Age and racial differences in the presentation of gestational trophoblastic neoplasia.

OBJECTIVE: Gestational trophoblastic neoplasia are a group of diseases with few data given their rarity. The aim of this study was to determine the age and racial differences in the presentation and survival of patients with gestational trophoblastic neoplasia in the United States. METHODS: Data were collected from the National Cancer Database from January 2004 to December 2014. Chi-square tests, Cox regression, and Kaplan-Meier models were performed. Demographic characteristics included age at diagnosis, race, insurance status, facility location and type, community median income, high school dropout rate, education, income, and population density data. RESULTS: There were 1004 eligible patients including 64% white (n=645), 23% black (n=233), and 8.3% Asian patients (n=83). Median age was 30.8 (range 14-59) years. Stage I, II, III, IV, and unknown were diagnosed in 32%, 5.4%, 30%, 18%, and 15% of patients, respectively, with 5-year survival of 99%, 93%, 94%, 72%, and 95%, respectively (p<0.001). Compared with national birth rates, those with gestational trophoblastic neoplasia were overrepresented at younger (age 10-19 years: 8.2% vs 4.8%) and older ages (age 40-54 years: 17% vs 3.3%). The extremes of age at presentation were more pronounced in black patients with gestational trophoblastic neoplasia (age 10-19 years: 11% vs 6.9%, 40-54 years: 18% vs 3.2%), and black patients constituted 23% of patients compared with 15% of births nationwide. Some 59% of patients were treated at Academic/Research Programs. Only 6/448 (1.3%) facilities treated more than one patient per year, and only 9% (n=92) of patients were treated at one of these high-volume facilities. On multivariable analysis, older age, higher Charlson/Deyo co-morbidity score, and higher stage disease were independently associated with worse survival (all p<0.001). CONCLUSIONS: Gestational trophoblastic neoplasia was disproportionately higher in those at extremes of age and in black women as compared with United States national data. The lack of centralization of care justifies the need to develop regional centers of excellence for this rare malignancy.

Gestational trophoblastic disease in a Greenlandic Inuit: diagnosis and treatment in a remote area.

We report on a 21-year-old pregnant Greenlandic Inuit woman, who presented at a small local hospital in Northern Greenland. The patient suffered from lower abdominal pain, irregular bleeding and vomiting. urine-human chorionic gonadotropin (U-hCG) was positive. Ultrasonography showed the typical 'snow-storm' images of a mole pregnancy. Owing to the fact that local physicians were able to perform an ultrasound, proper diagnosis could be established, and the patient was transferred to the regional hospital, located nearly 1200 km away. At the regional hospital, uterine evacuation was performed under general anaesthesia. Blood analysis showed that serum hCG returned to undetectable levels, and the patient recovered uneventfully. Our case shows that ultrasonography is a valuable diagnostic tool also in remote areas. In Greenland, geographical distances are large and weather conditions can be extreme, and in this report, we discuss how healthcare can be optimised in remote areas.

Effect of race/ethnicity on clinical presentation and risk of gestational trophoblastic neoplasia in patients with complete and partial molar pregnancy at a tertiary care referral center.

