Severe thyrotoxicosis as initial presentation of gastric choriocarcinoma: a case report.

BACKGROUND: Extragonadal choriocarcinoma is rare and can be associated with hyperthyroidism when producing very high levels of human chorionic gonadotropin. CASE PRESENTATION: A 62-year-old Hispanic female presented with a 3-week history of shortness of breath, palpitations, extreme weakness, new-onset hot flashes, and right flank pain. Her physical examination was remarkable for tachycardia, hepatomegaly, hyperreflexia, and tremor; goiter was absent. Laboratory studies revealed increased lactate dehydrogenase, alkaline phosphatase, suppressed thyroid stimulating hormone, very elevated T4, and absent thyroid stimulating immunoglobulin. 18F-fluorodeoxyglucose positron emission tomography-computed tomography exhibited hepatomegaly with multiple large fluorodeoxyglucose-avid liver masses and a focus of fluorodeoxyglucose avidity in the stomach with no structural correlate. A thyroid scan (99mTcO 4 - ) showed diffusely increased tracer uptake. She was started on propranolol and methimazole. Upon stabilization of severe thyrotoxicosis, upper endoscopy was performed, showing a ~ 5 cm bleeding lesion in the greater stomach curvature body; biopsy was consistent with choriocarcinoma; beta-human chorionic gonadotropin hormone was 2,408,171 mIU/mL. The patient received methotrexate followed by etoposide and cisplatin. Methimazole was titrated down, and upon liver failure the medication was stopped. The thyrotoxicosis was effectively controlled with antithyroid drug and concurrent chemotherapy. At ~ 1.5 months after initial diagnosis, the patient died due to bleeding/acute liver failure with coagulation defects followed by multiple organ failure. CONCLUSIONS: Severe thyrotoxicosis can represent an unusual initial presentation of metastatic choriocarcinoma in the setting of extreme elevation of beta-human chorionic gonadotropin. Primary gastric choriocarcinoma is an aggressive malignancy with very poor outcomes. The co-occurrence of severe thyrotoxicosis with advanced primary gastric choriocarcinoma and imminent liver failure complicates management options.

Splenectomy for Solitary Splenic Metastasis in Recurrent Papillary Thyroid Cancer. A Case Report and Literature Review.

Thyroid cancer is the most common endocrine malignancy, presenting with 23 500 new cases per year in the United States. About 7-23% of the patients will present recurrent metastases disease during follow-up. The classic variant of papillary carcinoma is less aggressive compared to its other variants like diffuse sclerosing, tall cell or columnar cell, and insular variants, and the sites to which this metastasizes is already well identified. Metastasis to the spleen is an extremely rare manifestation of papillary thyroid cancer. To date, only 3 cases have been reported in the literature. Herein, we present a 52-year-old male, who developed spleen metastases, 2.4 years after total thyroidectomy and central neck dissection followed by radioactive iodine ablation and seven months after treatment with sorafenib for lung metastases. The splenic lesion was detected in surveillance studies. This case highlights that splenic metastasis, although rare, may occur even in a patient with a locoregional and systemic controlled thyroid cancer and that it can be treated safely with surgical resection.

[Parathyroid disease: The full spectrum, from adenoma to carcinoma. Report of 3 cases]. / Enfermedad paratiroidea: el espectro completo, de adenoma a carcinoma. Reporte de 3 casos.

BACKGROUND: Primary hyperparathyroidism is a disease characterised by the autonomous production of parathyroid hormone. The most common cause is an adenoma, followed by hyperplasia, and rarely carcinoma. CLINICAL CASES: Three cases are presented. The first case is associated with a brown tumour that was diagnosed as hyperplasia after study and surgery. The second case was related to pathological fractures, and a lower right adenoma 236 times bigger than a normal parathyroid was excised. The last case presented with abdominal pain and heartburn. Histopathology reported a carcinoma, which was removed using surgery en bloc. All patients have improved. CONCLUSION: Hyperparathyroidism symptoms are very difficult to identify and diagnose, thus a detailed and broad approach is needed when hyperparathyroidism is suspected.

