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Журнал клинических случаев

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Объем 9, Проблема 11 (2019)

История болезни

A Case of Partial Intersphinteric Rectal Resection in a Patient with Undefined Intestinal Polyposis

Costanzo A, Rampulla V and Petrelli F

The intersphinteric technique was initially described by Schiessel et al. in 1994. It has gradually spread to Europe and Asia: it has progressively established itself also through laparoscopy and robotics. The largest published series are Asian, and the last review is by Japanese author Kazuo Shirouzu et al. Indications: adenocarcinoma of the low rectum “c” or ycT1-T3N0/+, within 1 cm of the anorectal ring, without involvement of the intersphincter plane. Contraindications: G3-G4 tumours, cT4 (external sphincter invasion, puborectalis muscle, prostate, vagina), distant metastases, tumour fixity to rectal exploration, sphincteric hypotonia, psychiatric comorbidity, moderate-severe cardio-respiratory, renal or hepatic comorbidity.

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A Peculiar Case of Hearing Loss Followed Years Later by Tinnitus and Vertigo

Marcelli V and Tortoriello G

Vestibular paroxysmia (also known as disabling positional vertigo) is a clinical syndrome generated by a symptomatic neurovascular compression of the eighth cranial nerve. Although doubted by some authors, this syndrome must be suspected in presence of brief spells of positional vestibular symptoms associated with temporary or permanent but worsening cochlea-vestibular symptoms and signs, not explained by other diseases. We report a case of a 20-year-old girl affected by permanent sensorineural hearing loss on the right since the age of 4-5 years, who subsequently developed intermittent low pitch tinnitus on the right ear and spells of vertigo or dizziness generated by position or physical activity. Angio-MRI of the brain showed a singular neurovascular contact between the right vertebral artery and the right eighth cranial nerve. The excellent response to carbamazepine confirms the presence of this syndrome.

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Fulminant Type 1 Diabetes Mellitus Associated with Excessive Alcohol Use: A Case Report

Wu M, Zou Y, Zhu Z, Li L, Shi-bo L and Huang S

We present a case of fulminant type 1 diabetes that is associated with excessive alcohol use. The patient
was admitted due to our hospital due to refractory diarrhoea. His blood glucose level was 24.6 mmol/L and he
was positive for urine ketone bodies. Laboratory examinations meets all the diagnostic criteria for fulminant type 1
diabetes. Immediate treatment actively reduced the blood glucose level and corrected his electrolyte disturbance
and acid-base imbalance. Fulminant type 1 progresses rapidly and the prognosis is extremely poor. Therefore, early
diagnosis and treatment are important to the outcomes.

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Jugular Vein Thrombosis and Recurrent Pleural Effusions in OHSS

Chahal R and Pakrashi T

Background: Ovarian hyperstimulation syndrome remains a significant cause of morbidity in ovulation induction. It may be further complicated by thromboembolism and pleural effusions.

Case Report: A 30-year-old nulligravida underwent ovarian stimulation with OCP/Antagonist protocol for in vitro fertilization. Estradiol levels peaked at 2411 pg/mL. Post-stimulation course was complicated by recurrent pleural effusions and an occlusive right internal jugular vein thrombosis. The patient underwent repeated thoracenteses with a drainage of a total of 8350 cc of pleural fluid. The patient received therapeutic anticoagulation and delivered a viable infant at term. Imaging at six weeks postpartum revealed a chronic, non-occlusive deep venous thrombosis involving both the right internal jugular and right subclavian veins.

Conclusion: OHSS results in dynamic changes that contribute to thrombosis and massive fluid shifts. Further research is needed to optimize prevention strategies.

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Two Cases of Scalp Eccrine Porocarcinoma with Review of Literature

Won JH, Je-Ho M and Kim BJ

Eccrine Porocarcinoma (EPC) is a rare malignant adnexal neoplasm arising from the eccrine sweat glands. Head and neck, lower extremities are the most common primary locations for the development of EPC. Scalp is a rare site of involvement, with less than 20 cases reported so far. This study aims to report two cases of scalp EPC, which showed different clinical courses that could be predicted by histopathologic features. In one case, a 62-year-old woman with EPC was treated by local excision with 30 mm margin. A mitotic count of 16/10 high-power field, invasion depth >7 mm, and infiltrative tumor margins were observed, which are suggested as poor prognostic factors. Pulmonary metastasis and local recurrence occurred rapidly after 4 months of disease-free interval. In the other case, local excision with 10 mm margin was performed in a 45-year-old woman with EPC. Poor prognostic histopathologic features were absent, and there was no evidence of the disease during the 2-year follow-up period.

