Multiple intestinal atresias are rare and its treatment is challenging. Here, we present a case of multiple gastro-intestinal atresia associated with choledochal cyst posing us a surgical challenge.  A full-term, normally vaginal delivered, 10-hour-old male baby, born out of consanguineous marriage, presented with bilious vomiting and mild upper abdominal fullness. Antenatal USG had shown gross polyhydramnios and dilated bowel loops. A 3-year-old sibling is known case of classical galactosemia. Abdominal examination revealed epigastric fullness. Abdominal roentgenograph showed single dilated gastric bubble with no distal gas and few calcifications in lower abdomen . Exploratory laparotomy revealed multiple atresias involving duodenum, jejunum, ileum and colon. There were intra-luminal calcifications between the atretic segments. The stomach and duodenum were grossly dilated. Most of the jejunum and ileum were atretic with no lumen and inter atretic segment filled with calcified putty-like material. The large gut from caecum to transverse colon was filled with calcified material. There were further atresia present in the descending and sigmoid colon. Additionally, there was a large choledochal cyst measuring 5x4 cms . We initially planned for multiple resection and anastomoses, but in view of the long atretic segments with no lumen, calcified intra-luminal contents and intra-operative multiple bronchospasms, only resection of proximal jejunum was done. End duodenostomy and jejunostomy were done. Rest of the jejunum, ileum and large bowel till transverse colon were left as such and descending colostomy was done. Tube-drainage of choledochal cyst was done . A central line for total parenteral nutrition was also placed in internal jugular vein. Post-operatively, the baby was kept on total parenteral nutrition (TPN) for three weeks. The baby had persistent gastric distention and repeat x-ray showed a single bubble (missed pyloric atresia). On day 24, the baby expired due to TPN induced nosocomial sepsis, with blood culture positive for acinetobacter. Multiple intestinal atresia (MIA) is different from duodenal and jejunal atresia because of the extent of involvement from stomach to anus; and present a unique constellation of clinical, radiologic and pathologic findings secondary to malformative process occurring during early intrauterine life.[1] It has been seen in children with common ancestry.[2] Severe combined immunodeficiency is also associated.[3] Transmission pattern in several reported families with MIA had been known to be consistent with autosomal recessive inheritance. TTC7A was the likely causal gene for MIA.[4] Associated anomalies include malrotation, cystic dilatation of bile duct, omphalocele, cardiac anomalies, and congenital cystic adenomatoid malformation.[5] The index case had associated choledochal cyst. We hypothesize that in view of both proximal and distal obstruction of duodenum, the back-pressure led to cystic dilation of common bile duct and thus formation of choledochal cyst.MIA poses a serious surgical challenge. Surgical treatment has included tapering enteroplasty and resection and primary anastomoses.[6] Standard surgical therapy may not allow maximal preservation of residual bowel because of trimming process required for a safe and secure anastomosis. In addition, multiple anastomoses may require excessive operating time, jeopardizing the newborn’s clinical status. Hatch and Schaller described a “shish-kebab” technique in 1986, to perforate multiple membranous obstructions as an alternative to multiple resections.[7] Use of intraluminal Silastic stents to support multiple hand-sewn anastomoses is also described in literature.[8] However, none of them are feasible in these neonates in which whole of intestine is atretic with no intervening segment of normal intestine. Major cause of morbidity and mortality is short gut syndrome. There are no survivals reported in literature for the neonates with MIA. [5]Source of Support: NilConflict of Interest: The author is editor of the journal but the manuscript is handled independently by other editors and he is not involved in decision making of the manuscript.


Actinomycosis is an anaerobic infection that involves the craniofacial region and its colonization has rarely been reported in the developmental odontogenic cysts. In the present report, a case of odontogenic keratocyst  with the colonization of actinomyces is introduced and its significance is discussed.   Actinomyces is an anaerobic, gram-positive and filamentous bacterium which is found in normal oral flora.-2 It is In healthy people, the bacteria may be colonized in tonsillar crypts, dental plaque, calculus, nonvital teeth, gingival sulcus , and periodontal pocket. The orgaThere have been many reports of actinomycosis infection in association with radicular cysts. Besides,A 17 year-old male patient referred with chief complaint of swelling on right side of his face since 4 months ago. The patient had a history of surgical treatment for KCOT in left and right sides of maxilla and mandible 4 years ago. Clinical examination revealed a swelling on the right side of mandible with intact mucosa. No defect was observed in any other organs and the patient did not show any signs of Gorlin syndrome.Radiographs revealed multiple well-defined radiolucent lesions in the posterior mandible and left side of the maxilla. The mandibular lesions were associated with the crown of impacted third molars without any evidence of root resorption .Excisional surgery of right and left mandibular lesions was performed under general anesthesia. The patient received routine postoperative treatment without any antibiotic therapy.Histopathologic examination revealed a cystic lesion lined by parakeratinized stratified squamous epithelium with palisaded basal layer a. The unSix months later, the surgeon reported normal healing. After 3 years, the patient did not show any pain or swelling, but he did not agree to undergo radiographic examination for further evaluation of other signs of recurrence. We presented a case of actinomycosis in association with recurrent KCOT. The infection was seen in a 17 year-old male. The previous studies have also reported a male tendency; although in older patients. In addition, the only similar reported case was found in a 39 year-old female.[Trauma, dental and surgical procedures are some of the recognized factors that enhance the risk of actinomycosis. Actinomycosis may present as an acute inflammation similar to an abscess with pain. In some The filamentous bacteria create a radiating, basophilic center with an eosinophilic periphery; these colonies are surrounded by PMNs and granulation tissue and a fibrotic band encases this area. Moreoveret al. have demonstrated a direct correlation between the number, relative surface area, clinical course of infection and the heavier bacterial load which may result in a longer period of antibiotic treatment.[This study found these colonies to be in association with one out of two non-syndromic multiple KCOTs. KCOT tends to grow within the medullary cavity of bone and does not cause obvious expansion. Swelling may be seen in infected cysts. Althoughreatment. The presreatment.In the present case, antibiotic therapy was not performed for actinomycosis and healing was found without any clinical complication. Although it has been proposed that all cases of true actinomycosis should be treated by surgical removal and antibiotic drugs for a long period, surgery In conclusion, actinomyces colonization may be found in association with recurrent pathologic lesions of the jaws such as developmental cysts following surgical treatments. However, it is possible to treat the lesion with standard surgery and without any additional treatment or considerations.