Immunohistochemical Expression of Nuclear ß-Catenin and STAT-6 in a Solitary Fibrous Tumor of the Soft Palate: A Case Report and Review of the Literature.

The solitary fibrous tumors (SFT) are rare tumors in the head and neck region and there have been only 5 cases reported in the literature in the soft palate. The current paper presents a unique case of a 62-year-old male with TFS arising in the soft palate. The tumor was highly cellular, composed of bland looking haphazardly arranged spindle cells. The signal transducer and activator of transcription (STAT)-6 and nuclear ß-catenin were reactive by immunohistochemistry (IHC). The current case highlights the importance of the STAT-6 and the ß-catenin as IHC markers to make a differential diagnosis with other entities. In summary, the paper presents the first reported case of a SFT of the soft palate in a male patient with nuclear expression of STAT-6 and ß-catenin.

S100A8/A9 proteins mediate neutrophilic inflammation and lung pathology during tuberculosis.

RATIONALE: A hallmark of pulmonary tuberculosis (TB) is the formation of granulomas. However, the immune factors that drive the formation of a protective granuloma during latent TB, and the factors that drive the formation of inflammatory granulomas during active TB, are not well defined. OBJECTIVES: The objective of this study was to identify the underlying immune mechanisms involved in formation of inflammatory granulomas seen during active TB. METHODS: The immune mediators involved in inflammatory granuloma formation during TB were assessed using human samples and experimental models of Mycobacterium tuberculosis infection, using molecular and immunologic techniques. MEASUREMENTS AND MAIN RESULTS: We demonstrate that in human patients with active TB and in nonhuman primate models of M. tuberculosis infection, neutrophils producing S100 proteins are dominant within the inflammatory lung granulomas seen during active TB. Using the mouse model of TB, we demonstrate that the exacerbated lung inflammation seen as a result of neutrophilic accumulation is dependent on S100A8/A9 proteins. S100A8/A9 proteins promote neutrophil accumulation by inducing production of proinflammatory chemokines and cytokines, and influencing leukocyte trafficking. Importantly, serum levels of S100A8/A9 proteins along with neutrophil-associated chemokines, such as keratinocyte chemoattractant, can be used as potential surrogate biomarkers to assess lung inflammation and disease severity in human TB. CONCLUSIONS: Our results thus show a major pathologic role for S100A8/A9 proteins in mediating neutrophil accumulation and inflammation associated with TB. Thus, targeting specific molecules, such as S100A8/A9 proteins, has the potential to decrease lung tissue damage without impacting protective immunity against TB.

CXCR5⁺ T helper cells mediate protective immunity against tuberculosis.

One third of the world's population is infected with Mycobacterium tuberculosis (Mtb). Although most infected people remain asymptomatic, they have a 10% lifetime risk of developing active tuberculosis (TB). Thus, the current challenge is to identify immune parameters that distinguish individuals with latent TB from those with active TB. Using human and experimental models of Mtb infection, we demonstrated that organized ectopic lymphoid structures containing CXCR5+ T cells were present in Mtb-infected lungs. In addition, we found that in experimental Mtb infection models, the presence of CXCR5+ T cells within ectopic lymphoid structures was associated with immune control. Furthermore, in a mouse model of Mtb infection, we showed that activated CD4+CXCR5+ T cells accumulated in Mtb-infected lungs and produced proinflammatory cytokines. Mice deficient in Cxcr5 had increased susceptibility to TB due to defective T cell localization within the lung parenchyma. We demonstrated that CXCR5 expression in T cells mediated correct T cell localization within TB granulomas, promoted efficient macrophage activation, protected against Mtb infection, and facilitated lymphoid follicle formation. These data demonstrate that CD4+CXCR5+ T cells play a protective role in the immune response against TB and highlight their potential use for future TB vaccine design and therapy.

[Bilateral tonsillar hypertrophy as the first manifestation of B cell-small lymphocytic lymphoma with interfolicular pattern]. / Hipertrofia amigdalina bilateral como primera manifestación de linfoma de linfocitos pequeños B con patrón interfolicular.

