Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 24 50 57 Fatemeh Rahimi-Sharbaf Fellowship of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Maternal Fetal and Neonatal Research Center, Yas Hospital, Tehran University of Medical Sciences, Tehran, Iran. Mahboobeh Shirazi Fellowship of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Maternal Fetal and Neonatal Research Center, Yas Hospital, Tehran University of Medical Sciences, Tehran, Iran. maasoumeh saleh Perinatology Fellowship, Department of Obstetrics and Gynecology, Yas Hospital, Tehran University of Medical Sciences, Tehran, Iran. Fatemeh Golshahi Fellowship of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Maternal Fetal and Neonatal Research Center, Yas Hospital, Tehran University of Medical Sciences, Tehran, Iran. 2021 04 15 2021 11 09 One of the most common causes of fetal anemia is red cell alloimmunization. The most common routes of maternal sensitization are via blood transfusion or fetomaternal hemorrhage. Antibodies can cross the placenta during pregnancies in alloimmunized women and, if the fetus is positive for these specific erythrocyte surface antigens, result in hemolysis of fetal erythrocytes and anemia. This in turn, can lead to potentially disastrous consequence for the fetus, such as Hydrops Fetalis (HF), a high-output cardiac failure syndrome. The standard treatment in fetuses with anemia is intrauterine transfusion (IUT). Mirror Syndrome (MS) is a rare condition in pregnancy, in which maternal edema in pregnancy is seen in association with severe fetal and/or placental hydrops. The pathogenesis, although not well established, mimics trophoblastic damage and maternal vascular endothelial dysfunction, as is also observed in preeclampsia, and hence, the two conditions may have a similar clinical presentation [1]. The clinical manifestations of this disease are complex. It is easily underdiagnosed and timely intervention is needed to prevent fetal and maternal morbidity. MS can be reversible when the underlying factors are identified and modified [2, 3]. If correction of the underlying fetal abnormality is not possible, the consensual treatment is to deliver the hydropic fetus and placenta, with improvement of the maternal condition shortly thereafter. We report a case of HF and MS secondary to Rh-D alloimmunization that did not respond to IUT. https://crcp.tums.ac.ir/index.php/crcp/article/view/516

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 58 60 Ghasem Farahmand Department of Neurology, Imam Khomeini Hospital Complex, Tehran University of Medical Sciences, Tehran, Iran. Hannah Magrouni Department of Neurology, Imam Khomeini Hospital Complex, Tehran University of Medical Sciences, Tehran, Iran. Vahid Zolfaghari Iranian Center of Neurological Research, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran. Sina Gharehjeh Department of Neurology, Imam Khomeini Hospital Complex, Tehran University of Medical Sciences, Tehran, Iran. sakine ranji -Burachaloo Iranian Center of Neurological Research, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran. 2021 04 18 2021 06 16 A 68- year – old male was admitted to ER of Referral Imam Khomeini hospital with abrupt commence of fever accompanied with debilitating non-radiating headache, photophobia, ataxia and cluster of confluent vesicles on right side of forehead with swelling of right eyelid. All his symptoms started after receiving Botulinum injection on the same week before admission and he had never had similar manifestations beforehand.His medical history was remarkable for hyperlipidemia, hypertension and right side Blepharospasm for which he received Botulinum injection every 6 months. He was on medication for the rest of his medical problems. He was not on any immunosuppressive medication and did not suffer from any disease weakening the immune system. His family history was unremarkable for any similar problems.In medical exam, he was ill but not toxic, his vital signs were blood pressure of 135/80 mmHg, heart rate of 76, respiratory rate of 17 and oral temperature of 38.5Ĉ. He had vesicles on his right forehead. Heart sounds were regular without murmur. Lungs were clear. Abdominal examinations were inconspicuous.In neurological examinations, he was confused and disoriented to time and place. Cranial nerves were without any pathological findings except for positive Marcus Gunn of affected side and blurring of right optic disk margin. No muscle atrophy was seen. Muscle force showed no weakness. He was ataxic. Sensory examination was normal. Reflexes were checked and were within normal limits and symmetric.Computer tomography (CT) scan of head was unremarkable and his MRI scan did not reveal any information compatible with his symptoms.Due to the sudden onset of fever with headache and confusion, encephalitis was suspected and empirical therapy with antibiotics was started and LP was performed which indicated pleocytosis in CSF. According to the vesicles on the skin and with suspicion of Varicella encephalitis, PCR was sent for diagnosis of VZV. Subsequently, his condition got dramatically better and symptoms diminished after acyclovir was started. https://crcp.tums.ac.ir/index.php/crcp/article/view/519

