Publications by authors named "Susama Patra"

51 Publications

Extra-Ventricular C11orf95-RELA Fusion Positive Anaplastic Ependymoma with Divergent Histological and Immunohistochemical Features: Report of a Rare Case.

Neurol India 2021 Jul-Aug;69(4):1060-1062

Department of Pathology and Neurosurgery, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

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http://dx.doi.org/10.4103/0028-3886.325311DOI Listing
September 2021

Ergotamine Induced Retroperitoneal Fibrosis.

Mediterr J Rheumatol 2021 Jun 30;32(2):168-173. Epub 2021 Jun 30.

Department of Gastroenterology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

Retroperitoneal fibrosis (RPF) is an uncommon disease characterised by the presence of fibroinflammatory reaction which starts around the infrarenal portion of the abdominal aorta in the retroperitoneum and frequently entrap the ureter causing obstructive uropathy. Approximately, two thirds of the cases are idiopathic, where aetiopathogenesis is not known. Ergotamine-induced RPF, although rare, is considered under secondary group. The fibrogenic process here is thought to be due to serotonergic activity. We report a case of RPF in a young female with obstructive uropathy who had history of long-term ergotamine intake for migraine. Histopathological evaluation revealed different evolving stages of necrotising vasculitis. In addition, the patient has responded to withdrawal of offending drug along with immunosuppressive therapy. We believe, apart from serotonergic activity, ergotamine can lead to RPF through a vasculitic process which has not been reported earlier.
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http://dx.doi.org/10.31138/mjr.32.2.168DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8369278PMC
June 2021

Bone marrow trephine immunohistochemistry is useful in characterizing malignancy-associated myelonecrosis: A retrospective observational study.

Int J Lab Hematol 2021 Aug 3. Epub 2021 Aug 3.

Department of Medical Oncology/Hematology, All India Institute of Medical Sciences, Bhubaneswar, India.

Objective: We aim to describe the utility of immunohistochemistry (IHC) in characterizing malignancy-associated myelonecrosis (MN) on bone marrow trephine biopsies (BMBx) as a part of initial workup.

Materials And Methods: Patten and intensity of antigenic immunoexpression in necrotic tumor cells on BMBx were evaluated in a series of cases using standardized avidin-biotin-complex immunoperoxidase technique after heat-induced epitope retrieval and compared the same with viable tumor cells wherever available.

Results: Fifteen out of 2494 (0.6%) cases (median age: 28 years; range: 4 to 66 years) had evidence of MN (extensive in eight, moderate in five, and focal in two) secondary to hematological (N = 9) and solid (N = 6) malignancies. Five (33.3%) had pancytopenia, and eight (53.3%) had difficult and/or hemodiluted aspirate. Antigenic expression for CD10, CD79a, CD3, CD7, and CD20 was retained by necrotic leukemic blasts or lymphoma cells; CD34, TdT, and PAX5 showed heterogeneous expression; and a weak Golgi zone (dot like) CD30 positivity was noted in Reed-Sternberg (RS) or RS-like giant cells. Necrotic epithelial metastases retained pancytokeratin in all and showed variable positivity for prostate-specific antigen, carcinoembryonic antigen, CK20, ER, PR, and GATA3. Necrotic neuroblastomas (N = 2) retained positivity for synaptophysin and chromogranin, whereas retained nuclear positivity for NKX2.2 in necrotic Ewing family of tumor (N = 1) aided in early diagnosis.

Conclusion: Myelonecrosis may retain tumor antigenicity, and immunohistochemistry using selected panel of antibodies should be tried in such challenging cases for an early presumptive diagnosis and further decision making.
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http://dx.doi.org/10.1111/ijlh.13671DOI Listing
August 2021

Pure primary ovarian carcinoid tumor with carcinoid syndrome and cervical carcinoma: A rare concoction of dual primary malignancies.

