pISSN: 2538-2683
eISSN: 2538-2691
Editor-in-Chief:
Shahin Akhondzadeh
Editor-in-charge:
Seyed Farshad Allameh
Current Issue
Vol 10 No 1 (2025): January-February
Case Report(s)
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Temporomandibular joint (TMJ) ankylosis is a debilitating condition characterized by the fusion of the mandibular condyle to the glenoid fossa, leading to restricted mandibular motion. We report the case of a 26-year-old female with left-sided TMJ ankylosis, presenting with limitation in mouth opening and degenerative changes. Preoperative imaging and planning facilitated the fabrication of patient-specific cutting guides and custom alloplastic components. Surgical intervention involved careful osteotomies, resection of the ankylotic mass, and placement of custom joint components into the surgically prepared fossa. Postoperatively, the patient underwent intermaxillary fixation for 2 months, and was then referred to a physiotherapist, ultimately achieving a mouth opening of 30 mm. Follow-up at 1 and 3 months showed stable improvement, with no signs of reankylosis. Customized TMJ reconstruction, combined with an autogenous fat graft, offers a viable
solution for restoring mandibular mobility and improving quality of life in patients with TMJ ankylosis. -
Scimitar syndrome is a rare congenital cardiac anomaly characterized by abnormal drainage of the right pulmonary veins into the inferior vena cava (IVC). Here, we report the case of a 26-year-old female diagnosed with scimitar syndrome (SS), with a history of imperforate anus and vaginal agenesis, consistent with a VACTERL association. The imperforate anus was identified and treated in infancy, whereas the vaginal agenesis was diagnosed at menarche. Additionally, the patient presented with a large atrial septal defect (ASD) and a hypoplastic right lung alongside SS. This case underscores the potential correlation between SS, vaginal agenesis, and imperforate anus as features within the VACTERL spectrum.
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Dentigerous cysts are developmental odontogenic lesions and represent the second most common type of jaw cysts, typically associated with the crowns of unerupted or impacted teeth. They are frequently discovered incidentally during routine radiographic evaluations, particularly in younger individuals. The present study describes a rare case of a 62-year-old edentulous female who presented with purulent discharge beneath
her maxillary complete denture. Clinical and radiographic examinations, including cone-beam computed tomography (CBCT), revealed an infected dentigerous cyst associated with an impacted maxillary canine and an unusual pattern of coronal tooth resorption. This finding is atypical, as dentigerous cysts are more commonly linked to root resorption. The case underscores the diagnostic value of CBCT in evaluating atypical presentations of odontogenic cysts and highlights the importance of early detection and management to prevent complications such as infection and structural compromise. -
Intravenous leiomyomatosis (IVL) is an unusual smooth muscle tumor characterized by benign smooth muscle cells growing within blood vessels, often reaching the inferior vena cava, right atrium, and pulmonary vessels. The best treatment is complete surgical removal, and the outlook is typically positive, with recurrence occurring rarely. This report discusses a 38-year-old woman who was unexpectedly found to have intravenous leiomyomatosis extending from her uterus to the inferior vena cava and right atrium. She underwent one-stage surgery, which included total hysterectomy, bilateral salpingo-oophorectomy, and removal of the lesions from the inferior vena cava and right atrium, all without the need for thoracotomy.
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We report a 37-year-old woman with recurrent episodes of excessive daytime sleepiness, bizarre behavior, social delays, and confusion lasting 3–5 days. Between episodes, she experienced only mild migraine-like headaches. Genetic testing identified an SCN1A mutation, which may cause central sleep apnea, basilar-type migraine, and a channelopathy-related encephalopathic state in her. This is the first report linking an SCN1A mutation with central sleep apnea. We recommend future cohort studies to clearly examine the association between SCN1A mutation and central sleep apnea.
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Krukenberg tumors, metastatic ovarian carcinomas primarily originating from gastrointestinal malignancies, present significant diagnostic and therapeutic challenges during pregnancy. We report a case of a 39-year-old pregnant woman initially diagnosed with autoimmune pancreatitis (AIP) and cholestasis of pregnancy, later found to have metastatic signet ring cell carcinoma of the stomach with ovarian involvement. This case highlights the complexities in differentiating malignancy from benign hepatobiliary disorders during pregnancy, emphasizing the need for a multidisciplinary approach. Early integration of tumor markers, advanced imaging, and HISORt criteria can facilitate timely diagnosis and improve clinical outcomes.
