Successful Treatment of Inflammatory Linear Verrucous Epidermal Nevus with Tacrolimus and Fluocinonide

2006 ◽  
Vol 10 (1) ◽  
pp. 45-47 ◽  
Author(s):  
Diya F. Mutasim

Background: Inflammatory linear verrucous epidermal nevus (ILVEN) is a relatively rare disorder that is characterized by an early age at onset; severely pruritic linear papules and plaques; histologic features resembling spongiotic dermatitis, psoriasis, or lichenified dermatitis; and poor response to treatment. Objective: To report the successful treatment of ILVEN with potent topical steroid and tacrolimus ointments. Methods: An 11-year-old girl presented with a 1-year history of markedly pruritic, progressive linear eruption that extended from the right foot to the right buttock. She had failed treatment with pimecrolimus, calcipotriol, mometasone furoate, triamcinolone, tazarotene, and alpha-hydroxy acid. Histologic examination revealed the findings of spongiotic dermatitis. Results: The lesions resolved with fluocinonide ointment and tacrolimus 0.1% ointment. Conclusion: The combination of two therapeutic agents with different mechanisms of action likely resulted in the successful treatment of this usually resistant condition.

2016 ◽  
Vol 2016 ◽  
pp. 1-4
Author(s):  
Punit Pruthi ◽  
Pramod Arora ◽  
Manoj Mittal ◽  
Anugrah Nair ◽  
Waqia Sultana

Venipuncture is one of the most commonly done medical procedures. We report a unique case of a 23-year-old young male who presented with features suggestive of inflammatory arthritis. The symptoms, which initially started on the right side, also involved the other side after a few weeks. Although the patient’s symptoms and signs were simulating inflammatory arthritis, he had atypical features like poor response to anti-inflammatory medicines and normal laboratory parameters. His musculoskeletal ultrasonography was also not suggestive of arthritis. His history was reviewed and on direct questioning he revealed a history of venipuncture for blood sample withdrawal, done from right antecubital region for routine health check on the day prior to the onset of symptoms. Complex regional pain syndrome was suspected and triple-phase radioisotope bone scan was done which was highly suggestive of this diagnosis. The patient was managed with multidimensional approach and responded very well to the treatment. Complex regional pain syndrome is usually not thought of in the initial differential diagnosis of inflammatory arthritis. In this report we highlight the need to elicit the often overlooked history of trivial trauma like venipuncture, especially in atypical cases of arthritis. Also the role of newer diagnostic modalities in such cases is emphasized.


2015 ◽  
Vol 2015 ◽  
pp. 1-4 ◽  
Author(s):  
Hae Won Jung ◽  
Chong Rae Cho ◽  
Ji Yoon Ryoo ◽  
Hyun Kyo Lee ◽  
So Young Ha ◽  
...  

Actinomyces meyeriis an uncommon cause of human actinomycosis. Here, we report a rare case of empyema caused byA. meyeri. A 49-year-old male presented with a history of 10 days of dyspnea and chest pain. A large amount of loculated pleural effusion was present on the right side and multiple lung nodules were documented on radiological studies. A chest tube was inserted and purulent pleural fluid was drained.A. meyeriwas isolated in anaerobic cultures of the pleural fluid. The infection was alleviated in response to treatment with intravenous penicillin G (20 million IU daily) and oral amoxicillin (500 mg every 8 hours) for 4 months, demonstrating that short-term antibiotic treatment was effective.


2022 ◽  
pp. 104063872110650
Author(s):  
Julie B. Engiles ◽  
Francisco A. Uzal ◽  
Mauricio A. Navarro ◽  
Virginia B. Reef ◽  
Susan J. Bender

Phlegmonous gastritis was diagnosed in 2 yearling fillies that were presented with a 1-wk history of fever, lethargy, and hypoproteinemia, associated with a previous diagnosis of equine proliferative enteropathy based on clinical signs and PCR assay detection of Lawsonia intracellularis in fecal samples. Abdominal ultrasound revealed enlargement of the stomach and expansion of its submucosal layer with hypoechoic fluid, as well as thickened hypomotile small intestinal segments. Given the poor prognosis and poor response to treatment, both horses were euthanized, one on the day of presentation and the other after 3 wk of intensive medical management including a combination of antimicrobials, analgesics, and intravenous colloids. At autopsy, acute mural gastritis characterized by severe submucosal edema with suppurative inflammation (i.e., phlegmonous gastritis) and necroulcerative enteritis compatible with the necrotizing form of equine proliferative enteropathy were identified in both horses. The gastric inflammation was associated with thrombosis and mixed bacterial populations, including Clostridium perfringens, that were confined to the submucosa without evidence of mucosal involvement; toxin genes compatible with C. perfringens type C were identified in one case. Human phlegmonous gastritis is an uncommon, often-fatal pyogenic infection that is often associated with mucosal injury, bacteremia, or immunocompromise. Our finding of this unusual gastric lesion in 2 horses with similar signalment, clinical disease, and spectrum of postmortem lesions suggests a similar etiopathogenesis that possibly involves local, regional, or distant hematogenous origin, and should be considered a potential complication of gastrointestinal mucosal compromise in horses.


