Giant immature teratoma in a 15 year old causing anti-n-methyl-d-aspartate (anti-nmda) receptor encephalitis-like and Guillain-Barré Syndrome as paraneoplastic manifestations: A case report / Philippine Journal of Obstetrics and Gynecology

@#Germ cell tumor is the most prevalent ovarian tumor in young women between 10 and 30 years of age.[1] However, immature teratomas account for only 20% of the malignant ovarian tumors found in the adolescent age group.[2] More uncommon is the occurrence of immature teratoma causing anti‑N‑methyl‑D‑aspartate receptor (Anti‑NMDAR) encephalitis and Guillain‑Barré Syndrome (GBS). This is a case of a 15‑year‑old female, nulligravid, who initially presented sudden behavioral change and symmetrical weakness of both lower and upper extremities with concomitant seizure episodes and with palpable lower abdominal mass. The patient was diagnosed to have GBS and treated with intravenous immunoglobulin causing resolution of neurologic symptoms months after. During the management of GBS, the patient noted increasing abdominal girth. Tumor markers showed elevated alpha‑fetoprotein, CA125, and lactate dehydrogenase. An imaging study revealed predominantly solid ovarian mass hence malignancy was considered. Once the medical condition stabilized, the patient underwent fertility‑sparing surgery with the final histopathologic result of immature teratoma. The aim of this report is to present a case of anti‑NMDAR encephalitis and GBS as paraneoplastic manifestation of immature teratoma.

Cistoadenoma volumoso associado a teratoma ovariano maduro, com evolução para pseudomixoma peritoneal: relato de caso / Giant cystadenoma associated with mature ovarian teratoma with progression to pseudomyxoma peritonei: a case report

Massas anexiais representam grande desafio diagnóstico, devido a seu caráter geralmente assintomático¹. Traz-se caso peculiar de paciente que evoluiu com sintomas exuberantes de rápida evolução, situação característica de cistoadenomas, tumores benignos de origem epitelial²,³. Realizada laparotomia, com achados macroscópicos que revelaram conteúdo mucinoso e presença de pelos e neovascularização em seu interior. O anatomopatológico confirmou o diagnóstico de cistoadenoma associado a teratoma ovariano, sendo assim considerado um tumor de colisão ovariana.

Adnexal masses represent a major diagnostic challenge due to its usually asymptomatic characteritcs¹. We present a peculiar case of a patient who evolved with exuberant symptoms of rapid evolution, characteristic of cystadenomas, benign tumors of epithelial origin²,³. Laparotomy was performed, with macroscopic findings that revealed mucinous content and the presence of hairs and neovascularization inside. Pathology confirmed the diagnosis of cystadenoma associated with ovarian teratoma, thus being considered an ovarian collision tumor.

Anti-NMDA receptor encephalitis caused by ovarian teratoma / 中华内分泌外科杂志

Ovarian teratoma is agynecological disease,which has been found to be closely related to antiNMDA receptor encephalitis in recent years.Anti-NMDA receptor encephalitis is a kind of autoimmune encephalitis.Its etiology,pathogenesis,diagnosis and treatment are still not clear,and the disease has various clinical manifestations and special test methods,which are often ignored by clinicians.The prognosis is poor if the best treatment timing is missed,so early treatment is very important.At present,the main treatment methods include surgical treatment and immunotherapy.Most people can be cured,but the disease has a certain recurrence rate.The recurrence mechanism and treatment need further investigation.Reports on maternal anti-NMDAR encephalitis are rare,but we need to pay lots of attention to it as it is related to maternal and fetal outcomes.

Teratoma inmaduro (maligno) de ovario / Ovarian immature (malignant) teratoma

Resumen El teratoma inmaduro se describió por primera vez en 1960 y puede ser puro o estar mezclado con un componente maduro. Es la segunda neoplasia maligna más común de células germinales de todos los cánceres de ovario (representa <1%). Alrededor del 50% de los teratomas inmaduros puros del ovario ocurren en mujeres entre las edades de 10 y 20 años. Se debe considerar el tratamiento para preservar su fertilidad futura porque la mayoría de los tumores de células germinales de ovario son curables con la cirugía conservadora y la quimioterapia combinada de seguimiento. La mayoría de los pacientes diagnosticados con un teratoma inmaduro se curan de su enfermedad. Sin embargo en todas las pacientes, se recomienda un seguimiento cercano, particularmente en los primeros 2 años después del diagnóstico, donde existe una mayor probabilidad de recurrencia.