Indian J Pathol Microbiol 2021 Jul-Sep;64(3):563-567

Department of Pathology and Laboratory Medicine, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

Carcinoid tumors are defined as rare slow-growing neuroendocrine tumors. A majority of primary ovarian carcinoids occur in association with mature cystic teratoma or are metastatic to the ovary. A 48-year-old post-menopausal woman presented with progressive facial puffiness, and intractable diarrhea. Radiological imaging suggested a 10 × 9 × 9.2 cm right ovarian mass. She underwent a total abdominal hysterectomy and bilateral salpingo-oophorectomy. Histopathological examination revealed primary pure carcinoid tumor of the ovary with the synchronous presence of early invasive squamous cell carcinoma of the cervix, along with the icthyosis uteri. The patient is doing well on 5 years of follow-up post-surgery without any recurrence or metastasis. In this report, we intend to highlight the rare association of cervical carcinoma and ichthyosis uteri with this tumor. In addition, we present a short review of the literature, over a decade of ovarian carcinoids associated with carcinoid heart disease.
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http://dx.doi.org/10.4103/IJPM.IJPM_1379_20DOI Listing
August 2021

Management of Helicobacter pylori infection: The Bhubaneswar Consensus Report of the Indian Society of Gastroenterology.

Indian J Gastroenterol 2021 Jul 5. Epub 2021 Jul 5.

Department of Surgery, Jawaharlal Institute of Postgraduate Medical Education and Research, Pondicherry, 605 006, India.

The Indian Society of Gastroenterology (ISG) felt the need to organize a consensus on Helicobacter pylori (H. pylori) infection and to update the current management of H. pylori infection; hence, ISG constituted the ISG's Task Force on Helicobacter pylori. The Task Force on H. pylori undertook an exercise to produce consensus statements on H. pylori infection. Twenty-five experts from different parts of India, including gastroenterologists, pathologists, surgeons, epidemiologists, pediatricians, and microbiologists participated in the meeting. The participants were allocated to one of following sections for the meeting: Epidemiology of H. pylori infection in India and H. pylori associated conditions; diagnosis; treatment and retreatment; H. pylori and gastric cancer, and H. pylori prevention/public health. Each group reviewed all published literature on H. pylori infection with special reference to the Indian scenario and prepared appropriate statements on different aspects for voting and consensus development. This consensus, which was produced through a modified Delphi process including two rounds of face-to-face meetings, reflects our current understanding and recommendations for the diagnosis and management of H. pylori infection. These consensus should serve as a reference for not only guiding treatment of H. pylori infection but also to guide future research on the subject.
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http://dx.doi.org/10.1007/s12664-021-01186-4DOI Listing
July 2021

associated gastritis: Report of a rare case.

Indian J Pathol Microbiol 2021 Jun;64(Supplement):S97-S99

Department of Pathology and Lab Medicine, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

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http://dx.doi.org/10.4103/IJPM.IJPM_629_20DOI Listing
June 2021

Diminution of the Primary Cilia From the Intrahepatic Cholangiocytes in a Pediatric Choledochal Cyst.

Appl Immunohistochem Mol Morphol 2021 Jun 3. Epub 2021 Jun 3.

Departments of Pathology and Lab Medicine Pediatric Surgery Pediatrics, AIIMS, Bhubaneswar, Odisha, India.

Choledochal cyst (CC) is a cystic disease predominantly involving the extrahepatic biliary tree. Biliary atresia (BA), another disorder of the extrahepatic biliary tree, is sometimes considered to be in the same spectrum as pediatric CC. Recently, the absence and/or the structural abnormality of the primary cilia in the intrahepatic cholangiocytes have been implicated in the pathogenesis of BA. We aimed to evaluate the expression of primary ciliary proteins in the intrahepatic cholangiocytes in cases of pediatric CC and compare it with normal control and BA. We performed immunohistochemistry for primary ciliary proteins (acetylated-α-tubulin and double-cortin domain containing 2) on the liver biopsies of control liver (n=5), pediatric CC (n=13), and BA (n=14). We also compared the expression with various clinical, biochemical, histopathologic (portal fibroinflammation and ductal plate malformation), and immunohistochemical (proliferative index) data. There was significant loss of primary cilia from the intrahepatic cholangiocytes in cases of CC and BA as compared with the normal control by both immunostains (CC: P=0.003 and 0.001, respectively; BA: P=0.001 and 0.001, respectively). There was no significant difference between the CC and BA in terms of ciliary protein loss. The loss of the ciliary proteins occurred irrespective of the proliferative (MIB-1 labeling) index, portal fibroinflammation, or ductal plate malformation. The loss of cilia did not correlate with the clinical follow-up in cases of pediatric CC. The loss of primary cilia from the intrahepatic cholangiocytes may be crucial in the etiopathogenesis of pediatric CC.
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http://dx.doi.org/10.1097/PAI.0000000000000953DOI Listing
June 2021

The Clinico-histopathological Spectrum of Tumors and Tumor-Like Lesions in Adult Intussusception.

