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Volume 72, Issue 12 (3-2015)
Background: Tumoral calcinosis is a hereditary disorder of metabolic dysfunction of phosphate regulation. It is an idiopathic calcinosis that characterized by the deposition of calcium phosphate in periarticular tissues that causes typically lobulated, well demarcated calcification around large joints particularly the extensor surfaces. It is usually painless. It is common in puberty age and adolescents. The involvement of the hand phalanges is very rare that can make a mistake in diagnosis if it is infected. Tumoral calcinosis is seen the same in both sexes. The electrolyte levels of calcium and phosphorus is normal and sometimes is hyperphosphatemia. It is the first report of tumoral calcinosis in Iran. Case report: A 7-year-old girl presented with redness, yellowish discharge and painful swelling of the left hip and the third web space of left hand admitted to Vali-e-Asr Hospital, Tehran, Iran, in 2013. The onset of the disease was 3.5 years ago. She did not mention the family history of the disease. The pain was at the left hip first. Six months later the third and fourth phalanges of the left hand was swollen. Physical examination revealed an erythematous mass in the extensor surfaces of the third and fourth metacarpals of the left hand. It was tender in palpation. The smear and culture of discharge was staphylococcus aureus. X-rays revealed calcification of the third and fourth metacarpals of the left hand. The entire lesion was managed by surgical excision. Successful postoperative medical management in the form of low calcium and low phosphorus diet and oral cloxacillin was performed. Conclusion: Tumoral calcinosis involves rarely the interphalangeal joints of hand. Because of its compression over adjacent nerves, it is painful. Sometimes it has a sterile discharge and rarely superimposed infections may occur. Radiologists can play a major role in early diagnosis and probable complications.
Volume 73, Issue 2 (5-2015)
Background: Rupture of uterus is a catastrophic complication associated with significant maternal and fetal morbidity and mortality. The prevalence of an unscarred uterine rupture is very rare. Although the most important complication of dilatation and curettage is perforation of uterus, dilatation and curettage is not introduced as an important cause of uterine rupture. Case presentation: Here we present a case of uterine rupture in a pregnant woman that was admitted in Tehran General Women Hospital, in December 2014, with reducing fetal movement in her 41th weeks of pregnancy. She did not have any risk factors for rupture of uterus including cephalo-pelvic disproportion and polyhydramnios, also there was no history of uterine surgery such as myomectomy and uterine abnormality repair. A term dead male neonate was delivered by cesarean section due to arrest of descending in stage 2 of labor. The baby weighed 3400 gr and had anomaly in ears, larynx, uvula and soft palate. Its chromosomal study depicted both trisomy and monosomy for chromosome 13 and 21. Mother had a history of illegal curettage and trauma to the uterus in her first pregnancy two years ago. She did not say to us this history and abortion during admission. After cesarean section we saw that in the left side of the posterior wall of uterus was ruptured and baby was died. At surgical exploration, moderate hemoperitoneum was evident. Fetus was already dead at the time of extraction. Total estimated blood loss was 100 ml, the patient was transfused with two units of packed cell. The woman was discharged on the fifth postoperative day in good condition. Conclusion: Effective contraception and safe curettage can reduce maternal mortality and morbidity. Also special attention to a history of curettage to predict uterine rupture is critical.
