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PURPOSE: Preoperative pain is known as one of the most powerful risk factors for chronic postoperative inguinal pain (CPIP), while its pathogenesis has not been fully elucidated. The aim of the present study was to evaluate patients with preoperative pain from the pathological perspective and discuss the potential pathogenesis of CPIP in those patients. METHODS: This was a single-institutional retrospective study. The study population was inguinal hernia patients with preoperative pain who underwent open anterior hernia repair for primary inguinal hernia with pragmatic ilioinguinal neurectomy during surgery between March 2021 and March 2023. The primary and secondary outcomes were proportion of collagen deposition and mucus accumulation within ilioinguinal nerve in those patients, respectively, which were evaluated histologically using Image J software. RESULTS: Forty patients were evaluated. Median value of proportion of intraneural collagen deposition was 38.3% (27.7-95.9). These values were positively correlated with the duration of pain (r2=0.468, P<0.001). Median value of proportion of mucus accumulation in ilioinguinal nerve was 50.1% (0-82.0). These values had no correlation with any clinicopathological variables. CONCLUSIONS: In the present study population, all patients with preoperative pain had intraneural fibrosis within ilioinguinal nerve, and its degree had a positive correlation with the pain duration.
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Hérnia Inguinal , Humanos , Hérnia Inguinal/complicações , Hérnia Inguinal/cirurgia , Hérnia Inguinal/diagnóstico , Estudos Retrospectivos , Dor Pós-Operatória/etiologia , Herniorrafia/efeitos adversos , Fibrose , ColágenoRESUMO
A 79-year-old man presented with subacute onset of dementia. Brain magnetic resonance imaging revealed leukoencephalopathy in the posterior lobes with presence of microbleeds. Although clinical manifestation suggested a diagnosis of leukoencephalopathy associated with cerebral amyloid angiopathy (CAA), the patient died of sudden rupture of an aneurysm of the thoracic aorta two months after the onset of dementia. Autopsy revealed pathological features of advanced-stage Alzheimer's disease. Immunohistochemistry for amyloid-ß revealed CAA mainly affecting arteries but not capillaries. Klüver-Barrera staining revealed white matter edema predominantly in the occipital lobes without ischemic changes. Perivascular cuffing was found to be sparse, but there was no evidence of angiitis. Pathological findings suggest that leukoencephalopathy was caused by the disruption of the blood-brain barrier rather than ischemia. Because the present patient died before immunotherapy, his neuropathological findings could reflect the pathomechanism of the acute stage of leukoencephalopathy with CAA.
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Doença de Alzheimer , Angiopatia Amiloide Cerebral , Leucoencefalopatias , Substância Branca , Idoso , Doença de Alzheimer/patologia , Peptídeos beta-Amiloides/metabolismo , Encéfalo/patologia , Angiopatia Amiloide Cerebral/complicações , Angiopatia Amiloide Cerebral/diagnóstico por imagem , Edema/complicações , Edema/patologia , Humanos , Leucoencefalopatias/patologia , Imageamento por Ressonância Magnética , Masculino , Substância Branca/patologiaRESUMO
ABSTRACT: The diagnosis of pilomatricoma, the most common matrical tumor, is generally straightforward; however, it exhibits diverse histology associated with various morphological stages and several clinical variants, and matrical differentiation can occur in various neoplastic diseases. A 56-year-old man was admitted to our hospital to resect an 11.0-cm skin tumor on his right shoulder. Because of its large size and surface irregularities, including multiple erosions and ulcers, cutaneous malignancies were clinically suspected. Histologically, the tumor formed numerous nodules with marked matrical differentiation in the superficial to deep dermis. Although the tumor was macroscopically asymmetrical and irregular, each nodule was microscopically round-shaped and consisted of basaloid cells without marked atypia, atypical mitoses, or lymphovascular invasion. Immunohistochemically, the tumor cells were positive for beta-catenin, LEF-1, and PHLDA-1, consistent with their pilomatrical differentiation. We diagnosed the case as a giant pilomatrical tumor with uncertain malignant potential, considering its "contradictory" features, namely, the worrisome histoarchitecture, such as the asymmetrical silhouette, but bland-looking cytological appearance. Unlike typical pilomatrical tumors, this tumor contained numerous epidermal components with features similar to those of the dermal components, resulting in a unique macroscopic and histological appearance. Our case broadens the known histological diversity of pilomatrical tumors.
