Figure 1. pelvic MRI with
rectovaginal metastatic lesion (There is one signal area posterior of
endocervical)
Cancer Therapy Vol 2, 575-578, 2004
Complete remission of an unusual
location of metastatic gestational trophoblastic
neoplasia GTN:a case report
Nadereh Behtash1,*, Malihe
Hasanzadeh1, Parviz Hanjani2
1Fellow of
Gynecology Oncology, Tehran University of Medical Sciences.
2Professor
of Gynecology Oncology, Gynecology Oncology Department, Temple University,
Philadelphia, Pennsylvania.
__________________________________________________________________________________
*Correspondence: Nadereh Behtash, Associate
Professor, Gynecologist Oncologist, Tehran University of Medical Sciences.
Gynecology Oncology Department, Vali-e-Asr Hospital, Imam Khomeini Hospital
Complex, Keshavarz Blvd., Tehran 14194, Iran; Phone: #98-21-6939320; Fax:
#98-21-6937321; E-mail: valrec2@yahoo.com,
nadbehtash@yahoo.com
Key words: Gestational
trophoblastic neoplasia, Rectovaginal septum, Pelvic metastasis, Vaginal
metastasis, Actinomycin-D.
Abbreviations: gestational
trophoblastic neoplasia, (GTN); gestational trophoblastic tumor, (GTT)
Summary
Metastatic
gestational trophoblastic neoplasia, (GTN) occurs in 4% of patients, after
evacuation of a complete mole and infrequently after other pregnancies. The
most common metastatic sites are the lung (80%), vagina (30%), brain (10%),
liver (10%). We couldn't find any report of metastasis in rectovaginal septum
in literature. To our knowledge this is the first case of metastatic GTN in
rectovaginal septum. Four weeks after evacuation of a molar pregnancy in a
28-year-old woman, a 45-55 mm solid mass was palpable deep in rectovaginal
septum. Rising titer of serum B-HCG and pelvic MRI, confirmed metastatic
lesion. After 5 courses of single agent chemotherapy, serum B-HCG returned to
normal, and pelvic MRI showed no abnormality. Although very rare, GTN can
metastases to rectovaginal septum. Careful pelvic examination can help
detection of metastatic site in the absence of metastasis in other common
sites. This large deep seated pelvic metastasis responded to single and
alternate agent chemotherapy.
Metastatic gestational
trophoblastic neoplasia (GTN) occurs in 4% of patients after evacuation of a
complete mole and infrequently after other pregnancies (Berkowitz and
Goldstein, 1996)
The most common metastatic sites are the lung (80%),
vagina (30%), and brain (10%) (Berek and Hacker, 2000). Following evacuation of
a hydatiform mole, about 20% of women will subsequently undergo further
treatment for suspected persistent gestational trophoblastic tumor (GTT) (Lurain
et al, 1985).
Vaginal metastases are usually highly vascular and may
appear reddened or violaceous. They can bleed vigorously if sampled for biopsy
(Berek and Hacker, 2000)
Metastases to the vagina may occur in the fornices or
suburethra and may produce irregular bleeding or a purulent discharge (Berek
and Novak, 2002). Yingna et al, (2002) reported that 73% of vaginal metastasis
were located in lower part and 59.05% were located on the anterior wall and 25%
were in the fornices (Yingna et al, 2002).
A search
of MEDLINE data base using the PUBMED retrieval service, with the keywords
"Gestational Trophoblastic Neoplasia" (GTN), "rectovaginal
septum", "vaginal metastasis" and "pelvic metastases"
for the period from 1966 to July 2004, showed no previously described case of
metastatic GTN to rectovaginal septum. To our knowledge, the present case is
the first case of GTN with involvement of rectovaginal septum. Also, we found
all reported vaginal metastatic of GTN have mucosal surface involvement.
A 28- year-old, Gravid 2, Para 1, was referred to
Gynecology oncology services of Vali-Asr hospital in Oct. 2003 due to molar
pregnancy.
At 8 weeks of gestation, following persistent
bleeding, pelvic ultrasonography revealed, molar pregnancy.
After complete blood chemistry and chest X-ray,
abdominal sonography, she underwent suction curettage at September
2003.Pathology report was mole hydatiform. Weekly serum B-HCG titer showed
decreasing level for 3 weeks, then it began to rise.