Volume 73, Issue 7 (10-2015)
Background: Standard treatment of Gestational Trophoblastic Disease (GTD) is chemotherapy. Single-agent chemotherapy regime including Methotrexate (MTX) or Actinomycin. Single-agent is widely used in treatment of persistent trophoblastic disease. We reported an uncommon toxicity of low-dose single-agent methotrexate in a patient. Case Presentation: A 20-year-old woman, primary gravid after two months missed period and spotting with diagnosis of incomplete abortion with uterine size equivalent of ten weeks pregnancy (8-10 cm) underwent evacuation curettage. In serial follow-up, based on rise of beta-hCG titer and absence of metastatic disease, it was categorized as low-risk persistent trophoblastic disease. She was referred to gynecology oncology center of Ghaem Hospital, Mashhad University of Medical Sciences in May 2014. Because of rise of beta-hCG titer, after complete metastatic work-up and lack of disease in other sites, persistent disease was diagnosed and candidate for chemotherapy (single agent low-dose). The patient received first course of therapy with MTX (50 mg/m², intra muscular). Unfortunately, after two days of treatment she developed uncommon severe toxicity, fever, severe nausea and vomiting, tachycardia, and generalized weakness. Also, we found hematologic abnormality (WBC: -14000-15000 µI, platelet- 540 µI and sever neutropenia), and abnormal rising in liver function test (SGOT, SGPT) (three to four times) and renal function test (BUN and Creatinine) (two times). In addition, she had disseminated erosive lesion in all of body especially in face. Due to the fatal side effects of chemotherapy, she was admitted to intensive care unit (ICU). Fortunately, after two to three weeks, she was improved by conservative management. After few weeks beta-hCG titer was in normal limit. However she had normal serial beta-hCG in one year of follow-up. Conclusion: It is important to emphasis unpredictable side effects of chemotherapy with low-dose methotrexate.
Volume 74, Issue 10 (1-2017)
Background: Autosomal recessive polycystic kidney disorder (ARPCKD) is one of the most prevalent hereditary disorders in neonates and children. Its frequency is between 1/6000 to 1/55000 births. In the most severe cases, it can be diagnosed prenatally by the presence of enlarged, echogenic kidneys and oligohydramnios. However, in the milder forms, clinical manifestations are usually detected in neonatal and childhood period. PKHD1 gene located on chromosome 6 is linked with this disorder. About half of detected mutations in this gene are missense ones. The largest protein product of this gene is called the FPC/polyductin complex (FPC). It is a single-membrane spanning protein whose absence leads to abnormal ciliogenesis in the kidneys.
Case presentation: Here we present a 5-year-old female patient affected with ARPCKD. She has been born to a non-consanguineous healthy Iranian parents. No similar disorder has been seen in the family. Prenatal history has been normal. In order to find the genetic background, DNA was extracted from patient's peripheral blood lymphocytes. PKHD1 gene exons and exon-intron boundaries were sequenced using next generation sequencing platform. Two novel variants have been detected in compound heterozygote state in the patient (c.6591C>A, c.8222C>A). Bioinformatics tools predicted these variants to be pathogenic.
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Conclusion: In the present study, we detected two novel variants in PKHD1 gene in a patient with ARPCKD. The relatively mild phenotype of this patient is in accordance with the missense mutations found. Molecular genetic tools can help in accurate risk assessment as well as precise genotype-phenotype correlation establishment in families affected with such disorder to decrease the birth of affected individuals through preimplantation genetic diagnosis or better management of disorder. |
Volume 75, Issue 2 (5-2017)
Background: Osteoblastoma is one of the rarest primary benign bone tumors which accounts for 1% of all bone neoplasms and 3.5% of benign bone tumors, with the potential for local invasion and recurrence. Osteoblastoma is not homogeneous. Differences in histological details have led to the division of these lesions into subtypes. The histologic features in most cases are distinctive, there are various changes that make the diagnosis challenging. Although involvement of this disease is more in the spines or small long bones, but it has been reported involvement of jaws in this disease. Correct diagnosis is very important because it may be misdiagnosed osteosarcoma with hypercellular cases.
Case presentation: In this case report, it is described patient a 7-year-old girl with a lesion in the posterior maxilla and bone resorption in May 2016, Sari, Mazandaran province, Iran. Differential diagnosis of the radiological and clinical findings suggested an osteosarcoma. In terms of pathology, osteoblastoma was included a well-vascularized connective stroma tissue with plenty of veins which osteoid and primitive woven bone can be seen actively. In microscopy diagnosis, samples were shown cortical bone with a natural appearance, prominent osteoblasts and surrounding granulation tissue containing blood vessels that confirmed maxilla osteoblastoma. The lesion was examined by histopathology method for final recognition and the results revealed osteoblastoma.