J Gastrointest Cancer 2021 May 20. Epub 2021 May 20.

Dept of Pathology and Lab Medicine, AIIMS, Bhubaneswar, PIN-751019, India.

Background: Intussusception is an uncommon cause of intestinal obstruction in adults. The etiology of this disease differs from the children. Thus, its management depends on the possible etiology and is different from pediatric cases.

Aims: We aimed to study the clinico-histopathological spectrum of the tumors and tumor-like lesions in the intussusception in adults.

Material And Methods: A retrospective review of the adult (> 18 years) intussusception cases was performed. The clinical data and follow-up were obtained. The histopathology was reviewed along with the special stains and immunohistochemistry for ascertaining a histopathological diagnosis.

Results: Fifteen cases of adult intussusception were identified from 107 resected specimens of adult intestinal obstruction. The mean age was 44.5 years with a male/female ratio of 1.1:1. A definitive pathology could be ascertained in 80% of the cases (n = 12/15). Eight cases had benign non-neoplastic etiology (53.3%) (33.3% tumor-like lesions) while seven cases (46.7%) had neoplastic etiology (20% benign neoplastic; 26.7% malignant neoplastic). All cases of colonic or enterocolic intussusceptions were associated with neoplasia whereas 90% of the enteric intussusceptions occurred due to benign non-neoplastic causes.  CONCLUSIONS: Non-neoplastic causes are predominant in the enteric intussusceptions while neoplastic causes are more commonly associated with colonic or enterocolic intussusceptions. The post-operative histopathological examination concludes on the adequacy of the index surgery or the provision of further management if required.
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http://dx.doi.org/10.1007/s12029-021-00647-5DOI Listing
May 2021

Minimally invasive esophagectomy: Preservation of arch of Azygos vein in prone position.

J Minim Access Surg 2021 Jul-Sep;17(3):405-407

Department of Radiodiagnosis, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

Minimally invasive esophagectomy (MIE) for oesophageal cancer has gained wide popularity in recent years due to its improved morbidity and mortality outcomes. We describe our modified technique of MIE in prone position with preservation of the arch of azygos vein. In our experience with 14 patients, the mean operative duration was 378 min (standard deviation [SD] 378 ± 59 min) and the mean blood loss was 390 ml (SD 390 ± 142 ml). The mean lymph node count was 28 (range 17-54). The Visick score was I in 12 (85.7%) patients and II in 2 (14.3%) patients at follow-up. The preservation of azygos vein arch is a technically feasible procedure and may be associated with a better quality of life outcome.
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http://dx.doi.org/10.4103/jmas.JMAS_267_20DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8270036PMC
April 2021

Sigmoid Colonic Stricture Mimicking Hirschsprung Disease in a Cytomegalovirus Positive Immunocompetent Infant.

Indian J Pediatr 2021 07 20;88(7):724. Epub 2021 Apr 20.

Department of Pediatric Surgery, All India Institute of Medical Sciences, Bhubaneswar, Odisha, 751019, India.

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http://dx.doi.org/10.1007/s12098-021-03730-8DOI Listing
July 2021

Krukenberg Tumor of Ovary During Pregnancy: Learning From A Mistake.

Int J Gynecol Pathol 2020 Nov 24. Epub 2020 Nov 24.

Departments of Pathology and Lab Medicine (S.J., S.M., S.P.) Obstetrics and Gynecology, (P.S., S.K.J.) AIIMS, Bhubaneswar, Odisha, India.

