Hepatic Inflammatory Myofibroblastic Tumor Detected in the Fetal Period That Caused an Oncologic Emergency

Hirotaka Kato; Yasuyuki Mitani; Taro Goda; Masaki Ueno; Shinya Hayami; Hiroshi Tsujimoto; Shinji Kounami; Takayuki Ichikawa; Hiroki Yamaue

doi:10.1159/000511052

Hepatic Inflammatory Myofibroblastic Tumor Detected in the Fetal Period That Caused an Oncologic Emergency

Case Reports in Oncology ◽

10.1159/000511052 ◽

2020 ◽

Vol 13 (3) ◽

pp. 1513-1519

Author(s):

Hirotaka Kato ◽

Yasuyuki Mitani ◽

Taro Goda ◽

Masaki Ueno ◽

Shinya Hayami ◽

...

Keyword(s):

Inflammatory Myofibroblastic Tumor ◽

Emergency Operation ◽

Cystic Mass ◽

Abdominal Distention ◽

Tumor Biopsy ◽

Emergency Cesarean ◽

Subcutaneous Edema ◽

Male Neonate ◽

Circulation Dynamics ◽

Inflammatory Effect

A huge abdominal cystic lesion with ascites was detected in a male neonate at 31 weeks of gestation. Increasing ascites and the appearance of subcutaneous edema were detected, which caused fetal hydrops. The patient was delivered by emergency cesarean section at 33 weeks of gestation. The birth weight was 2,407 g, and the Apgar score was 8/9 points (1-/5-min values). Breathing at birth was stable, but the patient presented with remarkable abdominal distention due to the ascites. Later, the patient presented with tachypnea, and breathing gradually worsened, so an emergency operation was performed. There were no intraoperative findings within the small intestine, but there was a large amount of ascites and a cystic mass arising from the liver. The patient’s breathing and circulation dynamics could only be stabilized by ascites removal, so only a tumor biopsy was performed. The pathological findings led to the diagnosis of an inflammatory myofibroblastic tumor, and steroids were administered early after surgery for the purpose of an anti-inflammatory effect and tumor shrinkage. The abdominal distention was alleviated, and blood examinations showed a reduced inflammatory response. There was no apparent shrinkage of the tumor, however; thus, radical surgical treatment was performed on day 24. The postoperative course was uneventful, so the patient was discharged on day 36. Seven years after the operation there has been no recurrence or distant metastasis.

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Case Report: Cytoreductive Surgery and Hyperthermic Intraperitoneal Chemotherapy Application in Intraperitoneally Disseminated Inflammatory Myofibroblastic Tumor and in the Youngest Patient in the World: New Indication and Modification of Technique

Frontiers in Surgery ◽

10.3389/fsurg.2021.746700 ◽

2021 ◽

Vol 8 ◽

Author(s):

Hanna Garnier ◽

Maciej Murawski ◽

Tomasz Jastrzebski ◽

Katarzyna Pawinska-Wasikowska ◽

Walentyna Balwierz ◽

...

Keyword(s):