Abstract The immature teratoma was first described in 1960 and cay be pure or mixed with a mature component. It is the second most common malignant germ cell neoplasm of all ovarian cancers (representing <1%). About 50% of pure immature teratomas of the ovary occur in women between the ages of 10 and 20 years. Treatment should be considered to preserve future fertility because the majority of ovarian germ cell tumors are curable with conservative surgery and combined combination chemotherapy. Most patients diagnosed with an immature teratoma are cured of their disease. However, in all patients, we recommend close follow-up, particularly in the first 2 years after diagnosis, where there is a greater chance of recurrence.

Recidiva de teratoma ovárico tras encefalitis anti-NMDA y manejo de la fertilidad: caso clínico / Ovarian teratoma recurrence after anti-NMDA encephalitis and fertility management: a clinical case

RESUMEN CASO CLÍNICO: la encefalitis anti receptor N-metil D-aspartato (NMDAR) es un trastorno autoinmune con un amplio espectro de síntomas neuropsiquiátricos. Se presenta el caso de una mujer de 22 años con una encefalitis anti-NMDA que cursó con amnesia, crisis parciales complejas y alteraciones del comportamiento asociado a un teratoma ovárico. La evolución fue adecuada con cirugía e inmunosupresores. A los tres años se objetivó un teratoma contralateral, sin recidiva de encefalitis; que fue extirpado tras estimulación ovárica para criopreservación de ovocitos. CONCLUSIONES: el teratoma ovárico debe ser sospechado ante la presencia de una encefalitis atípica. Antes de realizar cirugías ováricas repetidas, debe valorarse la opción de vitrificación ovocitaria si los deseos genésicos de la mujer no están cumplidos.

ABSTRACT CLINICAL CASE: anti-N-methyl-D-aspartate-receptor (NMDAR) encephalitis is an autoimmune disorder with a wide sprectrum of neuropsyquiatric symtoms. A case of a 22 years old woman with NMDAR encephalitis presenting with amnesia, focal complex seizures and behavior disturbances associated with an ovarian teratoma is reported. Three years after the first episode, a contralateral teratoma was observed, with no recurrence of the encephalitis; teratoma excision was performed, after controlled ovarian stimulation for oocyte cryopreservation. CONCLUSIONS: ovarian teratoma must be suspected when atypical encephalitis occurs. Before performing repetead ovarian surgeries, oocyte vitrificaction must be considered as a fertility-sparing option in women who have not completed their childbearing wishes.

The use of human mature cystic ovarian teratoma as a model to study the development of human lymphatics / 대한해부학회지

The angiogenic theory to the development of human lymphatics is not clear. The objective of this study was to investigate the development of human lymphatics. Semi-thin and thin paraffin sections from human mature cystic ovarian teratoma tissues were studied using light and electron microscopy. Lymphatics were formed by the differentiation of mesenchymal cells that gradually acquired morphological features of endothelial cells. It is suggested that in human mature cystic ovarian teratoma the lymphatic endothelium develops from mesenchymal cells, and not from cells derived from mature endothelium of a preexisting vein or lymphatic.

Treatment of anti-N-methyl-D-aspartate receptor encephalitis associated with ovarian teratoma / 上海交通大学学报（医学版）

The paper reported three young women who collapsed into sudden abnormal behavior and convulsions and fell into unconsciousness. After admission, they were found to suffer from ovarian teratomas with anti-NMDAR antibody positive in cerebrospinal fluid or serum. The patients were diagnosed as anti-NMDAR encephalitis associated with ovarian teratoma, and teratoma resection was performed.