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 61 64 Maryam Saeedi Department of Pediatrics, Division of Neonatology, Children’s Medical Center, Tehran University of Medical Sciences, Tehran, Iran. Razieh Sangsari Department of Pediatrics, Division of Neonatology, Children’s Medical Center, Tehran University of Medical Sciences, Tehran, Iran. kayvan Mirnia Department of Pediatrics, Division of Neonatology, Children’s Medical Center, Tehran University of Medical Sciences, Tehran, Iran. 2021 04 23 2021 08 02 After the Ministry of the health of Iran officially announced widespread of COVID-19 on 19 February 2020, our attention focused on novel coronavirus. In our case, a2-day- old neonate shows symptoms of sepsis. The main presentation was hypothermia and desaturation. The mother was COVID-19 positive with an active cough. The PCR of the neonate was negative. We don’t claim that the neonate is affected by COVID-19, but this may be an atypical form of sepsis in neonates with positive mothers following COVID-19. A2-day- old female neonate with a gestational age of 40 weeks and a birth weight of 2370 grams was born via the cesarean route from a mother who was a 34-year-old primigravida woman without any history of disease during pregnancy. Two days before delivery, the mother had malaise and dry cough. She was diagnosed as a COVID-19 positive case based on RT-PCR after delivery. On the second day after birth, the parents brought the baby to the emergency room of the children’s medical center hospital with complaints of poor feeding, poor sucking and decreased urination. Physical examination revealed the following signs; hypothermia; T=36℃, diminished primitive reflexes, hypotonia, and oxygen desaturation until 85% without respiratory distress that increased to 98% with oxy hood. We admitted and treated her early-onset sepsis and discharged in excellent condition.Early-onset sepsis as defined is a clinical state that is transferred from mother. The presentations in our case maybe a new form of clinical sepsis following a mother with COVID-19. We don’t claim that our case is COVID-19 positive but in neonates with affected mother’s insidious symptoms should be in concern. https://crcp.tums.ac.ir/index.php/crcp/article/view/520

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 65 67 Mehmet Yildirim Department of General Surgery, University of Health Sciences, Izmir Bozyaka Education and Research Hospital, Turkey. Asuman Argon University of Health Sciences, Izmir Bozyaka Education and Research Hospital, Depertmant of Pathology, Turkey. Ahmet Çekiç University of Health Sciences, Izmir Bozyaka Education and Research Hospital, Department of General Surgery, Turkey. 2021 05 03 2021 05 23 Ectopic liver is defined as the presence of normally organized liver tissue outside its normal location. It is frequently found in the gallbladder. Ectopic liver is normally as- ymptomatic and only discovered incidentally. We report a case of 51- year- old wom- an with ectopic liver and we also review the characterization of this rare entity .This report adds another case to the very rarely reported ectopic liver located in the gall- bladder. https://crcp.tums.ac.ir/index.php/crcp/article/view/527

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 68 74 Salman Ghaffari Associate professor of orthopedic surgery, Orthopedic Research Center, Mazandaran University of Medical Sciences, Sari, Iran. Mehran Razavipour Associate professor of orthopedic surgery, Orthopedic Research Center, Mazandaran University of Medical Sciences, Sari, Iran. Soroosh Fateh Resident of orthopedic surgery, Orthopedic Research Center, Mazandaran University of Medical Sciences, Sari, Iran. 2021 05 15 2021 05 30 We report a rare case of bilateral subtrochanteric femoral fracture related to prolonged treatment with Alendr onate. Bisphosphonates are the most common drugs used for treatment of osteoporosis and prevention of the risk of osteoporosis-related fractures. Atypical femoral fractures (AFF) have been reported in patients who have been on long term treatment with bisphosphona te especially Alendr onate. our case was a 68- year- old woman who sustained bilateral subtroc- hanteric femoral fracture after a simple falling down. She had a past medical history of osteoporosis and had been on treatment with alendronate for 5 years. Radiographs showed bilateral fractures in femur subtrochanteric area. We decided to use double plating technique and we used angled blade plate and locking plate (LCP). Alendronate was discontinued and Teriparatide was started. Careful evaluation of hip or thigh pain in patients who are on prolonged courses of bisphosphonates is essential. Our method of fixation with double plating can be used in pa tients with poor bone quality. https://crcp.tums.ac.ir/index.php/crcp/article/view/541