B-cell small lymphocytic lymphoma typically involves nodal or extranodal tissues as a diffuse proliferation with proliferation centers (pseudofollicules) obliterating normal architecture. But there are unusual patterns of involvement including interfollicular pattern that can be difficult to recognize histologically and probably represent partial or early involvement by neoplasm. Tonsillar lymphoma usually presents either as a unilaterally enlarged palatine tonsil or as an ulcerative and fungating lesion over the tonsillar area. Most lymphomas that involve the tonsil are diffuse large B cell lymphomas and primary low-grade lymphomas are exceptional. We present a primary B-cell small lymphocytic lymphoma affecting palatine tonsils with interfollicular pattern in a 54 year-old man that clinically presented with symmetric / bilateral tonsillar enlargement and sleep apnea.

Immunohistochemical characterization of Renaut bodies in superficial digital nerves: further evidence supporting their perineurial cell origin.

Renaut bodies are well-demarcated cylindrical (circular in cross section) hyaline structures attached to the inner layer of the perineurium that can be found in normal and pathological nerves of various animals and humans. They are composed of spidery fibroblasts and perineurial cells immersed in an extracellular matrix that contains randomly oriented collagen fibers and elastin precursors but not axons or Schwann cells. Frequently, they are associated with thickened subperineurial capillaries. As Renaut bodies are mainly located at sites of nerve compression, it is hypothesized they may act as 'protective cushions' for endoneurial components, and that they may be formed as a secondary reaction to trauma. Herein, we report the presence of Renaut bodies within numerous small dermal nerve trunks in an amputated finger. By immunohistochemistry, Renaut bodies expressed markers identical to those of perineurial cells (epithelial membrane antigen, Glut-1, and claudin-1), supporting the concept of a closely associated perineurial but not endoneurial origin. In addition, expression of hypoxia-inducible factor 1 alpha (which has been shown to increase Glut-1 transcription), neurofibromatosis 1 gene related product and NF-2, were also detected in these peripheral nerve structures.

[Gastric wall in benign cystic teratoma of the ovary]. / Pared gástrica en teratoma quístico benigno de ovario.

We present the clinicopathlogical features of an ovarian mature cystic teratoma with a fully developed gastric wall containing an extensive peptic ulcer. Complete gastric wall, was the only teratomatous tissue present. There are only two previous reported cases of gastric mucosa with a peptic ulcer in an ovarian teratoma.

[Küttner's tumour (chronic sclerosing sialadenitis). Clinical, pathological, and immunohistochemical study in 8 cases of a little-known entity]. / Tumor de Küttner (sialoadenitis crónica esclerosante). Estudio clinicopatológico e inmunohistoquímico de 8 casos de una entidad poco reconocida.

OBJECTIVE: To present the clinical-pathological characteristics of Küttner's tumour (KT) or chronic sclerosing sialadenitis. MATERIAL AND METHOD: We studied 8 cases of KT that were graduated histologically according to the Seifert grading system. An immunohistochemistry panel including CD20, CD3, and keratin AE1-3 was performed. RESULTS: All cases involved the sub-maxillary gland. Microscopically the cases were in stages 2, 3, and 4. There was a chronic inflammatory infiltrate, marked fibrosis, variable degrees of parenchymal atrophy, PAS+ intraluminal material, and microliths. The infiltrate was polymorphic and polyclonal, without lymphoepithelial lesions. CONCLUSIONS: Although it has a typical clinical and pathological presentation and was described more than a century ago, KT remains under-diagnosed and often reported as non-specific inflammatory infiltrate or as "low grade lymphoma". In this latter diagnosis lies the greatest importance for recognizing KT, as confusion with lymphoma would result in a different therapeutic approach.

[Pure mucinous (colloid) carcinoma of the male breast. An uncommon subtype]. / Carcinoma mucinoso (coloide) puro de la glándula mamaria masculina. Presentación de un caso de una variante poco común.

