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OJHAS Vol. 8, Issue 2: (2009
Apr-Jun) |
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Placental site Trophoblastic Tumor with Pulmonary and Brain Metastases |
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Anuradha Phadikar,
Associate
Professor, Dept. of G & O, Medical College, Kolkata
Asit Ranjan Deb
Associate
Professor, Dept. of Radiotherapy, NRS Medical College, Kolkata
Chandana Das
Associate Professor,
Dept of G & O, North Bengal Medical College, Darjeeling,
Picklu Chaudhuri
Assistant
Professor, Dept.of G & O, Chittaranjan Seva Sadan, Kolkata,
Aishik Majumdar
Post Graduate Trainee, NRS Medical college, Kolkata. |
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Address For Correspondence |
Narayani Apartment,GF-14,
Jardabagan, Jyangra,
Baguiati, Kolkata-700059,
West Bengal, India.
E-mail:
anuradha_phadikar@rediffmail.com |
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Phadikar A, Deb AR, Das C, Chaudhuri P, Majumdar A. Placental site Trophoblastic Tumor with Pulmonary and Brain metastases. Online J Health Allied Scs.
2009;8(2):11 |
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Submitted: May 25, 2009; Accepted
Jul 25, 2009 Published: Sep 8, 2009 |
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| Abstract: |
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A
rare case of Placental Site Trophoblastic Tumor with multiple metastases was
managed with initial chemotherapy (EMA-CO) and radiotherapy followed by surgery
with good prognosis.
Key Words: PSTT,
EMA-CO
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Placental
site trophoblastic tumor
(PSTT) is a very rare form of gestational trophoblastic disease (GTD),
consisting predominantly of intermediate trophoblast and has some unique
features such as low serum concentration of β-hCG and high human placental lactogen (hPL), relative insensitivity to chemotherapy and late metastasis.1
A 25 years old,
Para 2+0 with two normal deliveries, having the last child birth 4 years
back, was referred from periphery to the outpatient department of G&O in NRS Medical
College and Hospital, as a diagnosed case of PSTT. At the time of admission, her
chief complaints were irregular vaginal bleeding, cough and headache for last one year. There was no preceding history
of amenorrhea.
On examination, she was thin built, severely anaemic with a BP
of 100/60 mm of Hg and chest clinically normal. Abdominal examination revealed a firm, irregular, suprapubic mass corresponding to 14-16 weeks of gestation with restricted mobility. Cervix and vagina appeared normal on speculum examination. Bimanual
vaginal examination established a bulky uterus of 14-16 weeks size with the os open and moderate vaginal bleeding. A cystic mass about 8x6 cm
was felt through the left fornix, adherent with uterus.
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| Fig
1: CT Scan showing Brain Metastasis |
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Fig
2: Histopathology of PSTT |
Investigations revealed that her haemoglobin was 2.4gm%
and LFT, urea and creatinine were within normal
limits. Ultrasound examination of the whole abdomen and pelvis showed one heterogeneous
mass (8.1x6.4cm) in the anterior and upper part of the uterus, indenting the bladder wall.
On color Doppler study the mass was found to be highly vascular. Both ovaries were slightly
enlarged with multiple cysts. Chest X-ray showed bilateral multiple opacities.
CT scan of the brain revealed two enhancing nodules, one at the left basi–frontal region (7x8mm) and another at right
the post parietal region (11x13 mm) with peri-lesional edema and minimal regional mass effect.
Endometrial biopsy confirmed PSTT. Her β-hCG level on admission was 9700 mIU/ml while the previous β-hCG was
945mIU/ml. On the basis of clinical, laboratory, radiological and histopathological findings, the case was diagnosed
as metastatic PSTT.
After resuscitating her with 6 units of blood transfusion and taking into consideration
of her poor general condition and brain metastasis, we started chemotherapy (CT) on her.