Sertoli-Leydig cell tumor of the ovary with heterologous differentiation is a relatively uncommon tumor that occurs in females of variable age range. Krukenberg tumor (KT) is a relatively more common tumor of the ovary although only a few cases of KT occur during pregnancy making it an equally uncommon tumor. We received a unilateral ovarian mass in a 25-yr-old primigravida which we reported as Sertoli-Leydig cell tumor with heterologous (intestinal) differentiation based on its clinical and histomorphologic features. However, on further investigations, a gastric mass was found which turned out to be signet-ring cell adenocarcinoma. We rectified our diagnosis of ovarian mass as KT. We retrospectively analyzed the reasons for our mistake and concluded that the rarity coupled with the nonclassic clinical features and histomorphology of KT during pregnancy pose definite challenges to the correct diagnosis. This report highlights the diagnostic challenges faced by us along with the ways to circumvent them in the future.
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http://dx.doi.org/10.1097/PGP.0000000000000733DOI Listing
November 2020

Concomitant echinoderm microtubule-associated protein-like 4-anaplastic lymphoma kinase rearrangement and epidermal growth factor receptor mutation in non-small cell lung cancer patients from eastern India.

J Cancer Res Ther 2020 Jul-Sep;16(4):850-854

Department of Pathology, AIIMS, Bhubaneswar, Odisha, India.

Background: In non-small cell lung cancer common driver mutations such as epidermal growth factor receptor (EGFR) and anaplastic lymphoma kinase (ALK) are usually mutually exclusive. This study aimed to elucidate the concurrence of EGFR mutation and ALK rearrangement in eastern India patients with primary lung adenocarcinoma and assess the response of EGFR tyrosine kinase inhibitor (TKI) therapy after 6 months in primary lung adenocarcinoma.

Methods: We retrospectively analyzed 198 adenocarcinomas for EGFR and ALK mutations. EGFR and ALK tests were done by real-time polymerase chain reaction and immunohistochemistry (IHC) techniques, respectively. Radiological response was assessed by Response Evaluation Criteria in Solid Tumors (version 1.1).

Results: EGFR/ALK co-alteration was found in 4 adenocarcinoma patients. All were males with advanced disease. Younger patients had exon 19 deletion whereas older ones showed exon 21 mutation. The initial option of ALK-TKI in all four patients was excluded straightaway due to the high-cost burden of ALK-TKI. Two of them showed a partial response while other two had stable disease after 6 months of EGFR TKI therapy.

Conclusion: EGFR/ALK co-alterations in adenocarcinomas albeit rare do exist. The challenge of monetary hurdle in developing countries with ALK TKI therapy can be handled by giving only EGFR TKI in these cases of concomitant mutations. Future perspective in research could be finding an agent with the potential of dual inhibition of ALK and EGFR.
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http://dx.doi.org/10.4103/jcrt.JCRT_678_18DOI Listing
November 2020

Expression of CD56 is Not Limited to Biliary Atresia and Correlates with the Degree of Fibrosis in Pediatric Cholestatic Diseases.

Fetal Pediatr Pathol 2020 Jun 8:1-11. Epub 2020 Jun 8.

Pathology and Lab Medicine, All India Institute of Medical Sciences Bhubaneswar, Bhubaneswar, India.

: CD56 immunostain is used as an adjunct to aid in the preoperative diagnosis of biliary atresia (BA) by liver biopsy. We aimed to study the expression of CD56 in different pediatric cholestatic diseases thereby evaluating its utility in the diagnosis of BA.: We performed immunohistochemistry for CD56 on 35 cases of pediatric cholestatic diseases and five age-matched controls. CD56 expression was assessed by a multiplication score (percentage positivity x intensity) in the biliary epithelium.: The multiplication score between BA and choledochal cyst was not significantly different. High scores were also encountered in other cholestatic disorders. The score showed a significant negative association with serum albumin and a significant positive correlation with the serum ALT level. Very significant positive correlation between the score and portal fibrosis was obtained.: CD56 expression is an infidel marker for the histological diagnosis of BA and rather provides a clue to the disease status in pediatric cholestatic diseases.
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http://dx.doi.org/10.1080/15513815.2020.1765917DOI Listing
June 2020

Acquired Pure Red Cell Aplasia Following Recombinant Erythropoietin (Darbepoetin-alfa) Therapy.

Indian J Nephrol 2020 Mar-Apr;30(2):113-116. Epub 2019 Sep 6.

Department of Pathology and Laboratory Medicine, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

Acquired pure red cell aplasia (PRCA) following use of recombinant erythropoietin (rEPO) is distinctly rare and sporadically reported in the literature. We discuss a case of PRCA following the usage of rEPO (darbepoetin-α) during the management of anemia of chronic kidney disease in an elderly male subject with review of literature and a brief insight into proposed pathophysiologic mechanism, diagnosis, and management.
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http://dx.doi.org/10.4103/ijn.IJN_129_19DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7132853PMC
September 2019

Histopathologic Spectrum of Neuromuscular and Vascular Hamartoma With Special Reference to the Vasculopathic Phenomenon.