Pure mucinous (colloid, mucous) carcinoma of the male breast is an extremely rare neoplasm. We report the case of a 74-year-old male who displayed a rapidly growing retroareolar tumor of the left breast. The patient underwent modified radical mastectomy. The tumor was well demarcated, had a soft consistency with a glistening gelatinous appearance. Histologically, the neoplasm corresponded to a pure mucinous carcinoma which is one of the most unusual subtYpes, accounting for less than 2% of male breast carcinomas.

Carcinoma mucinoso (coloide) puro de la glándula mamaria masculina: presentación de un caso de una variante poco común / Pure mucinous (colloid) carcinoma of the male breast: an uncommon subtype

El carcinoma mucinoso (colide/mucoso) de la glándula mamaria masculina es una neoplasia poco frecuente. Se describe el caso de un hombre de 74 años de edad que presentó tumor retroareolar de crecimiento rápido. Se realizó mastectomía radical modificada. Microscópicamente el tumor estaba bien delimitado y con consistencia gelatinosa. Histológicamente la neoplasia correspondió a carcinoma mucinoso puro, tumor que representa menos del 1% de los carcinomas que afectan la glándula mamaria en el hombre.

Pure mucinous (colloid, mucous) carcinoma of the male breast is an extremely rare neoplasm. We report the case of a 74-year-old male who displayed a rapidly growing retroareolar tumor of the left breast. The patient underwent modified radical mastectomy. The tumor was well demarcated, had a soft consistency with a glistening gelatinous appearance. Histologically, the neoplasm corresponded to a pure mucinous carcinoma which is one of the most unusual subtYpes, accounting for less than 2% of male breast carcinomas.

Tumor de Küttner (sialoadenitis crónica esclerosante). Estudio clinicopatológico e inmunohistoquímico de 8 casos de una entidad poco reconocida / Küttner's Tumour (Chronic Sclerosing Sialadenitis). Clinical, pathological, and immunohistochemical study in 8 cases of a little-known entity

Objetivo: Presentar las características clinicopatológicas del tumor de Küttner (TK) o sialoadenitis crónica esclerosante. Material y método: Se estudiaron 8 casos de TK. Se graduaron los casos histológicamente de acuerdo con la clasificación de Seifert. Se realizó una batería de anticuerpos contra CD20, CD3 y queratina AE1-3. Resultados: Todos los casos afectaron a la glándula submaxilar. Microscópicamente se encontraban en estadios 2, 3 y 4. Había inflamación crónica folicular y fibrosis acentuada, con atrofia parenquimatosa variable, material PAS+ intraluminal y microcalcificaciones. El infiltrado inflamatorio fue polimorfo, policlonal y sin lesiones linfoepiteliales. Conclusiones: A pesar de su descripción hace más de un siglo y su presentación clínica e histopatológica característica, el TK es poco diagnosticado y a menudo se informa como proceso inflamatorio inespecífico o como linfoma de bajo grado. En este último diagnóstico radica la mayor importancia del reconocimiento del TK, ya que resultaría en un manejo terapéutico diferente

Objective: To present the clinical-pathological characteristics of Küttner's tumour (KT) or chronic sclerosing sialadenitis. Material and method: We studied 8 cases of KT that were graduated histologically according to the Seifert grading system. An immunohistochemistry panel including CD20, CD3, and keratin AE1-3 was performed. Results: All cases involved the sub-maxillary gland. Microscopically the cases were in stages 2, 3, and 4. There was a chronic inflammatory infiltrate, marked fibrosis, variable degrees of parenchymal atrophy, PAS+ intraluminal material, and microliths. The infiltrate was polymorphic and polyclonal, without lymphoepithelial lesions. Conclusions: Although it has a typical clinical and pathological presentation and was described more than a century ago, KT remains under-diagnosed and often reported as non-specific inflammatory infiltrate or as "low grade lymphoma". In this latter diagnosis lies the greatest importance for recognizing KT, as confusion with lymphoma would result in a different therapeutic approach

[Expression of p53 and BCI-2 as prognostic markers and for anatomical location in gastrointestinal stromal tumors (GIST). Clinico-pathological and immunohistochemistry study of 19 cases]. / Expresión de p53 y Bcl-2 como marcadores pronósticos y de localización anatómica en tumores del estroma gastrointestinal. Estudio clínico-patológico e inmunohistoquímico de 19 casos.

