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Endometriosis-associated Maternal Pregnancy Complications - Case Report and Literature Review.

ABSTRACT: The incidence of endometriosis is increasing. Particularly during pregnancy and labour, clinicians should be alert to possible endometriosis-associated complications or complications of previous endometriosis treatment, despite a low relative risk. In addition to an increased rate of early miscarriage, complications such as spontaneous bowel perforation, rupture of ovarian cysts, uterine rupture and intraabdominal bleeding from decidualised endometriosis lesions or previous surgery are described in the literature. Unfavourable neonatal outcomes have also been discussed. We report on an irreducible ovarian torsion in the 16th week of pregnancy following extensive endometriosis surgery, and an intraabdominal haemorrhage due to endometriosis of the bowel in the 29th week of pregnancy.

SUBMITTER: Petresin J

PROVIDER: S-EPMC4999325 | biostudies-literature | 2016 Aug

REPOSITORIES: biostudies-literature

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Endometriosis-associated Maternal Pregnancy Complications - Case Report and Literature Review.

Petresin J J Wolf J J Emir S S Müller A A Boosz A S AS

Geburtshilfe und Frauenheilkunde 20160801 8

The incidence of endometriosis is increasing. Particularly during pregnancy and labour, clinicians should be alert to possible endometriosis-associated complications or complications of previous endometriosis treatment, despite a low relative risk. In addition to an increased rate of early miscarriage, complications such as spontaneous bowel perforation, rupture of ovarian cysts, uterine rupture and intraabdominal bleeding from decidualised endometriosis lesions or previous surgery are described ...[more]

PMID: 27570252

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Project description:BackgroundExtrapelvic endometriosis is defined as the presence of ectopic endometrial tissue in structures outside the pelvis. Although extra-pelvic endometriosis is generally considered benign conditions, malignant potential within endometriotic foci occurs even after definitive surgery. Malignant transformation of hepatic endometriosis is extremely rare. Preoperative diagnosis of this cancer is difficult, and no guidelines on the optimal management currently exist. Here, we present a case report of malignant transformation of hepatic endometriosis and a brief literature review to highlight the current knowledge of the prevalence, clinical features, diagnosis, and management of this condition.Case presentationA 50-year-old woman with a 2-year duration of progressive right upper quadrant abdominal pain was admitted to the hospital. She underwent hysterectomy and bilateral salpingo-oophorectomy for benign conditions 4 years prior. Tumor markers demonstrated elevated carbohydrate antigen (CA)-199 112U/mL (normal range: 0-35U/mL) only. Radiological imaging suggested the presence of a 10.7 × 7.7-cm mass in the right lobe of the liver extending to the diaphragm. The intraoperative frozen sections suggested malignant tumor. Right hepatectomy with infiltrating diaphragm resection was performed. The final pathology with immunohistochemistry staining confirmed endometrioid adenocarcinoma in the liver originating from preexisting hepatic endometriosis. After the multidisciplinary team meeting, the consensus was surgery followed by adjuvant chemotherapy. To our knowledge, this is the first case of Chinese woman of a malignant liver tumor originating from endometriosis ever reported by reviewing the current English medical literature.ConclusionThough rare, extrapelvic endometriosis-associated cancers should be considered as differentiated diagnosis even after hysterectomy and bilateral salpingo-oophorectomy. This case highlights the importance of collaborative efforts across multiple disciplines for accurate diagnosis and appropriate treatment of malignant transformation of hepatic endometriosis.

