scholarly journals Metformin induced bullous pemphigoid, the first case

Author(s):  
Sudeb Mukherjee

<p class="abstract">Bullous pemphigoid is a polymorphic autoimmune subepidermal blistering disease seen in elderly individuals. Exact etiology of this autoimmune process is not known. Induced bullous pemphigoid has been associated with several drugs but metformin is not one of them. Here I have reported one such rare case of metformin induced Bullous Pemphigoid (BP).</p>

2020 ◽  
Vol 267 (10) ◽  
pp. 2865-2870
Author(s):  
Torstein R. Meling ◽  
Aria Nouri ◽  
Adrien May ◽  
Nils Guinand ◽  
Maria Isabel Vargas ◽  
...  

Abstract Introduction CNS cavernomas are a type of raspberry-shaped vascular malformations that are typically asymptomatic, but can result in haemorrhage, neurological injury, and seizures. Here, we present a rare case of a brainstem cavernoma that was surgically resected whereafter an upbeat nystagmus presented postoperatively. Case report A 42-year old man presented with sudden-onset nausea, vomiting, vertigo, blurred vision, marked imbalance and difficulty swallowing. Neurological evaluation showed bilateral ataxia, generalized hyperreflexia with left-sided predominance, predominantly horizontal gaze evoked nystagmus on right and left gaze, slight left labial asymmetry, uvula deviation to the right, and tongue deviation to the left. MRI demonstrated a 13-mm cavernoma with haemorrhage and oedema in the medulla oblongata. Surgery was performed via a minimal-invasive, midline approach. Complete excision was confirmed on postoperative MRI. The patient recovered well and became almost neurologically intact. However, he complained of mainly vertical oscillopsia. The videonystagmography revealed a new-onset spontaneous upbeat nystagmus in all gaze directions, not suppressed by fixation. An injury of the rarely described intercalatus nucleus/nucleus of Roller is thought to be the cause. Conclusion Upbeat nystagmus can be related to several lesions of the brainstem, including the medial longitudinal fasciculus, the pons, and the dorsal medulla. To our knowledge, this is the first case of an iatrogenic lesion of the nucleus intercalatus/nucleus of Roller resulting in an upbeat vertical nystagmus. For neurologists, it is important to be aware of the function of this nucleus for assessment of clinical manifestations due to lesions within this region.


2021 ◽  
pp. 106689692098834
Author(s):  
Raquel Machado-Neves ◽  
Bernardo Teixeira ◽  
Elsa Fonseca ◽  
Pedro Valente ◽  
Joaquim Lindoro ◽  
...  

Most malignant tumors of the penis are squamous cell carcinomas (SCC), being divided in 2 groups, one human papillomavirus (HPV)-related and another non-HPV-related, with lymphoepithelioma-like carcinoma (LELC) being one of the rarest HPV-related SCC. In this article, we report a case of a 50-year-old man who presented testicular swelling and pain for the past 3 months. A penile mass was identified, and the patient was submitted to a total penectomy. The penectomy specimen showed an ulcerated lesion at the glans reaching the cavernous bodies. Microscopic examination showed undifferentiated epithelial cells with syncytial growth pattern mix with a dense lymphoplasmacytic infiltrate, consistent with LELC. The tumor cells expressed p16 and all 3 different clones of PDL1 (22C3, SP263, and SP142). The patient is alive and well with a follow-up of 3 months. To our knowledge, this is the third LELC of the penis reported in literature and the first case reported with PDL1 expression.


2021 ◽  
Vol 49 (2) ◽  
pp. 030006052098773
Author(s):  
Kai Xuan Lim ◽  
Karl Wu

Schwannoma or neurilemmoma is a common soft tissue neoplasm arising from the neural sheath of Schwann cells. However, intraosseous schwannoma is rare, accounting for less than 0.2% of primary bone tumours. Several variants of schwannoma have been reported; among them, intraosseous schwannoma with ancient change is extremely rare. This current report presents an extremely rare case of ancient intraosseous neurilemmoma. The patient presented with right elbow pain and disability. A radiolucent, well-defined, lobulated lesion with a thin sclerotic rim in the proximal ulnar metaphysis that had caused a pathological fracture was noted. The mass was surgically excised using marginal resection and bone curettage was undertaken. The bone deficit was grafted with hydroxyapatite and β-tricalcium phosphate and augmented with bone cement. There were no signs of any recurrence after 3 years. This is the first case of an ancient intraosseous schwannoma of the proximal ulna. Although rare, intraosseous schwannoma should be considered in the differential diagnosis of radiographically benign-appearing osseous tumours in the bone. The cement technique is recommended for the treatment of intraosseous schwannoma.


2021 ◽  
Vol 10 (9) ◽  
pp. 1916
Author(s):  
Ágnes Kinyó ◽  
Anita Hanyecz ◽  
Zsuzsanna Lengyel ◽  
Dalma Várszegi ◽  
Péter Oláh ◽  
...  