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Pilomatrixoma/patologia , Neoplasias Cutâneas/patologia , Diferenciação Celular , Doenças do Cabelo , Humanos , Masculino , Pessoa de Meia-IdadeRESUMO
The neonatal Fc receptor (FcRn) plays a role in trafficking IgG and albumin and is thought to mediate intravenous immunoglobulin (IVIG) therapy for certain diseases. IVIG can be used for the treatment of human Langerhans cell histiocytosis (LCH); however, the mechanism remains unclear. The expression and function of FcRn protein have not been studied in LCH, though the expression of FcRn messenger RNA (mRNA) have been reported. In this report, we confirmed the expression of FcRn in 26 of 30 pathological cases (86.7%) diagnosed immunohistochemically as LCH. The expression was independent of age, gender, location, multi- or single-system, and the status of BRAFV600E immunostaining. We also confirmed the expression of FcRn mRNA and protein in the human LCH-like cell line, ELD-1. FcRn suppressed albumin consumption and growth of IVIG preparation-treated ELD-1 cells, but not of IVIG preparation-untreated or FcRn-knockdown ELD-1 cells. In addition, FITC-conjugated albumin was taken into Rab11-positive recycle vesicles in mock ELD-1 cells but not in FcRn-knockdown ELD-1 cells. IVIG preparation prolonged this status in mock ELD-1 cells. Therefore, ELD-1 recycled albumin via FcRn and albumin was not used for metabolism. Our results increase our understanding of the molecular mechanism of IVIG treatment of LCH.
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Histiocitose de Células de Langerhans , Antígenos de Histocompatibilidade Classe I/metabolismo , Imunoglobulinas Intravenosas , Receptores Fc/metabolismo , Adolescente , Adulto , Albuminas/metabolismo , Linhagem Celular , Criança , Pré-Escolar , Feminino , Fluoresceína-5-Isotiocianato/análogos & derivados , Fluoresceína-5-Isotiocianato/metabolismo , Histiocitose de Células de Langerhans/tratamento farmacológico , Histiocitose de Células de Langerhans/metabolismo , Humanos , Imunoglobulinas Intravenosas/metabolismo , Imunoglobulinas Intravenosas/farmacologia , Lactente , Recém-Nascido , Masculino , Albumina Sérica/metabolismoRESUMO
Acromegaly is characterized by autonomous excessive growth hormone (GH) secretion, generally due to GH-producing pituitary adenoma, and is associated with various systemic comorbidities including diabetes mellitus. Polycystic kidney disease (PKD) is characterized by the growth of numerous cysts in the kidneys that deteriorate renal function. While possible renal effects of excessive GH exposure have been a current issue in experimental medicine, only five cases of coexisting acromegaly and PKD have been reported previously, and little is known regarding the influence of acromegaly on renal disease. We treated a 50-year-old male with diabetes mellitus who showed a sudden and rapid decline of renal function along with increasing proteinuria, which led to diagnoses of PKD and acromegaly. His urinary protein levels were increased together with excessive GH secretion and worsening glycemic control. An increase of total kidney volume was also noted. Transsphenoidal surgery for the pituitary adenoma was successfully performed. Marked improvement of hyperglycemia and proteinuria were observed after the surgery, but renal function was unchanged. The patient's clinical course suggested common aspects of excessive GH secretion as an accelerating factor of the progression of diabetic nephropathy and PKD via direct and indirect pathways. Although coexisting acromegaly and PKD is clinically rare, vigilance for early diagnosis of acromegaly is appropriate in patients with diabetes and/or PKD, especially in those showing unexpected exacerbation of renal dysfunction.
