Multiple Small Nodules Research Articles

560P Circulating tumor DNA guided therapeutic strategies for colorectal cancer patients with multiple small pulmonary-limited nodules: Updated results from the open-label, prospective, phase II cohort study

560P Circulating tumor DNA guided therapeutic strategies for colorectal cancer patients with multiple small pulmonary-limited nodules: Updated results from the open-label, prospective, phase II cohort study

Rare Presentation, Critical Diagnosis: Primary Actinomycosis of the Foot

Actinomycosis, an uncommon granulomatous infection caused by the Actinomyces species, rarely targets as primary involvement the limb and is often linked to traumatic incidents. In this report, we present the case of a 44-year-old female who developed multiple small nodules on her left foot over approximately 12 months. Some nodules exhibited firmness and a violet hue, while others discharged a yellowish fluid. The patient had no significant comorbidities. Despite thorough blood paraclinical assessments, including complete blood count, serological HIV testing, and QuantiFERON-TB Gold testing, no abnormalities were detected. Bacteriological examinations and cultures of the discharge yielded negative results. Dermatoscopic examination revealed ovoid yellowish structures, with confocal microscopy highlighting granulomas. A subsequent skin biopsy confirmed characteristic changes indicative of actinomycosis. Although systemic antibiotic therapy with penicillin derivatives was initially considered, the patient’s documented allergic history to this medication class, verified through allergological testing, prompted the initiation of doxycycline treatment. Notably, significant improvement was observed at the 3-month follow-up. This case underscores the importance of reporting rare instances of actinomycosis due to its diagnostic complexity and management challenges.

Non-tuberculosis Mycobacterial Pulmonary Disease Caused by Mycobacterium kiyosense, a New Species.

Non-tuberculosis mycobacterial (NTM) pulmonary disease (NTM-PD) is quite common, and newly identified species are being reported increasingly frequently thanks to advances in identification technologies. A 56-year-old woman had mild sputum production showed bronchiectasis with multiple small nodules, consistent with NTM-PD, on chest computed tomography. Mycobacterial species were isolated from the specimens; however, conventional methods could not identify the species. We conducted whole-genome sequencing and identified the NTM isolates as Mycobacterium kiyosense, a species newly registered in 2023 from Japan. She was diagnosed with NTM-PD caused by M. kiyosense and received watchful waiting.

Double CYP11B1/CYP11B2 Immunohistochemistry and Detection of KCNJ5 Mutations in Primary Aldosteronism.

The search for somatic mutations in adrenals resected from patients with primary aldosteronism (PA) is performed by Sanger sequencing, often implemented with immunohistochemistry (IHC)-guidance focused on aldosterone-producing (CYP11B2-positive) areas. To investigate the impact of double IHC for CYP11B1 and CYP11B2 on Sanger and next-generation sequencing (NGS). We investigated 127 consecutive adrenal aldosterone-producing adenomas from consenting surgically cured PA patients using double IHC for CYP11B1 and CYP11B2, by Sanger sequencing and NGS. Double IHC for CYP11B2 and CYP11B1 revealed 3 distinct patterns: CYP11B2-positive adenoma (pattern 1), mixed CYP11B1/CYP11B2-positive adenoma (pattern 2), and adrenals with multiple small CYP11B2-positive nodules (pattern 3). Sanger sequencing allowed detection of KCNJ5 mutations in 44% of the adrenals; NGS revealed such mutations in 10% of those negative at Sanger and additional mutations in 61% of the cases. Importantly the rate of KCNJ5 mutations differed across patterns: 17.8% in pattern 1, 71.4% in pattern 2, and 10.7% in pattern 3 (χ2 = 22.492, P < .001). NGS allowed detection of mutations in many adrenals that tested negative at Sanger sequencing. Moreover, the different distribution of KCNJ5 mutations across IHC patterns indicates that IHC-guided sequencing protocols selecting CYP11B2-positive areas could furnish results that might not be representative of the entire mutational status of the excised adrenal, which is important at a time when KCNJ5 mutations are suggested to drive management of patients with aldosterone-producing adenomas.