Leiomyosarcoma is a rare tumor of the smooth muscle, but relatively frequent in the stomach and the small intestine. The mesocolic site is rare. Globally, leimyosarcoma represents less than 0, 1% of the malignant tumors found in the colon and the anus. Because of the similarities with other digestive tumors, namely mesenchymatous or benign tumors of the smooth muscle, the diagnosis of a pleomorphic sarcoma remains difficult even at the histological stage. Surgery is the mainstay of the therapy. We report a case of leiomyosarcoma of the mesocolon and discuss about its main characteristics in the view of the current literature about this pathological condition. Soft tissue sarcomas (STS) are an extremely diverse group of tumors occurring in every region of the human body and affecting all ages with significant morbidity and mortality. Soft tissue sarcomas include more than 35 different neoplasms derived from fat, muscle, nerve, fibrous, vascular, or deep skin tissue, and can be classified by their tissue of origin or their anatomical location . The WorA - 62-year- old female patient, who had past medical history of well treated pulmonary tuberculosis 6 years ago, that hospitalized because of a chronic abdominal pain for 10months. The patient reported weight loss about 6-kg during her illness. The physical examination found palpable per-umbilical mass (10 cm of diameter), hard and mobile relatively to the deeper and superficial neighboring structures. No lymph node or ascites was found. Laboratory exams: complete blood count was in normal range and tumor markers were negative.Computer tomography scanning of the abdomen showed a round tissular mass with irregular edges, which makes 14 cm in its great axis, with central necrotic areas enhancement in the peripheral zones . A primiOn macroscopically exam, the tumor was 15cm of diameter, firm with necrotic areas. Histopathological examination combined with immunohistochemical studies established the diagnosis of pleomorphous sarcoma of the mesocolon. The postoperative course was uneventful. No adjuvant therapy was instituted.th decade of the life [Pleomorphous sarcomas represent approximately 10 to 15% of the sarcoma of the smooth tissues of the adult . The meathe life . There ithe life .The clinical symptomatology is not specific; can be abdominal pain or mass –9, alterPathological exam combined with immunohistochemical studies usually confirms the diagnosis of pleomorphous sarcomas are defined as mesenchymal tumors consisting on pleomorphic spindle- cells with different forms and size and the pleomorphic is high lightened by the nuclear atypical and the anyso caryopses .Durant the years 1980 , a balanMacroscopically, pleomorphous sarcoma presents as a white to grey-pinked hard and firm great mass, sometimes elastic or renittent. More it is more than 10 cm, the tumor is generally lobulated with smooth areas corresponding to intratumoral necrotic zones, and recovered by a complex channel of blood vessels.At the microscopic level, pleomorphous leiomyosarcoma contain pleomorphous cells, which are around and spindle shaped, extending as longs fasciles takings contact the ones to others in rights angles. Atypical cells are frequents because of the presence of giants cells, normal and abnormal mitosis. The cells have an abundant eosinophil and fibrillary cytoplasm. The nucleus muscle and desmine  .The differential diagnosis is essentially with others clinical entities such as undifferenciated pleomorphous sarcoma, gastro-intestinal tumors of the stroma, pleomorphous rhabdomyosarcoma. Immunohistochemistry studies allow performing the diagnosis .The benign or malignant nature of a pleomorphous sarcoma is not always easy to establish. Grading's system relying on histological parameters that have been described in order to better differentiate between the different tumoral grades and so the prognosis . Like foMoreover, it has been shown that myogenic differentiation was a deleterious prognostic factors . The treAfter surgical treatment, the course of pleomorphous sarcoma is characterized by the high frequency of local recurrences, as up as 44% to 85% , 11. ThePleomorphous leiomysarcoma of the mesocolon is a rare tumor. The positive diagnosis of this condition can be made on pathological exam combined with immunohistochemistry studies after surgery. The association of an adjuvant therapy remains controversial; however, adjuvant chemotherapy can be suggested in the case of a bad prognosis, a short time after the surgical procedure.


Introduction. Angiofollicular lymph node hyperplasia (Castleman's disease) is a nonmalignant lymphoproliferative disorder that generally involves the lymph nodes of young adults, most commonly in the mediastinum. Rarely, Castleman's disease may present in the parotid gland. The disease can be further classified into unicentric or multicentric forms, with considerable differences in presentation, treatment, and prognosis. Case(s). We present cases of two pediatric patients, aged 7 and 11, who both presented with a slow-growing, painless parotid mass. In each case, the mass was excised via a superficial parotidectomy and the diagnosis made postoperatively upon further pathologic examination. At 6 months of follow-up, both had fully intact facial nerve function and no evidence of recurrence. Discussion. Castleman's disease presents a diagnostic challenge in the head and neck region, as radiographic characteristics and fine needle aspiration results are often inconclusive. Definitive diagnosis requires surgical excision for pathologic examination. The unicentric form generally presents as a painless mass and can be successfully treated with complete excision. The multicentric form is associated with constitutional symptoms and its treatment remains controversial. Conclusion. Although rare, clinicians should be aware of both forms of Castleman's disease when creating a differential diagnosis for parotid masses. Dr. Benjamin Castleman first described the nonmalignant lymphoproliferative disorder of angiofollicular lymph node hyperplasia in a group of patients with thymoma-like masses of the anterior mediastinum in 1956 .The pathogenesis of Castleman's disease remains unclear. Current hypotheses speculate chronic low grade inflammation, hamartomatous process, immunodeficient state, or pathogenic autoimmunity as potential etiology .CD generally occurs in young adults  and has Clinically, CD may present in either unicentric (UCD) or multicentric (MCD) form. The UCD form generally presents as an asymptomatic painless mass and presents less frequently with compressive symptoms or cosmetic concern.The MCD, or systemic form, has been linked to HHV-8 infection and presents with generalized lymphadenopathy, abnormal laboratory values, malaise, night sweats, hepatosplenomegaly, weight loss, and an aggressive clinical course . The sysCD involving the parotid gland is rare, and, to our knowledge, there are 29 reported cases in the literature of CD presenting as a parotid mass, 24 cases of UCD and 5 cases of MCD. –4, 6–23.A 7-year-old male presented with a painless right facial mass for one year. On examination he had a 3 cm right parotid mass that was firm, nonpulsatile, and nontender with no overlying skin changes. His facial nerve was intact. He had no constitutional symptoms. Fine need aspiration of the mass demonstrated diffusely small lymphocytes admixed with the occasional macrophage, suggestive of a reactive lymph node. A MRI contrast study revealed a well-circumscribed homogenously T1 isointense and T2 hyperintense mass replacing nearly the entire right parotid gland and involving the deep lobe of the gland that measured 3.0 × 3.1 × 3.6 cm  hyaline vascular and (2) plasma cellular . Hyaline hyaline Each type of CD is thought to represent the extremes of a spectrum. The plasma cell type represents an early and active stage, and the hyaline cell type represents a late and quiescent stage . MCD is The standard treatment for unicentric CD is total excision. Surgery for MCD is limited to tissue sampling for diagnosis. For patients with unresectable lesions, radiation therapy has achieved favorable responses , 25. TreIn summary, UCD may rarely present as a diagnostically challenging asymptomatic parotid mass. Clinicians should be aware of CD when forming a differential diagnosis, especially in the pediatric and young adult population. Surgical excision is both diagnostic and curative for UCD. MCD generally presents with systemic manifestations and treatment options for this entity remain controversial.


