A rare case of accessory maxilla and bilateral Tessier no. 7 clefts, a 10-year follow-up

2013 ◽  
Vol 41 (6) ◽  
pp. 527-531 ◽  
Author(s):  
Ali Borzabadi-Farahani ◽  
Stephen L.-K. Yen ◽  
Cameron Francis ◽  
Pedro A. Lara-Sanchez ◽  
Jeffrey Hammoudeh
Keyword(s):  
Rare Case

scholarly journals A life-threatening spontaneous ascending aortic rupture due to a small penetrating aortic ulcer

2021 ◽  
Vol 9 ◽  
pp. 2050313X2110377
Author(s):  
Yasuhito Nakamura ◽  
Kiyoshi Doi ◽  
Syojiro Yamaguchi ◽  
Etsuji Umeda ◽  
Osamu Sakai ◽  
...  
Keyword(s):  
Rare Case ◽  
Aortic Wall ◽  
Aortic Rupture ◽  
Ascending Aorta ◽  
Bleeding Site ◽  
Penetrating Aortic Ulcer ◽  
Postoperative Course ◽  
Life Threatening ◽  
Aortic Dissections

We reported a rare case of spontaneous frank rupture of a small (4 mm) penetrating aortic ulcer in the ascending aorta resulted in catastrophic bleeding. The ulcer only created a pinhole wound in the adventitia without saccular aneurysms, intramural hematomas, or aortic dissections. Notably, the wound could be directly closed because the aortic wall was intact only 5 mm away from the bleeding site. The postoperative course was uneventful, and the patient was discharged on the 11th postoperative day. After 8 months, follow-up computed tomography showed no abnormality of the aortic wall at the repair site.

A rare case of hypokalaemia and hypophosphataemia secondary to geophagia

2021 ◽  
Vol 14 (5) ◽  
pp. e239322
Author(s):  
Charmaine Schmidt ◽  
Jonathan Oxley Oxland ◽  
Robert Freercks
Keyword(s):  
Creatine Kinase ◽  
Iron Deficiency ◽  
Phosphate Binder ◽  
Rare Case ◽  
Potassium Level ◽  
Serum Potassium Level ◽  
Electrolyte Abnormality ◽  
Distal Muscle

We report a case of severe hypokalaemia and moderate hypophosphataemia from clay ingestion. A 60-year-old woman presented with flaccid paralysis. Investigations revealed a serum potassium level of 1.8 mmol/L, phosphate level of 0.56 mmol/L and creatine kinase level of 30 747 IU/L. She had marked proximal and distal muscle weakness due to severe hypokalaemia and concurrent hypophosphataemia, which likely contributed to the onset of rhabdomyolysis. The patient subsequently admitted to significant pica, most likely secondary to an associated iron deficiency. We conclude that the ingested clay acted as a potassium and phosphate binder. Although we did not investigate the content of the clay in this case, it has been reported that clay can bind potassium in vitro and is rich in minerals such as aluminium that could play a role in the binding of phosphate, although the exact mechanism remains unclear. The patient recovered fully and outpatient follow-up at 6 months and again at 40 months confirmed no electrolyte abnormality, myopathy nor any further geophagia.

Penile Lymphoepithelioma-Like Carcinoma: A Rare Case With PD-L1 Expression

2021 ◽  
pp. 106689692098834
Author(s):  
Raquel Machado-Neves ◽  
Bernardo Teixeira ◽  
Elsa Fonseca ◽  
Pedro Valente ◽  
Joaquim Lindoro ◽  
...  
Keyword(s):  
Human Papillomavirus ◽  
Tumor Cells ◽  
Rare Case ◽  
Malignant Tumors ◽  
Squamous Cell Carcinomas ◽  
The Third ◽  
The Past ◽  
First Case ◽  
Lymphoplasmacytic Infiltrate

Most malignant tumors of the penis are squamous cell carcinomas (SCC), being divided in 2 groups, one human papillomavirus (HPV)-related and another non-HPV-related, with lymphoepithelioma-like carcinoma (LELC) being one of the rarest HPV-related SCC. In this article, we report a case of a 50-year-old man who presented testicular swelling and pain for the past 3 months. A penile mass was identified, and the patient was submitted to a total penectomy. The penectomy specimen showed an ulcerated lesion at the glans reaching the cavernous bodies. Microscopic examination showed undifferentiated epithelial cells with syncytial growth pattern mix with a dense lymphoplasmacytic infiltrate, consistent with LELC. The tumor cells expressed p16 and all 3 different clones of PDL1 (22C3, SP263, and SP142). The patient is alive and well with a follow-up of 3 months. To our knowledge, this is the third LELC of the penis reported in literature and the first case reported with PDL1 expression.