A novel entity of HIPK2::YAP1 pulmonary fibromatosis

BackgroundPulmonary fibromatosis (PF) is a specific variant of fibromatosis, which is rarely reported occurring in the lung. PF with HIPK2-YAP1 fusion was a novel entity.Case presentationIn this report, a 66-year-old male with PF had been smoking over 40 years. Multiple cords and small nodules in both lungs had been detected in a health examination two years earlier at our hospital. But approximately twofold enlarged in the lingual segment of the upper lobe in the left lung were disclosed in this year. Immunohistochemical analysis demonstrated that the vimentin and β-Catenin were positive in the largest nodule. After underwent a DNA/RNA panel next-generation sequencing (NGS), missense mutations and HIPK2-YAP1 fusion were found in this sample. Ultimately, the case diagnosis as PF with HIPK2-YAP1 fusion after multidisciplinary treatment. Currently, the patient is doing well and recurrence-free at 14 months post-surgery.ConclusionsIt’s difficult for patients with complex morphology to make accurate diagnosis solely based on morphology and immunohistochemistry. But molecular detection is an effective method for further determining pathological subtypes.

Localized multisystem inflammatory syndrome in children (mis-c), faster recovery outcome: a case report

Backgrounds: MIS-C cases emerged approximately 4–5 weeks on average after the peak prevalence of COVID-19. The symptoms could be asymptomatic or mildly symptomatic with fever and cough. It was unknown if these infants’ exhibited symptoms of Kawasaki disease (KD) or toxic shock syndrome (TSS), or whether they represented a condition associated with the continuing COVID-19 epidemic. Objective: To describe clinical outcome and examination results of localized multisystem inflammatory syndrome in children. Case presentation: A 23-month-old boy was brought to the pediatric allergy immunology clinic as a referral from a pediatrician with complaints of lump on right preauricular region, which was redden and painful, feels warm, and difficulty opening mouth. There were also multiple small nodules in peri anal area. There was fever for 1 week before admitted, accompanied by a runny nose with clear secretions with no cough. Fever already resolved at presentation. Tonsils are enlarged to T3/T4 without hyperemic nor injection. The presence of fever, conjunctivitis and cracked lips mimicking Kawasaki disease, but electrocardiography and echocardiography were normal. The MISC panel was shown support for MISC, wherein CBC showed presence of lymphopenia, anemia, neutrophilia, and thrombocytosis. Other panel MISC like the erythrocytes sedimentation rate, quantitative CRP, D-dimer, and quantitative IgG SARS-COV-2 antibodies it was reported to be increased. The patient was treated with intravenous high doses methylprednisolone pulse 10 mg/kg, then tapered off before stopped. The lump was disappeared after the second dose of HDMP, and patient was discharged after 7 days of admission. Conclusion: We reported a localized Multisystem Inflammatory Syndrome (MIS-C) treated with high dose methylprednisolone resulting in fast recovery.

Camper’s and Scarpa’s fasciae: Anatomic landmark to differentiate between lymph node and abdominal wall involvement in a case of lymphoma

Cutaneous involvement of MALT lymphoma can either be primary or secondary, Secondary spread to the skin is considered disseminated extra-nodal disease involvement, which is classifies as a stage IV disease. We described a case of a 44-year-old woman with MALT lymphoma of the lacrimal glands with biopsy-proven lymphoma involvement in the subcutaneous tissue of the anterior abdominal wall. This involvement is manifested as multiple small discrete solid enhancing subcutaneous nodules. Although such findings on conventional CT scans are usually non-specific and may present in various systemic conditions, the knowledge of the patient’s prior clinical history should alert interpreting radiologists to be aware of cutaneous and subcutaneous involvement of lymphoma. This awareness can help plan the use of imaging-guided tissue biopsy for a minimally-invasive and safe acquisition of tissue specimen for histopathological diagnosis.

Plexiform fibrohistiocytic tumor of elbow: A rare case report

Plexiform fibrohistiocytic tumor is a very rare tumor of soft tissue that affects usually adolescents and children. On histopathological examination it shows multiple small nodules or fascicles that are arranged in plexiform pattern, hence its name. Here we are presenting a case of occurrence of PFT in bilateral elbows. Patient presented with gradually increasing in size and painless swellings for 7 years. Excision was done and on the basis of histopathological and immunohistochemical examination diagnosis of PFT was reached.

