A 13-Year-Old Presenting With Recurrent Angioma Serpiginosum

2018 ◽  
Vol 22 (5) ◽  
pp. 511-513 ◽  
Author(s):  
Michael Bishara ◽  
Michael Jiaravuthisan ◽  
Miriam Weinstein
Keyword(s):  
Blood Vessels ◽  
Clinical Spectrum ◽  
Disease Entity ◽  
Upper Arm ◽  
Vascular Abnormality ◽  
Histological Features ◽  
Case Summary ◽  
First Case ◽  
Dermal Papillae

Background: Angioma serpiginosum (AS) is a rare vascular abnormality consisting of proliferation and dilation of superficial blood vessels in the skin. AS typically presents in the first 2 decades of life and remains stable with time. Case Summary: We report the case of a 13-year-old female with an acquired, recurrent erythematous lesion with serpiginous borders on her left upper arm. Over several years, the lesion reappeared then disappeared 12 to 15 times. At one point, she developed a tender red nodule within the lesion; consequently, a biopsy was taken, revealing dilated telangiectatic vessels in the dermal papillae with mild, focal extravasation of erythrocytes. Conclusion: This case demonstrates a lesion with suggestive clinical and histological features of AS. However, this may be the first case of recurrent AS with a symptomatic episode. This case may contribute to the expanding clinical spectrum of this interesting disease entity.

scholarly journals Clinical, Laboratory and Histological Features of Dipeptidyl Peptidase-4 Inhibitor Related Noninflammatory Bullous Pemphigoid

2021 ◽  
Vol 10 (9) ◽  
pp. 1916
Author(s):  
Ágnes Kinyó ◽  
Anita Hanyecz ◽  
Zsuzsanna Lengyel ◽  
Dalma Várszegi ◽  
Péter Oláh ◽  
...  
Keyword(s):  
Bullous Pemphigoid ◽  
Clinical Laboratory ◽  
Case Series ◽  
Dipeptidyl Peptidase ◽  
Area Index ◽  
Histological Features ◽  
Dipeptidyl Peptidase 4 ◽  
First Case ◽  
Dipeptidyl Peptidase 4 Inhibitor ◽  
The Mean

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.

scholarly journals Acute upper airway obstruction as a life-threatening complication of ventral bulla osteotomy: report of two consecutive cases

2021 ◽  
Vol 7 (1) ◽  
pp. 205511692110059
Author(s):  
Michal Vlasin ◽  
Richard Artingstall ◽  
Barbora Mala
Keyword(s):  
Airway Obstruction ◽  
Clinical Signs ◽  
Upper Airway ◽  
Upper Airway Obstruction ◽  
Single Session ◽  
Inflammatory Polyp ◽  
Case Summary ◽  
First Case ◽  
Life Threatening

Case summary This paper presents two cases of acute postoperative upper airway obstruction following ventral bulla osteotomy (VBO) in cats. The first cat underwent a unilateral left-sided VBO for a suspected inflammatory polyp. The second cat underwent a single-session bilateral VBO procedure for bilateral otitis media. In the first case, immediate re-intubation and a gradual lightening of the anaesthetic plane resolved the clinical signs; in the second case, the patient deteriorated and went into acute cardiorespiratory arrest and received cardiopulmonary resuscitation. Both patients recovered well and were discharged home 3 days after surgery. Both cases were reported to show no further clinical signs on postoperative follow-up 3 weeks and 4 months after surgery, respectively. Relevance and novel information Upper airway obstruction should be regarded as a potential complication of VBO in cats.

Syncopal Episodes Associated with Cisapride and Concurrent Drugs

1998 ◽  
Vol 32 (6) ◽  
pp. 648-651 ◽  
Author(s):  
Vanessa Seals Gray
Keyword(s):  
English Language ◽  
Motor Vehicle ◽  
Concomitant Administration ◽  
Qt Prolongation ◽  
Azole Antifungals ◽  
Vehicle Accidents ◽  
Cardiac Dysrhythmias ◽  
Case Summary ◽  
First Case ◽  
Patients At Risk

OBJECTIVE: To report a case of QT prolongation and syncopal episodes resulting from concomitant use of cisapride and agents known to inhibit its metabolism. CASE SUMMARY: A 53-year-old white woman was involved in two motor vehicle accidents on the same day after experiencing syncopal episodes. Cardiac and neurologic evaluations were negative; the syncopal episodes were attributed to QT prolongation associated with the concomitant use of cisapride and agents known to inhibit its metabolism. DISCUSSION: This is the first case published in the English-language literature describing QT prolongation resulting from the concomitant use of cisapride and agents known to inhibit its metabolism. Clarithromycin inhibits CYP3A4, the isoenzyme responsible for the metabolism of cisapride. Concomitant administration of cisapride with agents known to inhibit CYP3A4 (i.e., azole antifungals, erythromycin, clarithromycin) may result in elevated cisapride concentrations. Elevated cisapride concentrations have been associated with QT prolongation, syncopal episodes, and cardiac dysrhythmias. CONCLUSIONS: Acquired QT prolongation is a well-recognized adverse effect of several drugs. Recognition of newer drugs and drug combinations that place patients at risk for this potentially fatal adverse event is imperative for appropriate monitoring and prevention.

