scholarly journals Brown Bowel Syndrome Is a Rare and Commonly Missed Disease: A Case Report and Literature Review

2021 ◽  
Vol 2021 ◽  
pp. 1-5
Author(s):  
Joaquin Ponce-Zepeda ◽  
Wenchang Guo ◽  
Giorgioni Carmen ◽  
Daniel Moon Kim ◽  
Gregory C. Albers ◽  
...  

Background. Brown bowel syndrome (BBS) is a rare gastrointestinal condition, and vitamin E deficiency has been considered to be a main contributor. However, vitamin E deficiency has been found in only a few patients throughout the published literature studies and its cutoff lab value for diagnosis is not entirely clarified. Case Presentation. A 56-year-old female patient with a history of congenital bowel obstruction (repaired at birth) presented with bloating, abdominal pain, and chronic diarrhea. Endoscopy identified unremarkable gastrointestinal mucosa except a few small polyps in the colon. A partial obstruction was detected by a small bowel follow-through series and then confirmed by CT scan. The resected small bowel was significantly dilated with a thickened brown wall and extensive serosal adhesion. Microscopic examination revealed unremarkable mucosa, but dense granular brown pigments were identified in the cytoplasm of the smooth muscle cells in the muscularis propria. These deposits resulted to be lipofuscin, and BBS was diagnosed. The patient was asymptomatic at 9-month follow-up after surgery without vitamin E supplement. Conclusion. Mitochondrial damage with lipofuscin deposition is at the root of BBS pathogenesis. Any etiology associated with mitochondrial damage can cause this disease, and vitamin E deficiency is just one of them. Dysmotility from extensive serosal adhesion could be a possible etiology for this patient. Due to overlapping symptoms, lipofuscin deposition primarily in the muscularis propria, and unclear serum value of vitamin E, this syndrome is often missed in routine clinical practice from the superficial biopsy. A transmural biopsy is necessary for a definite diagnosis.

2019 ◽  
Vol 25 (1) ◽  
Author(s):  
Danielle Whiting ◽  
Ian Rudd ◽  
Amit Goel ◽  
Seshadri Sriprasad ◽  
Sanjeev Madaan

Abstract Background Angiomyolipomas are rare mesenchymal tumours arising from the perivascular epithelioid cells consisting of variable amounts of adipose, thick-walled blood vessels and smooth muscle cells. These benign tumours commonly occur in the kidney with only a few case reports of adrenal angiomyolipomas which have the potential to reach a large size and haemorrhage. Case presentation A 45-year-old lady presented with a 3-week history of right loin pain, nausea and vomiting. A CT scan revealed a right adrenal angiomyolipoma measuring 6.3 × 6.8 cm with associated haemorrhage. The lesion was successfully treated with right open adrenalectomy, and histology confirmed the diagnosis of adrenal angiomyolipoma. The patient remained well with no evidence of recurrence at the 36-month follow-up. Conclusion Adrenal angiomyolipomas are rare benign tumours that have the ability to reach a large size and potential to bleed. Here, we report the second case of spontaneous haemorrhage in an adrenal angiomyolipoma, which was successfully treated with open adrenalectomy.


2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Sara L Weidmayer ◽  
Hakan Demirci

Abstract Background The natural course of a vortex vein varix, though not well understood, has been known to remain stable. However, here we report a novel case of a vortex vein varix that resolved after an extended period of monitoring. Case presentation An asymptomatic 96-year-old Caucasian man was found to have a vortex vein varix. At his previous examination 13 months prior, his fundus was normal. At 13 months of observation, his vortex vein varix become clinically undetectable. Further follow-up confirmed continued absence of the varix. Conclusion This case demonstrates the development then clinical resolution of a vortex vein varix with no clear identifiable factors for its evolution. This case is novel and offers new insight into the natural history of some vortex vein varices, implicating venous congestion as an instigator and venous collateralization as its alleviator, suggesting that vortex vein varices are likely more common than previously reported since some may be temporary and under-identified.