Int J Surg Pathol 2020 Jun 24;28(4):382-392. Epub 2019 Nov 24.

All India Institute of Medical Sciences, Bhubaneswar, India.

Neuromuscular and vascular hamartoma (NMVH) is an unusual lesion presenting as intestinal obstruction by stricture formation. It is characterized by a hamartomatous mass comprising haphazardly arranged mesenchymal tissue native to the intestinal mucosa and submucosa. We aimed to characterize the clinicohistopathological spectrum of NMVH in adult subjects with a search for an etiological clue. We reviewed 84 resected specimens (adult cases) of intestinal obstruction in our institute and diagnosed 4 cases with NMVH. A panel of special stains (Masson trichrome, Verhoeff-van-Gieson, and periodic acid-Schiff) and immunohistochemistry (smooth muscle actin, S100, Bcl, CD34, vimentin, desmin, CD117, and CD3) were performed in all cases. All cases of NMVH showed characteristic hamartomatous mounds comprising haphazardly arranged smooth muscle, nerves, ganglia, vessels, and collagen with overlying mucosal ulceration. Adjacent bowel showed submucosal fibrosis, muscularis mucosae thickening, and duplication along with vasculopathy. A typical vasculopathy ("vessel-in-vessel" appearance) was seen in the submucosal and/or subserosal veins. Besides, different other forms of vasculopathic changes like obliterative venopathy and concentric myohypertrophy were also seen. One case had vasculitis and the patient died despite successful surgery. One other case was associated with lymphocytic ganglioneuronitis and granulomatous etiology. We conclude that NMVH can be multifactorial in origin although ischemia resulting from vasculopathy appears to be directly causative. The characteristic vasculopathy in the submucosal location may aid in the diagnosis of NMVH in small biopsy samples.
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http://dx.doi.org/10.1177/1066896919890129DOI Listing
June 2020

Regional variation of human pancreatic islets dimension and its impact on beta cells in Indian population.

Islets 2019 19;11(6):141-151. Epub 2019 Nov 19.

Department of Anatomy, All India Institute of Medical Sciences, Bhubaneswar, India.

: Islet of Langerhans, the endocrine pancreas plays a significant role in glucose metabolism. Obesity and insulin resistance are the major factors responsible for beta cell dysfunction. Asian Indian population has increased susceptibility to diabetes in spite of having lower BMI. The morphology of islets plays a significant role in beta cell function. The present study was designed for better understanding the morphology, composition and distribution of islets in different parts of the pancreas and its impact on beta cell proportion. : We observed islet morphology and beta cell area proportion by Large-scale computer-assisted analysis in 20 adult human pancreases in non-diabetic Indian population. Immunohistochemical staining with anti-synaptophysin and anti-insulin antibody was used to detect islet and beta cells respectively. Whole slide images were analyzed using ImageJ software. : Endocrine proportion were heterogeneously increasing from head to tail with maximum islet and beta cell distribution in the tail region. Larger islets were predominately confined to the tail region. The islets in Indian population were relatively smaller in size, but they have more beta cells (20%) when compared to American population. : The beta cells of larger islets are functionally more active than the smaller islets via paracrine effect. Thus, reduction in the number of larger islets may be one of the probable reasons for increased susceptibility of Indians to diabetes even at lower BMI. Knowledge about the regional distribution of islets will help the surgeons to preserve the islet rich regions during surgery.
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http://dx.doi.org/10.1080/19382014.2019.1686323DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6930023PMC
May 2020

Pleomorphic hyalinizing angiectatic tumor of the mesorectum: a rare case and review of literature.

Autops Case Rep 2019 Jul-Sep;9(3):e2019097. Epub 2019 Jul 19.

All India Institute of Medical Sciences, Department of Anesthesiology. Bhubaneswar, Orissa, India.