scholarly journals A Case of Meningococcal Pyomyositis in an Otherwise Healthy Adult

2016 ◽  
Vol 3 (3) ◽  
Author(s):  
Monique T. Barakat ◽  
Kiran Gajurel ◽  
Katrina Fischer ◽  
Kathryn Stevens ◽  
Errol Ozdalga ◽  
...  
Keyword(s):  
Neisseria Meningitidis ◽  
English Language ◽  
Healthy Adult ◽  
Clinical Spectrum ◽  
Nasopharyngeal Colonization ◽  
Etiologic Role ◽  
First Case ◽  
Invasive Diseases ◽  
Life Threatening

Abstract The clinical spectrum of Neisseria meningitidis can range from nasopharyngeal colonization to life-threatening invasive diseases such as meningitis. However, its etiologic role in invasive pyomyositis (PM) has never been reported before in the English language. In this study, we report the first case of PM in the English language and the second case in the literature caused by N meningitidis.

Nitroprusside Treatment of Erythromelalgia in an Adolescent Female

1997 ◽  
Vol 31 (5) ◽  
pp. 590-592 ◽  
Author(s):  
Jonathan D Stone ◽  
Michael P Rivey ◽  
Douglas R Allington
Keyword(s):  
Rare Disease ◽  
Pediatric Patients ◽  
Aspirin Therapy ◽  
Adolescent Female ◽  
Adolescent Patient ◽  
Disease Entity ◽  
Case Summary ◽  
First Choice

OBJECTIVE: TO report a case of erythromelalgia in an adolescent patient successfully treated with nitroprusside. CASE SUMMARY: A 15-year-old girl with erythromelalgia resistant to aspirin therapy received an infusion of nitroprusside. The response of the erythromelalgia to nitroprusside was dramatic, with complete pain resolution within 17 hours after the start of therapy. No relapse of erythromelalgia was seen when nitroprusside was discontinued and the patient remained well after 6 months. DISCUSSION: This case adds to existing literature substantiating the benefit of nitroprusside for the treatment of erythromelalgia in pediatric patients. Erythromelalgia in children may represent a different disease entity than that seen in adults, which is commonly responsive to aspirin therapy. The pathogenesis of erythromelalgia is unclear and precludes formulating a proposed mechanism by which nitroprusside has benefit in children. CONCLUSIONS: Nitroprusside is valuable for erythromelalgia resistant to aspirin therapy in pediatric patients. Because of unanswered questions regarding the disease, aspirin remains the agent of first choice in all patients with this rare disease.

A diagnostic pitfall in hepatic mesenchymal hamartoma mimicking hepatoblastoma: A case report

2020 ◽  
Author(s):  
Zhihong Chen ◽  
Lutong Fang ◽  
Ying Zhang ◽  
Yuanzi Ye ◽  
Wenshu Ji ◽  
...  
Keyword(s):  
Blood Vessels ◽  
Female Child ◽  
Point Of View ◽  
Mesenchymal Hamartoma ◽  
Diagnostic Pitfall ◽  
First Case ◽  
Contrast Enhanced ◽  
Enhanced Ct ◽  
Vascular Channels ◽  
The Right

Abstract Background: Hepatic mesenchymal hamartoma (HMH) is an uncommon benign tumor in children. While mesenchymal hamartomas may be angiomatous and blood vessels may be identified, HMH with a malignant tumor symptom on the contrast-enhanced hepatic computed tomography angiography (CTA) has not been described. Here, we present the first case of HMH mimicking hepatoblastoma on the hepatic CTA from pathological point of view and review the imaging and histological features of this unique lesion. Case presentation: A 2-year-old female child was found a distention in the right abdomen and was admitted to our hospital. The Hepatic CTA showed that the blood vessels were thickened, the tumor blood vessels were clustered in the tumor. According to the hepatic CT findings, the tumor was considered to be malignant, possibly a hepatoblastoma. Microscopic examination showed a tumor arranged in lobules, composed of loose myxoid mesenchyme surrounding ductal structures, with intervening vascular channels. The Immunohistochemical staining revealed positive CK7 and CD34 for the bile duct elements and the lining endothelial cells of the vascular channels. There were abundant blood vessels around the nodules and the margins of remaining hepatocytes, but few in the central region. The blood vessels are small, thin-walled vessels and presented like capillaries and venules. Conclusions: A histological diagnosis of Hepatic Mesenchymal Hamartoma was confirmed by the microscopic examinations. This case adds learning points to radiologists when heterogeneous reinforcement on enhanced CT scan was presented in such a large childhood liver tumor.

scholarly journals Acquired smooth muscle hamartoma: A case report on the lower extremity with hidrosis

