

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