Early-stage granulosa cell tumor with pelvic recurrence and lung metastasis 16 years later: A case report and review of the literature

ObjectiveOnly a few cases of early-stage ovarian granulosa cell tumor (GCT) with lung metastasis have been previously documented in the literature. We present a case of stage IA adult-type ovarian GCT with late pelvic recurrence and lung metastasis and review the relevant literature.Case reportA 71-year-old female who underwent total abdominal hysterectomy and bilateral salpingo-oophorectomy for an ovarian GCT in her early 50s presented with pelvic recurrence 16 years later. Lung metastasis was accidentally discovered during a preoperative computed tomography scan of the pelvic tumor. The patient received surgical resection and adjuvant chemotherapy. She remains alive and healthy without evidence of disease after 12 months of follow up.ConclusionThis case illustrates the importance of long-term follow-up for early-stage GCTs. It also highlights the requirement of extended examination for possible extra-abdominal/pelvic tumors.     

Bone metastasis from a granulosa cell tumor of the ovary

BACKGROUND: Granulosa cell tumors (GCT) of the ovary generally have a good prognosis. Recurrences tend to be late and are usually abdominopelvic. Bone metastases are extremely rare. CASE: A case of recurrent GCT with vertebral metastasis is presented. Radiologic studies were helpful in documenting the presence of an invasive tumor destroying the vertebral body of T7. Bone scintigraphy excluded other metastatic sites. Diagnosis could not be established by CT-scan-directed fine-needle aspiration cytology or trocar biopsies. Since the lesion was isolated and resectable, aggressive surgery with complete tumoral excision was performed followed by local radiation therapy. Megestrol acetate was given as systemic treatment. CONCLUSION: Multiple treatments of GCT may alter the pattern of recurrence. Every symptom should be thoroughly evaluated. Bone metastases may be treated aggressively.     

Embryonal rhabdomyosarcoma of the uterine corpus in a 76-year-old patient

BACKGROUND: Primary embryonal rhabdomyosarcoma of the uterine corpus is an extremely rare tumor. A case of rhabdomyosarcoma originating in the uterine myoma is presented with a review of the literature. CASE: A 76-year-old female presented with a half-year history of abdominal swelling and fever of unknown origin. A 15 x 15 x 17 cm myoma-like tumor was noted on the anterior wall of the uterine myometrium. The patient underwent total abdominal hysterectomy and bilateral salpingo-oophorectomy. The histology and immunohistochemistry aided in the final diagnosis of a pure embryonal rhabdomyosarcoma of the uterine corpus. This patient received three courses of CYVADIC chemotherapy consisting of cyclophosphamide, vincristine, doxorubicin, and dacarbazine after the surgery and is now alive with no evidence of disease 10 months from her surgery. CONCLUSION: This case is extremely rare with respect to the uterine corpus origin, especially the myoma nodule and the age of the patient.     

Granular cell tumors of the vulva

Granular cell tumor (GCT), although nearly ubiquitous, is seen infrequently in the vulva. A review of the surgical pathology files from Grady Memorial Hospital, Atlanta, Georgia, from 1983 through 1987 identified eight cases of vulvar GCT. Five of the eight patients had more than one skin and soft tissue lesion. Two of the five had biopsy-proven multicentric GCT with a unique clinical course. One of the patients was a 32-year-old woman with multiple vulvar, lingual, laryngeal, bronchial and pulmonary GCT, necessitating multiple excisions and ultimately pneumonectomy. The second patient had multiple GCTs in the vulva and inguinal area and finally in both lungs, resulting in her death at age 39. No dependable microscopic features could be identified to distinguish benign GCT from its more aggressive variant. However, Feulgen DNA histomorphometry demonstrated aneuploidy in the patient with apparent lung metastases, whereas the tumors from patients with a benign course as well as from the patient with multiorgan involvement were diploid. In three of five patients who could be interviewed there was a history of soft tissue tumors in members of the family. The multifocal nature and possible familial component of GCT need to be explored further.     