2019 ◽  
Vol 7 ◽  
pp. 2050313X1989383
Author(s):  
Malika A Ladha ◽  
Todd Remington
Keyword(s):  
Smooth Muscle ◽  
Case Report ◽  
Lower Extremity ◽  
Smooth Muscle Cells ◽  
English Literature ◽  
Surgical Excision ◽  
Muscle Cells ◽  
Clinical Spectrum ◽  
Rare Entity ◽  
First Case

Smooth muscle hamartomas are benign dermal proliferations of smooth muscle cells. Smooth muscle hamartomas are sub-divided into congenital or acquired; the latter is a rare entity with less than 20 cases being reported in the English literature. Most often asymptomatic, acquired smooth muscle hamartomas follow an indolent course. Treatment in the form of surgical excision can be utilized for symptomatic or cosmetic purposes. Here, we report the first case of an acquired smooth muscle hamartomas of the shin which also uniquely presented with hidrosis. This case highlights the varied clinical spectrum of acquired smooth muscle hamartomas.

Fatal Neutropenia and Thrombocytopenia Associated with Ticlopidine

1994 ◽  
Vol 28 (11) ◽  
pp. 1236-1238 ◽  
Author(s):  
James A. Carlson ◽  
Jon E. Maesner
Keyword(s):  
Renal Function ◽  
Absolute Neutrophil Count ◽  
Neutrophil Count ◽  
Impaired Renal Function ◽  
Healthcare Professionals ◽  
Severe Neutropenia ◽  
Gram Negative ◽  
Case Summary ◽  
First Case

OBJECTIVE: To report the first case of ticlopidine-associated neutropenia resulting in sepsis and death. CASE SUMMARY: An 83-year-old Filipino man was started on ticlopidine 250 mg bid. By the seventh week of therapy his absolute neutrophil count (ANC) had dropped to 2700 from 7600 × 106 cells/L. The ticlopidine was stopped. Six days later, he was admitted to the hospital. He died 18 hours later of gram-negative sepsis. DISCUSSION: Although ticlopidine therapy was discontinued four days after the patient's ANC was 2700 × 106 cells/L, the ANC dropped to and remained at 0 until his death eight days later. This may be associated with the patient's decreased clearance of ticlopidine given his age and impaired renal function. This is the first reported case of moderate or severe neutropenia in a nonwhite patient and the first reported case of sepsis and death caused by ticlopidine CONCLUSIONS: Healthcare professionals must be aware of the possibility of severe neutropenia and death caused by ticlopidine, even when the manufacturers' monitoring guidelines are followed.

Oculogyric Crisis Possibly Related to Pentazocine

1993 ◽  
Vol 27 (7-8) ◽  
pp. 874-876 ◽  
Author(s):  
Aaron H. Burstein ◽  
Terence Fullerton
Keyword(s):  
Temporal Relationship ◽  
Opiate Receptors ◽  
Data Sources ◽  
Review Of Literature ◽  
Patient Case ◽  
Case Summary ◽  
First Case ◽  
Oculogyric Crisis ◽  
Plausible Mechanism ◽  
Relationship Of

OBJECTIVE: To report and describe the apparent first case of acute oculogyric crisis following administration of pentazocine, and to discuss the possible mechanism for this reaction. DATA SOURCES/CASE SUMMARY: Patient case and relevant review of literature. The patient, a 39-year-old woman, developed acute oculogyric crisis following administration of Talacen (pentazocine and acetaminophen) for pain relief. The crisis resolved after discontinuation of the medication and administration of intravenous diphenhydramine 50 mg. CONCLUSIONS: Based on the temporal relationship of drug administration to occurrence of the event, pentazocine is implicated as the cause of this acute oculogyric crisis. A plausible mechanism for precipitation of this crisis is the agonism of pentazocine on sigma opiate receptors, with postulated subsequent modulation of dopamine receptors.

scholarly journals SARS–CoV-2 isolation from an appendix

2020 ◽  
Vol 2020 (8) ◽  
Author(s):  
Shahzaib Ahmad ◽  
Rahim Nadeem Ahmed ◽  
Poonam Jani ◽  
Mattee Ullah ◽  
Hossam Aboulgheit
Keyword(s):  
Polymerase Chain Reaction ◽  
Abdominal Pain ◽  
Respiratory Symptoms ◽  
Tissue Sample ◽  
Gastrointestinal Symptoms ◽  
Shortness Of Breath ◽  
Chain Reaction ◽  
Histological Features ◽  
First Case ◽  
Polymerase Chain

Abstract Efforts to recognize SARS–CoV-2 infection have focused on respiratory symptoms such as cough and shortness of breath. Although it is also well known that SARS–CoV-2 infection can cause gastrointestinal symptoms such as abdominal pain, nausea, vomiting and diarrhoea, there are emerging reports of SARS–CoV-2 infection causing surgical pathology. We present the first case report of SARS–CoV-2 infection directly causing acute appendicitis, first suspected due to highly atypical histological features and later confirmed as polymerase chain reaction positive appendicular tissue sample.

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