2021 ◽  
Vol 108 (Supplement_6) ◽  
Author(s):  
P Gungadin ◽  
A Taib ◽  
M Ahmed ◽  
A Sultana

Abstract Introduction Small bowel obstruction can be caused by multiple factors. We describe an unusual case of small bowel obstruction secondary to three rare factors: gallstone ileus, peritoneal encapsulation and congenital adhesional band. Case Presentation A seventy-nine-year-old male presented with a four-day history of obstipation and abdominal pain. CT abdomen pelvis revealed small bowel obstruction secondary to gallstone ileus. The patient was managed by laparotomy. The intraoperative findings revealed the presence of a congenital peritoneal encapsulation with an adhesional band and gallstone proximal to the ileo-caecal valve. Although there was some dusky small bowel, this recovered following the release of the band. Discussion Peritoneal Encapsulation is a rare congenital pathology resulting in the formation of an accessory peritoneal membrane around the small bowel. This condition is asymptomatic and rarely presents as small bowel obstruction. The diagnosis is often made at laparotomy. There are less than 60 cases reported in literature. Gallstone ileus is another rare entity caused by an inflamed gallbladder adhering to part of the bowel resulting in a fistula. Conclusions The rarity of these conditions mean that they are poorly understood. A combination of this triad of gall stone ileus in the presence of peritoneal encapsulation and congenital band has not been reported before. Knowledge of this would raise awareness, facilitate diagnosis and management of patients.


Author(s):  
Zahra Tavoli ◽  
Ali Montazeri

Introduction: Uterus didelphys with obstructed hemivagina associated with ipsilateral renal agenesis (OHVIRA syndrome) is a rare female urogenital malformation and delay in its diagnosis could lead to several complications. Case presentation: A 21-year-old virgin woman was admitted to the emergency department (ED) with severe abdominal pain, without fever and vaginal discharge. She reported a history of cyclic abdominopelvic pain and dysmenorrhea for 5 years. The primary diagnosis (OHVIRA syndrome) was made using ultrasonography, spiral computed tomography (CT) and magnetic resonance imaging (MRI). In addition, laparoscopy was performed to confirm diagnosis and drain hematosalpinx. Then, hysteroscopy was carried out for septum resection and catheter insertion. At one-month follow-up the ultrasonography showed normal left hemicavity of uterus associated with significant decrease in dysmenorrhea. Conclusion: Being aware of OHVIRA syndrome and clinical suspicion of this rare anomaly are essential for making a timely diagnosis, preventing complications, relieving symptoms, and preserving future fertility.


FACE ◽  
2021 ◽  
pp. 273250162110536
Author(s):  
Joshua Harrison ◽  
Samantha Marley ◽  
Shawhin Shahriari ◽  
Christian Bowers ◽  
Anil Shetty

We report a rare case of an extramedullary plasmacytoma (EMP) in the frontal sinus with an indolent clinical presentation. Although a history of trauma was absent, the initial diagnosis was a mucocele, based on the radiological findings. Upon surgical excision, the patient was found to have an EMP. EMP, a form of solitary plasmacytoma, has a significantly high rate of conversion to multiple myeloma. This mandates long-term follow-up, even after successful radiotherapy and/or resection. While radiation therapy is generally considered a first line treatment for EMP, surgical intervention may provide optimal treatment in complicated cases. This case presentation highlights the prognosis of patients diagnosed with EMP.


2021 ◽  
Author(s):  
Tao Sun ◽  
Wentao Wang ◽  
Longshuang He ◽  
Yu Su ◽  
Ning Li ◽  
...  