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Acromegalia/complicações , Complicações do Diabetes/complicações , Doenças Renais Policísticas/complicações , Acromegalia/diagnóstico , Acromegalia/patologia , Adenoma/complicações , Adenoma/diagnóstico , Adenoma/metabolismo , Adenoma/patologia , Complicações do Diabetes/diagnóstico , Complicações do Diabetes/patologia , Diabetes Mellitus/diagnóstico , Diabetes Mellitus/patologia , Progressão da Doença , Adenoma Hipofisário Secretor de Hormônio do Crescimento/complicações , Adenoma Hipofisário Secretor de Hormônio do Crescimento/diagnóstico , Adenoma Hipofisário Secretor de Hormônio do Crescimento/metabolismo , Adenoma Hipofisário Secretor de Hormônio do Crescimento/patologia , Hormônio do Crescimento Humano/metabolismo , Humanos , Masculino , Pessoa de Meia-Idade , Doenças Renais Policísticas/diagnóstico , Doenças Renais Policísticas/patologiaRESUMO
Langerhans cell neoplasms, which include Langerhans cell histiocytosis and Langerhans cell sarcoma, are tumors that originate from dendritic cells. Langerhans cell sarcoma is defined as a high-grade neoplasm with overtly malignant cytological features and the Langerhans cell-like phenotype, and generally has a poorer prognosis and more aggressive phenotype than Langerhans cell histiocytosis. Insulin-like growth factor 2 messenger RNA-binding protein 3 (IGF2BP3 or IMP3) is an oncofetal protein that is expressed in various cancer types; its expression is often associated with a poor prognosis and aggressive phenotype. Here, we used immunohistochemistry to evaluate IGF2BP3 expression in Langerhans cell neoplasms. IGF2BP3 expression was scored as negative (< 1%) or positive (≥ 1%) by immunohistochemistry. All 4 patients with Langerhans cell sarcoma (100%) and 6 of 22 pediatric (age < 18 years) patients with Langerhans cell histiocytosis (27.3%) had positive results for IGF2BP3; however, 16 of 22 pediatric patients with Langerhans cell histiocytosis (72.7%) and all 15 adult (age ≥ 18 years) patients with Langerhans cell histiocytosis (100%) had a negative result. Among patients with Langerhans cell histiocytosis, IGF2BP3 expression was independent of sex, location, prognosis, and BRAF V600E staining results. Taken together, these results indicate that IGF2BP3 expression may be a helpful marker for distinguishing Langerhans cell sarcoma from Langerhans cell histiocytosis in adult patients.
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Histiocitose de Células de Langerhans/metabolismo , Sarcoma de Células de Langerhans/metabolismo , Proteínas de Ligação a RNA/metabolismo , Adolescente , Adulto , Biomarcadores/metabolismo , Biomarcadores Tumorais/metabolismo , Criança , Pré-Escolar , Diagnóstico Diferencial , Feminino , Histiocitose de Células de Langerhans/diagnóstico , Humanos , Imuno-Histoquímica , Lactente , Recém-Nascido , Sarcoma de Células de Langerhans/diagnóstico , Masculino , Pessoa de Meia-Idade , Adulto JovemRESUMO
CD72 is a transmembrane protein belonging to the C-type lectin family that is expressed by various hematopoietic cells. When bound to its natural ligand, CD100 (semaphorin 4D), CD72 inhibits the KIT-mediated responses of human mast cells, but not IgE/FcεRI-mediated mast cell degranulation. We extended these findings to examine the role of CD72 in mouse mast cells. CD72 expression was detected in mouse bone marrow-derived mast cells (mBMMCs). As for human mast cells, an agonistic antibody against CD72 (K10.6) suppressed the KIT-mediated cell growth of, IL-6 production by and chemotaxis of mBMMCs. However, in contrast to human mast cells, the IgE-triggered degranulation of mBMMCs was suppressed by K10.6. K10.6 did not affect the phosphorylation of SHP-1 in mBMMCs, although SHP-1 mediated the inhibitory effects of CD72 in human mast cells. Administration of K10.6 induced phosphorylation of the ubiquitin ligase Cbl-b and decreased the expression of KIT and FcεRIα on the surface of murine mast cells. We also observed expression of CD72 in a mouse neoplastic cell line, P815, harboring gain-of-function mutations in KIT genes. In addition, we found that K10.6 activated Cbl-b, down-regulated KIT expression and suppressed the mutated KIT-driven growth of these cells. Thus, the mechanism by which CD72 mediates inhibitory effects in mast cells is species-dependent.