Intestinal obstruction caused by neoplasms in two cows / Obstrução intestinal causada por neoplasias em duas vacas

ABSTRACT: In cattle, abdominal tumors leading to partial or total intestinal obstruction are rare. Here, it is described the clinico-pathological findings of two cows with intestinal obstruction by neoplasms. A crossbred cow presented apathy, weight loss, constipation, enophthalmos, ruminal and intestinal hypomotility, and abdominal distension in the right ventral quadrant. Pathological examination revealed bilateral ovarian teratoma. The other case was a cow that in the past 2 years had several episodes of squamous cell carcinoma in the third eyelid and vulva. During the 6 days prior to investigation, clinical signs of constipation and tenesmus were noted. Nodular and friable masses were located in the vulva, right third eyelid and cranially to the right kidney that were identified as poorly differentiated metastatic carcinoma of the adrenal cortex. To our knowledge, these are probably the first reports of ovarian teratoma and adrenal carcinoma causing clinical signs of intestinal obstruction in cattle.

RESUMO: Em bovinos, tumores abdominais que levam à obstrução intestinal parcial ou total são raros. Neste relato, descrevemos os achados clínico-patológicos de duas vacas com obstrução intestinal por neoplasias. Uma vaca mestiça apresentava apatia, perda de peso, constipação, enoftalmia, hipomotilidade ruminal e intestinal e distensão abdominal no quadrante ventral direito. O exame patológico revelou teratoma ovariano bilateral. O outro caso foi uma vaca que, nos últimos 2 anos, teve episódios de carcinoma de células escamosas na terceira pálpebra e vulva. Durante 6 dias antes da investigação, foram observados constipação e tenesmo. Massas nodulares e friáveis foram localizados na vulva, terceira pálpebra direita e cranial ao rim direito, que foram identificadas como carcinoma metastático pouco diferenciado do córtex adrenal. Ao nosso conhecimento, estes são os primeiros relatos de teratoma ovariano e carcinoma adrenal causando sinais clínicos de obstrução intestinal em bovinos.

Mature Cystic Teratoma of Ovary with Abnormally High Levels of Ca19-9: A Case Report / Teratoma maduro cístico do ovário com níveis aberrantes de Ca19-9: caso clínico

Abstract Mature cystic teratomas, or dermoid tumors, are the most common benign ovarian neoplasms in young women. Malignant transformation is rare, and occurs in less than 2% of the cases. The heterogeneous histological composition of these tumors may be responsible for the occasional elevation of various tumor markers, such as Ca19-9 and Ca125. We describe one case of mature cystic teratoma in a 50-year old woman with the second highest level of Ca19-9 (8922.76 UI/mL) described in the literature. We concluded that abnormal levels of Ca19-9 are not necessarily associated with ovarian malignancy, and may lead to unnecessary medical intervention and patient anxiety. Therefore, the clinical features, imaging studies and antigen testing should be interpreted carefully, and should not limit the surgical approach.

Resumo Os teratomas maduros císticos do ovário, ou tumores dermoides, são as neoplasias benignas mais frequentes em mulheres jovens. A sua transformação maligna é rara, e ocorre emmenos de 2% dos casos. A composição histológica heterogénea destes tumores pode ser responsável pela ocasional elevação de marcadores tumorais, como o Ca19-9 e o Ca125. Descrevemos umcaso de teratoma maduro cístico do ovário numa paciente de 50 anos com o segundo valor mais elevado de Ca19-9 (8922,76 UI/mL) descrito na literatura. Concluímos que níveis anormalmente elevados de Ca19-9 não estão necessariamente associados a tumores malignos, e podem conduzir a intervenções médicas desnecessárias e contribuir para o aumento da ansiedade da paciente. Portanto, as características clínicas, os estudos imagiológicos e os marcadores tumorais devem ser interpretados cuidadosamente, e não devem limitar o tipo de conduta cirúrgica.

