scholarly journals Intralymphatic Histiocytosis: An Unusual Presentation

2021 ◽  
pp. 1-6
Author(s):  
Timothy J. Blackwell ◽  
Zachary Ingersoll ◽  
Martin Blackwell

Intralymphatic histiocytosis (ILH) is a rare cutaneous condition initially described in 1994 by O’Grady et al. It often appears as a red to violaceous, livedoid patch or plaque usually on the extremities. We present a 71-year-old female with a history of psoriasis, 50 pack years smoking and recent Legionnaires disease who came to us complaining of a red to violaceous, blanching, edematous, mildly tender lesion covering the left lower lip and extending to the chin and anterior neck. After multiple biopsies, ILH was confirmed and the patient was initially started on tacrolimus 0.1% ointment b.i.d., but there was no response. Then, she was started on oral pentoxifylline and intermittent topical steroids, as well as continuing the topical tacrolimus. There was again no response, so now she is taking a TNF-ɑ inhibitor as it appears to be a granulomatous process. These ILH cases are very rare and there is limited literature that describes one treatment as a cure. Treatment of ILH is very difficult, but several different therapies have been reported with varying success. If the disease is secondary to an underlying inflammatory disease or malignancy, then treatment of the primary disorder can lead to resolution of the ILH.

2007 ◽  
Vol 11 (6) ◽  
pp. 222-225 ◽  
Author(s):  
Marta J. Cenkowski ◽  
Shane Silver

Background: Erosive pustular dermatosis of the scalp (EPDS) is a rare condition characterized by chronic, sterile, pustular erosions leading to scarring alopecia. Although the etiology is unknown, it appears to be associated with ultraviolet light exposure and trauma. Histologic findings include nonspecific atrophy of the epidermis and chronic inflammation. Case History: A 71-year-old female presented with a 1-year history of a boggy, erythematous, friable plaque on the vertex of her scalp. A diagnosis of EPDS was made based on presentation, negative cultures, and histologic findings. Initial therapy with intralesional and topical steroids and oral antibiotics resolved the inflamed plaques; however, steroid-induced atrophy became prominent after 5 months of use. The treatment was discontinued, resulting in recurrence of disease. Topical tacrolimus 0.1% ointment was initiated, which has been successfully controlling the lesions with reversal of skin atrophy and clinical evidence of hair growth. Conclusion: This is the fourth reported case of the successful treatment of EPDS with topical tacrolimus for the resolution of atrophy and the prevention of relapse of inflammation. Although its long-term use warrants close follow-up for side effects, tacrolimus may constitute a novel therapeutic option for the treatment of EPDS.


Author(s):  
Sahar H. Alsharif ◽  
Reda H. Saifaldeen ◽  
Logain G. Alghanemi

<p class="abstract">Granuloma faciale (GF) is a chronic condition characterized by asymptomatic erythematous plaque with prominent telangiectasia presenting usually over the face. Although the condition is benign, its treatment is often unsatisfactory. Therapeutic modalities that have been tried include topical steroids and topical tacrolimus sometimes enhanced with topical dapsone. Others include intralesional corticosteroids, antimalarials, isoniazid and pulsed-dye laser. We report a case of a 58 years old female with a 1 year history of a solitary slowly progressive plaque over the nose. Diagnosis of GF was made based on the histopathological findings. The patient was started on the combination of topical tacrolimus, intralesional corticosteroids injection and oral doxycycline for 3 months. The patient showed gradual improvement in 3 months without any side effects. This case supports previous papers of successful treatment of GF with topical tacrolimus. There was no recurrence at follow-up 18 months later. It also supports the use of combination therapy especially in resistant cases.</p>


Author(s):  
K Souaid ◽  
T Klejtman ◽  
N Kramkimel ◽  
C Isnard ◽  
N Dupin ◽  
...  