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Antígenos CD/imunologia , Antígenos de Diferenciação de Linfócitos B/imunologia , Regulação para Baixo/imunologia , Mastócitos/imunologia , Proteínas Proto-Oncogênicas c-kit/imunologia , Receptores de IgE/imunologia , Proteínas Adaptadoras de Transdução de Sinal , Animais , Anticorpos Neutralizantes/farmacologia , Linhagem Celular Tumoral , Regulação para Baixo/efeitos dos fármacos , Humanos , Mastócitos/citologia , Camundongos , Células NIH 3T3 , Proteínas Proto-Oncogênicas c-cbl , Semaforinas/imunologiaRESUMO
The incidence and mortality rate of pancreatic cancer are increasing worldwide. Regional lymph nodes, liver, lung, and peritoneum are common sites of metastasis from pancreatic cancer, but the gastrointestinal tract is rare as a metastatic organ from pancreatic cancer. An 80-year-old man was referred to our department for a hypovascular pancreatic mass on contrast-enhanced computed tomography (CECT). Endoscopic ultrasound-guided fine needle aspiration revealed adenocarcinoma, and he was diagnosed with pancreatic cancer. No lymph nodes or distant metastases were detected by either CECT or gadolinium-enhanced magnetic resonance imaging, and we evaluated this case as borderline resectable. However, total colonoscopy for positive fecal occult blood tests revealed a reddish and hemorrhagic mucosal thickening in the ascending and sigmoid colon and rectum, which was inconsistent with primary colorectal cancer. Biopsy specimens from these sites revealed cytokeratin (CK)7-positive and CK20- and CDX2-negative adenocarcinoma, consistent with cancer of pancreatic origin. The patient underwent palliative chemotherapy with gemcitabine but died from COVID-19 infection eight months after diagnosis. Performing total colonoscopy as a preoperative screening is important for accurate cancer staging of patients with possible resectable pancreatic cancer.
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Adenocarcinoma , Neoplasias Pancreáticas , Masculino , Humanos , Idoso de 80 Anos ou mais , Neoplasias Pancreáticas/patologia , Aspiração por Agulha Fina Guiada por Ultrassom Endoscópico , Linfonodos/patologia , Adenocarcinoma/patologia , Intestino Grosso/patologiaRESUMO
This case report describes a 78-year-old man initially treated for pneumonia and lung abscess who was resistant to antimicrobial treatment and was eventually diagnosed with ciliated adenocarcinoma. Ciliated adenocarcinoma, a rare non-terminal respiratory unit (TRU)-type lung adenocarcinoma, presents a unique diagnostic challenge because of its similarity to pneumonia and lung abscesses. Morphologically, the ciliated adenocarcinoma in this case appeared to be a non-TRU type adenocarcinoma, with partial mucous epithelium, no visible extracellular mucus, thyroid transcription factor (TTF)-1 negativity, and mucin (MUC) 5AC positivity on immunostaining. The patient was considered to have ciliated adenocarcinoma based on the fact that the mucous epithelium was partial and extracellular mucus was not prominent. This case emphasizes the importance of considering malignancy in patients with non-resolving pulmonary infections.