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Conclusion: Although jaw osteoblastoma is a rare tumor and presents with microscopic features that can mimic a variety of other types of malignant entities. It is important to avoid of other non-specific clinical and radiographic protests that led to the recognition problems. The importance of interdisciplinary cooperation between the surgeon, radiologist and pathologist must be pressured. |
Volume 75, Issue 3 (6-2017)
Background: Cerebral arteriovenous malformations are rare congenital anomalies presenting as different symptoms depending on their size and the age of patient. Congestive heart failure is a rare condition in neonatal period and is most common due to structural heart defects, but rarely may be a result of peripheral shunts such as cerebral arteriovenous malformation.
Case presentation: A term male newborn infant who was delivered by Caesarean Section in Chamran Hospital, Ferdows, South Khorasan Province, June 2016. The infant was admitted to neonatal care unit due to nonreactive nonstress (NST) with normal Apgar score. In first postpartum visit, a systolic heart murmur was detected. Echocardiography showed small atrial septal defect secundum type and patent foramen ovale (PFO). He presented clinical manifestations of heart failure after 72 hours of birth. Antibiotic and treatment of heart failure was started. Following excluding most common etiologies of heart failure such as sepsis, anemia and arrhythmias, for detecting less common conditions such as cerebral vascular aneurism a transfontanelle ultrasonography was performed which showed dilated cerebral venous system. Magnetic resonance imaging (MRI) and Magnetic resonance venography (MRV) revealed a large congenital cerebral arterio-venous malformation (CAVM), in right cerebral hemisphere. Finally, he was expired 9 days after birth due to severe heart failure before any definitive treatment for closing CAVM could be done.
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Conclusion: CAVM are extremely rare vascular anomalies in newborns which may present occasionally as congestive heart failure in neonatal period. So after excluding other most common etiologies of heart failure such as structural heart defects, screening CAVMs should be done. Inspite of early diagnosis, usually they have extremely poor prognosis. |
Volume 75, Issue 6 (9-2017)
Case presentation: A 37-years old woman was admitted in an University Hospital of Mashhad University of Medical Sciences, Iran, in 2016 two weeks after genital surgery. The chief complaints were fever, painful tenderness, ulcerative lesion and inflammatory papule on surgical site and thigh. She suffered of fever despite received oral and then wide spectrum intravenous antibiotic therapy. Blood cultures and wound culture were negative. In addition to two deferent intravenous antibiotics, topical wound debridement was performed. Despite this course of treatment which did not improve the lesion, biopsy was performed. Histopathology features of biopsy specimen indicated prominent neutrophils mixed inflammation and lymphocytic vasculitis indicated as pyoderma gangrenous. The patient's medical history included associated ulcerative colitis from 18 years ago and she was under irregular oral receiving of Asacol (mesalamine). Intravenous corticosteroid therapy was administered which led to response of skin of right thigh and surgical site inflammation. After 6-months follow-up, the patient is still in good condition.
| Conclusion: Based on major variable clinical manifestations and no diagnostic serologic test of pyoderma gangrenosum, diagnosis of this disease is difficult. Increased awareness about PG and exclusion of other etiologies such as inflammatory and immunologic disease will aid in prompting of pyoderma gangrenosum diagnosis and proper management of the disease. |
Volume 76, Issue 11 (2-2019)
Case Presentation: A 26-year-old virgin woman with abdominal pain in the hypogastric region and lower and right quadrant of the abdomen, with gastric fullness and without nausea and vomiting referred to an emergent unit of Ghaem Hospital (an academic hospital of Mashhad University of Medical Sciences) in June 2017. In an ultrasound, a cyst of 95×105 mm in right adnexa of uterus was seen. Due to continued abdominal pain, laparotomy was performed for the patient. At the time of laparotomy, the ovaries, uterus and adnexa were completely normal. A 10×15 cm cyst was seen with a thin wall which adhering to the omentum and the small end of the stomach. Frozen section biopsy reported hydatid cyst.