Pleomorphic hyalinizing angiectatic tumor (PHAT) is a rare tumor of uncertain tissue origin. Although it has been classified as a benign tumor under the WHO classification, it is locally aggressive, and multiple recurrences have been reported. PHAT commonly involves the lower extremities; however, various unusual sites of origin have been reported. We present the case of a 30-year-old female with dysmenorrhea, who presented a presacral mass on imaging. The core biopsy confirmed the diagnosis of PHAT. She underwent laparotomy and excision. Histopathology confirmed the presence of a tumor comprised of aggregates of ectatic vessels with perivascular hyalinization. An immunohistochemical study showed diffuse CD34 positivity, but S100, MDM2, and smooth muscle actin negativity. After surgical procedures, the patient is disease free as at the 12-month follow-up. Only 120 cases have been published in the English literature to date. Our study is only the third case of PHAT arising from the pelvis to be reported. Though considered to be a rare condition, the diagnosis of PHAT should always be considered in the differential diagnosis of well-defined hypervascular soft tissue mass in the pelvis. The typical histopathological findings along with immunohistochemistry should clinch the diagnosis.
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http://dx.doi.org/10.4322/acr.2019.097DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6655857PMC
July 2019

Epithelioid Malignant Peripheral Nerve Sheath Tumor (Epithelioid-MPNST) Presenting as Bleeding Rectal Polyp: A Case Report with Systematic Literature Review.

Am J Case Rep 2019 Aug 10;20:1175-1181. Epub 2019 Aug 10.

Department of Gastroenterology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

BACKGROUND Malignant peripheral nerve sheath tumor (MPNST) with pure epithelioid histology MPNST (epithelioid-MPNST) is a distinctly rare variant of MPNST described by World Health Organization. Occurrence of MPNST in the tubular gut is extremely rare with nearly 20 cases reported in the last 40 years. CASE REPORT In this case report, we described 2 such cases of epithelioid-MPNST occurring in a 65-year male and 47-year-old female, who both presented with bleeding rectal polyps. We also report on our comprehensive systematic literature review. CONCLUSIONS To the best of our knowledge, these are possibly the first two cases of pure epithelioid-MPNST at an unusual site. This needs to be differentiated from lesions more commonly encountered at the related anatomical site, such as malignant melanoma, poorly differentiated carcinoma, lymphoma, or even a gastrointestinal stromal tumor, by appropriate panel of antibodies.
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http://dx.doi.org/10.12659/AJCR.916251DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6698063PMC
August 2019

Malignant Extra-Gastrointestinal Stromal Tumor of the Mesentery.

Surg J (N Y) 2019 Jul 1;5(3):e65-e68. Epub 2019 Aug 1.

Department of General Surgery, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

Gastrointestinal stromal tumors (GISTs), the commonest mesenchymal tumors of gastrointestinal tract are often described to take origin from the interstitial cells of Cajal (ICC) or its precursor cells. Rarely these tumors do arise in structures other than the alimentary tract like omentum, mesentery, retroperitoneum, etc., of varying malignant potential and are known as extra-gastrointestinal stromal tumors (eGISTs). This is a case report of a 70-year-old female with multicentric malignant eGISTs arising in the mesentery of ileum. On laparotomy, a large mass of 20 × 15 cm was found in the small bowel mesentery without involvement of the adjacent ileum, with multiple other small nodules resembling lymph nodes, present adjacent to it. Histopathological study of the excised lump, confirmed the mass to be malignant eGIST without involvement of the adjacent ileum, with cluster differentiation (CD)117 positive and of high-risk stratification. The mesenteric nodule was confirmed on histopathology to be malignant eGIST, similar to that of that of the primary, without any lymphoid tissue. Adjuvant imatinib mesylate treatment was started immediately postoperation with the patient doing well at 1 year of follow-up. We report this case, due to the rare occurrence of multifocal malignant eGISTS of small bowel mesentery, which is yet to be reported. The existing literature is unclear regarding the clinicopathology and management of multifocal malignant stromal tumors of the mesentery.
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http://dx.doi.org/10.1055/s-0039-1693040DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6675587PMC
July 2019

Neonatal hemochromatosis in a newborn with Down syndrome.

Fetal Pediatr Pathol 2020 Feb 19;39(1):62-70. Epub 2019 Jun 19.

Pathology and Lab Medicine, AIIMS Bhubaneswar, Bhubaneswar, India.