Nephroblastomatosis (NB) has been considered as a precursor of Wilms tumor (WT). The natural history of NB seems to present significant variation as some lesions may regress spontaneously, while others may grow and expand or relapse and develop into WT later in childhood. Although, most investigators suggest adjutant chemotherapy, the effect and duration of treatment are not well established. Children with diffuse perilobar NB, Beckwith-Wiedemann syndrome, and hemihypertrophy seem to particularly benefit from treatment. We discuss our experience on two cases of NB and we review the literature for the management of this rare condition. Nephroblastomatosis (NB) defines the presence of diffuse or multiple nephrogenic rests (NRs). NRs are clusters of embryonic metanephric cells, which normally disappear after 36 weeks of gestational age. These lesions have been considered as precursors of Wilms tumor (WT). They can be present in about 1% of unselected infant kidneys at postmortem biopsies, while they are found in about 40% of kidneys with unilateral WTs and in nearly 100% of kidneys with bilateral WTs .NB can occur in any age, but it is most frequent in infants. NB in about of 40% of cases is bilateral, while unilateral presentation may be implicated with the presence of microscopic NRs on the contralateral kidney with increased risk of WT development. Limited publications have assessed the clinical course and the effect of management decisions on the outcome of children with NB. Most available data derive from small number of cases. In the current paper we discuss our experience on two cases of perilobar NB (PLNB) presented in our department with an interval of 20 years and we review challenging issues for the management of this rare condition.A 3.5-months-old girl was admitted to our department with right-sided hemihypertrophy. Screening with abdominal ultrasonography showed an enlarged right kidney with a large hypoechoic region presenting no corticomedullary differentiation as well as multifocal hypoechoic parenchymal foci bilaterally in both kidneys, suggesting PLNB. Magnetic Resonance Imaging (MRI) revealed multiple hypodense and nonenhancing cortical masses at both kidneys; the largest with a diameter of 2.65 cm was localized at the enlarged right kidney and presented reduced diffusion and faint enhancing tissue at periphery Figures . As all 3 to 3,26 cm3) and the patient received further cycles of vincristine and actinomycin D every 14 days for the next 3 months. Follow-up ultrasound at 4 months of treatment showed additional decrease of lesions dimensions , intralobar (ILNB), combined, and universal . NodularAs nephroblastomatosis is a preneoplastic condition, administration of chemotherapy could be considered under the concept of decreasing the volume of lesions and reducing the number of cells with malignant potential and subsequently the risk of malignant transformation , 7. TreaObservation and close follow-up may be an option although epidemiologic evidence may not favor such decision. The main arguments in favor of nontreatment are the possible side effects of chemotherapy applied for nonmalignant condition, which has usually a favorable prognosis even when WT is developed. Moreover, chemotherapy may enhance the selection of resistant tumors , 7. TherForty-one individual cases have been published in the literature since 1978, of which 26 were after the classification from Beckwith et al.  7–25]. . 25]. ObOne large series of 52 patients provides data on patients with long-term survival of HPLNB [actinomycin D and maybe partial nephrectomy after initial chemotherapy, especially if there is no response or if there is progression of disease or development of new lesions during therapy.Another interesting issue concerning the clinical course of PLNB is that the speed of the response to chemotherapy, which may suggest the duration of chemotherapy, presented significant variation among reported cases. In many cases prolonged chemotherapy is required to achieve regression of disease . These oIn conclusion, chemotherapy maybe the optimal treatment decision for pediatric patients with PLNB. Current evidence favor the individualization of treatment and close follow-up of the children with PLNB as suggested for individuals with increased risk for WT . Patient


Chordomas are known as rare primary malign tumours that have formed from primitive notochord remains. Sacral chordomas grow slowly but locally and aggressively. Chordomas are locally invasive and have low tendency to metastasis and have a poor prognosis in long-term follow-up. Metastasis may be seen in a rate of 5–40% of the chordomas. Metastasis of chordomas is common in liver, lung, lymph nodes, peritoneum, and brain. The treatment approaches, including surgery, have been discussed in the literature before. Susceptibility to radiotherapy and chemotherapy is controversial in these tumours. The success of surgical treatment affects survival directly. In this report, we will report a sacral chordoma case in which an intraperitoneal distant metastasis occurred and discuss the surgical approach. Chordomas are low-grade, rare, and primarily malign tumors that grow slowly. They rarely have distant metastasis, but instead more locally recurrent tumors grow from primitive notochordal cell remnants. The yearly incidence is estimated at 5/1,000,000; 50–60% of cases are seen in the sacrum, 25–35% at the skull base, and 10–15% in the thoracolumbar region [In this study, we report a case of sacral chordoma in a 53-year-old male with a distant metastasis to the peritoneal region; such a case has never been published before. Preoperatively, the patient was examined for metastasis, and none was found. The mass was removed via the posterior approach. Additional adjuvant radiotherapy was applied to the sacrococcygeal region. At early postoperative evaluation (one and three months), no local recurrence or distant metastasis was found. However, at the ninth month postoperatively, intraperitoneal distant metastases were seen and all metastases were completely extirpated by a general-surgery team. Postoperatively, conformal complex radiotherapy was applied. There was no metastasis or local recurrence within one year following the final surgery.A 53-year-old male was referred to our clinic with coccydynia and coccygeal effluence due to a local mass. Complaints had started three months previously. No neurological symptoms were evidenced in the neurological examination, and there was no urinary or fecal retention. Magnetic resonance imaging (MRI) of the sacrococcygeal spine showed a well-defined lobulated mass in the sacrococcygeal region . The sacSacral chordomas feature slow-growing, extradural tumors. They occur with atypical clinical findings. Pain is the cardinal symptom, whereas neurological deficits that vary with lesion size, shape, and location are also reported . PatholoChordomas are locally invasive, have a low tendency to metastasize, and have a poor prognosis in long-term follow-up. Metastasis is seen in 5–40% of chordoma cases. Metastasis of chordomas is common in the liver, lung, lymph nodes, peritoneum, and brain. Metastasis can be seen in 20% of chordomas in the clinical follow-up, as in the case with the current patient. Even more importantly, a large number of metastatic chordomas studies were published prior to the use of MRI, and all diagnoses were based on autopsy findings . In addiPeritoneal metastasis  is relatively common, particularly in tumors of the abdomen and pelvis; it generally implies a poor prognosis, often with a significant impact on palliation , 9. The The standard treatment for chordoma is surgery, but not even surgery can ensure survival; therefore, these patients are usually not completely cured and so most ultimately succumb. No certain surgical technique has been published, but the main purpose of surgical treatment is to facilitate resection as much as possible. Surgical techniques are defined as simple tumor resections and sacrectomy and posterior instrumentations. The posterior approach is mostly suitable for lesions that occur below the third sacral segment –13. We pThe size of the surgical resection is a major factor in an asymptomatic survey of these tumors. The surgical dissection must extend to the radiologically normal vertebra. However, these surgical dissections can create some important morbidities, like urine and fecal incontinence. It has been published that lumbosacral stabilizations are required following total sacrectomies , 11. In York et al. report an eight-month disease-free interval following subtotal resection, compared to 2.27 years following radical surgery . The disChordomas are locally invasive and have a low tendency to metastasize. As in the present case, metastasis may be seen in the clinical follow-up of chordoma cases. In clinical practice, distant metastasis to the peritoneal region is not as common as our case would suggest. The use of radical surgery can determine the efficiency of treatment in sacral chordomas, and it is clear that radical resection of the mass may prevent metastasis.