BACKGROUND: The reported incidence of molar pregnancy varies widely among different geographic locations. This variation has been attributed, at least in part, to racial/ethnic differences. While the incidence of molar pregnancies is decreasing, certain ethnic groups such as Hispanics, Asians, and American Indians continue to have an increased risk of developing gestational trophoblastic disease across the globe. OBJECTIVE: We sought to describe the potential effect of ethnicity/race on the presentation and clinical course of complete mole and partial mole. STUDY DESIGN: All patients followed up for complete mole and partial mole at a single institution referral center from 1994 through 2013 were identified. Variables including age, race, gravidity, parity, gestational age, presenting signs/symptoms, serum human chorionic gonadotropin values, and development of gestational trophoblastic neoplasia were extracted from medical records and patient surveys. Patients with complete mole and partial mole were categorized into race/ethnicity groups defined as white, black, Asian, or Hispanic. Due to low numbers of non-white patients with partial mole in each non-white category, patients with partial mole were grouped as white or non-white. Continuous variables were compared using the Kruskal-Wallis test and binary variables were compared using the Fisher exact test. RESULTS: A total of 167 complete mole patients with known race/ethnicity status were included (57.48% white, 14.97% Asian, 14.37% black, 13.17% Hispanic). Hispanics presented at younger age (median 24.5 years) compared to whites (median 32.0 years, P = .04) and Asians (median 31.0 years, P = .03). Blacks had higher gravidity than whites (P < .001) and Hispanics (P = .05). There was no significant difference in presenting symptoms, gestational age at diagnosis, and preevacuation serum human chorionic gonadotropin level by race/ethnicity. Hispanics were significantly less likely than whites to develop gestational trophoblastic neoplasia (absolute risk difference, 28.6%; 95% confidence interval, 8.1-39.2%; P = .02). A total of 144 patients with partial mole were analyzed. There were 108 white and 36 non-white patients. Median age was 31 years for white and 29 years for non-white patients (P = .006). Median gravidity was 2 for white and 3 for non-white patients (P < .001), and median parity was 0 for white patients and 1 for non-white patients (P = .003). There were no significant differences with respect to presenting signs and symptoms, gestational age, preevacuation human chorionic gonadotropin level, or risk of progression to gestational trophoblastic neoplasia. CONCLUSION: Hispanic patients with complete molar pregnancy had a significantly lower risk of developing gestational trophoblastic neoplasia than white patients. There were no significant differences among groups in terms of presenting symptoms, gestational age at diagnosis, or preevacuation human chorionic gonadotropin levels for either complete mole or partial mole patients.

Relationship between race and clinical characteristics, extent of disease, and response to chemotherapy in patients with low-risk gestational trophoblastic neoplasia.

OBJECTIVE: To evaluate the potential effects of race on clinical characteristics, extent of disease, and response to chemotherapy in women with postmolar low-risk gestational trophoblastic neoplasia (GTN). METHODS: This non-concurrent cohort study was undertaken including patients with FIGO-defined postmolar low-risk GTN treated with comparable doses and schedules of chemotherapy at the New England Trophoblastic Disease Center (NETDC) between 1973 and 2012. Racial groups investigated included whites, African American and Asians. Information on patient characteristics and response to chemotherapy (need for second line chemotherapy, reason for changing to an alternative chemotherapy, number of cycles/regimens, need for combination chemotherapy, and time to hCG remission) was obtained. RESULTS: Of 316 women, 274 (86.7%) were white, 19 (6%) African American, and 23 (7.3%) Asian. African Americans were significantly younger than white and Asian women (p=0.008). Disease presentation, and extent of disease, including antecedent molar histology, median time to persistence, median hCG level at persistence, rate of D&C at persistence, presence of metastatic disease, and FIGO stage and risk score were similar among races. Need for second line chemotherapy (p=0.023), and median number of regimens (p=0.035) were greater in Asian women than in other races. CONCLUSIONS: Low-risk GTN was more aggressive in Asian women, who were significantly more likely to need second line chemotherapy and a higher number of chemotherapy regimens to achieve complete remission than women of African American and Asian descent. Further studies involving racial differences related to clinical, biological and environmental characteristics are needed.

Two rare cases of methotrexate-induced pneumonitis and pleurisy in patients with gestational trophoblastic neoplasms.

BACKGROUND: Pneumonitis is a serious and unpredictable side-effect of treatment with methotrexate (MTX) that may result in a life-threatening outcome. Pulmonary toxicity of methotrexate in patients with a gestational trophoblastic neoplasm (GTN) has rarely been reported before. CASE REPORTS: For the first time two cases of methotrexate-induced pneumonitis and pleurisy in GTN patients of Chinese ethnicity are presented. Two patients were both categorized as the low-risk group, and underwent a single regimen therapy of methotrexate. Their symptoms, such as fever, chest pain, acute nonproductive cough, dyspnea and hypoxemia did not respond to antibiotics immediately. Treatment with corticosteroids may be helpful. CONCLUSION: Awareness of pneumonitis and pleurisy, potentially life-threatening complications of MTX, is very necessary and important to early recognition and treatment.

Changing trends in gestational trophoblastic disease.