Skin metastases from vulvar cancer: a fatal event

BACKGROUND: Cutaneous metastases from a vulval cancer are exceptional with only 4 reported cases in the literature. CASE: The present case is a patient who had a radical vulvectomy with bilateral groin node dissection for a vulvar cancer stage Ib. After 9 months she a local recurrence, which was treated with radiotherapy. An anterior exenteration was performed 8 months later for a second local recurrence. Three months thereafter she had skin metastases at her thighs and calves. Chemotherapy was started; yet the lesions slowly increased. The treatment was discontinued and she died 4 months later. CONCLUSIONS: Skin metastases must be considered a preterminal event with no well-established treatment. They could be caused by retrograde permeation of tumor cells after the destruction of the local draining system.     

Vertebral osteomyelitis and epidural abscess after laparoscopic uterus-preserving cervicosacropexy

Abdominal sacrocolpopexy is a standard surgical technique to repair apical vaginal prolapse. When approached laparoscopically, a uterus-preserving technique was suggested as potentially favorable for avoiding mesh-related complications. A 52-year-old Caucasian woman came to the emergency department with right-sided low back pain and vaginal discharge more than 1 year after undergoing a laparoscopic, uterus-preserving, cervicosacropexy with Mersilene (Ethicon Inc., Somerville, NJ) mesh and permanent Gore-Tex (W.L. Gore and Associates, Inc., Flagstaff, AZ) suture. Lumbar spine magnetic resonance imaging revealed diskitis and an epidural abscess at the L5 to S1 vertebral levels, and a fistulous tract was identified extending from the rectovaginal space to the affected vertebrae. The patient underwent a fine-needle aspiration of the epidural abscess and intravenous antibiotic therapy was started. However, because of a lack of symptom resolution she was taken to the operating department on hospital day 11 and underwent a total abdominal hysterectomy, left salpingectomy, right salpingo-oophorectomy, bilateral ureterolysis, and excision of an infected Mersilene mesh. Surgical findings were notable for 2 pinpoint areas in the upper vagina consistent with fistulae tracts communicating with the retroperitoneal space. The patient recovered well from her surgery, and was discharged to a rehabilitation facility. Vertebral osteomyelitis, with or without an epidural abscess, is a rare complication of sacrocolpopexy. The findings of this case suggest this complication likely occurred as a result of a permanent suture being placed entirely (or nearly entirely) through the vaginal mucosa resulting in fistulae formation, bacterial seeding of the Mersilene mesh, and subsequent osteomyelitis. This highlights the importance of mesh and suture selection, and determining the most appropriate sites for mesh attachment.     

Primary Bartholin gland carcinoma: a report of seven cases

This study reviews our experience with 7 patients with primary Bartholin gland cancer (BGC) treated at the Queensland Gynaecological Cancer Centre (QCGC) and compares this with previously published data. A retrospective clinicopathologic review of all patients with primary BGC treated at QCGC from 1988 to 2000 was performed. Of the 7 patients treated, all underwent primary surgery and 5 of the 7 patients received radiotherapy postoperatively. All patients presented with a local swelling or a lump. Two had associated discharge and 2 had associated pain. Of the 7 patients, 2, 3 and 2 respectively were classified as having Stage IB, II or III disease. Five of the 7 patients had squamous cell carcinoma (SCC), one had adenoid-cystic carcinoma and 1 had a small-cell neuroendocrine cancer of the Bartholin gland. None of the patients with SCC developed recurrent disease. The patient with adenoid-cystic carcinoma experienced local recurrences at 4 years and again at 5 years and 3 months. Nine years after primary treatment she was diagnosed with pulmonary metastases. The patient with small-cell neuroendocrine cancer of the Bartholin gland was considered tumour-free after operation. Thorough imaging, including a CT scan of her chest, abdomen and pelvis showed no evidence of disease. She died 1 year and three months after diagnosis from disseminated pulmonary disease. We present the first report of small cell neuroendocrine cancer of the Bartholin gland. Therapeutic principles in the management of vulval cancer at other sites appear to be appropriate for management of BGC.     