2016 ◽  
Vol 2016 ◽  
pp. 1-3 ◽  
Author(s):  
Mónica Loureiro ◽  
Renata Rothwell ◽  
Sofia Fonseca

Purpose.To describe a case of anterior nodular scleritis, preceded by an anterior hypertensive uveitis, which was primarily caused by varicella zoster virus (VZV).Case Report. A 54-year-old woman presented with anterior uveitis of the right eye presumably caused by herpetic viral disease and was successfully treated. Two months later, she developed a nodular scleritis and started oral nonsteroidal anti-inflammatory without effect. A complete laboratory workup revealed positivity for HLA-B27; the infectious workup was negative. Therapy was changed to oral prednisolone and an incomplete improvement occurred. Therefore, a diagnostic anterior paracentesis was performed and the polymerase chain reaction (PCR) analysis revealed VZV. She was treated with valacyclovir and the oral prednisolone began to decrease; however, a marked worsening of the scleritis occurred with the reduction of the daily dose; subsequently, methotrexate was introduced allowing the suspension of the prednisolone and led to clinical resolution of the scleritis.Conclusion.This report of anterior nodular scleritis caused by VZV argues in favor of an underlying immune-mediated component, requiring immunosuppressive therapy for clinical resolution. The PCR analysis of the aqueous humor was revealed to be a valuable technique and should be considered in cases of scleritis with poor response to treatment.


2021 ◽  
Vol 14 (10) ◽  
pp. e245733
Author(s):  
Guido Gabriele ◽  
Simone Benedetti ◽  
Fabiola Rossi ◽  
Paolo Gennaro

Odontogenic choristomas are a rare and recently classified entity, defined as neoplasms containing tissues of odontogenic derivation, found in abnormal locations and usually diagnosed in the early age. The authors report a unique case of a 79-year-old patient who presented to our attention for a skin lesion in the right cheek with no history of trauma or recent dental procedures, that underneath presented an indolent tooth-like structure inside the soft tissue of the right cheek, which at the histological study resulted to be an odontogenic choristoma. This rare histopathological diagnosis should be considered when a hard lesion is present in the head and neck region.


2020 ◽  
Author(s):  
Ken Yamashita ◽  
miki hiraoka ◽  
Shintaro Sugita ◽  
Takatoshi Yotsuyanagi

Abstract Backgroud: Concretions of the lacrimal drainage system can cause dacryocystitis. In the present study, our patient developed dacryocystisis with lacrimal concretions of a white soft mass. Rebamipide ophthalmic suspension had been applied for treatment of dry eye. To evaluate the pathogenic mechanism of the case, histological and chemical studies were performed.Case presentation: Our case was woman in her seventies. She had a medical history of rheumatoid arthritis. She was referred to our hospital for dacryocystitis after showing a poor response to treatment with antibiotic agents. A head computed tomography (CT) scan showed ductal high-density deposits along the lacrimal sacs. During dacryocystotomy, the hypertrophy of the lacrimal sacs was found replete with pus and cottage cheese-like white substances. The extracts were surgically removed and histological and chemical analysis was performed. The histological examination showed granulation tissues and acellular amorphous material with crystal-like structures. Positive staining by Alcian blue and Kossa was found in crystal-like legions. The extract obtained from the concretions showed the same fluorescence band and UV absorption spectrum in thin layer chromatography (TLC) and spectrometry, respectively, as rebamipide.Conclusion: Our findings suggest that the obstruction by lacrimal concretions containing rebamipide resulted in dacryocystitis in this case. In addition, it is speculated that in patients who have impaired tear secretion, refractory dacryocystitis may be evoked following the accumulation of rebamipide in the lacrimal sacs.


2020 ◽  
pp. 112067212092434
Author(s):  
Shotaro Asano ◽  
Victor Koh Teck Chang ◽  
Maria Cecilia Domingo Aquino ◽  
Paul Chew Tec Kuan

Purpose The aim of this study was to report the use of micropulse trans-scleral cyclophotocoagulation as an adjunct therapy for two cases of medically uncontrolled intraocular pressure spikes due to anterior segment inflammation. Case description: Case 1 had previous cataract surgery and exhibited an intraocular pressure spike due to phacoantigenic uveitis (right eye intraocular pressure = 52 mmHg). Despite medical treatment, the right eye intraocular pressure remained high (43 mmHg), thus micropulse trans-scleral cyclophotocoagulation was carried out as a rescue therapy. After micropulse trans-scleral cyclophotocoagulation, the intraocular pressure at 1 day and 3 weeks was 9 and 16 mmHg, respectively. Case 2 had a history of previous blunt ocular trauma and 180° of angle recession. Both eyes were pseudophakia and underwent right eye Nd:YAG laser capsulotomy for posterior capsular opacification. Immediately after the procedure, the right eye intraocular pressure increased to 64 mmHg. Due to poor response to medical therapy, rescue micropulse trans-scleral cyclophotocoagulation was performed. After micropulse trans-scleral cyclophotocoagulation, the intraocular pressure at 1 day and 2 months was 12 and 21 mmHg, respectively. Conclusion Micropulse trans-scleral cyclophotocoagulation successfully decreased intraocular pressure in both cases of acute rise in intraocular pressure. Micropulse trans-scleral cyclophotocoagulation can potentially be useful as a rescue procedure to safely reduce medically uncontrollable intraocular pressure spike due to anterior segment inflammation.


