Intrapulmonary mature cystic teratoma presenting with haemoptysis treated with right middle lobectomy in a low-resource setting.

A teenage girl presented to our remote rural hospital with history of massive haemoptysis. A CT scan showed a spherical lesion in the right lung. Exploratory thoracotomy was performed with a provisional diagnosis of pulmonary hydatid cyst. Intraoperative findings were consistent with intrapulmonary teratoma and this was confirmed by a histopathological diagnosis of mature teratoma. Four years later, the patient presented again with recurrence of haemoptysis and a right middle lobectomy was performed. We present a report of successful management of this patient with 20-year follow-up since the first surgery, describe the pathology of this rare condition and the procedures used to treat it. We also discuss the social and economic factors that affect decision making in the management of rare conditions in remote rural areas among poor communities that are usually referred to tertiary hospitals and suggest modifications to protocols to provide effective treatment in spite of resource constraints.

Need for urodynamic evaluation as a regular follow-up tool in assessment of long-term urological outcomes in patients with sacrococcygeal teratoma.

AIM: To assess the long-term urologic outcomes in follow-up of patients of sacrococcygeal teratoma (SCT) using urodynamic study (UDS) in addition to clinical and radiologic evaluation. METHODS: A prospective study of clinical, radiological and urodynamic evaluation in patients with SCT who underwent resection between January 2002-June 2015 and were followed up till January 2016 was conducted. RESULTS: Total 57 patients, 42 (73.7%) females and 15 (26.3%) males with 35 (62.4%) following treatment for benign and 22 (38.5%) for malignant disease were included. Twenty-eight of 57 (49.12%) had urological problems. Clinical complaints in 21 (36.8%) patients included stress urinary incontinence-14 (66.7%), enuresis-9 (42.9%), and poor stream or dribbling of urine-6 (28.6%). Eight of 51 patients (15.7%) had abnormal ultrasound findings, which included contracted, trabeculated thick walled bladder (3), bilateral hydronephrosis (3) and significant post void residue (PVR) (6). Seven of 57 underwent micturating cystourethrogram (MCU), 5 had an abnormal report[significant PVR (4), small trabeculated bladder (3), reflux (2) and large capacity bladder (1)]. Urodynamic study was done in 27 patients, 18/27 (66.7%) had abnormalities. Six patients without any clinical or ultrasonographic abnormalities had abnormal UDS. Total 28 (49.12%) had urological comorbidities. Three patients had overactive bladder, five dysfunctional voiding, one underactive bladder and one had giggle incontinence. Children were managed by behaviour therapy and pharmacotherapy. CONCLUSION: Urodynamic evaluation could detect abnormalities in patients who had no urinary complaints or abnormality on ultrasound. The abnormalities have a potential for progressive upper tract damage. Urodynamics should be an integral part of urological surveillance in patients operated for SCT. TYPE OF STUDY: Prognostic study. LEVEL OF EVIDENCE: Level II (Prospective cohort study).

Assessment of the treatment approach and survival outcomes in a modern cohort of patients with atypical teratoid rhabdoid tumors using the National Cancer Database.

BACKGROUND: Atypical teratoid rhabdoid tumors (ATRTs) are rare brain tumors that occur primarily in children under the age of 3 years. This report evaluates the treatment approach and survival outcomes in a large cohort of patients treated in the United States. METHODS: Using the National Cancer Database, the analysis included all ATRT patients aged 0 to 18 years who were diagnosed between 2004 and 2012 and had complete treatment data. RESULTS: Three hundred sixty-one ATRT patients were evaluated. The 5-year overall survival (OS) rate was 29.9%, and it was significantly lower for children who were less than 3 years old (5-year OS, 27.7%) versus those who were 3 years old or older (5-year OS, 37.5%; P < .001). The best outcome was seen for patients with localized disease who received trimodality therapy (surgery, chemotherapy, and radiation therapy [RT]) with a 5-year OS rate of 46.8%. The utilization of trimodality therapy significantly increased during the study period (27.7% in 2004-2008 vs 45.1% in 2009-2012; P < .01), largely because of the increased use of RT. In a multivariate analysis, treatment that did not utilize trimodality therapy was associated with significantly worse OS (hazard ratio, 2.52; 95% confidence interval (1.82-3.51). Children aged 0 to 2 years were significantly less likely to receive trimodality therapy because of decreased utilization of RT in this age group. CONCLUSIONS: The use of trimodality therapy significantly increased during the study period and was associated with improved outcomes. For patients with localized disease who received trimodality therapy, the OS rate at 5 years approached 50%. However, further research into the optimal management of children less than 3 years old is needed because of their significantly worse OS in comparison with older children. Cancer 2017;123:682-687. © 2016 American Cancer Society.

Sacrococcygeal teratoma: case report.