Complete remission of an endometrial carcinoma with bilateral multiple pulmonary and extrapelvic metastases treated by surgery and chemotherapy consisting of paclitaxel and carboplatin

Successful management of a patient with endometrioid type, grade 2 endometrial carcinoma with bilateral multiple pulmonary and extrapelvic abdominal metastases has been reported. A 61-year-old woman with the preoperative diagnosis of stage IVB endometrial carcinoma underwent surgery followed by six cycles of chemotherapy consisting of paclitaxel (175 mg/m2) and carboplatin (area under curve 5). After the sixth course, there were no abnormal findings on chest and abdominal computed tomography. She has no evidence of disease recurrence 24 months after the induction of chemotherapy. Tumor markers are within normal limits. Endometrial carcinoma with pulmonary metastases, especially those with bilateral multiple pulmonary metastases associated with additional extrapulmonary spread can be successfully treated by extensive surgery followed by chemotherapy consisting of paclitaxel and carboplatin.     

Management of multiple level spondylolysis of the lumbar spine in young males: a report of six cases.

The most common procedure for surgical treatment of lumbar spondylolysis is intertransverse processes spinal arthrodesis with instrumentation. However, this procedure is associated with significant morbidity on long-term follow up. Direct repair of spondylolysis is an alternative method for preservation of motion segment and anatomic continuity in young adults with multiple level spondylolysis. From 1992 through 1998, six soldiers with a diagnosis of multiple level spondylolysis who complained of persistent low back pain were treated in our hospital. Spondylolysis involved two levels in five cases and three levels in one case. All patients underwent serial roentgenography of the lumbar spine and a staged pars interarticularis infiltration test with bupivacain 0.5%. Five patients underwent direct repair of the pars interarticularis defects with hook screws, translaminar screws, and bone graft, while one patient received conservative treatment. All surgically treated patients had either good or excellent results at a mean of 34.4 months follow-up. One patient who was treated nonsurgically still complained of sport restriction at 16 months follow-up. Direct repair of multiple pars interarticularis defects after a positive staged pars infiltration test can restore anatomic stability, relieve back pain, and preserve a greater range of motion of the lumbar spine in young patients with intact disc height.     

The side of groin node metastases in unilateral vulvar carcinoma

The purpose of this study was to analyze the occurrence of ipsilateral, bilateral and contralateral inguinofemoral node metastases in unilateral vulvar carcinoma. One hundred and eighty-five women with a T1 or T2 squamous cell carcinoma who underwent radical vulvectomy with bilateral inguinofemoral lymphadenectomy were surveyed. Inguinofemoral lymph node metastases were found in 23 (22.1%) out of the 104 patients with a unilateral primary tumor. These lymph node metastases were found solely on the ipsilateral side in 21 (91.3%) out of the 23 patients. One patient presented with bilateral extranodal growth in the groins. Another patient with a history of endometrial carcinoma had a right-sided vulvar tumor with contralateral groin node metastases. Half a year later, she was diagnosed with recurrent endometrial cancer on the right pelvic side-wall. Our study endorses clinical evidence that the preferential lymph flow is to the ipsilateral groin. Established lymph node metastases may disturb the normal lymph flow with contralateral metastases as a possible consequence.     

Recurrence and rapid metastasis formation of a granular cell tumor of the vulva

Histologically confirmed local recurrence of the rare entity of a granular cell tumor of the vulva was diagnosed in a 55-year-old patient with no signs of distant metastasis. Intraoperatively (local excision of mons pubis and inguinal lymphnodes), widespread regional metastasis with retroperitoneal lymphnode metastases were found. Postoperative restaging detected pulmonary, hepatic and skeletal metastases and the patient died of her disseminated disease within 4 months. Early diagnosis of the malignant phenotype of granular cell tumor using expression of S-100-protein, MIB-1, vimentin and p53 is suggested. Once a malignant granular cell tumor is diagnosed histologically, thorough staging procedure should be performed to exclude disseminated disease. If this is the case, radical surgery should be tried due to the lack of efficiency of radiotherapy and of systemic treatments.     