Pre-germ cell neoplasia in-situ in a patient with complete androgen insensitivity syndrome

Abstract Introduction/Objective Complete androgen insensitivity syndrome is a disorder of sex development in 46, XY individuals with external female genitalia and undescended testes, often presenting with inguinal hernia in infancy and/or primary amenorrhea in adolescents. Given the increased risk for the development of germ cell neoplasms in the ectopic testis, early gonadectomy or orchidopexy may be indicated. Methods/Case Report We report a 21-year-old patient assigned female at birth (46, XY) with a history of bilateral inguinal hernia repairs (age 2) and later with lack of menarche (age 16). Additional work-up showed an elevated testosterone level (394 ng/mL) and normal luteinizing and follicle stimulating hormone values. On pelvic ultrasound, retained intraabdominal gonads were identified, but female sex organs were absent. During a gonadal relocation procedure, the gonads appeared irregular in shape with multiple cysts and small nodules, and bilateral gonadectomy was performed. Grossly, the gonads measured 4.9 and 4.3 cm with a smooth external surface and a lobulated cut surface with multiple cysts, up to 1.6 cm, containing clear, serous fluid. Histologic sections showed most seminiferous tubules were lined only by Sertoli cells with only occasional spermatogonia. Leydig cell hyperplasia and areas of ovarian-like stroma were seen. OCT4 immunostain highlighted the nuclei of individually disposed germ cells in the base of tubules (singly dispersed, non-linear pattern), raising concern for germ-cell neoplasia in-situ (GCNIS). However, the histology and staining pattern were better interpreted as diagnostic of germ cells with delayed maturation and pre-GCNIS. Additionally, OCT4 showed rare single cells within the stroma and considered to represent residual undifferentiated gonadal tissue. After gonadectomy, the patient was started on hormone replacement therapy. Results (if a Case Study enter NA) NA Conclusion This case highlights the potential pitfalls in the interpretation of OCT4 immunostain to avoid misdiagnosis of germ cell neoplasia.