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A heterotopic pregnancy involves multiple embryos implanted in different locations, usually one in the uterus and another ectopically. This rare condition can pose significant risks to both the mother and the developing embryos. Heterotopic pregnancy is very rare. An interstitial heterotopic pregnancy is even rarer, representing
2%–4% of all ectopic pregnancies, but it has a much higher mortality rate overall because of the complexities involved in diagnosis and management. This case involves a rare instance of interstitial heterotopic pregnancy, likely resulting from assisted reproductive technology. Ultrasound revealed a live intrauterine pregnancy and an interstitial ectopic pregnancy sac. A 40-year-old woman, G5P1Ab1Ep2, was referred to the hospital triage in her 7th week of pregnancy due to abnormal ultrasound findings. Considering the clinical conditions and the possibility of uterine rupture with increasing gestational age, a decision was made to reduce the ectopic pregnancy by injecting KCl. Ultimately, a multidisciplinary approach involving obstetricians, gynecologists, and,
when necessary, maternal–fetal medicine specialists is vital to optimize outcomes for both the mother and the developing fetus. -
Double pylorus is a rare endoscopic finding, often secondary to chronic peptic ulcer disease and mucosal fistulization. It may become evident incidentally or during an investigation of upper gastrointestinal (GI) bleeding. Our case was a hemodynamically unstable woman who presented with hematemesis. Gastroduodenoscopy revealed a double pylorus with a visible vessel in the bulb region. We successfully controlled the bleeding endoscopically, and the patient was discharged in stable condition. Double pylorus may rarely be observed in patients with recurrent or complicated peptic ulcers. Recognition of this anomaly is essential for appropriate management.
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Pregnancy significantly alters the clinical presentation of various diseases, often masking or mimicking symptoms of serious underlying conditions. In high-risk pregnancies, overlooking nonspecific symptoms may delay the diagnosis of life-threatening disorders, resulting in poor maternal and fetal outcomes. This report describes two rare cases of pregnancy in women with complex medical histories. The first patient, a 34-year-old at 30 weeks’ gestation, was diagnosed with invasive ductal breast cancer with extensive bone and pulmonary metastases, as well as hypercalcemia-induced renal failure. Despite emergency dialysis, respiratory support, spinal anesthesia for delivery, and coordinated care from nephrology, endocrinology, obstetrics, and anesthesiology teams, her condition worsened postpartum. She developed respiratory acidosis and decreased consciousness, and required intubation. Although initially stabilized and her malignancy confirmed via
biopsy, she progressed to multi-organ failure and died from asystole 5 days postpartum, on January 22, 2025, despite resuscitation. The second patient, also 34, had a history of repaired congenital heart disease and right-sided invasive ductal carcinoma. Against medical advice, she pursued a high-risk spontaneous pregnancy, closely monitored by a multidisciplinary team. Her hypertension and gestational diabetes were managed with
medications and insulin, respectively. At 37 weeks, she underwent elective cesarean section at a tertiary center, delivering a healthy boy with normal weight and Apgar scores of 9 and 10. She was discharged hemodynamically stable from the ICU, approved for breastfeeding, and scheduled to start chemotherapy on March 10, 2025. These contrasting cases demonstrate how outcomes in high-risk pregnancies are profoundly influenced by timely diagnosis and coordinated care. Nonspecific symptoms during pregnancy should
prompt thorough evaluation, especially in the absence of antenatal care. A structured, team-based approach can significantly improve maternal and neonatal outcomes, even in medically complex scenarios. -
Hereditary angioedema (HAE) is a rare condition that may involve life-threatening upper airway swelling. A 19-year-old female with known C1 esterase inhibitor deficiency presented multiple times with lip and tongue swelling without stridor. She was electively intubated for airway protection during each episode and treated with Berinert®, resulting in symptom resolution. Hormonal and prophylactic therapy was initiated after repeated ICU admissions. This case highlights the need for high suspicion and early airway intervention in HAE, even in the absence of overt respiratory distress. Prompt airway management and tailored long-term treatment are essential in managing recurrent angioedema.