Hand-Schüller-Christian disease and Letterer-Siwe disease). Localized LCH accounts for approximately 60-70 % of all LCH cases. Osseous involvement is the most common manifestation and typically involves the flat bones, along with lesions of the skull, pelvis, and ribs. Localized LCH in bone shows a wide spectrum of clinical manifestations and radiologic features that may mimic those of infections as well as benign and malignant tumors. The diagnostic imaging findings of localized LCH are also diverse and challenging. The penumbra sign is a common and characteristic magnetic resonance imaging (MRI) feature of Brodie's abscess, but is rarely seen in localized LCH. In this report, we describe a case of localized LCH misdiagnosed as Brodie's abscess in a 2-year-old child based on clinical symptoms, laboratory findings, and pre-diagnostic MRI findings (penumbra sign). Therefore, the penumbra sign is not sufficient to clearly establish the diagnosis of Brodie's abscess, and the differential diagnosis of localized LCH should be considered when a child with an osteolytic lesion presents with a penumbra sign.Langerhans cell histiocytosis (LCH), formerly known as histiocytosis X, refers to a spectrum of diseases characterized by idiopathic proliferation of histiocytes that produce either focal  or systemic manifestations ( Localized Langerhans cell histiocytosis (LCH) of the bone is a relatively rare, primary disease of childhood characterized by clonal proliferation of histiocytic cells and variable biologic behavior . The skA 2-year-old girl was presented with progressive left-knee pain and swelling since 1 month. She had no history of fever, chills, or recent trauma. The patient's travel and family history were insignificant. Physical examination showed swelling, mild tenderness, and local redness in the anterior part of her left knee. Exophthalmos, skin rash, and mucosal ulcers were not observed. While the patient's white blood cell count and eosinophil count was normal, her erythrocyte sedimentation rate  and C-reactive protein  level were elevated.Conventional radiography revealed a 2.5-cm-diameter medullary lytic lesion located centrally in the proximal left tibial metaphysis Figure 1. The radA tentative diagnosis of Brodie's abscess was made based on the imaging findings and clinical information; therefore, open biopsy with frozen section procedure was performed, followed by intralesional curettage and bone grafting. Intraoperative findings showed an intraosseous cystic lesion containing clear fluid. Gram staining and bacterial culture yielded negative results. Histologic examination of the specimen showed multinucleated giant cells and mononuclear histiocytes aggregated with eosinophils and neutrophils Figure 3A. ImmunohLocalized LCH is an uncommon disease, occurring in less than 1 % of patients with biopsied primary bone tumors  and typThe plain radiographic appearance of localized LCH lesions depends on the phase of the disease and the site of involvement. In the acute stage, bony lesions may have an aggressive pattern of osteolysis and appear permeative with a wide zone of transition and a laminated periosteal reaction. In the chronic stage, lesions have a more benign appearance with well-defined sclerotic margins, a narrow zone of transition, and a mature or absent periosteal reaction . The plTypically, compared to radiography, MRI is superior in depicting the extent of the lesion, evaluating marrow edema, and assessing soft tissue extent; however, in this case, the MRI findings of localized osseous LCH was non-specific. The most common MR appearance of skeletal LCH is a focal lesion, surrounded by an extensive, ill-defined bone marrow and soft tissue reaction with low signal intensity on T1-weighted imaging and high signal intensity on T2-weighted imaging, considered to represent bone marrow and soft tissue edema, respectively . While As with many signs in imaging, the penumbra sign is not pathognomonic for Brodie's abscess and subacute osteomyelitis . Acute None declared.The authors declare that they have no conflict of interest.


Pulmonary metastases of thymomas are relatively rare. We report on two patients who underwent surgery for resection of pulmonary metastases.One patient was a 74-year-old man. A chest CT scan showed a mediastinal mass and a hilar nodule in the left lung. The patient underwent surgical resection of both of these lesions. The histological diagnosis was type A thymoma with intrapulmonary metastasis, classified as stage IVb. He did not receive any adjuvant therapy following the operation because the resection was complete. There has been no evidence of recurrence in four years.The other patient was a 68-year-old man with myasthenia gravis. At the age of 61 years, he underwent extended thymectomy with combined resection of the surrounding involved structures. The histological diagnosis was type B3 thymoma, stage III. Adjuvant radiation (40 Gy) was administered postoperatively; however, a pulmonary nodule occurred seven years following the initial operation . He subsequently underwent right lower lobectomy and a diagnosis of intrapulmonary metastasis of thymoma was made. There has been no evidence of recurrence in two years.Long-term follow-up is important to detect recurrence in any cases of thymoma. Lung metastases should be operated upon if they appear to be completely resectable and this can achieve long-term survival. Thymomas are relatively rare mediastinal tumors arising from epithelial cells and accounting for approximately 0.2 to 1.5% of all malignancies[18 F-fluorodeoxyglucose (FDG) uptake in both lesions. We made a preoperative diagnosis of thymoma with intrapulmonary metastasis or thymoma combined with primary lung cancer. Therefore, thymo-thymectomy and left upper lobectomy were performed via a median sternotomy with transsternal extension. Both resected tumors were shown histologically to be type A thymoma  approximately 10 years previously . The patient refused surgery, and he was conservatively followed-up by annual CT scan. At the age of 74 years, a chest CT scan revealed a mediastinal mass, approximately 3 cm in diameter, and a 2 cm well-defined hilar nodule in the left upper lobe  was performed postoperatively. A pulmonary nodule occurred seven years following the initial operation  (Figure Several factors have been identified to influence the prognosis of thymomas after resection, such as clinical Masaoka stage, WHO histology, completeness of resection, and tumor size.The first case in this report was cytologically benign, although the type A thymoma produced lung metastasis in the ten-year follow-up period. Generally, stage IVb thymoma is not an indication for surgery; however, surgery is recommended for a good prognosis if the tumor appears completely resectable. Kondo aThe second case was cytologically malignant; hence the type B3 thymoma produced a lung metastasis even after the initial surgery combined with adjuvant radiotherapy. There is still no standard guideline for the management of recurrent pulmonary metastases. Recurrence rates after resection of thymomas range from 5 to 50% and are related to the initial clinical stage. CompletLong-term follow-up for thymoma patients is essential because thymomas are potentially malignant tumors.Here, we have reported two cases of metastasis from thymomas and reviewed the current literature. Surgery is the recommended option for a good prognosis for limited stage IVb patients if the thymomas with lung metastases appear completely resectable.We emphasize the need for careful long-term follow-up of the patients who have undergone surgery to detect recurrences as early as possible. Complete resection of small numbers of recurrent pulmonary metastases may also result in a good prognosis.Written informed consent was obtained from the patients for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.18 F-fluorodeoxyglucose; PET-CT: positron emission tomography-computed tomography; WHO: World Health Organization.CT: computed tomography; FDG: The authors declare that they have no competing interest.MK carried out the CT examinations and drafted the manuscripts the two cases. MH, MK, and DK performed the operation on Case 1. MN and DK performed the initial operation on Case 2, and MK, DK and YT performed the second operation. MK has followed-up the operations. All authors read and approved the final manuscript.