2020 ◽  
Vol 11 (3) ◽  
pp. 595-599
Author(s):  
Saeed T. Alshahrani ◽  
J. Fernando Arevalo

A patient presented with complaints of a sudden decrease in vision, ocular redness, and pain in the right eye. The patient had a history of clear lens extraction with intraocular lens (IOL) implantation for myopia 2 years previously. He had been prescribed topical steroids for episodes of inflammation that occurred repeatedly every 1–2 months. With a presumptive diagnosis of chronic endophthalmitis, a 23-G transconjunctival sutureless pars plana vitrectomy (PPV) with delivery of intravitreal antibiotics was performed the next day. Culture sensitivity testing of the vitreous sample indicated <i>Pseudomonas stutzeri</i> that was sensitive to ceftazidime and gentamicin. Two weeks later, the patient presented with sudden loss of vision and all the signs of recurrent endophthalmitis. 23-G transconjunctival sutureless PPV was performed along with removal of the posterior chamber IOL through a corneal incision. Complete resolution was only achieved after removal of the IOL, resulting in excellent visual recovery. Due to its chronic and fulminating nature, <i>P. stutzeri</i> can induce endophthalmitis and should be considered in the differential diagnosis. Aseptic measures are the best prevention.


2021 ◽  
Vol 9 ◽  
pp. 2050313X2110030
Author(s):  
Asfandyar Mufti ◽  
Muskaan Sachdeva ◽  
Khalad Maliyar ◽  
Marissa Joseph

Background: Hereditary haemorrhagic telangiectasia is an autosomal dominant genetic disorder characterized by abnormalities in blood vessel formation. The clinical manifestations of patients affected with hereditary haemorrhagic telangiectasia include mucocutaneous telangiectasias and visceral arteriovenous malformations. Case Summary: We report the case of a 30-year-old female diagnosed with hereditary haemorrhagic telangiectasia presenting with the classic triad of recurrent epistaxis, mucocutaneous telangiectasias and family history of hereditary haemorrhagic telangiectasia with activin receptor-like kinase 1 mutation. Upon skin examination, she was noted to have telangiectasias under left naris, inner lower lip and surface of the tongue, and a vascular malformation on the right forearm. Conclusion: Although the skin involvement and epistaxis may be mild symptoms and signs of hereditary haemorrhagic telangiectasia, timely recognition of these can ensure vigilant monitoring of potential severe complications from cerebral and pulmonary visceral arteriovenous malformations.


Author(s):  
Savita Chaudhary ◽  
Chandni Jain ◽  
Gaurav Paliwal ◽  
Priyanka Shukla

<p class="abstract"><strong>Background:</strong> Uncontrolled use and abuse of topical steroids has led to increase in number of cases of superficial dermatophytosis of skin, hair and nail in pediatric age group as well. Our study aimed to analyse epidemiological and microbiological profile of steroid modified tinea (SMT) in pediatric age group.</p><p class="abstract"><strong>Methods:</strong> Clinically diagnosed tinea childhood patients with history of usage of topical steroids in children were included in our study. Detailed history was taken and clinical examination along with KOH mount and culture was done.<strong></strong></p><p class="abstract"><strong>Results:</strong> 112 patients were clinically diagnosed as tinea out of which 61cases gave the history of topical steroids and were included in our study. Most common age group was 12-18 years with female:male of ratio approximately 3:1 and disseminated and atypical form was the most common variety. KOH mount was positive in 73.2% cases and culture was positive in 69.6% cases. Most common species found out to be <em>Tinea mentagrophytes</em> followed by <em>Tinea rubrum</em>. Among non-dermatophyte group, <em>Candida</em> was the commonest.</p><p class="abstract"><strong>Conclusions:</strong> There is rise in incidence of dermatophytosis, especially steroid modified, atypical and disseminated.</p>