OBJECTIVE: To correlate the expression of p53 and BCl-2 with the clinical outcome and anatomic location of the gastrointestinal stromal tumours (GIST). BACKGROUND DATA: The GIST are the most common nonepithelial neoplasm of the gastrointestinal tract. In spite of the existence of a wide range of predictive factors, their clinical outcome is unpredictable. There are several studies that correlate the expression of p53 and Bcl-2 with the clinical outcome and anatomic location of the GIST. METHODS: We obtained 19 cases from the archives of the Department of Pathology of the ABC Medical Center, in Mexico City. GIST were classified into risk groups according to the Fletcher et al. classification. We performed an immunohistochemestry panel including CD117, CD34, actin, desmin, P-S100, p53 and BCl-2 and correlated their expression to the risk group and anatomical site of the tumors. RESULTS: There was less expression of p53 in the gastric tumors (27%) than in small bowel tumors (100%). There was greater expression of p53 in the high-risk tumors than in the very low-risk ones, regardless of the anatomical site. Bcl-2 expression was more expressed in the small intestine tumors (100%) than in those located in the duodenum (50%) The high risk tumors showed slightly more expression of Bcl-2 than the low risk ones (89% vs. 100%), despite the anatomical location. CONCLUSIONS: Both, p53 and Bcl-2 are important markers to establish the anatomical site of GIST and are also helpful to predict the clinical behavior of these tumors.

Enfermedad de Kikuchi-Fujimoto (linfadenitis histiocítica necrosante): Estudio clinicopatológico e inmunohistoquímico de 14 casos y su diagnóstico diferencial con otras linfadenitis necrosantes reactivas y neoplásicas / Kikuchi-Fujimoto disease (histiocytic necrotizing lymphadenopathy) clinicopathologic and immunohistochemical study of 14 cases and its differential diagnosis with other reactive and neoplastic necrotizing lymphadenopathies

Kikuchi-Fujimoto disease also known as histiocytic necrotizing lymphadenopaty (HNL) is a rare entity, originally described in Japanese population, although currently it has been described all over the world. It is more frequent in young women and it is usually located in cervical lymph nodes. We report 14 cases of HNL in Mexican population, their clinicopathological and immunohistochemical study as well as a comparative study with other necrotizing lymphadenopaties due to B or T-cell lymphomas, tuberculosis, Epstein Barr virus infection, and non-specific necrosis. In our study we found that there was more expression of the immunomarkers CD68, MPO, CD123 and antikerat in OSCAR in the cases of HNL in contrast with the lesser or even null expression of the same markers in the necrotized lymph nodes of the comparative study group.

La enfermedad de Kikuchi-Fujimoto o linfadenitis histiocítica necrosante (LHN) es una entidad poco frecuente, inicialmente descrita en población japonesa. Sin embargo, actualmente se ha descrito alrededor de todo el mundo. Es más frecuente en mujeres jóvenes y su localización habitual es en ganglios linfáticos cervicales. Presentamos estudio clinicopatológico de 14 casos de LHN en pacientes mexicanos, y los comparamos con linfadenitis necrosantes secundarias a linfomas B y T, tuberculosis, virus de Epstein Barr y necrosis inespecífica. Encontramos mayor expresión de los anticuerpos CD68, MPO, CD 123 y antiqueratina OSCAR en los casos de LHN comparados con la expresión baja o nula de los mismos anticuerpos en las necrosis ganglionares del grupo comparativo.

[Kikuchi-Fujimoto disease (histiocytic necrotizing lymphadenopathy) clinicopathologic and immunohistochemical study of 14 cases and its differential diagnosis with other reactive and neoplastic necrotizing lymphadenopathies]. / Enfermedad de Kikuchi-Fujimoto (linfadenitis histiocítica necrosante). Estudio clinicopatológico e inmunohistoquímico de 14 casos y su diagnóstico diferencial con otras linfadenitis necrosantes reactivas y neoplásicas.