Abrupt Onset of Severe Pain at Term: A Case Report

This case report involves an adolescent primigravida at term who was admitted with urinary complaints to the labor and delivery unit of a medical center. Within an hour, she suddenly began screaming and complaining of severe pain running from her anterior pelvis through her vagina and up her spine. Three days of very challenging comanagement of the patient, with several recurrences of acute pain, followed. Differential diagnoses that could explain this patient's symptoms are reviewed and discussed. Difficult management issues, including the stress of clinical management in the face of unidentified disease processes, are addressed. Lacking a certain diagnosis even retrospectively, the authors request comments from readers.     

Bilateral Ureteral Stent Removal after 15 Years: A Case Report

We present the first reported case of a patient with a forgotten ureteral stent. A 68-year-old woman had undergone radical hysterectomy for cervical cancer in 1997, at which time bilateral ureteral stents were placed. She was admitted to our hospital with stranguria, dysuria, and lower back pain of 10 days duration. Preoperative radiologic evaluation revealed the presence of ureteral stents, which had been in place since 1997. With the patient under general anesthesia, the bilateral stents were removed by urethral cystoscopy. No stone formation was noted. The procedure was easy and fast, and no intraoperative complications occurred.     

Abrupt onset of severe pain at term A case report

This case report involves an adolescent primigravida at term who was admitted with urinary complaints to the labor and delivery unit of a medical center. Within an hour, she suddenly began screaming and complaining of severe pain running from her anterior pelvis through her vagina and up her spine. Three days of very challenging comanagement of the patient, with several recurrences of acute pain, followed. Differential diagnoses that could explain this patient's symptoms are reviewed and discussed. Difficult management issues, including the stress of clinical management in the face of unidentified disease processes, are addressed. Lacking a certain diagnosis even retrospectively, the authors request comments from readers.     

Supervivencia tras el tratamiento de metástasis cerebral, única,tardía en una paciente afectada de cáncer de ovario

Central nervous system metastases from ovarian carcinoma are uncommon. We present the case of a 48-year-old woman with a bilateral serous papillary ovarian carcinoma. She received neoadjuvant chemotherapy and underwent suboptimal cytoreductive surgery and pelvic irradiation. Nine years later she had a pleural relapse of the disease and was treated with six cycles of carbotaxol. Eleven years after the initial diagnosis she presented an 8 cm brain metastasis and underwent resection followed by whole-brain radiotherapy with a boost on the tumor bed. The patient is currently asymptomatic and shows no sequels. In patients with an isolated brain metastasis, surgery followed by radiotherapy and systemic chemotherapy offers the best results.     

Robotic-assisted laparoscopic cytoreductive surgery for lobular carcinoma of the breast metastatic to the ovaries

The first case of robotic-assisted laparoscopic cytoreductive surgery for a metastatic malignant ovarian tumor is described. A 65-year-old woman who was treated for breast cancer 13 years earlier presented with a deep venous thrombosis in the right leg and a pelvic mass. Imaging tests showed bilateral solid ovarian tumors and an enlarged lymph node at the level of the right common iliac vessels but no evidence of any other tumor metastases. The patient was offered and accepted to undergo a da Vinci robotic-assisted bilateral adnexectomy with hysterectomy and pelvic lymph node sampling. The surgery took 200 minutes, and the patient was discharged uneventfully on the third postoperative day. Histologic examination showed strongly estrogen receptor-positive metastatic lobular carcinoma of the breast in both ovaries and fallopian tubes. She was further treated with adjuvant exemestane and is currently doing well. It is possible to treat selected patients with malignant ovarian tumors by robotic-assisted laparoscopy.     