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Veronica M Figueredo ◽  
Alejandro Diaz ◽  
Pedro Pagan Banchs

Abstract Background: Hyperinsulinism is the most common cause of persistent hypoglycemia. It results from different genetic defects, the most common being recessive and dominant mutations in the ABCC8/KCNJ11 genes. The majority of recessive mutations have a poor response to Diazoxide, while dominant mutations are highly variable in both clinical presentation and response to treatment. Prompt recognition and management is critical to avoid irreversible brain damage. Clinical case: A 38-week gestation male, born via emergent c-section due to decreased fetal movement, presented with neonatal hypoglycemia. Pregnancy was uncomplicated and mother had a normal OGTT. Patient had a history of suspected sepsis, seizures, pulmonary hypertension and respiratory distress requiring intubation. Blood glucose was undetectable at birth and required multiple dextrose 10% boluses. A critical sample with a glucose of 47 mg/dL showed an elevated insulin at 30.3 m IU/mL with undetectable ketone levels. Lactic acid, ammonia, cortisol, GH, plasma amino acids, acylcarnitine profile and uric organic acids where all normal for a hypoglycemic state. He required intravenous glucose infusion with GIR up to 17 mg/kg/min to maintain normoglycemia. A brain MRI at 11 days of life showed findings of white matter injury. Subsequent genetic testing identified a heterozygous c.4051G>A (p.Val1351Met) variant in ABCC8, classified as “of uncertain significance”. However, an entry in the ClinVar database (RCV000714711.1) exists from a research lab and was classified as likely pathogenic. Analysis of parental samples showed that the mother was heterozygous for the same genetic variant. She did not have a history of hypoglycemia. Patient was started on diazoxide (8 mg/kg/day) and chlorothiazide with resolution of hypoglycemia. At a follow up visit at 5 months of age, there was no history of hypoglycemia, and no need for adjustments of the diazoxide dose by weight (dose at that time of 7.4 mg/kg/day). Conclusion: The ABCC8 reported here is a dominant mutation causing hyperinsulinemic hypoglycemia responsive to diazoxide with a milder phenotype later in infancy. Longitudinal follow up of the case is warranted to understand the long term progress in patients with this particular mutation. Reference: Adam MP, Ardinger HH, Pagon RA, Wallace SE, et al. None. 1993. Familial hyperinsulinism.


2018 ◽  
Vol 2 (1) ◽  
pp. 80-84
Author(s):  
Chun-Hui Yi ◽  
Chen Chen ◽  
Allen N Sapadin ◽  
Robert G Phelps

A 71-year-old woman presented with a one-month history of pruritus, burning, flaky, and erythematous plaques. The lesions had a very narrow lineal distribution, extending from the lateral left sole to the left popliteal region. Microscopically, the skin biopsy sample revealed orthokeratosis, parakeratosis and traumatized Koebnerization. Elongation of rete ridges was not prominent. The patient had a good clinical response to oral prednisone. Linear psoriasis is a rare variant of psoriasis of unknown etiology. Approximately 23 reports have been published in the English literature. The main differential diagnosis is Inflammatory linear verrucous epidermal nevus (ILVEN). Thorough clinicopathologic findings and response to treatment are all required in the diagnosis and management of this rare form of psoriasis.


2021 ◽  
Vol 3 (4) ◽  
pp. 1047-1052
Author(s):  
Ken Yamashita ◽  
Miki Hiraoka ◽  
Shintaro Sugita ◽  
Takatoshi Yotsuyanagi

AbstractConcretions of the lacrimal drainage system can cause dacryocystitis. In the present study, our patient developed dacryocystisis with lacrimal concretions of a white soft mass. Rebamipide ophthalmic suspension had been applied for treatment of dry eye. To evaluate the pathogenic mechanism of the case, histological and chemical studies were performed. Our case was woman in her seventies. She had a medical history of rheumatoid arthritis. She was referred to our hospital for dacryocystitis after showing a poor response to treatment with antibiotic agents. A head computed tomography (CT) scan showed ductal high-density deposits along the lacrimal sacs. During dacryocystotomy, the hypertrophy of the lacrimal sacs was found replete with pus and cottage cheese-like white substances. The extracts were surgically removed, and histological and chemical analysis was performed. The histological examination showed granulation tissues and acellular amorphous material with crystal-like structures. Positive staining by Alcian blue and Kossa was found in crystal-like legions. The extract obtained from the concretions showed the same fluorescence band and UV absorption spectrum in thin layer chromatography (TLC) and spectrometry, respectively, as rebamipide. Our findings suggest that the obstruction by lacrimal concretions containing rebamipide resulted in dacryocystitis in this case. In addition, it is speculated that in patients who have impaired tear secretion, refractory dacryocystitis may be evoked following the accumulation of rebamipide in the lacrimal sacs.


Sign in / Sign up

Export Citation Format

Share Document