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Summary: Functioning gonadotroph tumors are rare neoplasms that can cause ovarian hyperstimulation syndrome (OHSS) in women of reproductive age. Here, we present a case of a follicle-stimulating hormone (FSH)-producing pituitary neuroendocrine tumor (PitNET) with irregular menstrual cycles and OHSS in a Japanese woman. A 34-year-old woman with bilateral multi-cystic ovarian mass was referred to our hospital for ovarian surgery. The imaging feature of magnetic resonance imaging (MRI) of the ovary and elevated estradiol levels with normal FSH and low luteinizing hormone (LH) levels led us to suspect the presence of a functioning gonadotroph PitNET. MRI revealed a 19-mm pituitary tumor, and increased tracer uptake was observed in the pituitary lesion on 111In-pentetreotide scintigraphy. Transsphenoidal tumor resection resulted in the resolution of the ovarian enlargement, normalization of her menstrual cycles, and spontaneous pregnancy. Immunohistochemistry (IHC) of the resected tumor for pituitary transcription factors, including steroidogenesis factor 1 (SF1) and estrogen receptor alpha, demonstrated positive immunoreactivity, whereas IHC for pituitary-specific positive transcription factor 1 was negative, suggesting that the tumor belonged to the SF1 lineage of PitNETs (gonadotroph tumor). The tumor cells showed positive expression of FSHß, while LHß was mostly negative. Consistent with the high pituitary tumor uptake observed on 111In-pentetreotide scintigraphy, the pituitary tumor showed positive expression of somatostatin receptor 2A. Detailed clinical and histological evaluations will provide useful information to understand these rare functioning gonadotroph tumors better. Learning points: Functioning gonadotroph tumors are very rare neuroendocrine tumors of pituitary origin. Women of reproductive age presenting with bilateral multi-cystic ovarian enlargement, irregular menstrual cycles, and hyperestrogenemia under unsuppressed follicle-stimulating hormone (FSH) levels should be evaluated for FSH-producing tumor. Raising awareness of OHSS due to functioning gonadotroph tumors is crucial to prevent unnecessary ovarian surgery. Comprehensive histological analysis may provide useful information to better understand the characteristics of functioning gonadotroph tumors.
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Intravascular large B-cell lymphoma (IVLBCL) is a rare type of aggressive extranodal large B-cell lymphoma characterized by the selective growth of lymphoma cells within the lumina of blood vessels, particularly capillaries. IVLBCL lacks mass formation, and its diagnosis can be challenging. We analyzed the utility of insulin-like growth factor II mRNA-binding protein 3 (IMP3) immunohistochemistry for IVLBCL diagnosis in various organs. Double staining with paired box 5 (PAX5) was performed for validation. Overall, 152 pathological specimens (111 positive and 41 negative for IVLBCL) obtained from 88 patients with a diagnosis of IVLBCL were stained for IMP3 and IMP3/PAX5. As negative controls, 40 pathology specimens from 38 patients with no history of IVLBCL or other B-cell lymphomas were stained for IMP3, which comprised 31 benign pathological specimens from 29 patients in whom malignancy was suspected, 7 cases of appendicitis with intravascular and/or intralymphatic lymphoid proliferations, and 2 cases of intravascular natural killer/T-cell lymphoma. All mononuclear cells with cytoplasmic staining were considered positive for IMP3 expression, but expression restricted to germinal center B cells was excluded from evaluation. All 111 IVLBCL pathological specimens were positive for IMP3 and IMP3/PAX5. In addition, 11 of the 41 specimens originally diagnosed as IVLBCL-negative showed IMP3/PAX5 double-positive cells, raising the suspicion of IVLBCL. However, of the 40 negative control samples, IMP3-positive non-germinal center B cells were detected in only 2 samples ( P = 0.0131) and no intravascular IMP3-positive B cells suspicious for IVLBCL were identified. Altogether, IMP3 immunohistochemistry is a highly sensitive marker of IVLBCL and can be a helpful adjunct for IVLBCL diagnosis.