Long-term survival in an ovarian cancer patient with brain metastases

BACKGROUND: Central nervous system (CNS) metastases from ovarian adenocarcinoma are uncommon. The long-term prognosis for these patients is poor, with studies reporting a mean survival of less than 12 months. CASE: We present a case involving a 57-year-old woman diagnosed and treated for primary ovarian cancer in 1994. She underwent optimal cytoreductive surgery and received adjuvant chemotherapy. In 1996, she was diagnosed with a right cerebellar metastatic lesion, and treated with surgery and whole-brain radiotherapy. She is currently 7 years post-treatment of her brain metastasis without evidence of recurrent disease. CONCLUSION: Brain metastases from primary ovarian cancer are a relatively rare finding. These patients have a poor prognosis, with studies reporting a mean survival of 12 months. However, the patient in this report remains disease-free since her treatment for metastatic disease. Aggressive surgical and radiation treatment for patients with isolated CNS metastases is reasonable.     

External hemipelvectomy as treatment for solitary coxofemoral metastasis from endometrial carcinoma: case report and review of the literature

The best treatment for bone metastasis from endometrial cancer as a presenting feature is unclear. We report the first case in the literature of coxofemoral metastases from endometrial cancer treated by surgical approach. Then, after a careful review of the literature, we discuss the best therapeutic option for this subset of patients. A 62-year-old woman with pain, erythema and swelling of the left leg and no history of postmenopausal bleeding underwent biopsy of the leg, which revealed a moderately differentiated endometrial carcinoma, infiltrating muscle and adipose tissues. There were no other sites of distal spread. A literature review was conducted by searching the items 'endometrial cancer' and 'bone metastasis' in MEDLINE and EnBase up to September 2010. The patient was treated with neoadjuvant chemotherapy, but she did not show a clinical response. By considering her prognosis and quality of life, we decided to perform for the first time a total abdominal hysterectomy with bilateral salpingo-oophorectomy in addition to an external hemipelvectomy with a limb amputation and partial ilium and pubic preservation. Thirty months after the procedure the patient is still alive. No other similar results are present in the literature. Patients in good clinical condition with a single bone metastasis of endometrial cancer should be treated aggressively with surgery, as survival can be extended with an acceptable quality of life.     

Bilateral primary squamous cell carcinoma of the ovary: a case report of isolated metastasis to the lateral pelvic wall

Primary squamous cell carcinoma of the ovary is rare. The majority of cases arise most commonly from the lining of a dermoid cyst, and less often in endometriosis or a Brenner tumor. A 40-year-old woman underwent exploratory laparotomy and was found to have a right ovarian tumor adherent to the lateral pelvic wall with no ascites. She underwent total abdominal hysterectomy with bilateral salpingo-oophorectomy, pelvic lymphadenectomy, infracolic omentectomy, appendectomy, and right nephrectomy for bilateral primary squamous cell carcinoma of the ovary. She was started on multiagent chemotherapy. On follow-up after two years the patient had died of cerebral metastases. To our knowledge in this report we present the first case in the English literature of bilateral pure squamous cell carcinoma of the ovary.     

Groin recurrence after micrometastasis in a sentinel lymph node in a patient with vulvar cancer

BACKGROUND: The sentinel lymph node concept is attractive in vulvar cancer because of the potential to avoid the morbidity associated with formal groin dissection. CASE: An 84-year-old patient with a T2 carcinoma of the anterior vulva underwent surgery including bilateral sentinel node excision after identification with technetium-labeled nanocolloid. Frozen section histology showed a tumor deposit <1 mm in diameter in a left groin node whereas four nodes in the right groin were apparently negative. Completion lymphadenectomy was performed only for the left groin. Final histology including serial sectioning showed a micrometastasis in one of seven nodes from the right groin; no further treatment was given. Sixteen months postoperatively the patient developed a recurrence in the right groin; the left groin was free of tumor. CONCLUSION: This case indicates that groins with a micrometastasis detected by sentinel lymph node excision require further treatment.