: Neonatal hemochromatosis (NH) is a cause of neonatal/pediatric acute liver failure. Liver dysfunction/failure in Down syndrome had been described in relation to increased susceptibility to infection and transient myeloproliferative disease (TMD). The occurrence of NH in Down syndrome is described in only a few case reports. : A complete autopsy have been performed in a 79-day-old infant with severe liver dysfunction. TMD was suspected antemortem following a report of peripheral blood leukocytosis with 14% atypical cells. : The complete autopsy revealed NH-phenotype to be the cause of liver dysfunction and subsequent death. : Though TMD is a common cause of liver dysfunction in Down syndrome, NH should also be considered in its differential.
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http://dx.doi.org/10.1080/15513815.2019.1627630DOI Listing
February 2020

Cystic biliary atresia or atretic choledochal cyst: A continuum in infantile obstructive cholangiopathy.

Fetal Pediatr Pathol 2019 Dec 17;38(6):477-483. Epub 2019 Jun 17.

Pediatric Surgery, All India Institute of Medical Sciences, Bhubaneswar, India.

Both cystic biliary atresia and choledochal cyst present as infantile obstructive cholangiopathy. : We detail an infant with congenital biliary dilatation and obstructive cholangiopathy where clinicoradiological features (antenatally imaged subhepatic cyst, early onset jaundice, intrahepatic dilated biliary radicals) suggested a choledochal cyst but operative findings (lack of a distal communication of cyst with duodenum) and histomorphological features (cicatricial collagen and myofibroblastic hyperplasia in the cyst wall; ductal plate malformation, ductular cholestasis and strong expression of CD56 in the liver) were those associated with biliary atresia. : The observations support the contention that BA and CC may be interim entities in a continuum of manifestation of the same pathology.
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http://dx.doi.org/10.1080/15513815.2019.1627621DOI Listing
December 2019

Meningioma associated with Gorlin-Goltz syndrome and a short review of literature.

Neurol India 2019 Mar-Apr;67(2):595-598

Department of Pathology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

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http://dx.doi.org/10.4103/0028-3886.257991DOI Listing
December 2019

Diagnostic challenge in a metastatic pulmonary adenocarcinoma with neuroendocrine differentiation to breast and skin.

Cytopathology 2019 09 14;30(5):541-544. Epub 2019 May 14.

Department of Pathology and Lab Medicine, AIIMS, Bhubaneswar, India.

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http://dx.doi.org/10.1111/cyt.12703DOI Listing
September 2019

Solitary vallecular neurofibroma presenting with respiratory obstruction: an unusual presentation.

BMJ Case Rep 2018 Oct 7;2018. Epub 2018 Oct 7.

Department of Pathology, All India Institute of Medical Sciences Bhubaneswar, Bhubaneswar, Odisha, India.

An occurrence of vallecular neurofibroma with airway obstruction is extremely rare and to the best of our knowledge, no case report has been documented in the literature to date. Although microlaryngeal surgery with cold knife excision is the standard surgical procedure offered to patients of vallecular lesions, it can be successfully managed with the help of endoscopic coblation as later can provide a bloodless surgical field preventing the unmet need for the tracheostomy and reducing the postoperative morbidities.
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http://dx.doi.org/10.1136/bcr-2018-225330DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6194364PMC
October 2018

Hemophagocytic lymphohistiocytosis: A rare, potentially fatal complication in subcutaneous panniculitis like T cell lymphoma.

Indian J Dermatol Venereol Leprol 2019 Sep-Oct;85(5):481-485

Department of Dermatology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

Subcutaneous panniculitis-like T cell lymphoma is a rare subtype of cutaneous lymphomas with distinct clinical, histological and immunophenotypic characteristics, as well as an indolent clinical course. Rarely, it may be complicated with hemophagocytic lymphohistiocytosis: a hyperinflammatory syndrome which, if not diagnosed early, carries a dismal outcome. In this article, we describe a case of subcutaneous panniculitis-like T cell lymphoma in a middle-aged female patient which was complicated with secondary hemophagocytic lymphohistiocytosis with a favorable outcome following etoposide-based therapy. The various histological mimics of subcutaneous panniculitis-like T cell lymphoma and the management options are also briefly discussed.
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http://dx.doi.org/10.4103/ijdvl.IJDVL_277_17DOI Listing
May 2020

Oropharyngeal paraganglioma presenting with stridor: an unusual presentation.