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Biomarcadores Tumorais , Imuno-Histoquímica , Linfoma Difuso de Grandes Células B , Proteínas de Ligação a RNA , Humanos , Biomarcadores Tumorais/análise , Proteínas de Ligação a RNA/análise , Masculino , Feminino , Linfoma Difuso de Grandes Células B/patologia , Linfoma Difuso de Grandes Células B/diagnóstico , Pessoa de Meia-Idade , Idoso , Adulto , Idoso de 80 Anos ou mais , Neoplasias Vasculares/patologia , Neoplasias Vasculares/química , Valor Preditivo dos Testes , Reprodutibilidade dos Testes , Adulto JovemRESUMO
Metaplastic thymoma (MT), a rare subtype of thymic epithelial tumors (TETs), harbors YAP1::MAML2 fusions. Poroma, a skin tumor, also carries these fusions and exhibits a unique staining pattern for YAP1 immunohistochemistry (IHC), namely, a YAP1 N-terminus (YAP1[N])-positive but YAP1 C-terminus (YAP1[C])-negative pattern. In this context, MT was recently reported to lack YAP1(C) expression exclusively among TET subtypes. However, a lack of information about YAP1(N) expression in that study and another report that wild-type YAP1 expression was diminished in type B3 thymoma and thymic carcinoma warrants further studies for YAP1 expression in TETs. Thus, we immunohistochemically examined YAP1(N) and YAP1(C) staining patterns in our TET samples, including 14 cases of MT. In addition, 11 of the 14 MT cases were genetically analyzed with the formalin-fixed paraffin-embedded tissues if they harbored YAP1::MAML2 fusions. MT consistently exhibited YAP1(N)-positive and YAP(C)-negative staining, whereas type B3 thymoma and thymic carcinoma showed relatively heterogeneous staining patterns for YAP1(N) and YAP1(C) and were sometimes negative for both antibodies. Furthermore, a lower expression of YAP1 was found in type B3 compared to B2 thymomas. Among genetically analyzed 11 MT cases, 6 cases showed YAP1::MAML2 fusions, whereas the analysis failed in 5 very old cases due to poor RNA quality. These results indicate that IHC of both YAP1(N) and YAP1(C) is recommended to obtain staining patterns almost unique to MT. The biological significance of YAP1 in high-grade TETs warrants further investigation.
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Double somatic mutations in CTNNB1 and GNA11/Q have recently been identified in a small subset of aldosterone-producing adenomas (APAs). As a possible pathogenesis of APA due to these mutations, an association with pregnancy, menopause, or puberty has been proposed. However, because of its rarity, characteristics of APA with these mutations have not been well characterized. A 46-year-old Japanese woman presented with hypertension and hypokalemia. She had two pregnancies in the past but had no history of pregnancy-induced hypertension. She had regular menstrual cycle at presentation and was diagnosed as having primary aldosteronism after endocrinologic examinations. Computed tomography revealed a 2 cm right adrenal mass. Adrenal venous sampling demonstrated excess aldosterone production from the right adrenal gland. She underwent right laparoscopic adrenalectomy. The resected right adrenal tumor was histologically diagnosed as adrenocortical adenoma and subsequent immunohistochemistry (IHC) revealed diffuse immunoreactivity of aldosterone synthase (CYP11B2) and visinin like 1, a marker of the zona glomerulosa (ZG), whereas 11ß-hydroxylase, a steroidogenic enzyme for cortisol biosynthesis, was mostly negative. CYP11B2 IHC-guided targeted next-generation sequencing identified somatic CTNNB1 (p.D32Y) and GNA11 (p.Q209H) mutations. Immunofluorescence staining of the tumor also revealed the presence of activated ß-catenin, consistent with features of the normal ZG. The expression patterns of steroidogenic enzymes and related proteins indicated ZG features of the tumor cells. PA was clinically and biochemically cured after surgery. In conclusion, our study indicated that CTNNB1 and GNA11-mutated APA has characteristics of the ZG. The disease could occur in adults with no clear association with pregnancy or menopause.