Metastatic cancers of the paratesticular tissue are very rare; however, the most frequent primary site of spermatic cord metastasis is the gastrointestinal tract.We recently observed two cases of late-onset metastatic adenocarcinoma of the spermatic cord. Both patients complained of groin discomfort with a palpable mass in the scrotum and inguinal area. Radical orchiectomy and adjuvant chemotherapy were performed in both patients. Although the prognosis of patients with metastatic adenocarcinoma of the spermatic cord is typically poor, the prognosis of our patients was favorable after follow-up for 14 to 18 months.In patients with groin discomfort or swelling and a history of gastric cancer, metastatic adenocarcinoma should be included in the differential diagnosis for early detection of tumors. Gastric cancer is the second most common cause of cancer-related deaths worldwide. In Korea, gastric cancer is the most common cancer, affecting over 25,000 individuals annually . Sites oA 49-year-old man was referred to the Outpatient Department at Soonchunhyang University Cheonan Hospital  with a palpable mass in his right scrotum and discomfort in the spermatic cord persisting for 2 months. The mass was characterized with an insidious onset; it gradually increased in size. Seven years previously, the patient had undergone subtotal gastrectomy with gastrojejunostomy, segmental resection of the transverse colon, and end-to-end anastomosis for advanced gastric cancer. Histologic examination of the specimen revealed mucinous adenocarcinoma with signet ring cell carcinoma . The patient had received adjuvant chemotherapy with six cycles of 5-fluorouracil and cisplatin. The patient had been followed-up for 7 years on an outpatient basis, and he had not shown any sign of recurrence.On physical examination, a 3- × 2-cm mass was palpated above the right testis. The mass was hard, nodular, and non-tender. Scrotal ultrasonography revealed a 3- × 2-cm-sized heterogeneous mass with irregular contours in the right epididymal head, and it extended into the spermatic cord Figure a.The patient underwent transinguinal exploration. Frozen biopsy revealed a mucinous carcinoma of the epididymis. The specimen was removed via radical inguinal orchiectomy. Grossly, the specimen was an ill-defined grayish-white solid mass (4.0 × 1.5 × 1.5 cm) in the epididymis and spermatic cord Figure a. HistopWe performed systemic reevaluation including gastroduodenoscopy, total colonoscopy, abdominal computed tomography (CT), and total-body positron emission tomography (PET), none of which could confirm another primary tumor. The levels of tumor markers such as PSA, carcinoembryonic antigen (CEA), beta-human chorionic gonadotrophin (β-HCG), alpha-fetoprotein (AFP), carbohydrate antigen 19–9 (CA19-9), and cancer antigen 125 (CA125) were normal. The patient received eight courses of adjuvant chemotherapy with the FOLFOX regimen . He has been doing well with no evidence of recurrence after radical orchiectomy at 26 months of follow-up.A 60-year-old man presented to the Urology Department at Soonchunhyang University Seoul Hospital with left inguinal pain for 1 month. The patient had a history of T3M3M0 gastric adenocarcinoma and moderate differentiation 6 years previously. He had been treated with total gastrectomy followed by three cycles of capecitabine and cisplatin chemotherapy. After chemotherapy, the patient had no evidence of recurrence of cancer at the 6-year follow-up. Results of laboratory tests including the levels of the tumor markers, including PSA, CEA, CA19-9, β-HCG, AFP, and CA125, were within the normal range. Physical examination revealed moderate right scrotal tenderness and inguinal tenderness. Chest radiography did not reveal anything abnormal; the results of hematological and biochemical tests were within normal limits. Ultrasonography, pelvic CT, and magnetic resonance imaging (MRI) revealed a soft tissue mass with irregular margins in the spermatic cord (Figure Metastatic tumors of the spermatic cord are rare . The moset al. [Moon et al.  have recIn patients with advanced gastric carcinoma undergoing radical gastrectomy and adjuvant chemotherapy, approximately half the patients survive for > 5 years [The prognosis of a metastatic tumor of the epididymis and spermatic cord is typically unfavorable , as metaMetastatic lesions of the spermatic cord should be included in the differential diagnosis of a scrotal or inguinal mass with discomfort, especially in patients with a history of gastrointestinal cancer, to promptly achieve a targeted treatment.Written informed consent was obtained from the patients for publication of these case reports and any accompanying images.AFP: Alpha-fetoprotein; CA125: Cancer antigen 125; CA19-9: Carbohydrate antigen 19–9; CEA: Carcinoembryonic antigen; CT: Computed tomography; H & E: Hematoxylin and eosin; MRI: Magnetic resonance imaging; PET: Positron emission tomography; PSA: Prostate specific antigen; β-HCG: Beta-human chorionic gonadotrophin.The authors declare that they have no competing interests.JHK and DSK wrote the paper, HDC carried out the pathologic confirmation, MSL have designed the concept and supervised. All authors read and approved the final manuscript.


Background. The benign metastasizing leiomyoma is an exceptionally rare entity; it presents with ectopic leiomyoma nodules with a benign pattern. Symptoms vary according to the anatomic location. The diagnosis is histopathological, usually in patients with history of hysterectomy. Case Presentation. A 36-year-old female with 2-month history of left knee pain was diagnosed with bone fibrosarcoma. A CT scan showed pulmonary nodules. The patient started neoadjuvant chemotherapy. Conservative surgery of pelvic limb was achieved. A new CT scan reported pulmonary nodules that remained in relation to the previous CT. A nodule resection by thoracotomy and TOB (transoperative biopsy) was performed. The final pathology report described benign proliferative lesions consistent with benign metastatic leiomyoma. Conclusions. Benign metastatic leiomyoma is a rare condition presenting with uterine and extrauterine nodules most commonly in the lung. The diagnosis is histopathological. The surgical procedure must be reserved for selected patients. The benign metastasizing leiomyoma is an exceptionally rare entity with few cases reported in the world literature; it is associated with ectopic (extrauterine) leiomyoma nodules with a benign pattern in the lungs . Most paA 36-year-old multiparous Mexican woman, married, living in Monterrey, NL, with past medical history of two plastic surgeries, cesarean, and an abortion. She presents with 2-month history of left knee pain, progressive in intensity from a mild discomfort to a severe disabling pain. It increased with walking. The physical examination revealed increased volume of the knee, tenderness, and edema, the X-ray showed a permeative pattern in distal femur Figures  without Benign metastasizing leiomyoma is a rare condition characterized by the presence of extrauterine leiomyoma nodules with the lung being the most common site, although, it has also been reported in skin, pelvis, abdomen, muscle, omentum, inferior vena cava, right atrium, brain, and bone ; until 1This condition presents with development of leiomyoma lung tumors, well-differentiated and histologically benign behavior due to its low mitotic rate, absence of nuclear pleomorphism, absence of local invasion, and indolent curse. Molecular studies suggest that this is a monoclonal process similar to ordinary leiomyomas . There aThe disease is asymptomatic and often discovered incidentally; if symptoms are present they would be chest pain, dyspnea, and cough. Pathological and immunohistochemical studies are necessary to exclude lymphangioleiomyomatosis; benign metastasizing leiomyoma is characterized by having less, smaller, and more limited interstitial lung and visceral muscle destruction . The hisThere are variants of clinically and histologically benign uterine leiomyomas. In particular, benign metastasizing leiomyoma is a rare condition with ectopic nodules of uterine leiomyoma in the lung. The diagnosis is histopathological after an incidental finding since its presentation is asymptomatic. The gold standard treatment is based on hormonal therapy. The surgical procedure must be reserved for selected patients.


