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1.
J Surg Case Rep ; 2024(1): rjad695, 2024 Jan.
Article in English | MEDLINE | ID: mdl-38186753

ABSTRACT

Inflammatory fibroid polyps (IFP) are rare benign neoplasms most commonly occurring within the respiratory tract but are rarely also observed in the gastro-intestinal tract. Herein we present the case of a 73-year-old female presenting with ileo-ileal intussusception secondary to IFP. The patient was treated with emergency laparotomy with segmental bowel resection and primary anastomosis. Histopathological analysis of the excised bowel segment initially revealed a low-grade, mural based spindle cell neoplasm with surrounding benign, reactive lymphadenopathy. Immunohistochemical analysis demonstrated that the lesional cells stained positive for Vimentin, Smooth Muscle Actin (SMA), and CD34. On secondary analysis of the specimen, the morphology and immunohistochemical profile of the mass was in keeping with IFP. No invasive malignancy was identified. Such cases have been previously reported under the pseudonym 'the great mimicker', due to their striking similarity to malignant processes. This case report aims to add to the small body of research reporting such atypical presentations.

2.
Eur J Case Rep Intern Med ; 9(9): 003537, 2022.
Article in English | MEDLINE | ID: mdl-36299853

ABSTRACT

A 22-year-old woman presented with a 12-year history of intensifying paroxysms of anxiety, palpitations and recurrent syncope following micturition. The patient was referred to endocrinology upon discovery of hypertension. An extended family history revealed metastatic phaeochromocytoma and paraganglioma in two grand-uncles. Clinical examination revealed hypertension, with a mean 24-hour ambulatory blood pressure of 150/100 mmHg. Supine plasma normetanephrines were markedly elevated with a raised 3-methoxytyramine, while plasma metanephrines were normal. Computed tomography identified a 4.4 cm mass at the right inferolateral margin of the bladder wall. Scintigraphic imaging confirmed unifocal bladder lesion uptake with no additional metastatic lesions. Following pre-operative alpha blockade, the patient underwent a partial cystectomy. Histology confirmed a paraganglioma, and SDHB staining was lost in neoplastic cells consistent with an SDHB-related paraganglioma. Plasma normetanephrine, 3-methoxytyramine and blood pressure returned to normal postoperatively. Genetic screening identified a germline heterozygous SDHB gene variant c.723C>G. Bladder paragangliomas are a rare but important differential to consider when investigating post-micturition syncope. An extended family history should be sought and suspicion for a genetic cause should be raised, especially when the condition presents at a young age. This is the first reported case describing phaeochromocytoma or paraganglioma with the SDHB gene variant c.723C>G. LEARNING POINTS: Bladder paragangliomas are a rare neuroendocrine tumour which should be considered when assessing patients with haematuria and hypertension, headache, palpitations, sweating and facial pallor with micturition.This case highlights the importance of a thorough clinical history with an extended family history and examination in the setting of micturition syncope, which can rarely occur with bladder paraganglioma.Young age at presentation, a family history of phaeochromocytoma and paraganglioma (PPGL), unusual paraganglioma location, mutifocality and aggressive disease should raise the suspicion for a genetic predisposition to PPGL.

3.
J Surg Case Rep ; 2021(12): rjab536, 2021 Dec.
Article in English | MEDLINE | ID: mdl-34925756

ABSTRACT

Actinomycosis is an invasive infection, which can affect numerous anatomical sites, though rarely the kidney. The rate of nephrectomy is high despite antibiotic therapy. A 51 year old presented with a Proteus mirabilis renal abscess 9 years following a similar renal abscess. The abscess persisted despite appropriate antibiotic treatment and radiological drainage. In addition to P. mirabilis, Actinomyces species was isolated on polymicrobial abscess culture after 6 weeks antibiotic therapy. Despite appropriate antibiotics, nephrectomy was required. Histology confirmed actinomycosis. Actinomycosis should be considered in chronic, destructive infections, especially if failure to respond to appropriate antimicrobials. However, Actinomyces species may be missed by routine culture techniques. Because of the polymicrobial nature of abscesses, good communication with the laboratory is essential to ensure that cultures are prolonged and the isolation of one pathogen does not hinder the isolation of others.

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