Conclusion: Considering that hydatid cyst is endemic in Iran, the presence of cystic mass in the peritoneal cavity, hydatid cyst should be considered. To prevent complications and relapse, it is best to avoid cyst rupture during surgery as much as possible.
Volume 77, Issue 6 (9-2019)
Case presentation: A 52 years old man referred to Shohada-e-Tajrish Hospital, Tehran, with diagnosis of kidney stones candidate for percutaneous nephrolithotomy (PCNL). The patient underwent general anesthesia and after 40 minutes, the surgeon requested injection of tranexamic acid because of bleeding, but unintentionally the patient received 100 unites of crystalline insulin by nurse anesthesia. Vital signs were stable, the patient's blood glucose was 85 mg/dl and he had no sweat. Then the therapeutic intervention consisted of administering a bolus dose of 50 cc 50% dextrose water (DW) and then infusion of 50% dextrose water over that time. The patient was monitored for 10 hours in recovery and also received 1 mg of glucagon. The blood glucose was checked frequently. Fortunately, there were not any detectable hypoglycemic attacks (blood glucose less than 70 mg/dl) during that time. Throughout the first three hours in ICU, he suffered from severe hypoglycemic episodes and treated by DW 50% (bolus stat and infusion) and after stabilization of vital signs he transferred to ward.
Conclusion: The mortality of iatrogenic hypoglycemia is lower than other causes of hypoglycemia. However, on time diagnosis and aggressive treatment can prevent serious complications. In addition, proper communication between health care providers and precise checking of drugs labels before injection can dramatically decrease these events.
Volume 77, Issue 8 (11-2019)
Case presentation: Herein, we present a young girl who was referred to us in our tertiary level hospital, Firoozgar Hospital, Tehran, Iran, in october, 2017 just with pain and swelling of the left Bartholin gland. At first, it was suspected to be a cyst or abscess of Bartholin gland, she did not have any history or symptoms of infection on comprehensive physical examinations such as pneumonia, meningitis, Nevertheless, the ultimate diagnosis of AML was made after generalized and precise systemic examination and laboratory findings were done. According to the guidelines for the treatment of AML, systemic chemotherapy with multiple drugs was given immediately but unfortunately, she died due to severe septicemia which was resistant to broad-spectrum antibiotics and disseminated intravascular coagulation.
Conclusion: Based on our searching, this is the first case. Because we expected other more common symptoms of acute lukemia, systematic and precise generalized examination must be performed gently in all of the patients even in women just with genital symptoms as their chief compliant for instance, pain and swelling of Bartholin gland. Finally, not focusing just on the perineal site and detailed examination for all parts of the body may reveal an accurate diagnosis of the main underlying disease.
Volume 77, Issue 9 (12-2019)
Case Presentation: The patient was an unmarried 32-year-old man, who was hospitalized in Rasul-Akram Hospital in December 2018 for severe bleeding caused by GSM. He was diagnosed with type I bipolar disorder, which was treated from the late adolescence. Also, a history of drug abuse (amphetamine, opium, cannabis, alcohol) and multiple sexual relationships with different partners was reported. He abused tramadol tablets daily nowadays. His IQ was normal. He had lots of abrasions, scars and lacerations due to suicidal attempts in the past years. Two months before GSM, his auditory hallucinations about genital self-mutilation had started. He committed GSM after having sexual intercourse and was transferred to our hospital by an ambulance with severe hemorrhage, scrotum damage, and presentation of both testes, for which he received appropriate treatments.
Conclusion: It seems that a set of factors, such as patient's history of psychosis following bipolar disorder, substance abuse, mental retardation, multiple sexual relationships, and strong emotional relationship with the mother can contribute to self-injury that may lead to genital self-mutilation. It should be noted that genital self-mutilation cases are less likely to be reported in Iran, considering the cultural and religious background. Therefore, the pathology of this devastating phenomenon should be more thoroughly examined.