We present a male newborn child with a sacrococcygeal mass who was sent to clinic 46 of the Mexican Social Security Institute located in Gomez Palacio, Durango, Mexico for pediatric/neonatal surgical resolution. The mass was detected on gestation week 24 in the sacrococcygeal area and was initially interpreted as a myelomeningocele. On gestation week 32, the mass had grown, so the diagnosis of cystic hygroma was posed. The child was born at 38 weeks of gestational age with a large tumor in the sacrococcygeal area. Images were obtained, and tumor resection was performed without complications. Pathologic examination confirmed the diagnosis of sacrococcygeal teratoma. The postoperative course was uneventful and there were no further complications.

Se presenta el caso de un recién nacido del género masculino que es enviado a la clínica 46 del Instituto Mexicano del Seguro Social de Gómez Palacio, Durango, México para manejo por cirugía pediátrica y neonatología, por la presencia de una masa en región sacrococígea que fue detectada en la semana 24 de gestación como probable mielomeningocele. A las 32 semanas de gestación se observó un mayor crecimiento y se sospechó de un higroma quístico. Se programa cesárea a las 38 semanas de gestación y, después de exámenes imagenológicos, se realiza resección del tumor sin complicaciones. El estudio anatomopatológico confirmó el diagnóstico de teratoma sacrococcígeo. La evolución posoperatoria inmediata y su condición en la actualidad, son satisfactorias.

Assessment of pain and stress intensity among women with ovarian endometriomas versus teratomas.

BACKGROUND: In recent years, numerous studies have considered endometriosis to be a subclinical, local inflammatory process in the pelvic peritoneum, the main symptom of which is pain. OBJECTIVES: To assess pain intensity and pain-related stress in women with ovarian endometriomas versus teratomas. METHODS: In total, 860 women (18 to 38 years of age) treated laparoscopically for lesions in the adnexa between September 2006 and November 2013 were included in the present study. After an intraoperative review of their histopathological lesions, the patients were divided into two study groups: group E (n=480), with histopathologically confirmed ovarian endometriomas; and group T (n=380), after laparoscopic treatment of ovarian teratomas. A questionnaire was generated for the study and completed by each group. Statistical analysis was performed using the Mann-Whitney U test (P≤0.05). RESULTS: Median pain scores for group E versus group T were as follows: pain during menstruation, 6 versus 3 (P=0.001); pain outside of menstruation (in professional life), 2 versus 2 (P=0.014); and pain during sexual intercourse, 3 versus 1 (P=0.006). Pain-related stress scores were higher in group T versus group E (5 versus 3; P=0.007). CONCLUSION: Ovarian endometriomas caused more pain than ovarian teratomas, likely due to the endometrial tissue component and not a mass effect. The assessment of pain and pain-related stress associated with the pelvis minor showed a high level of pain intensity and lower level of pain-related stress among patients with ovarian endometriomas.

Multicentre risk-adapted management for stage I non-seminomatous germ cell tumours.

BACKGROUND: The Spanish Germ Cell Group is composed of 60 centres. Our challenge was to define a surveillance protocol that would be safe and suitable regardless of population size or geographic coverage. METHODS: From January 1994 to January 2004, 589 patients with stage I non-seminomatous germ cell tumours entered a risk-adapted surveillance protocol after orchiectomy. Patients with vascular or local invasion of adjacent structures (231/589; 39%) received two cycles of BE400P (bleomycin 30 U/week, etoposide 100 mg/m2 x4, cisplatinum 25 mg/m2 x4). Other patients (358/589; 61%) were kept on close follow-up (chest X-ray; serum tumour markers: first year every 2 months, second year every 3 months, third year every 4 months; abdominal computed tomography scans at every other outpatient control). The outcomes selected for the study were feasibility, relapse rate and number of patients lost to follow-up and mortality. RESULTS: Median follow-up was 40 months. In the surveillance group, 21 patients were lost to follow-up. In the chemotherapy group, two patients relapsed at 12 and 14.5 months and they are presently free of disease. In the surveillance group, 71 (19%) patients relapsed, of which 55 (71%) relapsed within the first year. Five (1.4%) patients died of their cancer. Factors associated with relapse were embryonal carcinoma and vascular invasion in patients who refused chemotherapy. CONCLUSIONS: Our risk-adapted surveillance protocol provided a low rate of recurrences.

Sacrococcygeal teratoma: prenatal assessment, fetal intervention, and outcome.

