Unusual Presentation of Pulmonary Interstitial Glycogenosis: A Case Report Study

Introduction: Pulmonary interstitial glycogenosis (PIG) is a kind of children’s interstitial lung disease (ChILD). This is exclusively limited to neonates and infants. Often, PIG is diagnosed in the lung biopsy in a short time after birth (usually < 6 months). Most cases of PIG in infants are symptomatic within the first days to weeks of life. PIG expresses itself with diverse clinical symptoms such as tachypnea and hypoxia and may lead to acute respiratory failure in neonates. Case Presentation: In this case report study, we presented a 1.5-year-old boy with the chief complaint of stage 4 clubbing in fingers and toes. Mild chest deformity was observed in his physical exam. No evidence of respiratory and cardiac complications was observed. Initial lab tests and further specific studies were normal. His parents did not mention the history of any diseases in this patient. His chest X-ray (CXR) showed hyperinflated lungs, diffuse bilateral interstitial infiltration, and hazy opacities. Ground glass opacities (GGO) and interlobular septal thickening and cystic changes with reversed halo sign in both lungs were observed in his chest computerized tomography (CT). Finally, pulmonary biopsy showed a high level of glycogen-laden mesenchymal cells in the interstitium of alveoli, and vimentin-positive interstitial infiltration in lung parenchyma confirmed the PIG diagnosis. Conclusions: The new manifestation of PIG, which has been reported in this case, can be beneficial for future diagnoses of PIG.

Complex Fibroadenoma: a Cystic Neoplasia Report Case

Fibroadenoma is the most common breast tumor in young woman. It is considered a non-malignant tumor, showing hyaline component and a biphasic stroma and epithelial process, thus, it is similar to phyllode tumor revealing intralobular stroma. The cystic changes in a complex fibroadenoma may mimic a carcinoma, therefore it may represent some problems in images interpretation, and it may enlarge the risk of cancer development. This is a description of a case report with literature review. A 21-year-old female patientwith no history of breast cancer in family, presented a palpable lump in her right breast and clinical examination revealed a mobile, firm, circumscribed lesion in the right inner quadrant, measuring around 8 x 7 cm. An excision had been done under a stereotactic surgery and the structure revealed a nodular surface, measuring 8 x 6.5x 5.5cm. Cut section revealed heterogeneous aspect: cysts with colloid content and white-gray compact fibroblastic areas. Histopathological examination shows hypocellular stroma and few hypercellular areas, without atypia. Epithelial components presenting proliferation of intracanalicular and pericanalicular pattern ducts. Cystic proliferation with epithelial lining and apocrine characteristics without atypia. Because of the variation may be present inside the lesion it is difficult to establish the diagnosis without a biopsy, and a histopathological analysis. Moreover, it is necessary to know the microscope difference between fibroadenoma and the other sort of lesions; furthermore, that heterogeneity represent why that tumor is considered complex Keywords: Women's Health. Breast Neoplasms. Breast Cyst. ResumoFibroadenoma é o tumor de mama mais comum em mulheres jovens. É considerado um tumor não-maligno, apresentando um componente hialino e um estroma bifásico, e processo epitelial, por isso, é similar ao tumor filoide, revelando estroma intralobular. As alterações císticas no fibroadenoma complexo pode mimetizar um carcinoma, isso acarreta alguns problemas na interpretação das imagens, e pode aumentar o risco de desenvolvimento de câncer. Trata-se de um relato de caso de paciente de 21 anos de idade, sem histórico de câncer de mama na família, apresenta uma massa palpável em seio direito. Exame clinico revelou lesão móvel, firme, circunscrita no quadrante interno do seio. Excisão por cirurgia estereotáctica e a estrutura se mostra com superfície multinodular, medindo 8 x 6,5 x 5,5 cm. com aspecto heterogêneo, cistos com conteúdo coloide, e áreas fibrobásticas branco-acinzentada. Exame histológico demonstra-se estroma hipercelular, com áreas hipocelularizadas, sem atipia. Componentes epiteliais apresentando proliferação intercanalicular e intracanicular dos ductos. Proliferação cística com revestimento epitelial e características apócrinas sem atipia. Por conta da variação presente na lesão, pode ser difícil a estabelecimento de um diagnóstico se biópsia e análise histopatológica. Além disso, é fundamental o conhecimento das diferenças microscópicas entre o fibroadenoma e outros tipos de lesões; além disso, a heterogeneidade representa o motivo do tumor ser considerado complexo. Palavras-chave: Saúde da Mulher. Neoplasias da Mama. Cisto Mamário.