Abstract Background: Primary trigeminal neuralgia (TN), hemifacial spasm (HFS) and glossopharyngeal neuralgia (GN) are common diseases of nervous system, with similar pathogenesis and treatment strategies. Coexistent of such disease, especially coexistent of TN-HFS-GN simultaneously, is very rare. To date, only nine cases have been reported.Case Presentation: A 70-year-old male with a history of hypertension and diabetes complained of severe involuntary contraction for about 10 years, knife-like and lighting-like pain, which was restricted to the distribution of the second and third branches of trigeminal nerve and pharynx and root of tongue, for about 2 years. Coexistent of TN HFS and GN was diagnosed and MVD was carried out. After MVD, the patient completely free from symptoms and no recurrence and hypoesthesia were recorded in 18 months follow up.Conclusion: Here we report the tenth and oldest male patient with coexistent of TN-HFS-GN. Despite limited reports, MVD is the preferred choice for such diseases which can free patients from spasm and neuralgia.


2019 ◽  
Vol 64 (3) ◽  
pp. 116-118
Author(s):  
Özant Helvacı ◽  
Seyma Yıldız ◽  
Berfu Korucu ◽  
Ulver Derici ◽  
Turgay Arinsoy

Background Coeliac crisis is a life-threatening presentation of coeliac disease. Severe diarrhoea, weight loss, electrolyte imbalances and malnutrition are prominent features. Although mainly a disease of childhood, it can on the rare occasion be diagnosed in adults. Case presentation A 25-year-old female with severe generalised oedema, lower extremity weakness, hypokalemia and profound hypoalbuminemia was referred with an initial diagnosis of nephrotic syndrome. Three months previously she had given birth to a healthy child following an uneventful pregnancy. She did not have proteinuria. She had a history of diarrhoea with gluten-containing food since childhood but lacked a formal diagnosis of coeliac disease. A duodenal biopsy confirmed the suspected diagnosis. Coeliac crisis was diagnosed with life-threatening multisystem involvement. Introduction of a gluten-free diet abolished all disease symptoms and ameliorated laboratory parameters at six months’ follow-up. Conclusion Coeliac crisis is a rare, yet dangerous presentation of coeliac disease in adults. As this case suggests, it can present with generalised oedema and hypoalbuminemia mimicking nephrotic syndrome. Rapid diagnosis is the key to successful treatment.


2020 ◽  
Vol 48 (11) ◽  
pp. 030006052097144
Author(s):  
Yanan Xie ◽  
Jingzhe Han ◽  
Jinming Liu ◽  
Jie Hao ◽  
Xiuguang Zu ◽  
...  

Background Bidirectional ventricular tachycardia (BVT) is a rare, but serious, arrhythmia. Hypokalemia is commonly found in clinical practice, but hypokalemia-induced BVT has rarely been reported. Case presentation A 74-year-old male patient with the symptoms of chest distress and palpitations was admitted owing to frequent discharge of his implantable cardioverter defibrillator (ICD) for 4 days. Before admission, the patient experienced diarrhea after intake of crabs, and felt frequent discharge of his ICD with a total of approximately 17 discharges in 4 days. He had no history of digitalis use. The serum potassium level after admission was 3.1 mmol/L and an electrocardiogram was consistent with BVT. The diagnosis was ventricular tachycardia, electrical storm, and hypokalemia. His ventricular tachycardia was completely relieved after correction of hypokalemia. Conclusions After correction of hypokalemia in this patient, the episode of BVT was terminated and no recurrence of BVT was observed during long-term follow-up. Our findings suggest the diagnosis of hypokalemia-induced BVT.


2006 ◽  
Vol 72 (12) ◽  
pp. 1216-1217
Author(s):  
Hadi Najafian ◽  
Camille Eyvazzadeh

The wireless enteroscopy capsule (WEC) was approved for noninvasive visualization of small bowel. We report an unusual case of a previously healthy man with history of bowel resection and anastomosis who developed small bowel obstruction after ingestion of a WCE. At operation, an anastomotic stricture site was noted and the WEC was proximal to this stricture, causing obstruction. This case emphasizes the importance of a good history and physical examination, as well as vigilant follow-up and retrieval of WEC.


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