Cytoreductive Surgery ◽

Inflammatory Myofibroblastic Tumor ◽

Systemic Exposure ◽

Chemotherapeutic Agents ◽

Cystic Mass ◽

The World ◽

Key Factor ◽

Rare Lesion ◽

Crs And Hipec ◽

The Right

Introduction: Peritoneal metastases occur in cancers that spread to the peritoneal cavity and indicate the advanced stage of the disease. In children they are mainly seen in sarcomas, Gastrointestinal Stromal Tumors and primary disseminated ovarian tumors. Inflammatory Myofibroblastic Tumor (IMT) is a very rare lesion, characterized by an unpredictable clinical course. The absorption of chemotherapeutic agents through the peritoneal-plasma barrier (PPB) is minimized, thus HIPEC procedure limits the systemic exposure to chemotherapy and permits the administration of its higher doses. The main purpose of HIPEC is to remove the visible macroscopic disease in order to achieve complete cytoreduction (CRS).HIPEC Procedure in Children: Several papers deal with the CRS and HIPEC in children and adolescents, however pediatric experience is still limited. Thus far, the HIPEC procedure has been carried out on patients over 2 years old. The most common indication for the surgery and the best outcome was experienced by patients with desmoplastic small round cell tumor (DSRCT). Most patients received intraperitoneal cisplatin.HIPEC Modification: A 5-month-old infant was admitted to the Department of Pediatric Oncology due to the abdominal distention and blood in the stool. The Computed Tomography (CT) revealed a solid-cystic mass in the right abdominal area. The primary tumor and numerous peritoneal metastasis were removed and the Inflammatory Myofibroblastic Tumor (IMT) was diagnosed. The patient underwent subsequently CRS and modified HIPEC procedure. To avoid overheating of the infant, the intraperitoneal normothermic chemoperfusion was performed. Due to the low body weight a modified dosage of intraperitoneal doxorubicin was used. The child underwent standard postoperative chemotherapy and received crizotinib therapy. At 12 months follow-up since treatment completion the patient remains in complete remission. To our knowledge this is the youngest patient, the only infant and the first pediatric patient with IMT who underwent the modified HIPEC procedure in the world.Conclusions: CRS and HIPEC is technically possible also in infants. For its safe course patients selection and technique modification are necessary. Use of HIPEC should be also considered in intraperitoneally disseminated IMT. A complete cytoreductive surgery as the first HIPEC step seems to be the key factor in survival.

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Retroperitoneal Cystic Lymphangioma: About A Case and Review of The Literature

European Journal of Medical and Health Sciences ◽

10.24018/ejmed.2019.1.5.122 ◽

2019 ◽

Vol 1 (5) ◽

Author(s):

El Mahdi Graiouid ◽

Walid Bai ◽

Messian Gallouo ◽

Issam Jandou ◽

Yassine Larrache ◽

...

Keyword(s):

Clinical Examination ◽

Lymphatic Vessels ◽

Cystic Mass ◽

Cystic Lymphangioma ◽

Abdominal Distention ◽

Pathological Examination ◽

Review Of The Literature ◽

Surgical Exploration ◽

Venous Circulation ◽

Distended Abdomen

Cystic lymphangioma is a rare benign malformative tumor of lymphatic vessels in various locations. We report the observation of a 62-year-old man who had been consulting for major abdominal distention for 5 years. The clinical examination found a very distended abdomen with collateral venous circulation. The imaging was in favor of a cystic retro-peritoneal image. Surgical exploration showed a retroperitoneal cystic mass. Anatomo-pathological examination concluded that retroperitoneal cystic lymphangioma.

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A rare presentation of a pediatric neurenteric cyst as an intra-axial pontine lesion: A case report with a 5-year follow-up

Surgical Neurology International ◽

10.25259/sni_214_2019 ◽

2019 ◽

Vol 10 ◽

pp. 236

Author(s):

Yuzaburo Shimizu ◽

Naohide Fujita ◽

Osamu Akiyama ◽

Mario Suzuki ◽

Akihide Kondo

Keyword(s):

Histopathological Examination ◽

Final Diagnosis ◽

Cystic Mass ◽

Magnetic Resonance Images ◽

Neurenteric Cyst ◽

Pediatric Case ◽

Rare Presentation ◽

Tumor Biopsy ◽

Short Period ◽

Pontine Lesion

Background: Intracranial neurenteric cysts are rare, benign, and slow-growing tumors. However, we encountered a pediatric case that the cyst expansion occurred in a short period of time resulting in rapid deterioration of the patient’s symptoms. Case Description: A previously healthy 7-year-old girl had a week history of dysarthric speech and diplopia along with headaches. Her magnetic resonance images (MRI) showed an abnormal cystic mass in her brainstem. Her symptoms were deteriorated for 1 month and her second MRI revealed an enlargement of the cystic lesion. The tumor biopsy and cyst drainage were carried out and histopathological examination of the cyst wall showed columnar epithelium containing ciliated cells. The final diagnosis of her tumor was neurenteric cyst. Conclusion: We report a pediatric case of a neurenteric cyst in the brainstem, which expanded in a short period, and review this rare entity.