OBJECTIVE: The New Mexico Tumor Registry (NMTR) and Surveillance, Epidemiology and End Results (SEER) registries were utilized to determine (30+)-year trends in gestational trophoblastic disease and choriocarcinoma. STUDY DESIGN: Age-adjusted incidence rates of gestational trophoblastic disease per 100,000 woman-years (1973-2003) and ratios per live births and pregnancies were calculated using data abstracted from the NMTR and state vital records. SEER data (1973-2002) were used to calculate age-adjusted incidence rates, estimated annual percentage change (EAPC) and relative survival rates for choriocarcinoma. RESULTS: In New Mexico there were 1,153 cases affecting 377 non-Hispanic whites, 504 Hispanics and 241 American Indians, with respective incidence rates of 3.494, 5.150 and 9.991 (p < 0.0001). American Indian incidence rates decreased 53.3%, from 13.34 (1988-1992) to 6.23 (1998-2002). Within SEER (1973-2002), there were 504 gestational choriocarcinomas. The 30-year incidence rate was 0.132 and decreased by 37.7% (EAPC, -2.1% per year; p=0.0001)-by 40.1% for whites, 55.9% for blacks and 62.1% for others. However, over the previous 10 years, rates among blacks (0.097 vs. 0.259, p = 0.01) and for distant disease (0.044 vs. 0.071, p = 0.046) increased. CONCLUSION: Disparities in incidence rates by race/ethnicity in New Mexico are decreasing. An increase in rates among blacks and distant disease diagnosis may be the consequence of fewer regional trophoblastic centers in the United States.

Gestational trophoblastic neoplasia in American Indians.

OBJECTIVE: To analyze gestational trophoblastic neoplasia (GTN) trends among American Indians (AI) using population-based data. STUDY DESIGN: GTN incidence, by race and age, was calculated using data collected by the New Mexico Tumor Registry over 29 years (1973-2001). Live birth, pregnancy and women at risk were tabulated using data derived from the state's vital record annual reports and from the registry. Statistical methods included trends analysis and Poisson regression. There is no national registry in the United States for all GTN. Therefore, the Surveillance, Epidemiology and End Results (SEER) database was used to identify choriocarcinoma cases in American Indians between 1973 and 1999. RESULTS: Within New Mexico, 1,082 cases of GTN were identified among 752,374 live births and 904,831 pregnancies, with ratios of 1:695 and 1:836, respectively, affecting 234 AI, 355 non-Hispanic whites (NHW), 463 Hispanic whites (HW) and 30 other nonwhites. Ratios per live births (pregnancy), respectively, were significantly higher in AI (AI 1:439 [1:487], NHW 1:739 [1:949], HW 1:783 [1:903]), as was age-adjusted incidence per 100,000 woman-years (AI 10.62, NHW 3.53, HW 5.15; all P<.0001). Using Poisson models with live birth and woman-year denominators, AI were found to be at increased risk for all GTN histologic subsets (complete, partial and invasive hydatidiform mole and choriocarcinoma). Of 524 total gestational choriocarcinoma cases identified within SEER, 8 (1.8%) affected American Indians; of them, 7 were from New Mexico. CONCLUSION: In New Mexico, AI continue to be at higher risk of GTN than are other groups. Given the rarity of choriocarcinoma within SEER, especially among AI, the New Mexico dataset provides the best available estimate of trends in U.S. AI GTN risk.

Ethnic differences at risk for gestational trophoblastic disease in New Mexico: A 25-year population-based study.

OBJECTIVE: The purpose of this study was to compare gestational trophoblastic disease incidence rates with the use of population-based data. STUDY DESIGN: All incident cases between 1973 and 1997 and live birth, pregnancy, and women at risk were tabulated with the use of data that were derived from the New Mexico Tumor Registry and Vital Records and Health Statistics Annual Reports. Statistical methods included trends analyses, odds ratios, and Poisson regression. RESULTS: Of 939 total cases, 312 non-Hispanic white women, 399 Hispanic white women, 201 American Indian women, and 27 other women were affected. Age-adjusted incidence rates were significantly higher for American Indian women (11.16%) compared with non-Hispanic (3.57%) or Hispanic white women (5.32%); the probability value was <.001. When live birth (1:438 women) and pregnancy (1:486 women) denominators were considered, American Indian women alone were at increased risk, and the ratio increased by 56% over 25 years. American Indian women were also at increased risk for partial mole (relative risk, 4.03; 95% CI, 2.57-6.31), invasive mole (relative risk, 26.7; 95% CI, 7.81-93.14), and choriocarcinoma (relative risk, 6.29; 95% CI, 1.81-22.66) variants. CONCLUSION: American Indians are at increased risk relative to the other predominant ethnic groups in New Mexico. Age-adjusted standardization provided a reproducible measurement that may be applicable across other registries.