Bezoars are rare causes of gastrointestinal obstruction. Basically, they are of four types: trichobezoars, phytobezoars, pharmacobezoars, and lactobezoars. Some rare types of bezoars are also known. In this article a unique case of plastic bezoars is presented. We describe a girl aged 14 years who ingested large amounts of plastic material used for knitting chairs and charpoys. The conglomerate of plastic threads, entrapped food material and other debris, formed a huge mass occupying the whole stomach and extended into small bowel. Bezoars are rare causes of gastrointestinal obstruction. They mostly originate in the stomach, and occur mainly in patients with psychiatric ailments who chew and swallow their hair (trichobezoar), vegetable fibres (phytobezoar), persimmon fibres (diospyrobezoar), or tablets/semi liquid masses of drugs (pharmacobezoar). Industrial materials including wood trashes, polystyrene, plastic have been reported as the rare causes of bezoar formation. In this report, we describe a 14-year-old girl who developed obstructing Plastic bezoars of both the stomach and small intestine and literature review.A 14-year-old girl presented with upper abdominal pain and vomiting for 15 days and absolute constipation for 7 days. Examination revealed pallor, tachycardia and a 20 cm firm, smooth, non-tender lump palpable in the epigastrium, moving with respiration. An ultrasound of the abdomen confirmed the presence of a large intragastric mass and an abdominal computed tomography (CT) scan revealed a heterogeneous gastric mass.At laparotomy, a bezoar made up of plastic material extended throughout the length of the stomach  and smalth century, bezoars obtained from sacrificed animals had been widely used as antidote [The word bezoar is a word derived from the Arabic “bazahr” or “badzehr” which means an antidote or counter-poison. Till the 19Bezoars are the uncommon result of ingestion of poorly digestible or indigestible substances. Trichobezoars and phytobezoars are the two most common bezoars described in literature. Other unusual bezoars are pharmacobezoars, lactobe- zoars, metal bezoars, plastic bezoars, and sand bezoars. Trichobezoars are found in teenagers, especially in female patients with psychiatric problems. Phytobezoars are found more frequently and are incarcerated foodstuffs not digested in the intestine , 2. PlasPredisposing factors to bezoars, in addition to dietary behaviour, include previous gastric surgery, particularly partial gastrectomy or truncal vagotomy with pyloroplasty. In adults, bezoars are most frequently encountered after gastric operations. In children they are associated with pica, mental retardation, psychiatric disorder and coeliac disease , 4. BezoPatients usually present with recurrent vomiting, abdominal pain, and sometimes lump in the abdomen. The diagnosis often can be made on the basis of findings of conventional radiography and barium studies. On plain abdominal radiography, we found an opaque bezoar, which formed a perfect cast of the stomach. CT-scan could demonstrate a well-defined round mass which could be outlined by stomach or the bowel wall and present characteristic internal gas bubbles of bezoars .Endoscopic investigations could show all of gastric bezoars. Bezoars located in the esophagus or stomach should be treated conservatively in the first instance. Surgery is recommended in cases with massive and non-progressive foreign bodies, or complicated cases presenting with perforation, penetration, hemorrhage, or obstruction , 7.Plastic bezoars are rare causes of gastrointestinal obstruction, resulting from the ingestion of plastic material, especially by mentally retarded patients. Surgery is recommended in cases with massive and non-progressive foreign bodies, or complicated cases.


We report a case of intracerebral cystic schwannoma in the temporal fossa manifested as a gradually worsening headache in a 49-years-old woman. Computed Tomography (CT) and magnetic resonance imaging (MRI) showed a left temporal partly cystic, partly solid mass. The preoperative diagnosis was astrocytoma or glioblastoma multiforme (GBM), but microscopic examination of the mass showed the characteristic pattern with cellular Antony A component. Immunohistochemically, the tumor was positive for S-100 protein. These findings are consistent with a schwannoma. Intracerebral schwannomas not related to cranial nerves are rare and most reported cases involved young patients. Most of in 1966 , 4.A 49-year-old woman was admitted through the emergency department with a history of gradually worsening long standing headache and mild gait disturbance. There was no history of loss of consciousness, or visual deterioration. Neurological examination found mild right sided weakness and left 3rd nerve palsy. The rest of neurological examination was normal. The patient was admitted under neurosurgery. She experienced a single seizure episode during that time.Computed Tomography (CT) and magnetic resonance imaging (MRI) showed intra-axial mass occupying the anteromedial aspect of left temporal lobe with a predominantly cystic component and peripheral solid nodules. Post contrast imaging showed peripheral thick nodular enhancement of the mass . MultiplThe patient underwent a left fronto-temporal craniotomy with partial tumor removal. Only small fraction of the lesion that was adherent to the cavernous sinus was left behind. The pathological section confirmed a diagnosis of schwannoma. Patient did well in the immediate postoperative period with almost back to the preoperative neurological status.Pathologic sections show a non-infiltrating, solid, spindle cell neoplasm and formation of patterning Verocay bodies with perivascular pseudo-rosettes, which is strongly S-100 positive but with nonspecific or weak GFAP reactivity. Ultrastructure demonstrates pericellular basal lamina collagen aggregates. The findings are consistent with those of cellular schwannoma.Intracranial schwannomas account for 8% of central nervous system tumors . Most ofIntraparenchymal schwannomas are located in both supratentorial and infratentorial areas; 63% and 37% respectively. The frontal and temporal lobes are the most common reported sites to be involved. They were reported to originate from the ventricular and periventricular structures. They have also been described in the cerebellar hemispheres,vermis and brain stem , 5.et al reported a significant male to female ratio (M: F 3:1). Furthermore, the majority of cases have occurred in children and young adults, with average ages at presentation less than 21 years, unlike the vestibular schwannomas which usually occur in 5th decade unless associated with neurofibromatosis [Unlike vestibular schwannomas, the intraparenchymal schwannomas exhibit slight male predominance or even no female: male predominance. However Sharma omatosis , 7.et al in a review of 30 cases, calcification was found in 17%, cyst formation was found in 27%, and surrounding edema was found in 53% of the tumors. Khoo HM et al reported that calcification was noted in 29%, cyst formation in 68% and peritumoral edema in 87.5% of the cases. These characteristics appear to be more frequent in intracerebral schwannomas than in acoustic neurinomas and peripheral schwannomas [The characteristic features of intraparenchymal schwannomas include calcification, cysts formation, peritumoral edema, gliosis and a solid enhancing component. A variety of contrast enhancement patterns were described, including those of a cyst with a mural nodule and peripheral enhancement . Accordiwannomas .However these findings are not specific and may be seen in other primary CNS tumors. Preoperative