2020 ◽  
pp. bjophthalmol-2020-316196
Author(s):  
Ceyhun Arici ◽  
Burak Mergen

PurposeInvestigation of the efficacy and safety of 12 months of topical tacrolimus 0.03% ointment treatment against the subepithelial infiltrates (SEIs) due to adenoviral keratoconjunctivitis (AKC) resisting at least 2 years was aimed.MethodsThis case series included consecutive patients with SEIs secondary to AKC who were resistant to topical steroid and ciclosporin-A (CSA) treatment and treated with topical 0.03% tacrolimus (Protopic; Fujisawa Healthcare, Teva, Deerfield, Illinois, USA) for 12 months, at least 2 years after AKC. For the evaluation of treatment efficacy, best-corrected visual acuity (BCVA), Fantes score, corneal subepithelial infiltrate score (CSIS), Oxford score, Schirmer and tear breakup time results were evaluated. Intraocular pressure and complaints of the patients were followed for evaluating the safety profile of the treatment. The patients were followed after the baseline visit at the 1st, 3rd, 6th and 12th month.Results15 eyes of 11 patients with SEIs and 16 eyes of 16 healthy controls were included in this study. 1 patient (9.1%) could not tolerate the treatment. Significant improvements in BCVA, CSIS, Fantes score and Schirmer results were observed in the study group starting from the 3rd-month visit, and the improvements persisted until the end of 12 months of treatment.ConclusionTopical 0.03% tacrolimus might show efficacy against the SEIs persisting at least 2 years despite corticosteroid and/or CSA treatment without any prominent side effect. While at least a period of 3 months was necessary for a significant improvement in the BCVA, SEIs and Schirmer results, a period of 6 months was necessary for a decrease in Oxford score.


2017 ◽  
Vol 102 (5-6) ◽  
pp. 222-226
Author(s):  
Sang Yull Kang ◽  
Yo Na Kim ◽  
Seon Kwang Kim ◽  
Hyun Jo Youn ◽  
Sung Hoo Jung

Immunoglobulin (Ig) G4-related Hashimoto's thyroiditis is a newly discovered subtype of Hashimoto's thyroiditis and characterized by thyroid inflammation and marked fibrosis. IgG4-related Hashimoto's thyroiditis is very rare and there has been relatively little information available to date. A 46-year-old woman with a past history of thyroid dysfunction visited our outpatient clinic for severe anterior neck swelling. She complained of swallowing discomfort and pain due to severe goiter and was successfully treated with total thyroidectomy. Immunohistochemistry showed thyroid invasion by IgG4-positive cells and an IgG4/IgG ratio over 40%. The patient was diagnosed with IgG4-related Hashimoto's thyroiditis. We report a very rare case of IgG4-related Hashimoto's thyroiditis with severe goiter. A more comprehensive understanding of the IgG4-related Hashimoto's thyroiditis may help physicians to allow proper diagnosis and treatment of patients with severe goiter.


2020 ◽  
Vol 154 (Supplement_1) ◽  
pp. S98-S98
Author(s):  
C N Giraldo ◽  
D Myers ◽  
A Holmes ◽  
J Dodd ◽  
W Wendi

Abstract Introduction/Objective Cutaneous plasmacytosis (CP) is an uncommon condition typically affecting Asian males in the 3rd to 5th decades. It is thought to be a reactive process that classically presents with asymptomatic, red-brown, plaques and nodules on the face and neck. It has been associated with polyclonal hypergammaglobulinemia and systemic involvement. Histologically it is characterized by dense superficial and deep dermal infiltrates of mature plasma cells with polyclonal differentiation on in-situ hybridization (ISH). The differential diagnosis includes neoplastic plasma cell processes, characteristically with monoclonal plasma cell infiltrate, and mature B cell neoplasms with polyclonal plasma cell differentiation. Methods We report a case of a 69 year old Caucasian male who presented with asymptomatic, enlarging red-brown nodules on bilateral nasal ala. Histologic examination revealed dense, superficial plasma cell infiltrate, concerning for a plasma cell neoplasm. CD138 and Kappa/Lambda ISH demonstrated plasma cell polyclonality. Further workup ruled out infectious or systemic involvement and a plasma cell dyscrasia was ruled out by Hematology/Oncology. Results These findings supported the diagnosis of CP. Treatment with intralesional steroids showed initial improvement with regrowth of the nodules. To date, treatment with topical steroids and CO2 laser ablation are being considered. Conclusion CP is reported as type of pseudolymphoma, which is described as a reactive lymphoproliferation that histopathologically and/or clinically imitates cutaneous lymphoma. The pathogenesis is unknown, however, there are studies suggesting an association with increased interleukin-6, which is involved in the differentiation of B cells to mature plasma cells. The majority of patients with CP have a favorable prognosis. There has been variable success with both topical and intralesional treatment to include, cyclophosphamide, topical tacrolimus, prednisone, intralesional steroid therapy, topical psoralens combined with ultraviolet A exposure, and other chemotherapies. Familiarity with this rare entity is imperative to prevent misdiagnosis and overtreatment.


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