Kikuchi-Fujimoto disease also known as histiocytic necrotizing lymphadenopaty (HNL) is a rare entity, originally described in Japanese population, although currently it has been described all over the world. It is more frequent in young women and it is usually located in cervical lymph nodes. We report 14 cases of HNL in Mexican population, their clinicopathological and immunohistochemical study as well as a comparative study with other necrotizing lymphadenopaties due to B or T-cell lymphomas, tuberculosis, Epstein Barr virus infection, and non-specific necrosis. In our study we found that there was more expression of the immunomarkers CD68, MPO, CD123 and antikerat in OSCAR in the cases of HNL in contrast with the lesser or even null expression of the same markers in the necrotized lymph nodes of the comparative study group.

[Neuroendocrine gastric carcinoma. A case report and review of current management]. / Carcinoma neuroendocrino gástrico. Presentación de un caso clínico y revisión actual del manejo.

Neuroendocrine or carcinoid tumors of the gastrointestinal tract considered previously extremely rare, are diagnosed at present with increased frequency due to the better capacity to identify neuroendocrine system cells in normal and pathologic conditions. Occasionally, these tumors secrete a great variety of vasoactive substances, producing the carcinoid syndrome. Gastric carcinoids are classified, according to their degree of differentiation into well differentiated and poorly differentiated tumors, also called neuroendocrine carcinomas. Neuroendocrine gastric carcinomas or poorly differentiated gastric carcinoids are seen in 5-15% of all gastric carcinoids, mainly in older male patients. Generally they are large, very aggressive tumors with extensive local infiltration. Due to poor differentiation, they are not frequently associated with an endocrine syndrome. They can be located in any part of the stomach but are mainly seen in antrum. These tumors have an aggressive behavior and must be treated in a radical manner; recurrences are not uncommon. We report the case of a patient with a neuroendocrine gastric carcinoma treated with an en bloc subtotal gastrectomy and colectomy.

Case report: Ocular gnathostomiasis in northwestern Mexico.

A 42-year-old woman from Culiacan in the Sinaloa State of Mexico presented with a four-year history of migratory, pruritic, painful swellings of the face. Palpebral edema with conjunctival erythema developed when lesions developed near the eye. Routine eye examination showed a mobile worm in the anterior chamber. Following surgical removal, the parasite was identified by light and electron microscopy as an advanced third-stage larva of Gnathostoma sp. This prevalence of this helminthiasis may be increasing in areas where eating freshwater raw fish ("cebiche") is customary.

Tumores sólidos seudopapilares del páncreas / Solid pseudopapillary tumors of the pancreas

El informe de tumores sólidos seudopapilares del páncreas, lesiones de potencial maligno incierto o francamente malignos, ha venido incrementándose a partir de su más amplio reconocimiento y caracterización como entidad anatomoclínica distintiva. Se les estima una frecuencia del 2 por ciento entre todas las neoplasias exocrinas del páncreas. Crecen lentamente sin correlación exacta entre aspecto histológico y comportamiento biológico. Presentamos los casos de dos pacientes femeninas de 24 y 15 años (casos 1 y 2), con excelente evolución luego del diagnóstico posquirúrgico, en el Centro Médico ABC de la Ciudad de México, de tumores sólidos seudopapilares del páncreas. Acudieron al hospital, por dolor abdominal, en el segundo caso precedido de trauma leve. El primer caso fue predominantemente quístico y el segundo predominantemente sólido, con 14 y 12 cm de diámetro respectivo. La histología, semejante en ambos, consistió en zonas sólidas y áreas de aspecto papilar con cambios mixoides. La inmunotinción destacó positividad para vimentina, expresión variable para marcadores neuroendocrinos, citoqueratina, receptores de progesterona y un hallazgo interesante: complejos ductuloinsulares en páncreas residual. Conclusiones: Ocurrencia en mujeres jóvenes; aspecto sólido-quístico; fácil exéresis; gran tamaño e histología sólida-papilar sugieren diagnóstico de tumor sólido seudopapilar pancreático, pudiendo evitar resecciones quirúrgicas excesivas.