BMJ Case Rep 2018 Jul 3;2018. Epub 2018 Jul 3.

Department of Pathology, AIIMS, Bhubaneswar, Odisha, India.

Head and neck paraganglioma is a rare disease to encounter in clinical practice and involvement of the base of the tongue in the oropharynx is yet to be described in the current literature. Although various surgical approaches have been described in the literature, transcervical mandibular swing is an effective technique for adequate exposure and complete excision of the oropharyngeal paraganglioma with adequate control on bleeding especially when it is assisted by coblation.
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http://dx.doi.org/10.1136/bcr-2018-225607DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6040529PMC
July 2018

Primary pulmonary synovial sarcoma: A reappraisal.

J Cancer Res Ther 2018 Apr-Jun;14(3):481-489

Department of Pulmonary Medicine, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

Synovial sarcoma (SS) is a malignant mesenchymal tumor with variable epithelial differentiation that affects mostly young adults and can arise at any anatomic site. Primary intrathoracic SS is very rare accounting for <0.5% of all lung tumors. Most commonly, it arises from the lung followed by pleura and mediastinum. Primary pulmonary SS (PPSS) affects both sexes equally with no preference for any hemithorax. The morphology, immunostaining properties, cytogenetic features, and management strategy of PPSS are similar to that of soft tissue SS. Histologically, there are two main types of SS - monophasic and biphasic with a feature of poor differentiation seen in both types. Most patients present with large intrathoracic masses with or without ipsilateral pleural effusion. Bone invasion or mediastinal adenopathy is very rare. SS is characterized by a specific chromosomal translocation producing SS18-SSX fusion gene in more than 90% of cases. Identification of this fusion gene remains the gold standard for the diagnosis in the presence of consistent histology and immunophenotype. Multimodality treatment including wide excision, chemotherapy, and radiotherapy is the mainstay of therapy. SS is relatively chemosensitive, and ifosfamide-based regimen showed improved survival in metastatic disease. Generally, SS is considered as high-grade tumors with a poor prognosis. Novel therapies targeted at fusion oncogene, SS18-SSX-derived peptide vaccine, epidermal growth factor receptor, and vascular endothelial growth factor are the future hope in SS. We describe a prototype case and present an elaborate review on primary SS of lung.
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http://dx.doi.org/10.4103/0973-1482.204883DOI Listing
October 2018

Angioleiomyoma of Uterus Masquerading as Malignant Ovarian Tumor.

J Midlife Health 2017 Jul-Sep;8(3):145-147

Department of Pathology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.

Uterine angioleiomyoma is rare. A 40-year-old nulliparous woman presented with heavy menstrual bleeding (HMB) for the past 2 years and mass per abdomen with severe dysmenorrhea for three cycles. She had received 8 units of packed cell transfusion outside. Clinical examination revealed a huge 32-week-sized abdominopelvic mass with irregular margins. Ultrasonography (USG) of the abdomen and pelvis showed a large solid cystic mass on the right side of the abdomen with a well-defined hypoechoeic rounded lesion of size 5.7 cm × 5.0 cm, in the right lobe of the liver, with ovaries not being imaged separately. On color Doppler USG, there was moderate vascularity throughout. A provisional diagnosis of malignant ovarian tumor with hepatic metastasis was made. Her hemoglobin was 5.7 g/dl, and she had repeated episodes of HMB upon admission. She was transfused with 5 units of packed cells. Computed tomography (CT) showed a large fundal subserosal uterine fibroid on the right side, with a solid ovarian tumor measuring 5.0 cm × 4.5 cm on the left side, with ascitis, right-sided hydronephrosis, and a well-defined hypoechoeic lesion in the right lobe of the liver, suggestive of hepatic hemangioma. Tumor markers were within normal limits. In view of discrepancy in clinical findings, ultrasound, and CT report, CT-guided biopsy of the huge mass was done which revealed leiomyoma, with no evidence of mitosis, pleomorphism, or malignancy. Laparotomy with total abdominal hysterectomy and bilateral salpingo-oophorectomy was done. Histopathology revealed an angioleiomyoma uterus. At 1-year follow-up, she was asymptomatic, and the liver mass was stable.
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http://dx.doi.org/10.4103/jmh.JMH_47_17DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5625580PMC
October 2017
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