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Can Endoscopic Submucosal Dissection Combine with Variceal Ligation for Early Gastric Cancer and Esophageal Varices

Chen Y, Zhu Y, Chen F and Wang L

A liver cirrhotic patient presented with hematemesis was endoscopically found with esophageal varices accompanied with early gastric cancer. Endoscopic submucosal dissection and endoscopic variceal ligation were performed in one time. The early gastric cancer lesion was removed en bloc and varices banded without complication of bleeding and perforation. This case indicates that the strategy of performing the endoscopic mutation submucosal dissection and endoscopic variceal ligation at the same time may be safe and effective for the patient with decompensated stage liver cirrhosis.

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Wernicke Encephalopathy after Sleeve Gastrectomy: A Case Report

Nowak M, Niedziela N, Kubicka-Baczyk K and Adamczyk-Sowa M

The number of obese patient’s increases, and bariatric procedures have become more common. The total number of these procedures in Poland was 1958 in 2016. Malabsorption, rapid weight loss and vomiting can predispose patients to nutritional deficiencies following surgery. After bariatric surgery, the majority of vitamin and mineral deficiencies seem to be asymptomatic. However, in some cases both reversible and irreversible neurological symptoms may be observed. Punchai et al. observed that the incidence of neurological complications due to vitamin B deficiency was 0.7%. Wernicke Encephalopathy (WE) is an acute disorder resulting from thiamine deficiency and is characterized by neuropsychiatric symptoms such as ataxia, ophthalmoplegia and confusion. We present a case of a 29-year-old woman who developed WE after sleeve gastrectomy.

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An Unprecedented Case of Myasthenia Gravis Induced by Binimetinib

Alabdali M, Bril V, Morgan E, Pereira A and Maurice C

Low-grade serous ovarian cancer, representing 10% of all ovarian cancer diagnoses, is renown for its poor response rate to chemotherapy, despite its indolent course. The MILO study is an international, randomized phase III trial comparing binimetinib, a targeted agent, to standard chemotherapy in patients diagnosed with low-grade serous ovarian cancer. This oral drug is designed to down-regulate cancer cell proliferation and survival through the inhibition of MEK-1 and MEK-2, involved in the RAS/RAF/MEK/ERK signal cascade. We present the case of a 77-year-old female started on binimetinib in November 2014, in a clinical trial context. She developed head drop syndrome correlating exactly with the administration of this drug. The clinical presentation, evolution and electrodiagnostic studies were compatible with myasthenia gravis. Anti-acetylcholine receptor antibodies were not detected. Improvement was noted immediately after the MEK inhibitor was withdrawn.

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Primary Undifferentiated Pleomorphic Sarcoma of the Breast in a Male Patient: A Case Report and a Review of the Literature

Jaba S, Belhabib S, Kebdani T, Elkacemi H, Majjaoui S and Benjaafar N

Male breast cancer is a rare disease, accounting for less than 1% of all breast cancers. Like soft tissue sarcomas, breast sarcomas are a heterogeneous group of several subtypes: osteosarcoma, liposarcoma, fibrosarcoma, leiomyosarcoma, angiosarcoma, rhabdomyosarcoma, pleomorphic sarcoma, and sarcomas of uncertain differentiation. Undifferentiated pleomorphic sarcoma constitutes less than 5% of all sarcomas in adults. It is most frequently located in the extremities but has also been reported in the retroperitoneum and the abdomen, However, Localization in the breast is extremely rare, especially in patients with no history of radiation. In this report, we describe an unusual case of undifferentiated pleomorphic sarcoma of the breast in 60-years-old men who presented a tumor measuring over 4 cm with pain in the left breast. He noticed the mass 3 months previously. Breast ultrasound revealed a left tumor. The patient underwent total mastectomy without axillary lymph node dissection. Based on examination of the excised tumor, the initial pathologic diagnosis was atypical spindle-shaped cells with uncertain malignant potential. Histological findings with immunomarkers led to the final diagnosis of undifferentiated pleomorphic sarcoma. Following the simple mastectomy, the patient was given adjuvant hypofractionated radiotherapy. The follow up at 42 months was uneventful. This case highlights a rare and interesting variant of primary breast sarcoma presenting in a male patient. A review of the available literature with evaluation of the etiology, prognostic factors and treatment modalities of pleomorphic sarcoma of the breast are discussed.

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Propylthiouracil Treatment may be Associated with Agranulocytosis and Hepatotoxicity

Kemec Z, Yesilbagdan H, Gurel A, Demir M, Kacmaz H and Balci AM

Agranulocytosis is a rare adverse effect of propylthiouracil. The usual duration of treatment prior to the onset of agranulocytosis is approximately 1 to 4 months, and can be as long as 1 year. Agranulocytosis together with hepatotoxicity is an extremely rare side effect of propylthiouracil treatment. We present an unprecedented case of a hyperthyroidism patient who showed a strong reaction to propylthiouracil with obvious agranulocytosis and hepatotoxicity which developed approximately one month after administration. The patient’s symptoms and laboratory abnormalities resolved following withdrawal of offending drug. This case, along with a literature review, is aimed to increase the awareness of physicians of sudden onset of agranulocytosis and hepatotoxicity due to propylthiouracil.

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