She received 7 cycles of CT with EMA-CO regime, intra-thecal methotrexate (10mg twice
a week, 6 doses), cranial irradiation (10 exposures) and local irradiation of abdominal
mass (25 exposures) before we opted for surgery. Laparotomy with total abdominal
hysterectomy was undertaken. Uterus was found to be bulky and a necrotic solid growth, 5-6 cm in diameter, was
found protruding through the anterior uterine wall near left cornu which was adherent to urinary bladder. The mass was relatively
avascular and dissection from bladder wall was surprisingly easy with minimal bleeding.
However, the histopathology was inconclusive. She was put on four cycles of
postoperative chemotherapy. Although her β-hCG level touched baseline after two cycles,
two more cycles of CT were given to reduce the chance of recurrence.
Placental site trophoblastic tumor (PSTT) is a very rare and unique form of gestational
trophoblastic disease (GTD).¹ There are over just 200 cases reported in literature.
PSTT differs histologically and immunologically from gestational choriocarcinoma.1
Clinical behavior of PSTT varies and prediction of its biological behavior remains
difficult. WHO prognostic scoring system also does not correlate well with clinical course of PSTT. However, poor prognostic factors are an interval of >2 years from known antecedent pregnancy, Mitotic Index > 5/10 HPF
and extensive necrosis and extension outside the uterus. Increased uterine volume significantly increases the chance of metastasis.2 The most common presenting symptoms are irregular vaginal bleeding with or without preceding amenorrhea.3 Diagnosis of PSTT is confirmed by histopathology report. However, persistently low level of β-hCG or elevated hPL along with unexpected resistance to CT raises the suspicion of the disease. It can present with galactorrhoea, nephrotic syndrome or just raised serum β-HCG. Metastasis at presentation occurs in 10-15% of patients and recurrence develops in 10% cases. Surgery is the mainstay of treatment in non-metastatic PSTT.
Present day advances of CT has a distinct role in the management of PSTT. Radio-therapy is especially effective for metastatic disease.4
Conservative therapy by combination CT without hysterectomy may be an alternative
for patients desiring future fertility.¹ Bonnazzi et al6 reported that one of their
patients, treated medically only with EMA-CO, had complete recovery. J-H Nam et al5 have
reported two cases of PSTT treated successfully by CT followed by curettage without
definite surgery. The first line CT regimen is EMA-CO, as reports have shown complete
response with this regimen. For EMA-CO refractory cases, second line CT is EP/EMA.
The most recent data from the Charing Cross Hospital, UK and other centers suggest that EMA/EP is the most effective treatment for metastatic or recurrent PSTT. Clinical
outcome of PSTT, reported in the literature, are highly variable. All cases of metastasis
to vital organs e,g. brain, result in mortality despite all forms of treatment.¹
Our patient, in spite of poor prognostic factors (interval more than 4 years, brain
metastasis), tolerated the treatment well with complete remission and she is alive and
well till date, one year after completion of treatment. Because of rarity and its
successful outcome, this case deserves reporting.
- Seung Jo Kin. Placental Site
Trophoblastic Tumor. Best Practice & Research. Clinical Obstetrics and
Gynecology. 2003;17(6):969-984.
- Feltmate CM, Genest DR, Wise L et al.
Placental Site Trophoblastic Tumor: a 17- year experience at the New England
Trophoblastic Disease Center. Gynecol Oncol
Sept 2001;82(3);415-9.
- Gillespie AM, Liyim D, Goepel JR et
al. Placental Site Trophoblastic Tumor: a rare but potentially curable
cancer. Br. J of Cancer. 2000;82:1186-1190.
- Dessau R, Rustin GJ, Dent J et
al. Surgery and chemotherapy in the management of Placental Site Tumor. Gynecol Oncol. 1990;39(1):56-9.
- Nam J-H,
Kim J-H, Park
Y et al.
Placental Site Trophoblastic Tumor: Can it be treated by chemotherapy alone
without surgery? Inter
Gynecological Cancer 2003;7(5):381-387.
- Bonazzi C, Urso M, Dell ‘Anna T et al.
Placental Site Trophoblastic Tumor: an overview. Reprod Med. 2004;49(8):585-8.
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