Assessment of Clinical Features and Higher Brain Dysfunction in Anti-NMDA Receptor Encephalitis / The Japanese Journal of Rehabilitation Medicine

　We report five cases of anti-<i>N</i>-methyl-D-aspartate receptor (NMDAR) encephalitis. Five women (27-38 years), Who-presented with psychiatric symptoms, neurological complications, and decreased consciousness, were diagnosed with anti-NMDAR encephalitis after testing positive for serum anti-NMDAR antibodies. The mean（±SD）for hospitalization duration was 272.4（±144.8)days. All patients presented with respiratory failure due to central hypoventilation and required mechanical ventilation for 50.2（±13.1）days on average. Four patients showed no abnormal findings upon brain MRI, one showed high intensity lesions in the right temporal cortex and bilaterally in the hippocampus on T2 weighted images. Higher brain function assessment revealed an overall decrease in intelligence, attention, memory, and executive function in all patients. Temporal assessments revealed progressive improvement in these dysfunctions over several years. Four patients presented with deep venous thrombosis, articular contracture, ectopic ossification, and compression paralysis during the first immobility episode. Two had severely impaired communication and ability to perform activities of daily living when admitted for rehabilitation. However, eventually all the patients attained a premorbid state.<br>　Anti-NMDAR encephalitis possibly results from reversible synaptic dysfunction；therefore, it has a better functional prognosis compared with classical limbic encephalitis and other paraneoplastic neurologic syndromes. Previous studies found abnormalities in the limbic area on MRI in about 25％ of patients, although other findings were non-specific. Prevention of disuse syndrome due to prolonged immobility is important in acute phase rehabilitation. Our study shows that long-term temporal assessments of higher brain function are necessary and useful in the chronic stage.

Rehabilitation of Patient with Anti-N-methyl-D-aspartate Receptor Encephalitis with Memory Disturbance / The Japanese Journal of Rehabilitation Medicine

　Anti-N-methyl-D-aspartate（NMDA）receptor（NMDAR）encephalitis is an autoimmune disorder typically involving ovarian teratomas and a severe neuropsychiatric disorder involving prominent memory and behavioral deficits. The purpose of this study was to evaluate the clinical symptoms and outcomes of six patients（one male, five female；age：20 to 47 years）with autoantibody-associated NMDAR encephalitis, who underwent comprehensive rehabilitation in our hospital. The five female patients underwent ovarian teratoma removal. At the beginning of rehabilitation, the mean score on the Rivermead Behavioural Memory Test was 16.7, and prospective memory was preserved in two patients and was recovered in two patients. The mean Wechsler Memory Scale-Revised score of the six patients significantly increased after rehabilitation, which suggested good recovery, compared to that in the patients with herpes encephalitis. However, in the chronic state, two patients showed persistent short-term memory disturbances that required a compensatory approach and employment assistance. Four patients who were treated with convalescent cognitive rehabilitation 4.5 months after onset improved in 4 months and showed good prognoses. Three returned to work, one returned to school, and two lived independently. Patients with anti-NMDAR encephalitis respond to immunotherapy, but they exhibit persistent cognitive deficits that are related to the distribution and functional role of NMDARs in the human brain. Early intervention for the short-term memory disturbances in convalescent rehabilitation, chronic interventions for the persistent memory disturbances, and preservation of perceptive memory are important to facilitate future employment.

A Case of Paraneoplastic Limbic Encephalitis Presented with Schizophrenic Symptoms / 신경정신의학

Paraneoplastic limbic encephalitis associated with ovarian teratoma has variable clinical manifestations, including hallucination, abnormal behavior, amnesia, mental change, fever, chorea and dystonia, and often has antibodies to N-methyl-D-aspartate receptor (NMDAR), however cases without tumor or antibodies to NMDAR have also been reported. Here, we describe a 35-year-old female who was initially misdiagnosed as schizophrenia because she presented with acute onset of psychiatric symptoms, including visual hallucination, abnormal behavior, confusion, and memory impairment. Three days after admission, she developed high fever refractory to antibiotics. Brain MRI and examination of cerebrospinal fluid were normal. Computerized tomography of the pelvis showed an ovarian teratoma. The patient showed complete improvement after surgical removal of the ovarian teratoma and steroid therapy. We suggest that physicians should consider the possibility of paraneoplastic limbic encephalitis in patients who present with acute onset of psychiatric symptoms with high fever or movement disorder.