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 75 78 Alireza Akhavan Rezayat Department of Urology, School of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran. Amir Yarahmadi Department of Clinical Biochemistry, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran. AND Transplant Research Center, Shiraz University of Medical Sciences, Shiraz, Iran. Atena Aghaee Nuclear Medicine Research Center, Mashhad University of Medical Sciences, Mashhad, Iran. Farid Zeynali Kidney Transplantation Complications Research Center, Mashhad University of Medical Sciences, Mashhad, Iran. Salman Soltani Kidney Transplantation Complications Research Center, Mashhad University of Medical Sciences, Mashhad, Iran. 2021 05 20 2021 10 03 Ureteral fibroepithelial polyps (UFPs) are rare abnormality that may cause ureteral obstruction. Herein, we report a 35- year- old woman with flank pain, dysuria, frequency and transient total gross hematuria. A mass in the right ureter was reported on imaging and in endoscopic evaluation, small polypoid mass protruding from the right ureteral orifice in the bladder, and a huge ureteral mass originating from proximal ureter was observed. A right subcostal flank incision was done, a huge polypoid mass with a segment of ureteral was excised, and end-to-end anastomosis of the ureter was done. Histopathological examination showed a UFP without malignancy. https://crcp.tums.ac.ir/index.php/crcp/article/view/543

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 79 81 Reza Erfanian Otorhinolaryngology Research Center, Tehran University of Medical Sciences, Tehran, Iran. Farrokh Heidari Otorhinolaryngology Research Center, Tehran University of Medical Sciences, Tehran, Iran. Saeed Sohrabpour Otorhinolaryngology Research Center, Tehran University of Medical Sciences, Tehran, Iran. Saeed Shakiba Otorhinolaryngology Research Center, Tehran University of Medical Sciences, Tehran, Iran. 2021 05 21 2021 12 01 A 35- year- old woman presented with massive enlargement of the tongue. She had suffered this problem since a week ago when her tongue became enlarged with a sudden onset and eventually did not fit her mouth. Chronic tongue enlargement since childhood is noted. Patient had history of mental problems, namely cerebral palsy, and as she is not able to walk, she uses a wheelchair. Furthermore, due to severe scoliosis, right lung was atelectatic.Soon after initial assessments, she was carried to surgery room and as she could not lie down to supine position, was intubated in sitting position with a rigid laryngoscope. During the whole process, patient was sedated. Afterwards, tracheostomy was per- formed.After 5 days, no improvement in the size of tongue was observed. Diagnosis was revised in a medical commission and despite inflammatory pathology, the bulging tongue was resected.After recovery, patient could bear oral feeding and was dismissed with good general health. After a week, the devised airway created by tracheostomy was removed with- out any complication. During one-year follow up, no sequelae were observed. https://crcp.tums.ac.ir/index.php/crcp/article/view/544

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 82 85 katayoun katebi Department of Oral and Maxillofacial Medicine, Faculty of Dentistry, Tabriz University of Medical Sciences, Tabriz, Iran. Ali Taghavi Zenouz Department of Oral and Maxillofacial Medicine, Faculty of Dentistry, Tabriz University of Medical Sciences, Tabriz, Iran. Mohammad Ali Ghavimi Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Tabriz University of Medical Sciences, Tabriz, Iran. 2021 05 29 2021 12 01 Methotrexate is an anti metabolite drug that is used for treating several autoimmune diseases in low doses. It has been reported that oral ulcers occur in 11-17% of patients receiving low doses of Methotrexate. The purpose of this article is to report the Meth- otrexate-induced oral ulcers and their effects on patient’s quality of life. The patient was a 69-year-old woman with a complaint of oral ulcers. She was taking alendronate, Methotrexate, and hydroxychloroquine for osteoporosis and rheumatoid arthritis dis- eases. The possibility of bisphosphonate-induced osteonecrosis was recognized due to necrotic ulcers with mandibular bone exposure in the first therapy session. Then, a new necrotizing ulcer on the hard palate appeared in the next session. Methotrexate consumption was discontinued and folic acid was initiated. The oral ulcers were fully recovered in three weeks. Methotrexate-induced mucositis should be considered in the differential diagnosis of patients with oral ulcers who receive low-dose Metho- trexate. https://crcp.tums.ac.ir/index.php/crcp/article/view/547