Project description:IntroductionMulibrey nanism (MUL) is a rare disorder caused by TRIM37 gene variants characterized by growth failure, dysmorphic features, congestive heart failure (CHF), and an increased risk of Wilms' tumor. Although immune system impairment has been documented in MUL, the underlying mechanisms remain poorly understood.MethodsWe present a case of MUL with progressive lymphopenia and review similar cases from the literature.ResultsOur patient presented with prenatal onset growth restriction, characteristic dysmorphic features, and Wilms' tumor. She developed progressive lymphopenia starting at 10 years of age, leading to the initiation of intravenous immunoglobulin (IVIG) replacement therapy and infection prophylaxis. Genetic analysis detected a likely pathogenic variant on the maternal allele and copy number loss on the paternal allele in TRIM37. Subsequently a cardiac magnetic resonance imaging was conducted revealing signs of pericardial constriction raising concerns for intestinal lymphatic losses. The cessation of IVIG therapy did not coincide with any increase in the rate of infections. The patient exhibited a distinct immunological profile, characterized by hypogammaglobulinemia, impaired antibody responses, and skewed T-cell subsets with an altered CD4+/CD8+ ratio, consistent with previous reports. Normal thymocyte development assessed by artificial thymic organoid platform ruled out an early hematopoietic intrinsic defect of T-cell development.DiscussionThe immunological profile of MUL patients reported so far shares similarities with that described in protein-losing enteropathy secondary to CHF in Fontan circulation and primary intestinal lymphangiectasia. These similarities include hypogammaglobulinemia, significant T-cell deficiency with decreased CD4+ and CD8+ counts, altered CD4+/CD8+ ratios, and significantly modified CD4+ and CD8+ T-cell phenotypes toward effector and terminal differentiated T cells, accompanied by a loss of naïve CD45RA+ T lymphocytes. In MUL, CHF is a cardinal feature, occurring in a significant proportion of patients and influencing prognosis. Signs of CHF or constrictive pericarditis have been evident in the case reported here and in all cases of MUL with documented immune dysfunction reported so far. These observations raise intriguing connections between these conditions. However, further investigation is warranted to in-depth define the immunological defect, providing valuable insights into the pathophysiology and treatment strategies for this condition.

Project description:Peripartum pancreatitis is a rare clinical condition that occurs usually in the third trimester of pregnancy. Pancreatitis is usually secondary to gallstones, and it can lead to life-threatening and rare complications. We report a case of necrotizing postpartum pancreatitis that developed abdominal compartment syndrome (ACS) in early course, posterior reversible encephalopathy syndrome (PRES), and splanchnic and extrasplanchnic thrombosis later on. Case. 31-year-old female, one week after delivery, presented to the emergency department with abdominal pain, nausea and vomiting, tenderness in the epigastrium, and raised pancreatic enzymes. Ultrasound (USG) showed bulky pancreas with gallstones. She was diagnosed as having acute biliary pancreatitis and started to be hydrated and was supplemented with analgesia. Her condition deteriorated on the 2nd day, and she was shifted to the surgical intensive care unit (SICU) where she developed abdominal compartment syndrome (ACS), respiratory distress, and acute kidney injury, requiring endotracheal intubation and ventilation. Computerized tomography (CT) showed pancreatic necrosis with multiple fluid collections and significant left-sided pleural effusion. Percutaneous drainage of pleural effusion was done, and she was stabilized to be weaned off from mechanical ventilation. On day 15, she underwent USG-guided drainage of the pancreatic collection and ERCP (endoscopic retrograde cholangiopancreatography) on day 19. Post-ERCP, she had tonic colonic convulsions which were treated with benzodiazepines and phenytoin. It was diagnosed by imaging studies as posterior reversible encephalopathy syndrome (PRES). Her abdomen was still distended and tender; CT showed a significant pseudocyst with splanchnic and extrasplanchnic thrombosis. She had laparotomy, gastrocystostomy, and cholecystectomy on day 28th. She made uncomplicated recovery and discharged in good health. Conclusion. Peripartum pancreatitis can be complicated by ACS, PRES, and splanchnic and extrasplanchnic thrombosis.

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Endometriosis-associated Maternal Pregnancy Complications - Case Report and Literature Review.

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Endometriosis-associated Maternal Pregnancy Complications - Case Report and Literature Review.

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