In pelvic and rectovaginal exam a 45-55 mm soft
rounded mass in the rectovaginal space was detected. The inferior border of the
tumor was at 4 cm from the vaginal introitus and 4 cm from the anus.The tumor
was deeply seated in rectovaginal septum and it was definitely separate from
uterus and adenexa. Pelvic MRI revealed a lesion in the septum rectovaginal (Figure 1).
Chest X-ray, abdominal CT scan and brain CTscan were
normal. There was no other abnormal finding in metastatic workup.
The total WHO scores for the patient obtained: 5, as a
low risk patient (Kohorn, 2001) )(hcg >100000 :score 4 + tumor size 45 -55 mm : score 1 ).
We started single agent chemotherapy (MTX&FA/8
days) for the patient:
|
Methotrexate |
1
mg/gk |
IM
on day 1,3,5,7 |
|
Folinic
acid |
0/1
mg/m2 |
IM
on days 2,4,6,8 |
Repeated every 7 days if possible
She received 4 courses of MTX&FA regimen, B-HCG
titer, had persistent declining titer. After the end 4th courses of
chemotherapy, serum B-HCG titer began to rise.
The chemotherapy regimen switched to actinomycine-D:
|
Actinomycine |
1/25
mg/m2 |
IV
(repeat every 14 days if possible) |
At the end of the 1st course of
chemotherapy, B-HCG titer decreased to <5 mIu/ml.Two additional courses of actinomycine-D administrated as
consolidation therapy (Figure 2).
Pelvic MRI revealed no metastatic lesion in pelvis (Figure 3).
In regular follow up visit, there were no abnormal finding in physical exam, chest X-ray and pelvic MRI. Serial serum B-HCG remained < 5 at 20 weeks after treatment.
We report a rare case of metastatic GTN which has been
presented as a tumor on the rectovaginal septum. The vagina and pelvic, next to
the lungs is the second most common metastatic site in trophoblastic tumors
(Song et al, 1983).
The vaginal lesion is replete with abundant venous
plexus without valva.This puts the patients at high risk for significant,
repeated, and uncontrolled hemorrhage (Yingna et al, 2002)
In spite of numerous cases of GTN in our hospital
during last 15 year, we had no such patient with a metastatic lesion in
rectovaginal septum.
In our search we found, all reported vaginal metastases
of GTN have mucosal surface involvement, but in this case, the mucosal of
vagina was intact and tumor was deeply seated in rectovaginal septum.
This case illustrates that GTN could be considered in
a woman of reproductive age with rectovaginal mass .Serum concentration of
human chorionic gonadotropin may reveal the exact final
diagnosis.
Figure 1. pelvic MRI with
rectovaginal metastatic lesion (There is one signal area posterior of
endocervical)
Figure 2. B-HCG regression curve and
chemotherapy courses
Figure 3. pelvic MRI after treatment
without metastatic lesion
Trophoblastic neoplasia is known to be extremely
responsive to appropriate chemotherapy. Cure rates are high with chemotherapy
even in widely disseminated disease. In low risk patient even stage II and III,
single agent regimen ,Methotrexate or actinomycin-D has been recommended
(Lurain, 2002).
MTX with Folinic acid had 95% remission in non
metastatic patients compared with 56% success rate in low risk metastatic
disease. Toxicity is low, particularly in comparison with the standard methods
of giving MTX (Disaia and Creasman, 2002).
Our patient was treated with MTX &FA because this
regimen is safe and hepatic toxicity is lower compared other regimen.
Traditionally, the patients who fail to respond to MTX
have been switched to actinomycin-D and vice versa. All the patients with low
risk disease cured by alternate secondary chemotherapy, regardless of which
regimen was used.
Actinomycin-D, given as a single bolus every 14 days also
has an extremely high therapeutic ratio in women with nonmetastatic GTN and has
been successfully used in the therapy of a few patients with low risk
metastatic disease (Petrilli and Morrow, 1980).
We reported a case of rectovaginal septum metastasis in
a patient with GTN, which is very rarely seen. In this case, mucosa of the
vagina and rectum were intact and the mass was deeply seated in rectovaginal
septum. Regardless of the large size of the tumor and deep pelvic site, single
agent chemotherapy was effective in remission of the disease in this patient.
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