Primary Pulmonary Diffuse Large B-Cell Lymphoma - a Rare Case Report

Background: Diffuse large B cell lymphoma (DLBCL) is an aggressive hematological malignancy, and a common histological form of non-Hodgkin's lymphoma. However, its extra-nodal presentation is seen in 30-40% of cases with involvement of gastrointestinal tract, skin, testes, adrenals and bone. ( Lugogo NL et al., 2006) A pulmonary manifestation is an extremely rare entity. We present an atypical case of DLBCL presenting as pleuritic chest pain with imaging consistent with bilateral pulmonary nodules suggestive of metastasis. Case description: A healthy 87 year old male former smoker with a history of type 2 diabetes mellitus, hypertension presented to the emergency department with pleuritic chest pain. There was no associated cough, shortness of breath, palpitations, nausea, dizziness or diaphoresis. No recent infections or B symptoms. He had a good appetite. He had prostate cancer 20 years ago and had a radical prostatectomy. Upon presentation, work up showed mild anemia (Hb 11.1 g/dL). Cardiac work up for chest pain was unremarkable. Chest X-Ray revealed mass like opacities in each lung with largest in left mid lung measuring up to 6 cm. CT chest with IV contrast revealed multiple lung nodules bilaterally with 2 largest in left upper lobe measuring 5.6 x 3.7 cm and 5.7 x 3.3 cm. Multiple other small nodules are present in both lung fields consistent with diffuse lung metastasis. No lymphadenopathy was noted. (Image 1) PET CT scan was significant for bilateral FDG avid lung lesions. No evidence of other FDG avid disease. CT guided biopsy of the left lung mass surprisingly revealed diffuse large B cell non-Hodgkin's lymphoma, non-germinal center type. (Image 1) Immuno-histochemistry showed atypical lymphocytes positively stained with CD20, BCL6, BCL2, PAX5 and MUM1. C-myc had 50% positivity. Ki67 had 90% proliferative index. FISH analysis was negative for MYC, BCL2 and BCL6; hence there was no double hit lymphoma detected. According to Lunago classification, patient had advanced stage disease with IPI (prognosis index) of 2. His ECOG PS score was 0. He was initiated on R-Pola-CHP. Given his age, Cyclophosphamide and Adriamycin doses were reduced as done with R-mini CHOP. Vincristine was avoided given neurotoxicity. Discussion: Primary pulmonary B-cell lymphomas (PP-BCLs) represent less than 3-4% of all extra-nodal NHL. They primarily affect the lung without evidence of extrapulmonary disease at the time of diagnosis. PP-DLBCL accounts for only 10% cases of PP-BCLs. ( Sanguedolce et al., 2021) Most cases have atypical presentation. Likewise, in our patient, he manifested with atypical chest pain without any B symptoms. Radiographic imaging can show variable findings of consolidation, single/multiple nodules, reticular interstitial infiltrate or bilateral ground glass opacities. In view of the insidious onset and lack of specificity in clinical symptoms, primary pulmonary lymphomas are frequently misdiagnosed as refractory pneumonia, lung abscess, asthma, primary lung cancer, or lung metastasis. ( Liu et al., 2020) Patients with DLBCL are typically treated with R-CHOP (rituximab, cyclophosphamide, doxorubicin, vincristine and prednisone). However, with this regimen, only 60% of patients are cured and the rest might relapse. In the POLARIX phase 3 trial, polatuzumab vedotin (a monoclonal antibody to CD79b ubiquitously expressed on B cells) combined with R-CHP significantly improved 2 year progression-free survival compared to the routine R-CHOP therapy in previously untreated DLBCL patients. ( Tilly et al., 2022) Given our patient's advanced stage disease, he was initiated on the Pola-R-CHP regimen. Doses of cyclophosphamide and doxorubicin were reduced as per the R-mini CHOP dose-reduced regimen for a physically fit elderly individual. Patient continues on therapy at the time of this abstract. In conclusion, primary pulmonary DLBCL is a rare entity with only a few case reports in literature. It should be considered in patients presenting with large pulmonary masses, mimicking metastatic disease.

De novo thoracic endometriosis in a menopausal women

Endometriosis is very common in reproductive age group. Since it is an estrogen dependent problem it is generally believed that it denotes “active ovarian function” and is “healed” after menopause. Most commonly endometrial tissue is found in ovaries resulting in the formation of chocolate cysts but it can also be found in extrapelvic sites. In this case report we will discuss endometriosis after menopause. Pulmonary endometriosis is a rare form of thoracic endometriosis. We describe a case of a 54-year-old woman with surgical menopause with recurrent episodes of haemoptysis, chest pain and dyspnea. Her Chest CT revealed multiple small pulmonary nodules. Biopsy from left lung lesion suggested endometriosis.

Mucosa-Associated Microbiota Dysbiosis in the Terminal Ileum Correlates With Bowel Symptoms in Diarrhea-Predominant Irritable Bowel Syndrome.

The mucosa-associated microbiota (MAM) is not as frequently studied in diarrhea-predominant irritable bowel syndrome (IBS-D) compared with the fecal microbiota. In this study, we examined the MAM in the terminal ileum and its correlation with bowel symptoms in IBS-D. Mucosal biopsies of the terminal ileum from 25 patients with IBS-D and 25 healthy controls were collected for 16S ribosomal RNA gene sequencing. Correlation analysis was performed. Compared with healthy controls, the MAM in the terminal ileum showed a decreased alpha diversity in the IBS-D cohort (Chao1 and Shannon indexes, P < 0.05). And the overall MAM profile clustered separately into 2 groups (ADONIS [PERMANOVA, permutational multivariate analysis of variance], P < 0.05). At the phylum level, the relative abundance of Proteobacteria was significantly higher in the ileal MAM of patients with IBS-D while that of Firmicutes was significantly lower. At the genus level, the relative abundance of Pseudomonas was significantly higher in the IBS-D cohort, with lower Bacteroides and Ruminococcus . Moreover, 40.0% of patients with IBS-D had multiple small nodules (nodular lymphoid hyperplasia) on the mucosal surface of the terminal ileum, which indicated a low-grade inflammation. In patients with IBS-D with nodular lymphoid hyperplasia, the changes of Pseudomonas and Bacteroides were more overt. Correlation analysis revealed that the relative abundance of Pseudomonas positively correlated with abdominal pain and the severity of IBS. Patients with IBS-D showed a dysbiosis of MAM in the terminal ileum, which may be associated with bowel symptoms. Moreover, 40.0% of them displayed mucosal low-grade inflammation, with a more severe mucosal microbial disturbance.