Características anatomopatológicas del teratoma ovárico procedente de ooforectomías, en el Instituto de Gastroenterológico Boliviano-Japones / Anatomopathological characteristics of ovarian teratoma from oophorectomy, at the Instituto de Gastroenterológico Boliviano-Japonés

Objetivos: el objetivo de este estudio es conocer las características anatomopatológicas y la frecuencia del teratoma ovárico respecto a otras patologías no neoplásicas y neoplásicas de tipo no-teratoma en piezas quirúrgicas producto de ooforectomía en el departamento de patología del IGBJ. Métodos: para este fin se revisaron los registros de piezas quirúrgicas del Seguro Universal de Maternidad y Niñez (SUMI) del Instituto Gastroenterológico Boliviano Japonés. Resultados: se obtuvo como resultados que los teratomas ováricos corresponden a un 8% de todas las patologías de ovario, en el 95% de los casos es de presencia unilateral y el grupo etáreo con mayor afectación corresponde a mujeres de 21 a 30 años. Durante el proceso de recolección de datos, en algunos reportes histopatológicos no se identificó la variable de localización anatómica (derecha ó izquierda) esto por omisión en la solicitud de envió de muestras lo cual dificulto la obtención datos estadísticos más adecuados y exactos. Conclusiones: con lo que se pudo concluir que la incidencia de teratomas en nuestro medio coincide con la literatura expuesta a nivel internacional.

Objectives: the aim of this study was to determine the anatomical and pathological characteristics and frequency of ovarian teratoma compared to other non-neoplastic and neoplastic pathologies of type non- teratoma in product oophorectomy surgical specimens in the pathology department of IGBJ. Methods: for this purpose we reviewed the records of surgical specimens Universal Mother and Child Insurance of Boliviano Japanese Gastroenterological Institute. Results: it was obtained as ovarian teratomas results correspond to 8% of all ovarian pathologies in 95 % of cases are unilateral and presence age group most affected are women of 21-30 years. During the data collection process , in some histopathological reports did not identify the variable anatomic location (right vs. left) this omission in the application for sending samples making it difficult to obtain adequate statistical data and accurate. Conclusions: with it was concluded that the incidence of teratomas in our literature matches internationally exposed.

Primary malignant melanoma arising in a cystic teratoma

The malignant transformation of a cystic teratoma is a rare event, occurring in about 0.2% to 1.8%. Primary malignant melanoma arising from the ovary is extremely rare. A primary melanoma in an ovarian cystic teratoma originates most frequently at the dermoepidermal junction, similar to a cutaneous melanoma. Though there are no standard adjuvant regimens for the treatment of primary malignant melanoma of the ovary. We present another case report of malignant melanoma developing in cystic teratoma.

Malignización de teratoma maduro de ovario / Malignant transformation of ovarian mature teratoma

Presentar dos casos con malignización de teratoma maduro de ovario. Con clínica inicial inespecífica y aumento progresivo de la circunferencia abdominal. En ambos el ultrasonido y la TAC de abdomen revelaron imágenes de aspecto tumoral por lo que se realizaron otros estudios, no identificando enfermedad a distancia. La biopsia definitiva de la intraoperatoria del anexo derecho e izquierdo respectivamente de cada caso, concluyó adenocarcinoma bordeline mucinoso y adenocarcinoma borderline seroso en un teratoma maduro de ovario. El estadio clínico fue IIIC y recibieron quimioterapia adyuvante. Ambos se mantienen libres de enfermedad

The malignancy of an ovarian mature teratoma is a rare find. We present two cases with nonspecific initial clinical and progressive increase in abdominal girth. In both ultrasound and CT of the abdomen revealed tumor-like images so other studies were conducted to identify distant disease. The final biopsy Annex intraoperative right and left respectively of each case, borderline mucinous adenocarcinoma concluded borderline serous adenocarcinoma in a mature teratoma of the ovary. Clinical stage was IIIC and received adjuvant chemotherapy. Both patients remain free disease

Teratoma maduro ovariano bilateral: relato de caso / Bilateral ovarian teratoma: a case report

Apresenta-se o caso de uma mulher de 45 anos com massa abdominal no hipocôndrio direito. O estudo de imagem com tomografia computadorizada de abdômen forneceu o diagnóstico de teratoma ovariano bilateral. Acrescentam-se imagens obtidas com a ecografia e a ressonância magnética, que também podem ser úteis para o diagnóstico.