diagnosis is difficult. In fact, the preoperative diagnosis of our case was astrocytoma or GBM, and the final diagnosis of schwannoma was based on histological and immunochemistry examinations. In CT, these tumors show an isoattenuated or low-attenuation contrast-enhancing mass associated with a cystformation. The MRI characteristics are variable but usually they demonstrate a hypointense and hyperintense signal intensity on T1- and T2-weighted images, respectively. The salient radiological feature in the reported cases was a very frequent cystic component , 8, 9.et al as frankly hemorrhagic lesion.What make the presented case unique or unusual compared to the reviewed cases, is that it lacks calcification and peritumoral edema. The age also not typical, it is 49 years old which is much older than the mean age of reported cases in the literature. The mass showed fluid-fluid levels which indicate degradation of blood components, tied with only 1 other case reported by Casadei The histogenesis of intracranial schwannomas not arising from cranial nerves is still unclear. Since the Schwann cells are not normally present in the cerebral parenchyma, It is difficult to explain the origin of intraparenchymal brain schwannomas. Many theories have been proposed to explain the possible mechanism underlying the histogenesis and origin of these rare tumors. There are two common theories. One theory suggests a developmental origin, whereas the other suggests that they are more likely to be non-developmental in origin. According to the developmental theory, aberrant schwann cells in the brain parenchyma may occur due to the transformation of the mesenchymal pial cells, or from displaced neural crest cells that form the foci of schwann cells . Non-devThe radiological differential diagnosis of an intracerebral schwannoma includes many other neoplasms that may occur in children and young adults. These include piloctic astrocytoma, pleomorphic xanthoastrocytoma, ganglioglioma, meningioma and glioblastome multiforme. In order to differentiate between these tumors, exisional biopsy is usually needed. Further immunohistochemistry and/or electron microscopy are also needed to make the final diagnosis since some tumor like pilocytic astrocytoma and fibroblastic meningiomas resemble intracerebral schwannoma microscopically .The clinical manifestations of intracerebral parenchymal schwannoma depend mainly on the locations and the sizes of the tumors. The most common symptoms and signs include headache as in our case, seizures, and focal neurological deficits. The treatment of choice for these tumors is total excision but this depends on the location of the tumors . Completet al and Sharma M C et al reported no evidence of tumor recurrence after complete resection in a follow-up period of 2-24 months. However, Stefanco et al had reported a highly malignant primary intracerebral schwannoma [et al had reported a case of malignant cerebellar schwannoma [The intracerebral schwannoma are benign tumors. Casadei hwannoma . Also Sihwannoma .The intracerebral schwannoma are almost benign tumors with very rare incidence. It may resemble the astrocytoma or GBM. The treatment of choice for these tumors is total excision with complete relief of clinical symptoms and signs mostly achieved.


Teratomas are rarely seen in adults, and presacral region is an area where they rarely settle in. Similarly, only about 1% of teratomas show malignant transformation. Malignant transformation is often associated with the area where teratoma settles in. Malignant transformation of mediastinal teratomas is more frequent than the ones located in retroperitoneal area and gonad. They most commonly show rhabdomyosarcoma, primitive neuroectodermal tumor, enteric adenocarcinoma, and leukemia transformation. In teratomas showing malignant transformation, the clinical course is aggressive; and survival of patients with metastatic disease is very low. The primary treatment of teratomas with malignant transformations is surgical. Effect of radiotherapy and chemotherapy is not clear in patients, to whom surgical operation cannot be applied, or those who are with residual tumor, even if surgical operation can be applied to them, or those who are at metastatic stage. In this paper, we presented a 76-year-old male patient due to the histologic diagnosis of mucinous adenocarcinoma within teratoma, in whom approximately 7 cm presacral mass was found during the radiographic examination made by the reason of low back pain and pelvic pain. Tumors settled in presacral region are highly rare –3. Such Teratomas are the tumors most commonly seen in presacral area in infants and newborns. The rate of teratoma incidence in infants and newborns was 1/35,000–40,000, with the dominance of male gender, while the ratio was determined to be 1/40,000–63,000 in adults, with a higher incidence in women .While there were numerous reports on malignant transforming, including mucinous, adenocarcinoma, or squamous, from teratomas of ovary and testicle, no case of unknown primary mucinous adenocarcinoma within presacral teratoma in an adult male was found in English literature. In this paper, we presented the case of mucinous carcinoma that developed on the basis of presacral teratoma, which we diagnosed in a 76-year-old male patient, for the purpose of emphasizing the fact that both the settlement area and the type of malignant transformation were rare.The 76-year-old male patient presented to the outpatient clinic of general surgery, with the complaints of increasing and intensifying low back pain and pelvic pain, as well as constipation, suffered for the last two months. The history obtainedfrom the patient enabled us to ascertain that he had no different symptom related to other systems, especially weight loss, rectal bleeding, and abdominal pain; and he had no systemic disease history. No lymphadenopathy, organomegaly, abdominal mass symptom, skin lesion, abnormal pulmonary, or cardiovascular finding was detected during his physical examination. The patient's blood biochemistry and complete blood count were normal. There was no peculiarity in the rectal touch, except for presacral sensitivity and swelling detected during the examination. os coccyx level, a part of which shines both in T1 and in T2, a part of which gives high signals in T1, and which had intense involvement of peripheral and septal heterogeneous contrast from postcontrast study  19-9, CA 125, carcinoembriogenic antigen, and prostate specific antigen levels were within normal limits. Any finding related to malignancy was observed in the esophagus, stomach, and the colorectal during the endoscopic examination of the upper and lower gastrointestinal systems. The testicular ultrasonography was normal. No primary tumor or metastatic lesion was detected in terms of mucinous adenocarcinoma, by the performed positron-emission tomography/computerized tomography. Adjuvant systemic chemotherapy was recommended to the patients with negative surgical margins. The therapy was commenced with the biweekly application of the regimen containing irinotecan 185 mg/mOn microscopically examination the mass was located in the subcutaneous area and it had an appearance of a multiloculated cyst with some areas being more solid. The cystic areas were lined by either ciliated columnar epithelium with serous nature or mucinous epithelium. There were some large mucin pools. Some of these mucin pools contained some atypical cell groups which were considered malignant based on the morphological and immunohistochemical staining (diffuse MIB-1 staining and p53 positivity). The tumor was reported as a sacrococcygeal