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Adenoma , Adenoma Adrenocortical , Hiperaldosteronismo , Hipertensão , Adulto , Feminino , Gravidez , Humanos , Pessoa de Meia-Idade , Adenoma Adrenocortical/complicações , Adenoma Adrenocortical/genética , Adenoma Adrenocortical/cirurgia , Aldosterona/metabolismo , Citocromo P-450 CYP11B2/metabolismo , beta Catenina/genética , beta Catenina/metabolismo , Hiperaldosteronismo/genética , Hiperaldosteronismo/cirurgia , Adenoma/genética , Adenoma/cirurgia , Adenoma/metabolismo , Hipertensão/complicações , Mutação , Subunidades alfa de Proteínas de Ligação ao GTP/genética , Subunidades alfa de Proteínas de Ligação ao GTP/metabolismoRESUMO
The combination of exocrine and neuroendocrine neoplasms is rarely found in the pancreas. These combined lesions vary from a clonal tumor with mixed differentiation to the incidental co-existence of two or more independent tumors, but the differential diagnosis is sometimes difficult. Here we report a case of multiple endocrine neoplasia type 1 (MEN1) with extensive ductal and neuroendocrine neoplastic changes. These two types of tumors admixed markedly in some parts, which made it difficult to determine the pathological diagnosis based on histological findings. Cytogenetic analysis showed that loss of heterozygosity (LOH) of the MEN1 locus exists in neuroendocrine but not in exocrine neoplasms, indicating that independent mechanisms of tumorigenesis may occur in these two types of tumors. This case shows the usefulness of cytogenetic analysis for the diagnosis of combined tumors of the pancreas. Extensive exocrine neoplastic change, including pancreatic intraepithelial neoplasia (PanIN) in virtually all pancreatic ducts and a focus of intraductal papillary mucinous neoplasm (IPMN) with focal invasion, was a distinguishing feature of the present case. The possible association of ductal tumorigenesis and a MEN1 background is discussed.
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Carcinoma in Situ/patologia , Carcinoma Neuroendócrino/patologia , Perda de Heterozigosidade/genética , Neoplasia Endócrina Múltipla Tipo 1/patologia , Neoplasias Primárias Múltiplas/patologia , Neoplasias Pancreáticas/patologia , Idoso , Carcinoma in Situ/genética , Carcinoma Neuroendócrino/genética , Análise Citogenética/métodos , Feminino , Humanos , Neoplasia Endócrina Múltipla Tipo 1/genética , Mutação/genética , Neoplasias Primárias Múltiplas/genética , Pâncreas Exócrino/patologia , Ductos Pancreáticos/patologia , Neoplasias Pancreáticas/genéticaRESUMO
BACKGROUND: Programmed death-1 (PD-1) is a marker for human neoplastic T cells. Here, we evaluated whether or not PD-1 was also a marker for human mastocytosis, and explored the role of PD-1 in human mastocytosis cells. METHODS: Immunohistochemical analysis was used to evaluate the expression of PD-1 in clinical samples of human cutaneous mastocytosis. The expression of PD-1 in human mastocytosis cell lines was checked by RT-PCR, western blotting and flow cytometry. We stimulated human mastocytosis cell lines (LAD2 and HMC1.2) with recombinant ligand for PD-1, PD-L1 (rPD-L1), and tested the proliferative activity and the status of signal molecules by Cell Counting Kit-8 and ELISA, respectively. RESULTS: Ten of 30 human cutaneous mastocytosis cases (33.3%) expressed PD-1 protein. We also found that a human mastocytosis line LAD2 cells expressed PD-1 protein on their surfaces. The administration with rPD-L1 suppressed the stem cell factor-dependent growth of the LAD2 cells. And, rPD-L1 activated SHP-1 and SHP-2 simultaneously, and decreased the phosphorylation of AKT, in LAD2 cells. In contrast, we could not detect the expression of PD-1, and the significant effect of rPD-L1 on the mutated KIT-driven growth of HMC1.2 cells. CONCLUSIONS: PD-1 could be a marker for human cutaneous mastocytosis and regulate the growth of human PD-1-positive mastocytosis cells.