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 86 90 Marzieh Pazoki Department of Pulmonary Medicine, Sina Hospital, Tehran University of Medical Sciences, Tehran, Iran. Elham Nazar Department of Pathology, Sina Hospital, Tehran University of Medical Sciences, Tehran, Iran. Behnaz Jahanbin Department of Pathology, Imam Khomeini Complex Hospital, Tehran University of Medical Sciences, Tehran, Iran. 2021 05 31 2021 07 14 Benign metastasizing leiomyoma of the lung which is categorized by the growth of uterine leiomyoma in the lung is an extremely exceptional disease.We report a 39-year-old woman who had a previous history of uterine leiomyoma. Chest computed tomography (CT) scan exhibited round nodules well-defined margins in both lungs. Thoracoscopic partial resection of the lung was done. Pathological and immunohistochemical examination showed that neoplastic tissue composed of spindle-shaped smooth muscle cells, compatible with uterine leiomyoma. Thus, these tumors can be reflected to be lung metastases from a uterine leiomyoma (benign metastasizing leiomyoma).It is an uncommon condition but it would be careful in women of re- productive age with a past medical history of uterine leiomyoma, who existing with pulmonary nodules. https://crcp.tums.ac.ir/index.php/crcp/article/view/549

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 91 96 Reza Ershadi Department of Thoracic Surgery, Valiasr Hospital, Tehran University of Medical Sciences, Tehran, Iran. 2021 06 02 2021 07 03 Pulmonary echinococcosis most frequently occurs in the right and both lower lobes. Children are more likely to develop pulmonary rather than hepatic echinococcal cysts. Some evidence suggests that echinococcal cysts develop more rapidly in the lungs of children than those of adults, which may explain the more common appearance of pulmonary cysts in children. Younger patients have larger cysts owing to their greater tissue elasticity. Large pulmonary hydatid cysts are critical because they pose a higher risk of rupture. Herein, we present an eight-year-old girl presented with severe respi- ratory distress due to giant hydatid cyst of right lung with shift of the mediastinum at left. The lesion displaced the trachea at left. The patient was successfully treated by parenchymal-saving operation. In conclusion, Giant hydatid lung cysts represent a distinct clinical entity most frequently encountered in children and adolescents with more serious symptoms. Although non-complicated giant hydatid cysts have a good prognosis regardless of their size and can be safely treated by parenchyma-preserving surgery. https://crcp.tums.ac.ir/index.php/crcp/article/view/552

Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 7 2 2022 09 26 97 100 Zahra Nafei MD, Children Growth Disorder Research Center, Shahid Sadoughi University of Medical Sciences, Yazd, Iran. Marjan Jafari Department of Pediatrics, Shahid Sadoughi University of Medical Sciences, Yazd, Iran. 2021 12 19 2022 03 14 Congenital insensitivity to pain with anhidrosis (CIPA) is the subtype four of hereditary sensory and autonomic neuropathy (HASN IV), caused by a defect in the NTRK1 gene and presenting early in life. We report a ten-year-old boy with gait problems and an inability to put weight on his feet. Four days before the visit, a trauma entered his right knee during a football match. He had swelling and erythema in his right knee and multiple scars on his torso and limbs. Magnetic resonance imaging (MRI) offered osteomyelitis and soft tissue periosteal abscess. The patient underwent an operation, and based on the pathology results, myositis ossificans (MO) was reported. Moreover, he was treated with antibiotics and supportive measures and was discharged with partial recovery. According to our knowledge, this is the first report of MO due to recurrent trauma in children with CIPA syndrome. https://crcp.tums.ac.ir/index.php/crcp/article/view/642