Common and Uncommon CT Findings in CVID-Related GL-ILD: Correlations with Clinical Parameters, Therapeutic Decisions and Potential Implications in the Differential Diagnosis

PurposeTo investigate computed tomography (CT) findings of Granulomatous Lymphocytic Interstitial Lung Disease (GL-ILD) in Common Variable Immunodeficiency (CVID), also in comparison with non-GL-ILD abnormalities, correlating GL-ILD features with functional/immunological parameters and looking for GL-ILD therapy predictive elements.MethodsCT features of 38 GL-ILD and 38 matched non-GL-ILD subjects were retrospectively described. Correlations of GL-ILD features with functional/immunological features were assessed. A logistic regression was performed to find a predictive model of GL-ILD therapeutic decisions.ResultsMost common GL-ILD CT findings were bronchiectasis, non-perilymphatic nodules, consolidations, Ground Glass Opacities (GGO), bands and enlarged lymphnodes. GL-ILD was usually predominant in lower fields. Multiple small nodules (≤10 mm), consolidations, reticulations and fibrotic ILD are more indicative of GL-ILD. Bronchiectasis, GGO, Reticulations and fibrotic ILD correlated with decreased lung performance. Bronchiectasis, GGO and fibrotic ILD were associated with low IgA levels, whereas high CD4+ T cells percentage was related to GGO. Twenty out of 38 patients underwent GL-ILD therapy. A model combining Marginal Zone (MZ) B cells percentage, IgA levels, lower field consolidations and lymphnodes enlargement showed a good discriminatory capacity with regards to GL-ILD treatment.ConclusionsGL-ILD is a lower field predominant disease, commonly characterized by bronchiectasis, non-perilymphatic small nodules, consolidations, GGO and bands. Multiple small nodules, consolidations, reticulations and fibrotic ILD may suggest the presence of GL-ILD in CVID. MZ B cells percentage, IgA levels at diagnosis, lower field consolidations and mediastinal lymphnodes enlargement may predict the need of a specific GL-ILD therapy.

Circulating tumor DNA guided therapeutic strategies for colorectal cancer patients with multiple small pulmonary-limited nodules: An open-label, prospective, phase II cohort study.

e15645 Background: The clinical diagnosis and treatment of small pulmonary nodules (suspected to be lung metastases) in advanced colorectal cancer (CRC) patients remain controversy. Previous studies have shown that tumor-informed circulating tumor DNA (ctDNA) testing with blood sample can sensitively detect recurrence or metastasis of CRC patients. This study was designed to explore the clinical value of tumor-informed ctDNA test in metastatic CRC patients with small pulmonary-limited nodules. We aimed to identify the low-risk patients for de-escalation therapy, as well as high-risk patients with no evidence of disease (NED) for escalation therapy. Methods: This is an ongoing, open-label, prospective and phase II cohort study (NCT 05495672). According to the longest diameter of the pulmonary nodule, patients were divided into two cohorts: Cohort 1(0-1cm) and Cohort 2 (1-2cm). Tumor-informed ctDNA test (OriMIRACLE S) would be performed every three months until progression or 2 years. The treatment strategy of each patient is decided by a multidisciplinary team depending on ctDNA and lung CT scan results. In general, negative minimal residual disease (MRD) patients will receive only regular follow-up. Patients with positive MRD will receive intensive examination and local treatments for pulmonary nodules such as surgery, radio frequency ablation or stereotactic body radiotherapy combined with or without systemic therapeutic treatment, aiming to achieve NED or cure. Results: From June 2022 to December 2022, a total of 26 patients were screened in the group, one patient was eliminated (with extrapulmonary metastasis), and a total of 25 patients were successfully enrolled. At present, 1 patient died (by acute intestinal perforation), 4 patients were excluded from the group (with the emergence of extrapulmonary metastasis), and a total of 20 patients were in the group. The positive rate of MRD in baseline were 4/11(36.4%) for Cohort 1 and 10/14 (71.4%) for Cohort 2, respectively. Notably, in Cohort 1, 3 of 4 patients with positive MRD at baseline were examined extrapulmonary metastasis during the follow-up examination. Conclusions: Our preliminary results suggested that the positive rate of MRD were higher in Cohort 2 than Cohort 1. Patients with pulmonary nodule less than 1 cm tended to develop extrapulmonary metastasis if baseline MRD was positive. In such circumstances, more intensive follow-up examination is suggested to prevent early disease progression. Clinical trial information: NCT05495672 .