PURPOSE: To understand the natural history and define indications for fetal intervention in sacrococcygeal teratoma (SCT), the authors reviewed all cases of fetal SCT presenting for evaluation. METHODS: Prenatal diagnostic studies including ultrasound scan, magnetic resonance imaging (MRI), echocardiography and pre- and postnatal outcomes were reviewed in 30 cases of SCT that presented between September 1995 and January 2003. RESULTS: The mean gestational age (GA) at presentation was 23.9 weeks (range, 19 to 38.5) with 3 sets of twins (10%). Overall outcomes included 4 terminations, 5 fetal demises, 7 neonatal deaths, and 14 survivors. Significant obstetric complications occurred in 81% of the 26 continuing pregnancies: polyhydramnios (n = 7), oligohydramnios (n = 4), preterm labor (n = 13), preeclampsia (n = 4), gestational diabetes (n = 1), HELLP syndrome (n = 1), and hyperemesis (n = 1). Fetal intervention included cyst aspiration (n = 6), amnioreduction (n = 3), amnioinfusion (n = 1), and open fetal surgical resection (n = 4). Indications for cyst aspiration and amnioreduction were maternal discomfort, preterm labor, and prevention of tumor rupture at delivery. Although 15 SCTs were solid causing risk for cardiac failure, only 4 fetuses met criteria for fetal debulking based on ultrasonographic and echocardiographic evidence of impending high output failure and favorable anatomy at 21, 23.6, 25, and 26 weeks' gestation. Intraoperative events included maternal blood transfusion (n = 1), fetal blood transfusion (n = 2), chorioamniotic membrane separation (n = 2), and fetal arrest requiring successful cardiopulmonary resuscitation (CPR) (n = 1). In the fetal resection group, 3 of 4 survived with mean GA at delivery of 29 weeks (range, 27.6 to 31.7 weeks), mean birth weight of 1.3 kg, hospital stay ranging from 16 to 34 weeks, and follow-up ranging from 20 months to 6 years. Postnatal complications in the fetal surgery group included neonatal death (n = 1, secondary to premature closure of ductus arteriosus with cardiac failure), embolic event (n = 1, resulting in unilateral renal agenesis, jejunal atresia), chronic lung disease (n = 1), and tumor recurrence (n = 1). CONCLUSIONS: For fetal SCT, the rapidity at which cardiac compromise can develop and the high incidence of obstetric complications warrant close prenatal surveillance. Amnioreduction, cyst aspiration, and surgical debulking are potentially life-saving interventions.

Diagnostic value of US and CT in the assessment of ovarian teratomas and their qualifications for surgical treatment with laparoscopic method.

In the group of 17 women after surgery with histopathologically confirmed diagnosis of teratomas diagnostic usefulness of US and CT was analysed. Differentiated echostructure of tumours was correlated with different densities of pathologic tissues in CT examination, also after their contrast enhancement. In 2 patients teratomas of both ovaries were recognised, in the remaining 15 women tumours were unilateral. In total morphologic pictures of 19 teratomas were assessed, 9 times tumours with prevailing liquid component had the picture of a thick-walled cyst, 7 times these were hyperechogenic fatty foci and in 3 cases had the character of solid tumours. Teratomas had different morphologic pictures, but it was possible to show the prevalence of one element and classify the tumour to one of the three types of teratomas. It was found that US examination, due to its high frequency, lets early diagnose teratomas before they are accessible in the clinical exam, in the symptomless period or when they give indirect symptoms, usually dysuric ones. CT examination, due to its high sensitivity in recognising fatty tissue and calcifications, is necessary before laparoscopic operation requiring fragmentation of big tumours to determine their character.

Resection of small, residual retroperitoneal masses after chemotherapy for nonseminomatous testicular cancer: a decision analysis.

BACKGROUND: After chemotherapy for metastatic, nonseminomatous testicular cancer, small, retroperitoneal lymph nodes still harbor mature teratoma or viable cancer cells in less than half of patients with normal tumor markers. Surgical resection is an effective treatment to remove residual masses, but observation may also be considered. METHODS: Using a decision analysis model, the authors estimated the survival achieved with either resection or observation for patients with residual masses measuring 0-20 mm. Prognostic estimates were obtained from an international data set containing 362 patients with masses < or =20 mm and from 10 clinical experts. RESULTS: According to the model, resection prolonged life expectancy by more than 2 years for masses 11-20 mm and by more than 1 year for masses 0-10 mm. The estimated gains in 5-year survival were 4.3% and 2.7%, respectively. In a sensitivity analysis, these results appeared rather robust for changes in the estimates of the experts. The magnitude of the gain in survival, however, depended on the probabilities of the residual histologies, which could be estimated with several well-known predictors, and the assumed benefit of resection for residual mature teratoma or cancer. CONCLUSIONS: Resection may on average be beneficial for patients with small, residual masses. The expected benefit depends on the probability and risks of residual malignancy, regarding which further research is required. For decision-making regarding individual patients, the morbidity and costs of resection and a patient's individual preferences should be considered in addition to any assumed gain in survival.

MRI in the assessment of a newborn with cervical teratoma.