Volume 77, Issue 10 (1-2020)
Case Presentation: The patient was a 23-year-old woman with a gestational age of 29 weeks who presented with fever, headache, delirium, and cognitive impairment three days prior to admission following seizure. Early magnetic resonance imaging (MRI) showed an increase in the cortical signal in the temporal lobe and PCR assay for the detection of herpes virus was negative. Magnetic resonance venography also showed an increase in the left maxillary sinus with differential diagnosis of herpetic encephalitis. Considering the symptoms of the patient with a strong suspicion of herpes simplex encephalitis, the patient was treated with acyclovir, which was associated with improvement of clinical symptoms. Finally, normal delivery was performed after 36 weeks of gestation.
Conclusion: Given that herpes simplex virus encephalitis is a rare and potentially dangerous and complicated disease, it is imperative for a pregnant patient with atypical symptoms to be identified with an appropriate diagnostic and therapeutic measures.
Volume 77, Issue 11 (2-2020)
Case Presentation: The patient was a 3-year-old girl who was referred for prolonged fever (more than two weeks) and abdominal pain from another medical center, and she was hospitalized. In abdominal and pelvic ultrasound, splenomegaly was seen and in laboratory tests, she had hypereosinophilia. In the flow cytometry of bone marrow aspiration, the only finding was increased eosinophil level. Abdominal and thoracic a computerized tomography (CT) scans showed an increased size of para-aortic lymph nodes. On her examination, lymphadenopathy was present in the inguinal region. Therefore, a biopsy of an inguinal lymph node was performed to rule out lymphoma. Lymph node biopsy was negative for lymphoma. Fasciola serology was performed for the patient, and the stool exam was collected three times (for one day in between) to rule out parasitic disease, including Fasciola, etc. Due to weakly positive serology Fasciola hepatica, triclabendazole was started for the patient (it was given in two doses, 12 hours apart), despite the absence of Fasciola parasitic eggs in her stool. During hospitalization, the patient’s fever was stopped and by starting the use of mentioned drug, eosinophilia was reduced. The patient received a complete improvement in the follow-up.
Conclusion: In patients with hypereosinophilia, parasitic diseases such as fascioliasis should be considered even if the fecal specimen is negative for Fasciola eggs.
Volume 77, Issue 12 (3-2020)
Case Presentation: Two patients (1 male, 1 female) with an average age of 50 years underwent surgery, due to the history of tooth extraction. They had resorption of jaw bone for implant surgery. The teeth were 11 and 37. This study was performed in Al-Mortaza's Clinic, Qom province, Iran, from May 2016 to January 2017. 10 ml of blood were taken individually and centrifuged at 1300 revolutions per minute (rpm) for 8 minutes to separate the platelet-rich fibrin. Then, platelet-rich fibrin was combined with allogeneic bone to form sticky bone. Sticky bone prepared during implant surgery, with implant inside the patient's jaw bone was used.
Conclusion: The use of sticky bone to stimulate and induce bone resorption in toothless area was associated with increased implant's success. Sticky bone due to multiple growth factors, such as TGF-β1 and VEGF, usability is an appropriate and efficiency method for stimulation of bone resorption.
Volume 78, Issue 1 (4-2020)
Case presentation: A 51-year-old woman, gravida 2, para 2, presented with complaint of abnormal vaginal bleeding and discharge, with no abnormal finding in cervical cytology and sonography, so uneventfully a diagnostic error had happened in the assessment of her. After several months and multiple different treatment, the patient referred to the Oncology Department of Obstetrics and Gynecology Center, Ghaem Hospital, Mashhad, Iran in May 2017. Re-assessment was performed by biopsy and imaging, and the final pathologic diagnosis of diffuse large B-cell non-Hodgkin's lymphomas was confirmed.
Conclusion: Primary cervical lymphoma is an uncommon malignancy; the diagnosis could be missed simply by low suspicious due to low accuracy of Pap smear and imaging in this situation. So an accurate evaluation and pelvic examination, high suspicious and close communication between clinician and pathologist are needed. By timely diagnosis of patient in early stage and appropriate approach, the prognosis could be excellent most of the time.