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Mastocitose Cutânea/metabolismo , Receptor de Morte Celular Programada 1/metabolismo , Linhagem Celular , Proliferação de Células/efeitos dos fármacos , Expressão Gênica , Humanos , Mastocitose Cutânea/genética , Receptor de Morte Celular Programada 1/genética , Proteínas Recombinantes/genética , Proteínas Recombinantes/metabolismo , Proteínas Recombinantes/farmacologiaRESUMO
A colonic depressed lesion was found in a 77-year-old man and suspected as a neoplastic lesion using conventional endoscopy. Magnifying endoscopy, however, did not support it and biopsy revealed the lesion to be AL amyloidosis.
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We describe a 51-year-old otherwise healthy woman hospitalized for hypotension, fever, and weakness 4 days after the second-dose Covid-19 mRNA vaccine. Elevated inflammatory markers, natriuretic peptide levels and troponin levels, and slightly reduced left ventricular ejection fraction of 50% were noted. We also found the multiple organ damage, including mucocutaneous, gastrointestinal, and neurologic systems. In addition, we revealed the positive results for anti-nucleocapsid SARS-CoV-2 IgG, albeit negative for SARS-CoV-2 polymerase chain reaction testing, suggesting the prior asymptomatic Covid-19 infection. We finally diagnosed her as multisystem inflammatory syndrome after vaccination. Of note, we obtained myocardial specimen from the patients and demonstrated the lymphohistiocytic myocarditis, which is a rare form of myocarditis.
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Vacinas contra COVID-19 , COVID-19 , Miocardite , Feminino , Humanos , Pessoa de Meia-Idade , COVID-19/diagnóstico , Vacinas contra COVID-19/efeitos adversos , Miocardite/diagnóstico , Miocardite/etiologia , SARS-CoV-2 , Volume Sistólico , Função Ventricular EsquerdaRESUMO
The transformation to SCLC is a known mechanism of resistance against molecularly targeted therapies. This study reports a patient with untreated lung adenocarcinoma, characterized by a KRAS G12C mutation, which transformed into SCLC before treatment. Both the adenocarcinoma and SCLC components were responsive to sotorasib.
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Pulmonary cryptococcosis (PC) is a rare fungal lung infection that usually occurs in immunocompromised hosts. We report a rare case of PC with asymptomatic and mixed lesions of infiltrative and nodular shadows. A woman in her 50s with a 7-year history of methotrexate treatment for rheumatoid arthritis (RA) presented with abnormal chest shadows on annual examination. Chest CT revealed two tiny nodules and two infiltrative shadows in the right lower lobe of the lung. We suspected lung cancer, cryptogenic organising pneumonia or RA-interstitial lung disease. However, transbronchial lung biopsy and positivity for serum cryptococcal antigen confirmed the diagnosis of PC. We initiated treatment with fluconazole, which drastically reduced the chest shadows without any adverse events. Since PC can present with several pulmonary shadows, it should be considered as a differential diagnosis of any pulmonary lesion.
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Artrite Reumatoide , Criptococose , Pneumonia em Organização Criptogênica , Feminino , Humanos , Criptococose/diagnóstico , Criptococose/tratamento farmacológico , Hospedeiro Imunocomprometido , Tórax , Antígenos de Fungos , Artrite Reumatoide/complicações , Artrite Reumatoide/tratamento farmacológicoRESUMO
Background: Pulmonary hyalinizing granuloma (PHG) is a very rare pulmonary disease characterized by multiple fibrosclerotic inflammatory lung nodules. The disease is supposedly caused by an unusual immune response. Case presentation: We present a case involving a 53-year-old female with a history of lumpectomy surgery due to invasive ductal carcinoma who was admitted for slowly progressive pulmonary nodules. The patient's elevated serum IgG4 level and the pathological findings obtained in surgical biopsy indicated IgG4-related lung disease. The nodules continued to enlarge despite administration of corticosteroid therapy, and we performed a second surgical biopsy to obtain a correct diagnosis. The pathological findings obtained in the second biopsy were different and consistent with the features of PHG. Conclusions: In this report, the radiological follow-up data obtained after lumpectomy surgery demonstrate the very early stage of PHG and the following radiological changes over a decade, and the two surgical biopsies support us to realize the pathological change from previous diagnosed disease before PHG.