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A Huge Cystic Retroperitoneal Lymphangioma Presenting with Back Pain

Case Reports in Medicine ◽

10.1155/2016/1618393 ◽

2016 ◽

Vol 2016 ◽

pp. 1-4 ◽

Cited By ~ 4

Author(s):

Mahir Gachabayov ◽

Kubach Kubachev ◽

Elbrus Abdullaev ◽

Valentin Babyshin ◽

Dmitriy Neronov ◽

...

Keyword(s):

Back Pain ◽

Rare Disease ◽

Female Patient ◽

Clinical Presentation ◽

Cystic Mass ◽

Abdominal Distention ◽

Abdominal Tumors ◽

Rare Location ◽

History Of ◽

Retroperitoneal Lymphangioma

Retroperitoneal lymphangioma is a rare location and type of benign abdominal tumors. The clinical presentation of this rare disease is nonspecific, ranging from abdominal distention to sepsis. Here we present a 73-year-old female patient with 3-month history of back pain. USG and CT revealed a huge cystic mass which was surgically excised and appeared to be lymphangioma on histopathology.

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A Case Of Pancreatic Hamartoma Mimicking Malignancy: An Uncommon Pitfall

American Journal of Clinical Pathology ◽

10.1093/ajcp/aqaa161.160 ◽

2020 ◽

Vol 154 (Supplement_1) ◽

pp. S73-S74

Author(s):

S Dasaraju ◽

S Liu ◽

R Kelly ◽

W Mneimneh

Keyword(s):

Final Diagnosis ◽

Pancreatic Tissue ◽

Cystic Mass ◽

Uncinate Process ◽

Clinical Suspicion ◽

Neuroendocrine Cells ◽

Proliferation Index ◽

Ki 67 ◽

Tumor Biopsy ◽

Cd34 Immunostain

Abstract Casestudy Hamartomas are benign, disordered, tumor-like growth of cellular constituents resembling the tissue of its origin. Hamartomas are usually seen in lung, heart, kidney and spleen. Pancreatic hamartomas (PH) are extremely rare, accounting for <1% of all hamartomas. PH occurs at any age (median: 50 years) without gender predilection. PH presents as single or multiple, solid and/or cystic mass composed of exocrine tissues. Admixed neuroendocrine cells may be seen, but well-formed islets are unusual. PH stroma is typically positive for CD34 by immunohistochemistry. We present a case of a PH resected for the clinical suspicion of malignancy, with the final diagnosis established postoperatively. The case is that of a 74-year-old male with an incidental 2.3 x 1.7 x 1.1 cm hyperenhancing solid mass of the pancreatic uncinate process, found during anemia workup. The radiologic appearance was suspicious for a neuroendocrine tumor. Biopsy of the mass showed benign-appearing pancreatic ductal and acinar tissue. Given the clinical suspicion of malignancy, the patient elected to undergo a pancreaticoduodenectomy. Macroscopically the mass was well-circumscribed with solid, tan-white, firm cut surface. Microscopic examination revealed well- circumscribed proliferation of disorderly-arranged, well-differentiated, bland exocrine pancreatic tissue. Chromogranin, synaptophysin and CD56 immunostains did not highlight significant neuroendocrine component. Ki-67 proliferation index was low (1%). CD34 and CD117 immunostains were negative in the stroma. The findings were consistent with PH. Conclusion PH may mimic a malignant process of the pancreas. The preoperative diagnosis of PH is extremely challenging due to the lack of characteristic clinical and radiological features, therefore, the diagnosis of PH is often made on resection specimen. CD34 immunostain is not always helpful for the diagnosis as it may be negative in PH stroma. Although extremely rare, pancreatic hamartoma should be considered in the differential diagnosis of a pancreatic tumor.

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Chronic pyrrolizidine alkaloid toxicosis in a heifer

Brazilian Journal of Veterinary Pathology ◽

10.24070/bjvp.1983-0246.v14i3p206-209 ◽

2021 ◽

Vol 14 (3) ◽

pp. 206-209

Author(s):

Vanessa Barraza ◽

◽

Mariana Flores ◽

Keyword(s):

Body Condition ◽

Chronic Diarrhea ◽

Pyrrolizidine Alkaloid ◽

Clinical Signs ◽

Abdominal Distention ◽

Clinical Exam ◽

Neurological Signs ◽

Subcutaneous Edema ◽

Native Pasture

This heifer came from a group of cattle with chronic diarrhea and emaciation. Some of the animals also had neurological signs, predominantly aggressiveness. Two animals had already died spontaneously after worsening of the clinical signs. The farmer had kept these animals on native pasture during the winter, and he reported that the vegetation had been scarce in that period. On clinical exam, the animals were in bad body condition, with some presenting dyspnea, subcutaneous edema mainly affecting the dewlap, and abdominal distention.