Short-term follow-up HRCT Chest of COVID-19 survivors and association with persistent dyspnea

Background There is an increasing concern that a proportion of the survivors of COVID 19 might develop fibrotic and/or other non-reversible lung changes. The aim of this retrospective study was to review the imaging findings of HRCT of lungs in a cohort of COVID 19 survivors, coming for short-term clinical follow-up and to assess the relation of the observed HRCT changes with the presence of dyspnea. Results In total, 40 patients with residual CT findings were included in this study with a mean age of 44.3 years and male: female ratio of 3:2. The presence of residual ground-glass opacities (85%) and reticular opacities (80%) was the most common findings. 25% of the cases had cystic changes in their lung. The presence of dyspnea was significantly associated with male sex and a history of smoking. On HRCT, the presence of cystic changes, involvement of > 10 lung segments, and an HRCT severity score > 7 were significantly associated with dyspnea. Conclusion Survivors of COVID 19 demonstrate persistent changes in the lung on HRCT. We recommend that a follow-up HRCT should be performed in these patients to identify those with post-COVID sequelae.

Cytomorphological features of papillary thyroid carcinomas with the development of radioiodine-resistance

Background. The problem of diagnosis and treatment of papillary thyroid cancer (PTC) are radioiodine resistant metastases (RIRM). An urgent issue is the possibility of their early prediction and diagnosis based on the detection of cytomorphological features of the PTC and its metastases, which correlate with the development of radioiodine (RI) resistance. Purpose – detection of cytomorphological features of primary PTC and its metastases in patients who subsequently showed resistance to 131I therapy, compared with the control group of patients who achieved a positive effect of 131I therapy. Materials and Methods. A comparative analysis of morphological features in histological conclusions and the study of cytological characteristics in the fine-needle aspiration (FNA) smears of primary PTC and their primary metastases, as well RIRM оf experimental and control groups of patients were conducted. The experimental group consisted of 152 patients who showed resistance to RI therapy, the control group – 161 patients who achieved a positive effect of RI therapy. Statistical processing was done in Statistica 12. Results and discussion. It is shown that the frequency of follicular structures in the material of primary PTC of patients with a positive effect of 131I therapy is significantly higher, and the frequency of necrotic changes is significantly lower than in patients with the development of RIRM (p <0.05 by criterion χ2). The frequency of follicular structures in the histological material and FNA smears of primary metastases of patients with a positive effect of 131I therapy is significantly higher, and oxyphilic changes are significantly lower than in patients with the development of RIRM (p <0.05 by criterion χ2). Oxyphilic changes were observed significantly more often in the RIRM compared with primary metastases of the control group (p = 0.03). It was proved that cystic changes are significantly more common in FNA smears and histological material of RIRM in comparison with primary metastases of experimental and control groups of patients (p <0.05). Conclusions. TThe use of the presence of oxyphilic and cystic changes in the FNA smears and histological material of metastases of the PTC as prognostic factors of RI resistance is proposed. The presence of follicular structures in the materials of primary PTC and metastases can be a prognostic factor of effective RI therapy.

Clinical Findings of Melanoma-Associated Retinopathy with anti-TRPM1 Antibody

Introduction. We report the clinical features and clinical course of melanoma-associated retinopathy (MAR), in which autoantibodies against the transient receptor potential cation channel subfamily M member 1 (TRPM1) were detected. Case Presentation. A 74-year-old man was referred to our hospital for treatment of bilateral vision loss. The best-corrected visual acuity was 20/100 in the right eye and 20/200 in the left eye. His electroretinogram (ERG) showed a reduced b-wave and a normal dark-adapted a-wave in both eyes. Optical coherence tomography (OCT) revealed loss of the interdigitation zone in both eyes. We strongly suspected MAR based on the markedly reduced b-wave in the ERG and a history of intranasal melanoma. The diagnosis was confirmed after autoantibodies against TRPM1 were detected in his blood serum. Fifteen months later, his ERG remained unchanged, and OCT showed bilateral cystic changes in the internal nuclear layer. The visual acuity in both eyes also remained unchanged. Conclusions. Anti-TRPM1 autoantibodies were detected in a patient diagnosed with MAR who had negative flash ERG and retinal microstructural abnormalities, and the impairment did not recover during the follow-up period. Identification of anti-TRPM1 antibodies was helpful in confirming the diagnosis of MAR.

Comparative clinicopathological and immunohistochemical study of micronodular thymoma and micronodular thymic carcinoma with lymphoid stroma

AimTo elucidate the clinicopathological and immunohistochemical characteristics of micronodular thymomas (MNTs) and micronodular thymic carcinomas (MNCs) with lymphoid stroma.MethodsWe examined four cases of MNTs and three cases of MNCs pathologically and immunohistochemically.ResultsThere were prominent cystic changes infive of the seven cases. The neoplasms contained epithelial tumour cells arranged in a micronodular growth pattern lined by cystic walls and separated by abundant lymphoid stroma. Only the tumour cell component of MNCs showed signs of malignancy characterised by cytological atypia and increased mitotic activity. Neoplastic MNC epithelial cells showed strong positivity for CD5 and CD117. However, no immature lymphocytes (TdT-positive and CD99-positive) were present in and around the tumour nodules. None of the patients died or suffered from disease due to MNTs or MNCs.ConclusionMNTs and MNCs are rare and less aggressive forms of thymic tumours and can be differentially diagnosed by immunohistochemistry.