scholarly journals A case report of inappropriate inhibition of ventricular pacing due to a unique pacemaker electrogram storage feature

2021 ◽  
Vol 5 (3) ◽  
Author(s):  
Habib R Khan ◽  
William K Chan ◽  
Juliana Kanawati ◽  
Raymond Yee
Keyword(s):  
Rare Case ◽  
Storage Capacitor ◽  
Ventricular Pacing ◽  
Dual Chamber ◽  
Holter Monitor ◽  
Case Summary ◽  
Ventricular Lead ◽  
Internal Function ◽  
Third Degree Atrioventricular Block

Abstract Background Modern permanent pacemakers (PPMs) have individual features designed to identify cardiac rhythm abnormalities and improve their performance. Inappropriate pacing inhibition may be an undesired outcome from these features and cause symptoms in patients who require frequent pacing, leading to dizziness, and syncope. Inappropriate inhibition can be difficult to identify in circumstances that are intermittent and difficult to reproduce. Case summary A 57-year-old female underwent a mitral valve replacement (MVR) for severe mitral stenosis. One month following MVR, she presented with symptomatic third-degree atrioventricular block, and a dual-chamber PPM (Advisa™, Medtronic, Minneapolis, USA) was implanted and programmed DDD 50–130 b.p.m. At the 3-month follow-up, she reported frequent episodes of lightheadedness. She was found to have intermittent ventricular pacing inhibition on a 48-h Holter monitor due to an internal function of the Advisa™ series of PPMs that attempts to store an electrogram (EGM) every 1 h and 30 s. During the EGM storage, an amplified signal from the storage capacitor can result in oversensing by the ventricular channel and inappropriate pacing inhibition. Discussion To rectify the issue, the ventricular lead sensitivity value was increased from 0.9 mV to 1.2 mV. No instances of inappropriate ventricular pacing inhibition were noted on follow-up. To our knowledge, this is a rare case of inappropriate ventricular pacing inhibition caused by a combination of PPM self-adjusting sensitivity algorithm and oversensing every 1 h and 30 s from an amplified storage capacitor. Physicians should be aware of this possible complication and differentiate it from device or lead malfunction.

scholarly journals Closed loop obstruction and adhesive intestinal obstruction in perineal hernia

2020 ◽  
Vol 13 (12) ◽  
pp. e238112
Author(s):  
Ramprasad Rajebhosale ◽  
Mohammad Miah ◽  
Fraser Currie ◽  
Pradeep Thomas
Keyword(s):  
Small Bowel ◽  
Intestinal Obstruction ◽  
Rare Case ◽  
Transition Point ◽  
Closed Loop ◽  
Abdominoperineal Excision ◽  
Perineal Hernia ◽  
Bowel Gangrene ◽  
Adhesive Intestinal Obstruction

Perineal hernia with bowel gangrene is uncommon but known complication of laparoscopic extralevator abdominoperineal excision (ELAPE). We present a rare case of closed loop small bowel obstruction with bowel gangrene secondary to an incarcerated perineal hernia that developed 7 years after an ELAPE. Intraoperatively, we found a definitive transition point due to adhesions in pelvis and a closed loop obstruction of the distal small bowel at different site with gangrenous intestine. She was managed successfully surgically with adhesiolysis and fixation of defect with biological mesh. Prevalence of perineal hernias will rise in future because of the increasing cases of ELAPE, in which no repair of pelvic floor is performed. The need of follow-up of these operations and more reporting of such cases are important in increasing awareness of these complications. Patients should be made aware of such complications and should seek urgent medical care.

scholarly journals Mixed Cutaneous Infection Caused byMycobacterium szulgaiandMycobacterium intermediumin a Healthy Adult Female: A Rare Case Report

2015 ◽  
Vol 2015 ◽  
pp. 1-4 ◽  
Author(s):  
Amresh Kumar Singh ◽  
Rungmei S. K. Marak ◽  
Anand Kumar Maurya ◽  
Manaswini Das ◽  
Vijaya Lakshmi Nag ◽  
...  
Keyword(s):  
Case Report ◽  
Rare Case ◽  
Healthy Adult ◽  
Nontuberculous Mycobacteria ◽  
Anterior Abdominal Wall ◽  
Cutaneous Infection ◽  
Once Daily ◽  
The Past ◽  
Rare Case Report

Nontuberculous mycobacteria (NTMs) are ubiquitous and are being increasingly reported as human opportunistic infection. Cutaneous infection caused by mixed NTM is extremely rare. We encountered the case of a 46-year-old female, who presented with multiple discharging sinuses over the lower anterior abdominal wall (over a previous appendectomy scar) for the past 2 years. Microscopy and culture of the pus discharge were done to isolate and identify the etiological agent. Finally, GenoType Mycobacterium CM/AS assay proved it to be a mixed infection caused byMycobacterium szulgaiandM. intermedium. The patient was advised a combination of rifampicin 600 mg once daily, ethambutol 600 mg once daily, and clarithromycin 500 mg twice daily to be taken along with periodic follow-up based upon clinical response as well as microbiological response. We emphasize that infections by NTM must be considered in the etiology of nonhealing wounds or sinuses, especially at postsurgical sites.