subcutaneous teratoma with malignant mucinous adenocarcinoma . Status In this paper, we discussed our 76-year-old male patient diagnosed with mucinous carcinoma developing on teratoma, as a result of total presacral mass excision.Teratomas are tumors containing different types of cells developing from multiple germinal layers. Most of teratomas are seen in testicles and ovaries of adolescents , 7, 9. HPrimary presacral teratomas are highly rare in adults. Its malignant transformation rate is about 1% . In the Presacral teratomas are often asymptomatic in adults, and they are usually detected during clinical or radiological examination. Although 90% of sacrococcygeal teratomas are externally visible in neonatal period, most of the teratomas in this region are presented as pelvic mass in adults –4, 10. TPresacral cystic teratomas should be distinguished from ependymomas in men and from ovarian cancer in women. However, in both sexes, meningocele, rectal duplication, tail-gut cyst, neurogenic tumors, osseous lesions, renal cysts, the Mullerian ducts cysts, and epidermal cysts are the reasons for benign disorders that they may encounter. Besides these, also lymphangioma, sarcoma, mucinous adenocarcinoma, cystic mesothelioma, metastatic lymphadenopathy, ovaries or uterus metastatic tumors listed as Wilms tumor, and soft tissue malignancies should be distinguished , 13–17. Our patient was diagnosed with adenocarcinoma on the teratoma surface, during the histopathological examination. Malignant tumor finding was not observed in the testicle, colon, stomach, and pancreas during the radiological and endoscopic examinations intended for primer. Tumor markers were not guiding. Our patients was evaluated to be a patient with unknown primary cancer.No case of mucinous adenocarcinoma transformation in teratoma was found in the literature. However, two cases with retroperitoneal adenocarcinoma transformation have been reported , 16. MetAltman et al.  have claHistological features of teratomas showing malignant transformation have been very well defined. They are considered to be somatic components of histological features of a tumor with nonseminomatous germ cell, which cannot be distinguished from a somatic malignancy –17. MaliIn conclusion, it should be remembered that teratomas can be seen in presacral spaces in adults and may develop malignant transformation even though rarely; and histopathological examination should be carried out in detail, based on this thought. No standard systemic treatment intended for teratomas showing malignant transformation is clear yet; and therefore, sharing such rare cases may create an important database.


Isolated metachronous gastrointestinal metastases from advanced-stage lung cancer are rarely diagnosed on the basis of symptoms and resected. In this report, we present a case of resectable metachronous gallbladder and small intestinal metastases of lung cancer. An 86-year-old woman was treated for lung cancer with resection of the right inferior lobe. Five months after the surgery, she was re-admitted because of melena and anemia. Ultrasonography showed a gallbladder tumor with gastrointestinal hemorrhage, and laparoscopic-assisted cholecystectomy was subsequently performed. However, 2 months after this event, the patient presented again with melena and anemia and was diagnosed with a small intestinal tumor. Therefore, laparoscopic-assisted partial resection of the small intestine was performed. Immunohistochemical staining for thyroid transcription factor-1 and cytokeratin 7 confirmed that the two resected tumors were metachronous metastases of the primary lung cancer. The patient died of liver metastases 5 months after the last surgery. Our experience with this case suggests that surgical resection might not be curative but palliative for patients with isolated gallbladder and small intestinal metastases diagnosed on the basis of melena that is resistant to conservative treatment. Although gastrointestinal metastases occasionally develop from advanced-stage primary lung cancer, isolated metachronous gastrointestinal metastases are rarely diagnosed on the basis of the symptoms and resected . We receAn 86-year-old woman with a medical history of hypertension was treated for lung cancer with video-assisted thoracoscopic surgical resection of the right inferior lobe in September 2009. No distant metastasis was detected by preoperative positron emission tomography-computed tomography (PET-CT). Her histopathological diagnosis was adenocarcinoma with mixed solid adenocarcinoma and bronchioloalveolar carcinoma. The tumor’s Union for International Cancer Control (UICC) tumor-node-metastasis (TNM) classification was T2bN0M0 stage IIA. The patient received postoperative adjuvant chemotherapy with tegafur-uracil. However, 5 months after the surgery, in January 2010, she was urgently hospitalized because of melena and anemia. Laboratory data revealed her hemoglobin concentration to be 4.7 g/dL, which was lower than the 10.2 g/dL level measured a month prior to this event. No obvious source of bleeding was identified by both upper and lower gastrointestinal endoscopy. Ultrasonography revealed a gallbladder tumor (Figure In April 2010, 2 months after the gallbladder surgery, the patient experienced melena and anemia again. No abnormality was detected on either upper or lower gastrointestinal endoscopy. However, a contrast-enhanced CT scan revealed slight extravasation in the small intestine, which was also observed on angiography Figure a,b. Conset al. [The most common distant metastasis site of lung cancer is the liver, and metastases in other intra-abdominal organs are very rarely detected before death . Autopsyet al.  indicateet al. . Even whet al. ,5-7. Addet al. . Such suet al. ,8. Reporet al. ,10, and TTF-1 is a very sensitive and specific marker of the pulmonary origin of an adenocarcinoma, if a thyroid origin has been excluded -13. TherIn most cases, symptoms are not observed in the early stages . As a reIf a metastatic tumor from the primary lung cancer is isolated and the primary disease is well controlled, resection should be the first choice. However, surgical intervention might not be indicated in cases of multiple metastases and in case in which prognosis cannot be improved. Therefore, careful examination with imaging studies including chest CT, contrast-enhanced CT of the abdominopelvic region, or PET-CT should be conducted before a decision regarding surgery can be made ,23.In the present case, we could not perform PET-CT, but we examined carefully by using other imaging studies, including enhanced CT, upper and lower gastrointestinal endoscopy, endoscopic retrograde cholangiography, and angiography, to diagnose the respective isolated metachronous metastases.Although the gallbladder and small intestinal metastases could have been part of systemic disease, no other lesions were detected by either preoperative imaging studies or intraoperative examination. Therefore, each tumor was diagnosed as an isolated metastasis and resected by a minimally invasive procedure to resolve the patient’s gastrointestinal hemorrhage and anemia that were uncontrollable with conservative treatment. Although such resection procedures might not be curative, these procedures were worth performing to improve the patient’s quality of life ,8. To ouWe encountered a case of isolated resectable metachronous gallbladder and small intestinal metastases from a primary lung cancer, which were detected by gastrointestinal hemorrhage. Immunohistochemical staining was useful in establishing a connection with the primary cancer.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.