Primary leiomyosarcoma of the inferior vena cava in a pediatric case: a case report and literature review

BackgroundLeiomyosarcoma is classified as a soft tissue sarcoma. In adults, leiomyosarcoma is the most common malignancy affecting the vascular system; however, vascular leiomyosarcoma in children is extremely rare as most pediatric soft tissue tumors are rhabdomyosarcomas. The survival rate is very low, and incomplete resection is a poor prognostic factor. There is also a high rate of distant recurrence, with the lungs and liver being the most common sites of metastasis. There is no established effective chemotherapy, and complete surgical resection is the only potentially curative treatment for leiomyosarcoma.Case presentationA 15-year-old female patient with no significant medical history presented with severe upper abdominal pain and was admitted. Abdominal contrast-enhanced computed tomography and magnetic resonance imaging showed a large retroperitoneal tumor protruding into the lumen of the inferior vena cava behind the liver and multiple small nodules, and metastasis to the liver was suspected. The tumor was 6 × 4 × 5 cm in diameter, located just behind the hepatic hilar structures, and was suspected to infiltrate into the right portal vein. The tumor was diagnosed as a leiomyosarcoma through an open tumor biopsy. As the multiple liver metastases were located only in the right lobe of the liver on imaging, we performed tumor resection with right hepatectomy and replacement of the inferior vena cava (IVC). The postoperative course was uneventful; however, on postoperative day 51, distant metastatic recurrences were found in the remaining liver and right lung. The patient was immediately started on chemotherapy and trabectedin proved to be the most effective drug in the treatment regimen; however, severe side effects, such as hepatotoxicity, prevented timely administration, and the patient passed away 19 months after surgery.ConclusionsIVC resection and reconstruction combined with right hepatectomy were able to be safely performed even in a pediatric case. To improve the prognosis of leiomyosarcoma with multiple metastases, an effective treatment strategy combining surgical treatment and chemotherapy, including molecularly targeted drugs, should be established as early as possible.

Sarcomatoid urothelial carcinoma of the renal pelvis treated with immunotherapy

BackgroundSarcomatoid carcinoma is a rare, high-grade malignancy with epithelial and mesenchymal components. It may be a good candidate for immunotherapy because it is associated with overexpression of programmed cell death ligand 1. Sarcomatoid urothelial carcinoma (UC) of the upper urinary tract is extremely rare. Here we report the first case of sarcomatoid UC of the renal pelvis that responded to immunotherapy.Case presentationA 79-year-old man was referred to our hospital complaining of various symptoms, including anorexia and abdominal pain. A computed tomography scan revealed a right atrial tumor, a 9 cm left renal mass with a renal vein tumor thrombus, para-aortic lymphadenopathy, and multiple small lung nodules. The patient underwent resection of the right atrial tumor. Pathological analysis of the tumor did not lead to an accurate diagnosis even after several rounds of immunohistochemistry. He underwent a needle biopsy of the left kidney and was initially diagnosed with collecting duct carcinoma, a rare subtype of renal cell carcinoma (RCC). Following the initial diagnosis, immunotherapy with nivolumab and ipilimumab commenced. Thereafter, almost all lesions, including the left renal tumor, were reduced in size. However, he underwent a left nephrectomy approximately a year after beginning immunotherapy due to repeated left renal bleeding. Histological examination of the nephrectomy specimen revealed two forms of cancer—sarcomatoid UC and conventional high-grade UC. Two months after surgery, the patient was found to have new lung metastases. He underwent chemotherapy with gemcitabine and cisplatin, followed by immunotherapy with pembrolizumab. However, both treatments were ineffective. The patient died of cancer 19 months after his first admission.ConclusionsThe presented case of sarcomatoid UC of the renal pelvis that partially responded to immunotherapy suggests that immunotherapy can be a promising treatment for sarcomatoid UC.