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Patent Urachus with Bladder Prolapse

Journal of Nepal Paediatric Society ◽

10.3126/jnps.v34i1.7877 ◽

2014 ◽

Vol 34 (1) ◽

pp. 68-70 ◽

Cited By ~ 1

Author(s):

B Thapa ◽

MS Pun

Keyword(s):

Urinary Bladder ◽

Abdominal Wall ◽

Umbilical Cord ◽

Cystic Mass ◽

Solid Mass ◽

Antenatal Ultrasound ◽

Patent Urachus ◽

Male Neonate ◽

Mucosal Mass ◽

Bladder Prolapse

We report a case of bladder prolapse through a patent urachus in a term male neonate with a large, red, tubular, mucosa lined mass inferior to the umbilical cord. A cystic mass communicating with fetal urinary bladder was detected in an antenatal ultrasound in a 26 years primigravida at 18 and 26 weeks gestation. The cyst disappeared at 35 weeks and a new solid mass was noted at the fetal abdominal wall. After birth a protruded mucosal mass inferior to the umbilical cord was noted. Urethral catherisation confirmed communication with bladder. On the second day of life excision of urachus, repair, reduction of bladder and reconstruction of abdominal wall was performed. The patient voided well and was discharged on ninth day without any complication. DOI: http://dx.doi.org/10.3126/jnps.v34i1.7877 J Nepal Paediatr Soc 2014;34(1):68-70

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Reduction of paw edema and liver oxidative stress in carrageenan-induced acute inflammation by Lobaria pulmonaria and Parmelia caperata, lichen species, in mice

International Journal for Vitamin and Nutrition Research ◽

10.1024/0300-9831/a000620 ◽

2019 ◽

pp. 1-9

Author(s):

Samira Salem ◽

Essaid Leghouchi ◽

Rachid Soulimani ◽

Jaouad Bouayed

Keyword(s):

Time Course ◽

Acute Inflammation ◽

Lichen Species ◽

Paw Edema ◽

Sod Activity ◽

Edema Formation ◽

Lobaria Pulmonaria ◽

Anti Inflammatory ◽

Endogenous Antioxidant ◽

Inflammatory Effect

Abstract. Paw edema volume reduction is a useful marker in determining the anti-inflammatory effect of drugs and plant extracts in carrageenan-induced acute inflammation. In this study, the anti-inflammatory effect of Lobaria pulmonaria (LP) and Parmelia caperata (PC), two lichen species, was examined in carrageenan-induced mouse paw edema test. Compared to the controls in carrageenan-induced inflammation (n = 5/group), our results showed that pretreatment by single oral doses with PC extract (50–500 mg/kg) gives better results than LP extract (50–500 mg/kg) in terms of anti-edematous activity, as after 4 h of carrageenan subplantar injection, paw edema formation was inhibited at 82–99% by PC while at 35–49% by LP. The higher anti-inflammatory effect of PC, at all doses, was also observed on the time-course of carrageenan-induced paw edema, displaying profile closely similar to that obtained with diclofenac (25 mg/kg), an anti-inflammatory drug reference (all p < 0.001). Both LP and PC, at all doses, significantly ameliorated liver catalase (CAT) activity (all p < 0.05). However, superoxide dismutase (SOD) activity, glutathione peroxidase (GPx) activity and glutathione (GSH) levels were found increased in liver of PC- compared to LP-carrageenan-injected mice. Our findings demonstrated on one hand higher preventive effects of PC compared to LP in a mouse carrageenan-induced inflammatory model and suggested, on the other hand, that anti-inflammatory effects elicited by the two lichens were closely associated with the amelioration in the endogenous antioxidant status of liver.

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