Case of laser induced maculopathy and treatment outcome after use of corticosteroids and lutein.

Introduction:A 30yopatient presented sudden decrease of left vision and metamorphopsia after staring at a laser in a nightclub five days before. Results:At presentation left visual acuitywas 6/18-2 unaided improving to 6/12-2 pinhole.Dilated fundoscopy showed a yellow-orange foveolar lesion in the left eye. OCT showed an alteration of foveal anatomy involving predominantly the outer retinal layers, hyper-reflective vertical band and large cystoid change at the inner retina. There was an increase of foveolar thickness to 397µ.Treatment was with oral corticosteroids (Prednisolone 0.5mg/ Kg).At one week left visual acuityimprovedto 6/12+2.There were hardly any cystic changes, with less hyper-reflective band and disruption at the outer layer.Lutein capsules 20 mg daily were added at one week. At three weeks patient reported vision back to normal - the left visual acuity was 6/6-2 unaided. OCT showed near-complete restoration of macular structure. Conclusions: Positive clinical outcome with combined oral corticosteroids and lutein over a short period of time in a typical case – further review recommended of ideal treatment regimes for laser induced maculopathy.

Association of Body Mass Index With Severity and Lesion Location in Adolescents With Osteochondritis Dissecans of the Knee

Background: The association between body mass index (BMI) and severity of osteochondritis dissecans (OCD) of the knee at presentation is poorly understood. Hypothesis: We hypothesized that adolescents in higher BMI percentiles for age and sex would have OCD lesions that were more severe at their initial presentation and located more posteriorly on the condyle as compared with adolescents in lower BMI percentiles. Study Design: Cohort study; Level of evidence, 3. Methods: This study included patients aged 10 to 18 years who were treated for knee OCD at a tertiary care hospital from 2006 to 2017. Patients with noncondylar OCD or missing BMI data within 3 months of presentation were excluded. Patients were stratified per the Centers for Disease Control and Prevention guidelines as underweight, normal weight, overweight, or obese, and the groups were compared according to age, side of lesion, 4 markers of lesion severity (cystic changes, loose fragments, subchondral fluid, and subchondral edema), and surgical treatment. Lesion angle was measured in reference to a line parallel to the femoral axis drawn through the center of a best-fit circle covering the distal condyle. Data were analyzed using chi-square tests, relative risk, Student t tests, analysis of variance, and linear regression of cumulative running percentages. Bonferroni correction was performed when applicable. Results: A total of 77 patients met our inclusion criteria (mean age, 14.2 years; range, 10.1-18.8): 2 were underweight, 50 had normal BMI, 13 were overweight, and 12 were obese. We found correlations between BMI percentile and surgical treatment ( R 2 = .732), subchondral fluid ( R 2 = .716), subchondral edema ( R 2 = .63), loose fragments ( R 2 = .835), and the presence of at least 1 marker of lesion severity ( R 2 = .857) ( P < .0001 for all). No correlation was observed for cystic changes ( R 2 = .026). There were significant associations between BMI ≥80th percentile and subchondral edema (risk ratio, 2.5; 95% CI, 1.3-4.8), medial condylar lesions (risk ratio, 1.3; 95% CI, 1.01-1.7), and lesions more anterior on the condyle ( P < .05). Conclusion: Higher BMI in adolescents was strongly correlated with multiple markers of severity of knee OCD at initial presentation as well as with more anterior lesions.

Gastric Leiomyoma Misdiagnosed as a Left Cystic Adrenal Tumor

Gastrointestinal leiomyomas often occur in the esophagus, colon, and rectum, but are very rare in the stomach. The patient was referred to Jeju national university medical center for the treatment of a left adrenal tumor diagnosed 5 years ago. She had a long history of repeated left flank pain, nausea, and abdominal distension for the last few years. Annual follow-up computed tomography scan showed a no changed 5×5.5-cm sized multiloculated left adrenal cystic lesion. Laboratory tests revealed that the basal levels of plasma adrenocorticotropin, cortisol, aldosterone, renin, testosterone, adrenaline, noradrenaline, and dopamine were normal findings. The preoperative diagnosis was a left nonfunctioning adrenal neoplasm with symptom of discomfort. However, the tumor revealed as an exophytic growing leiomyoma with cystic changes of a stomach by surgery. The mass was found at the posterior wall of the fundus and about 5-cm-sized mass. Resection of the tumor was performed using laparoscopy. The pathologic diagnosis revealed a gastrointestinal leiomyoma with a benign cystic lesion lined by respiratory epithelium with an old hemorrhage and chronic gastritis. The patient had no evidence of recurrence during the 12-month follow-up. Gastric leiomyoma is unfamiliar to the usual urologists. It needs to take gastric leiomyoma into account if the left adrenal tumor is close to the stomach. Herein, we report the case of a 44-year-old woman with gastrointestinal leiomyoma misdiagnosing as a nonfunctioning adrenal tumor.