A rare case of recurrent hyperparathyroidism

Abstract A 58 year old man presented to our hospital three years ago with history of upper abdominal pain, recurrent vomiting and dehydration. On evaluation, pancreatitis with mass in the head of pancreas was diagnosed. Fine needle aspiration from pancreatic head mass was suggestive of low-grade adenocarcinoma. Whipple's procedure was planned and the surgical gastroenterology team did open pylorus resecting pancreatoduodenectomy and feeding jejunostomy for him in October 2016. But the biopsy was reported as chronic calcific pancreatitis. On workup, hypercalcemia (12.5 mg/dl) was noted and hyperparathyroidism due to right inferior parathyroid adenoma was diagnosed as PTH levels were elevated (iPTH-474 pg/ml). He had a slippage of the jejunostomy tube and was readmitted. Right inferior parathyroidectomy was done by the surgical oncologist along with the repair of slipped feeding jejunostomy tube by the surgical gastroenterology team in the same sitting in March 2017. Biopsy was reported as right inferior parathyroid adenoma. Post-surgery, he had hungry bone syndrome which stabilized after one month. After 6 months, he started noticing a small nodule in the right side of the neck which progressively increased in size from a small nodule to a size of 1*1 cm over a duration of one year, associated with multiple small nodules superiorly. He also complained of weight loss, constipation, generalized weakness, and bone pains. Hypercalcemia was noted and iPTH level was again elevated (564.7 pg/ml). He was admitted for evaluation for recurrence of parathyroid adenoma/ parathyroid carcinoma/ parathyromatosis. Ultrasonography of the neck showed a 4*3 mm hypoechoic lesion with internal vascularity noted posterior to right lobe of thyroid and medial to great vessels, and multiple heterogenous hypoechoic well-defined lesions noted in the subcutaneous plane on the right side of the neck, few noted along surface of right strap muscle and sternocleidomastoid muscle. FNAC of the subcutaneous nodule was suggestive of parathyroid adenoma. Parathyroid scan showed multiple MIBI-avid nodule seen anterior and lateral to inferior pole of thyroid gland. He was operated in February 2019 and five nodules were removed. Biopsy was suggestive of parathyroid carcinoma. After the surgery, his symptoms improved but Calcium(13mg/dl) and iPTH (453 pg/ml) levels were elevated at 3 months follow up. Biopsy was suggestive of parathyroid carcinoma. But in view of the previous surgery, earlier biopsy report of adenoma and no metastasis, he was diagnosed to have parathyromatosis. A follow up CT revealed residual nodules in the neck. A repeat surgery was done in August 2019 to remove the nodules as the patient complained of persistent generalized malaise and bone pains. Following surgery, his iPTH levels remained elevated (iPTH pre-op-492.4 pg/ml; post-op- 444.4 pg/ml). His symptoms improved mildly. He was started on medical management for hypercalcemia with Cinacalcet. Figure 1. Image of the neck of the patient showing multiple nodules.

Multinodular gastro intestinal stromal tumor in small intestine with perforation and Meckel’s diverticulitis- An unusual presentation