Teratoma of the head and neck is a rare lesion comprising 6 % of all teratomas, with only 3 % occurring in the cervical region [1]. Most are non-malignant lesions consisting of a variety of tissues of variable maturity, commonly with neuroepithelial and thyroid elements. They often present as a large cystic mass in the neck of a neonate or infant and frequently cause respiratory embarrassment due to local mass effect necessitating urgent surgical intervention. They may be difficult to distinguish from cystic hygromas, both clinically and radiologically. Imaging plays an important role in the assessment of these lesions, especially in preparation for surgery. We present a case of cervical teratoma and emphasise the role of MRI.

Surgical management of benign cystic teratoma. Laparoscopy vs. laparotomy.

OBJECTIVE: To compare laparoscopy to laparotomy for the surgical treatment of benign cystic teratomas. STUDY DESIGN: This was a retrospective, case series comparison of 20 patients who had surgery at Rochester General Hospital from June 1991 to January 1993 for benign cystic teratomas. Statistical comparisons were made by Student's t test or chi 2 analysis. RESULTS: Eight patients had surgery via laparoscopy and 12 via laparotomy. Laparoscopic oophorectomy and cystectomy resulted in significantly shorter hospital stays and decreased hospital costs, but the surgery time was significantly increased. The patients' fertility status influenced the choice of conservative cystectomy or nonconservative oophorectomy as the surgical procedure. There were no serious complications in any patients, including those with laparoscopic cystectomies and intraperitoneal spill. CONCLUSION: Oophorectomy and ovarian cystectomy via operative laparoscopy appear to be reasonable options for the surgical treatment of benign cystic teratoma.

[Non-seminomatous germ cell tumors of the testis. Role of the pathologist in the assessment of prognostic factors based on the specimen of orchiectomy. Proposal of an anatomopathological examination report defined by the Study Group of the Committee of Cancerology of the French Association of Urology]. / Tumeurs germinales non séminomateuses du testicule. Rôle de l'anatomopathologiste dans l'établissement des facteurs du prognostic sur la pièce d'orchidectomie. Proposition d'une fiche d'etude anatomopathologique établie par le Groupe de Travail du Comité de Cancérologie de l'Association Française d'Urologie.

Non-seminomatous germ cell tumors of the testis. Role of the pathologist in the definition of the prognostic factors based on examination of the orchidectomy specimen. Proposal of a pathological examination report form defined by the study Group of the Oncology Committee of the French Urological Association.

Neonatal sacrococcygeal teratoma: multiimaging modality assessment.

Neonatal sacrococcygeal teratoma (SCT) is a rare and potentially malignant tumor. We report on four cases of neonatal SCT, in which ultrasound (US), computed tomography (CT), and magnetic resonance imaging (MRI) were used preoperatively to accurately establish the extent of the tumor and its relationship to the surrounding anatomic structures. This approach facilitates complete surgical resection and optimal outcome.

Is routine primary retroperitoneal lymph node dissection still justified in patients with low stage non-seminomatous testicular cancer?

We present 8 years' experience of primary retroperitoneal lymph node dissection (RLND) in 190 patients with low stage non-seminoma; 154 patients had clinical stage I (CSI) and 36 had clinical stage IIa (CSIIa) disease. Of the 154 patients with CSI tumours, 33 had increased serum AFP and/or HCG before RLND (CSIM+) and 121 had normal tumour markers (CSIM-). Retroperitoneal lymph node metastases (pathological stage II) (PSII) were found in 38 of 121 patients with CSIM-, in 19 of 33 patients with CSIIM+ and in 26 of 36 patients with CSIIa. In a multivariate analysis, the presence of small vessel infiltration (demonstrated in histological sections of the primary tumour) and a prolonged tumour marker half-life were predictive factors for PSII. These 2 factors enabled a group of non-seminoma patients with CSI disease to be identified who had a 15% risk of retroperitoneal tumour growth (low risk group) as compared with a high risk group where 60 to 70% of patients had retroperitoneal lymph node metastases. Relapses occurred in 7 of 107 patients with PSI and in 6 of 83 patients with PSII disease; in the latter group, 5 relapses developed before the start of routine adjuvant chemotherapy; 6% of patients developed major post-operative complications. In addition, "dry ejaculation" was the principal side effect following RLND (unilateral RLND: 20/132 patients; bilateral RLND: 50/54 patients). The comparative cost to the health service during the first year of follow-up was estimated for low risk non-seminoma patients with CSI subjected to RLND and for those in whom a surveillance policy was adopted. The latter approach was preferable. It was concluded that a surveillance policy should be followed in low risk non-seminoma CSI patients provided that frequent follow-up is possible. A more active policy is recommended in high risk patients (e.g. adjuvant chemotherapy without RLND). Nerve-sparing RLND may be considered in patients with CSIIa disease and negative tumour markers.