Bullous pemphigoid (BP) is an autoimmune blistering disease of elderly patients that has shown increasing incidence in the last decades. Higher prevalence of BP may be due to more frequent use of provoking agents, such as antidiabetic dipeptidyl peptidase-4 inhibitor (DPP4i) drugs. Our aim was to assess DPP4i-induced bullous pemphigoid among our BP patients and characterize the clinical, laboratory and histological features of this drug-induced disease form. In our patient cohort, out of 127 BP patients (79 females (62.2%), 48 males (37.7%)), 14 (9 females and 5 males) were treated with DPP4i at the time of BP diagnosis. The Bullous Pemphigoid Disease Area Index (BPDAI) urticaria/erythema score was significantly lower, and the BPDAI damage score was significantly higher in DPP4i-BP patients compared to the nonDPP4i group. Both the mean absolute eosinophil number and the mean periblister eosinophil number was significantly lower in DPP4i-BP patients than in nonDPP4i cases (317.7 ± 0.204 vs. 894.0 ± 1.171 cells/μL, p < 0.0001; 6.75 ± 1.72 vs. 19.09 ± 3.1, p = 0.0012, respectively). Our results provide further evidence that DPP4i-associated BP differs significantly from classical BP, and presents with less distributed skin symptoms, mild erythema, normal or slightly elevated peripheral eosinophil count, and lower titers of BP180 autoantibodies. To our knowledge, this is the first case series of DPP4i-related BP with a non-inflammatory phenotype in European patients.


2019 ◽  
Vol 07 (09) ◽  
pp. E1163-E1165
Author(s):  
Leonardo Blas Jhon ◽  
Paloma Sánchez-Fayos ◽  
Maria Jesus Martín Relloso ◽  
Daniel Calero Barón ◽  
Juan Carlos Porres Cubero

AbstractPrimitive neuroectodermal tumors (PNET) are very rare tumors that belong to a family of malignant neoplasms of tiny round cells which are derived from the neural crest. This report discusses a rare case of an adult woman with esophageal PNET, confirmed by immunohistochemistry, that presented with metastasis to the pineal gland. To our knowledge, this is the first case report of a PNET with these features. Despite surgery and chemotherapeutic treatment, our case has shown disease progression.


2021 ◽  
pp. 197140092110428
Author(s):  
Trilochan Srivastava ◽  
Ashok Gandhi

We are describing a case of a 14-year-old girl who developed acute cerebral infarction which was documented on diffusion-weighted magnetic resonance imaging (DWI). On detailed evaluation, diagnosis of cerebral proliferative angiopathy (CPA) was made. Incidentally, she had capillary malformation on the forehead contralateral to cerebral vascular malformation. To our knowledge, this is the first case of CPA in the literature where DWI abnormality was due to acute cerebral infarction.


2021 ◽  
Author(s):  
Meriem Belheouane ◽  
Britt M Hermes ◽  
Nina Van Beek ◽  
Sandrine Benoit ◽  
Philippe Bernard ◽  
...  

Bullous pemphigoid (BP) is an autoimmune skin blistering disease afflicting mostly the elderly and is associated with significantly increased mortality. Here, we conducted the most extensive sampling effort of skin microbiota in BP to date to analyze whether intra-individual, body-site-specific, and/or geographical variation contributes to the emergence of BP. We find marked differences in the skin microbiota of BP patients compared to that of control subjects, and moreover that disease status rather than skin biogeography governs the skin microbiota composition in BP. Our data reveal a discernible transitional stage between normal and diseased skin in BP characterized by a loss of protective microbiota and an increase in sequences matching Staphylococcus aureus, a known inflammation-promoting species. Notably, S. aureus is ubiquitously associated with disease status, suggesting that this taxon is an important indicator of BP. Importantly, differences in a few key indicator taxa are able to reliably discriminate between BP patients and controls, characterized by their opposing abundance patterns. This may serve as valuable information for assessing disease risk and treatment outcomes. Future research will focus on the functional analysis of host-microbe and microbe-microbe interactions and the relevance of the host genome for microbiota abundances to identify novel BP treatment approaches.


Author(s):  
Rakesh Srivastava ◽  
Vini Tandon

<p class="abstract">Teratomas are embryonal neoplasm arises from totipotent germ cells. They are having tissues from all the three blastodermic layers. There are various locations of congenital teratoma. Naso-oropharyngeal site teratoma are either sessile or pedunculated. We describe a rare case of laryngeal teratoma in a five years old patient presented with change in voice and breathing difficulty. On flexible laryngoscopy, it appeared like supraglottic cyst but on CT scan it was confirmed as teratoma. Pre-operative tracheostomy and transoral carbon dioxide laser assisted excision done. Histopathological examination showed osteoid trabeculae, chondroid tissue with loose myxoid islands and adipose tissue. No recurrence of tumor on 18 months follow-up. This is the first case report of pediatric larynx teratoma reported in present century.</p>


2017 ◽  
Vol 7 (3) ◽  
Author(s):  
Kamal E.H. Mohamed ◽  
Rusha A.E. Ali

Primary breast lymphoma (PBL) represents 0.04-0.5% of all malignant breast tumors, &lt;1% of all patients with non-Hodgkin’s lymphomas and 1.7-2.2% of all patients with extra nodal lymphomas. Despite the high prevalence of breast cancer, primary breast lymphoma is very rare. We report a rare case of PBL, successfully treated with surgery, chemotherapy and radiotherapy. This is the first case of PBL to be reported from Sudan to our knowledge.


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