Gastrointestinal Stromal tumor are rare group of mesenchymal neoplasms originating from the wall of the digestive tract. Outcome strongly dependent on tumor size and mitotic activity. A 60-year male presented with abdominal pain and distention for 3 months. After initial evaluation, emergency laparotomy was done. Intraoperatively found to have multiple small nodules in peritoneum and mesenteric border of small intestine and rupture of a big nodule was observed causing peritonitis. Gross examination-Multiple nodular tumour mass in small intestine along with multiple haemorrhagic nodule of varying size seen in mesentry. Tumor arising from muscular layer arranged in interlacing fascicles, bundles, whorls and sheets. Tumor cells are elongated having spindle to elongated nucleus with vesicular to dense chromatin and inconspicuous nucleoli. Some of the nodules shows areas of haemorrhage and necrosis. Mitotic figure &amp;#62; 5/50 HPF seen. Histological grade-G2 High grade &amp;#62;5/50 HPF, TNM grade-PT4PNxPMx. Immunohistochemistry-Diffusely Positive for CD117. Focally positive for DOG1, CD34 &amp; SMA. Small intestinal GISTs comprise approximately 30% of all GISTs. Tumor size vary from small nodules to complex masses of around 20 cm. GIST is more common in patients with age &amp;#62;40 years. CD117 and DOG1 positivity are used to define these tumors. Novelty: Only few cases has been reported in small bowel GIST with perforation and multiple nodules.

Characterization of the mucosal microbiota in patients with nodular lymphoid hyperplasia with concurrent irritable bowel syndrome compared to healthy controls.

Nodular lymphoid hyperplasia (NLH) is known as a lymphoproliferative lesion in which multiple small nodules appear on the intestinal wall. It has been documented that patients who struggle with irritable bowel syndrome (IBS) are at greater risk of developing NLH. Here, we aimed to investigate the previously reported pathogens and the abundance of a selection of mucosal microbiota in IBS + NLH patients compared to IBS, and healthy controls. Terminal ileum biopsies were collected from 37 IBS + NLH, 37 IBS, and 29 healthy controls. Bacterial culture and PCR was performed to detect the presence of pathogens in biopsies. A qPCR assay was applied to assess the abundance of a selection of bacterial taxa. Totally, five bacterial isolates including two enteropathogenic and one enteroaggregative Escherichia coli (EPEC, EAEC), one enterotoxigenic Staphylococcus aureus (SEA), and one Yersinia enterocolitica strains were detected among the IBS + NLH cases. The relative abundance of Bacteroidetes and Streptococcus spp. in IBS + NLH patients was significantly less than IBS and healthy controls. Firmicutes, Pseudomonas spp., Haemophilus spp., and Campylobacter spp. were notably more abundant in IBS + NLH than in IBS patients. The abundance of Verrucomicrobia was higher in NLH + IBS than in healthy controls. Actinobacteria was also significantly more abundant among NLH + IBS patients than the controls. Our results demonstrated that mucosal microbiota composition in NLH + IBS patients slightly differs from that of IBS patients and healthy controls. Further research using large-scale cohorts are needed to enhance current understanding of the contribution of the mucosal microbiota to NLH pathogenesis with concurrent IBS.

Cutaneous metastasis of carcinomatous component of ovarian carcinosarcoma: A case report and review of the literature

Skin metastasis of ovarian cancer is extremely rare. We report an unusual case of ovarian carcinosarcoma with cutaneous metastasis of carcinomatous component that displayed distinct clinical manifestation. A 48-year-old woman presented to the dermatologist complaining of a new onset of erythematous, plaque-like skin rash with multiple small nodules on the left inner thigh, the area measuring 8 × 5cm. While the patient had no history of dermatologic conditions, she underwent a total hysterectomy and bilateral salpingo-oophorectomy, omentectomy, and lymph node dissection 16 months ago with a pathology confirmed stage IIIC ovarian carcinosarcoma. Of note, the carcinomatous component, mainly adenocarcinoma with hybrid features of seromucinous, endometrioid and minor high-grade serous carcinoma, involved bilateral fallopian tubes, omentum, and parametrium with extensive lymph node metastases. A skin biopsy specimen revealed an adenocarcinoma involving epidermis, dermis, and subcutaneous tissue with nodular contours, consistent with metastatic carcinomatous component of carcinosarcoma. Both carcinomatous component of primary ovarian carcinosarcoma and metastatic adenocarcinoma in the skin demonstrated Pax8, WT-1, and ER positivity and a mutation pattern of p53. The patient passed away 15 months after identification of skin metastasis. This case represents a unique example of cutaneous metastasis of ovarian carcinosarcoma with distinct clinical manifestation and detailed histopathological description. Alertness to the possibility of cutaneous metastasis, in combination with clinical history, morphological and immunohistochemical findings, is critical for a definitive classification.