A woman with 45 years old woman with abdominal mass in the right hypochondrium is presented. The imaging studies with computed tomography of the abdomen provided the diagnosis of bilateral ovarian teratoma. In addition, images obtained with ultrasound, and magnetic resonance imaging can also be useful for diagnosis.

Sclerosing peritonitis mimicking ovarian carcinoma: Associated with ovarian teratoma / 대한산부인과학회지

Sclerosing peritonitis is an unusual fibrosing condition predominantly involving the omentum and simulating carcinoma. The presenting signs and symptoms, imaging examination and cancer antigen 125 (CA-125) status in sclerosing peritonitis sometimes resemble those of ovarian cancer. Thus, the possibility of sclerosing peritonitis should be considered in the differential diagnosis of ovarian carcinoma. It may occur idiopathically and secondary to chronic peritoneal dialysis, the use of peritoneovenous shunt, practolol therapy, or in association with ovarian tumors such as ovarian teratoma. We report a case of peritonitis initially suspected as ovarian carcinoma but diagnosed as sclerosing peritonitis associated with teratoma.

A Case of Immature Ovarian Teratoma Combined with Paraneoplastic Limbic Encephalitis / 대한산부인과학회지

Paraneoplastic limbic encephalitis (PLE) is a rare neurologic disorder that can be combined with several systemic tumor. PLE is commonly associated with small cell lung carcinoma, but others are uncommon. The cause is unknown, but it is thought to be a autoimmune disorder developing secondary to carcinomatous process. Progressive neurologic symptoms usually predate the diagnosis of cancer, most symptoms usually improve after resection of tumor. PLE can combined with several tumors, but it is very rare with immature ovarian teratoma. We report a case of 31-year-old woman with PLE combined with an immature ovarian teratoma whose neurologic symptoms improved after resection of primary tumor.

A Case of Paraneoplastic Limbic Encephalitis Associated with Immature Ovarian Teratoma

Paraneoplastic limbic encephalitis is commonly associated with small cell lung carcinoma. A 31-year-old female presented with amnesia and bizarre behavior for 1 month. Brain T2-weighted MRI showed high signal intensity in right medial temporal region. Abdominal CT revealed a mass in right lower abdomen, and subsequent excisional biopsy disclosed immature ovarian teratoma. Most symptoms improved after resection of the tumor. Our case demonstrates that immature ovarian teratoma also can be associated with paraneoplastic limbic encephalitis with good prognosis.

CT Manifestations of Teratomas of the Ovary / 实用放射学杂志

Objective To study CT findings of teratomas of the ovary.Methods 25 cases of teratomas of the ovary proved pathologically were presented.All cases were female,with a mean age of 32.8 years.There were 21 cases of benign cystic teratoma,and 4 cases of malignant non-mature teratoma.CT scans were obtained in all cases.Results 12 tumors were presented in right ovary,and 13 in left ovary.The average maximum diameter was 81 mm .20 tumors were round or oval shape,and 5 tumors were irregular shape.The fat component inside the ovarian tumor was showed in 24 cases .Among 21 cases of benign cystic teratomas,the wall plug was demonstrated in 16 cases,solid mass in 3 cases,fat-fluid level in 2 cases.A inhomogenous density was found in 4 cases of malignant non-mature teratomas.Calcification or ossification was presented in 20 cases.The peritoneal metastasis was showed in 2 cases.Conclusion The ovarian teratoma can be considered if a ovarian tumor with fat comporent and calcification or ossification.