Intra-Peritoneal Pseudocyst as a Rare Complication After Peritoneal Dialysis: The Use of a Staged Surgical Approach in Management

2021 ◽  
pp. 000313482110474
Author(s):  
Ahmad Kharsa ◽  
Kayla Colvill ◽  
Heather Stevenson ◽  
Jeffrey Fair ◽  
Rupak Kulkarni ◽  
...  
Keyword(s):  
Peritoneal Dialysis ◽  
Surgical Approach ◽  
Rare Case ◽  
Diagnostic Laparoscopy ◽  
Rare Complication ◽  
Cyst Excision ◽  
Neoplastic Processes ◽  
Life Threatening ◽  
Pseudocyst Formation

Despite its numerous benefits, peritoneal dialysis (PD) can rarely result in dangerous and even life-threatening complications, including peritonitis, hernias, encapsulating peritoneal sclerosis (EPS), and rarely peritoneal pseudocysts. Herein, we present a rare case of a giant intra-peritoneal pseudocyst that presented four months following the discontinuation of a 5-year course of complicated PD. Despite the initially successful drainages, the patient’s symptoms continued to recur, and the imaging findings were concerning for underlying neoplastic processes. As such, a staged surgical approach was performed, starting with a diagnostic laparoscopy and was subsequently followed with cyst excision and marsupialization to the peritoneal cavity. While previous reports of such rare pseudocyst have been documented in the literature as a complication of PD, to our knowledge, this is the second case of pseudocyst formation to occur months after the discontinuation of PD therapy. This case emphasizes the importance of close follow-up in PD patients and showcases how a staged surgical approach can be utilized to accurately diagnose and manage such complicated cases.

Snapping thumb: a rare case of stenosing tenosynovitis of the extensor pollicis longus tendon

2021 ◽  
Vol 14 (6) ◽  
pp. e241306
Author(s):  
Andrea Lund ◽  
Pelle Hanberg ◽  
Anders Ditlev Foldager-Jensen ◽  
Maiken Stilling
Keyword(s):  
Rare Case ◽  
Diagnostic Tools ◽  
Extensor Pollicis Longus ◽  
The Third ◽  
Stenosing Tenosynovitis ◽  
Repetitive Loading ◽  
Extensor Compartment ◽  
Extensor Pollicis Longus Tendon ◽  
Surgical Release

Tenosynovitis of the extensor pollicis longus (EPL) is rarely reported in patients without rheumatoid arthritis but may lead to thumb snapping as a consequence of EPL stenosing tenosynovitis.This case presents painful thumb snapping that developed after a wrist trauma and repetitive loading. Ultrasound and MRI were used as diagnostic tools, before surgical release of the EPL in the third extensor compartment was performed. Neither EPL tenosynovitis nor thumb snapping were found at follow-up.

A rare case of spontaneous haemothorax in patient with haemophilia A

2021 ◽  
Vol 14 (4) ◽  
pp. e242412
Author(s):  
Suthaphong Tripoppoom ◽  
Nophol Leelayuwatanakul
Keyword(s):  
Chest Pain ◽  
Rare Case ◽  
Rare Event ◽  
Haemophilia A ◽  
Minor Trauma ◽  
Conservative Approach ◽  
Pleuritic Chest Pain ◽  
Good Clinical Outcome ◽  
History Of

Haemorrhage in patients with haemophilia is common after minor trauma but may occur spontaneously. Despite the diversity of bleeding sites, spontaneous haemothorax, on a non-traumatic basis, is an exceedingly rare event and only a few cases had been reported. We present a case of a 43-year-old man with a history of haemophilia A who had pleuritic chest pain for 1 day without significant history of trauma. Diagnostic thoracentesis showed bloody pleural fluid in which neither abnormal cell nor organism was found. He was treated by cryoprecipitate replacement and therapeutic thoracentesis for releasing haemothorax. After discharge, the patient returned for follow-up with complete radiological resolution. Regarding the consequences of retained haemothorax from conservative approach and the procedure-related bleeding of given therapeutic intervention in haemothorax making its management in patients with haemophilia to be more challenging. Our case illustrates a conservative treatment of spontaneous haemothorax in patient with haemophilia resulting in a good clinical outcome.

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