Volume 78, Issue 2 (5-2020)
Case Presentation: A 59 years old man, known case of diabetic nephropathy with a serum creatinine level of 1.2 mg/dL and proteinuria of about 3300 mg in 24 hours referred to Hypertension Clinic of Imam Khomeini Hospital Complex, Tehran University of Medical Sciences, for evaluation of resistant hypertension. The patient was under treatment by losar-H, diltiazem, and furosemide, which are not appropriate combinations of antihypertensive agents. The ambulatory blood pressure monitoring (ABPM) of the patient revealed daytime and nighttime hypertension and also the non-dipping status of blood pressure. Dihydropyridine CCB was prescribed in combination with RAS blocker (Exforge 50 mg/ 160 mg) and the diuretic was changed to thiazide-like agents. In the next step, spironolactone and carvedilol were added for better control of blood pressure. The patient referred to the sleep clinic for polysomnography and evaluation of obstructive sleep apnea. Following three months prescription of these medications, ABPM revealed relatively appropriate control of blood pressure, so the average 24-hour blood pressure decreased from 157/91 mm Hg to 136/83 mm Hg.
Conclusion: The complications and mortality of hypertension would be increased dramatically in patients with resistant hypertension. So, an appropriate diagnostic and therapeutic approaches should be considered for these patients. Choosing appropriate diuretic agents would be important in the management of resistant hypertension.
Volume 78, Issue 9 (12-2020)
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Background: Laryngeal tuberculosis (TB) is one of the most common complications of pulmonary tuberculosis, which increased for various reasons such as more prevalence of immune system suppression diseases, increasing the survival of the elderly, immigrants from high-risk areas and the appearance of atypical or resistant organisms over the past two decades, and its clinical pattern changed compared to the past. In contrast to the past that patients complained about dyspnea, coughing and other symptoms, today, the main complaints of these patients are hoarseness and Odynophagia. In reality, the prevalence of laryngeal TB without pulmonary manifestation was increased these days. The purpose of this study was to evaluate these symptoms in laryngeal TB.
Case Presentation: In this case report study, we examined a 77-year-old man who was admitted to the otorhinolaryngology department of Shafa hospital of Kerman (an academic hospital of Kerman University of Medical Sciences), Iran in April 2019 due to hoarseness since 6 months before admission and the results of evaluation confirmed the presence of laryngeal tuberculosis in this patient. The patient treated with Anti-TB drugs (Isoniazid, Rifampicin, Pyrizinamide, and Ethambutol) according to standard protocol and responded appropriately to this medication regimen. Conclusion: Although laryngeal tuberculosis is a rare disease, it still occurs. Therefore, by increasing the incidence of tuberculosis, changes in the clinical pattern and its spreading mechanism, physicians should always be aware of the unusual clinical features of laryngeal tuberculosis and the possibility of developing it for early diagnosis and treatment. It should be considered as a differential diagnosis in all laryngeal diseases to prevent the complications of the disease and decrease the risk of transferring it to other people. |
Volume 78, Issue 10 (1-2021)
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Case Presentation: A 24-years-old woman was referred to the academic dermatology clinic of Boo- Ali SINA Hospital in Sari, Iran, in November 2019 with a 9-month complaint of verrucous and pruritic plaque in the genital area that previously misdiagnosed as wart with no response to treatment. The dermatologist requested a review of the previous pathology documents for diagnosing dermatophytosis. The pathologists reexamined the relevant paraffin-embedded skin tissue block of the patient. By Periodic acid-Schiff staining, some fungal elements had been found in the horny layers of the epidermis, so dermatophytosis was confirmed. Based on this diagnosis, terbinafine was prescribed and the patient responded well to this treatment after four weeks.
Conclusion: Although the occurrence of genital dermatophytosis with the verrucous appearance and without involvement of groin is a rare finding, it should be considered in the differential diagnosis. In each case suspected of genital dermatophytosis, direct examination or fungal culture for definite diagnosis and prevention of delay in the appropriate treatment is required. |
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