Mucosal Involvement Research Articles

Acyclovir Induced Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis - A Rare Case Report

Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis are serious, life-threatening conditions often triggered by drug reactions, characterized by widespread skin detachment and mucosal involvement. This case report describes a 29-year-old woman who developed Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis after receiving prophylactic acyclovir for suspected herpes. The patient initially presented with oral ulcers that rapidly progressed to widespread blisters, ulcers, and severe skin peeling. A thorough diagnostic evaluation, including skin biopsy and the Naranjo algorithm, identified acyclovir as the causative agent. The patient was managed in intensive care with intravenous methylprednisolone 500mg per day for 3 consecutive days as pulse therapy, antibiotics, and other supportive treatments. This case underscores the importance of early recognition, prompt discontinuation of the offending drug, and a multidisciplinary treatment approach. It also highlights the critical need for heightened awareness among healthcare professionals regarding the potential for severe adverse reactions with commonly used medications like acyclovir, to improve patient safety and outcomes.

Toxic epidermal necrolysis: management strategies in burns units.

The most common severe exfoliative condition is toxic epidermal necrolysis (TEN), also known as Lyell's syndrome, for which patients may sometimes require admission to hospital burns units. This study analyses the experience of the authors and results in this condition at the Cruces University Hospital in Bilbao, Spain. Over the last 22 years, the authors carried out a retrospective analytical observational study of TEN cases at the hospital and analysed risk factors including age, associated comorbidities, percentage of body surface area affected, positive bacterial culture, and treatment strategies used to assess their potential influence on prognosis. The results indicated an association between mortality and age (ρ (rho)=0.60, 95% CI [0.29, 0.79], p<0,001), ocular (ρ=0.58, 95% CI [0.28, 0.78], p<0.001), oropharyngeal (ρ=0.64, 95% CI [0.36, 0.81], p<0.001), genitourinary (ρ=0.58 CI [0.28, 0.78], p<0.001) respiratory mucosa involvement (ρ=0.53, 95 % CI [0.28, 0.78], p<0.001) and bacteraemia (ρ=0.44, 95% CI [0.17, 0.64], p=0.020). Bacteraemia also showed a higher incidence in elderly patients (ρ=0.4, 95% CI [0.03, 0.67], p=0.033) and in those with a higher Score for Toxic Epidermal Necrolysis at admission (ρ=0.51, CI [0.17, 0.74], p=0.006). A reduction in mortality was found in patients who received systemic treatment; cyclosporine, etanercept or a combination of both (ρ=-0.44, Cl [-0.70, -0.09], p=0.018). TEN is a rare but serious exfoliative condition. Traditionally, management of denuded skin was seen as the main prognostic factor; yet, systemic damage, especially to the respiratory mucosa, significantly increases mortality risk. While life support is crucial and recovery is possible, preventing long-term sequelae relies on effective care protocols. Current treatments may offer benefits, though their efficacy remains unclear. Clinicians in burns units should develop standardised protocols and collaborate on long-term studies to enhance patient care.

Advances in Adhesive Materials for Oral and Maxillofacial Soft Tissue Diseases.

Oral diseases represent a prevalent global health burden, profoundly affecting patients' quality of life. Given the involvement of oral mucosa and muscles in diverse physiological functions, coupled with clinical aesthetics considerations, repairing oral and maxillofacial soft tissue defects poses a formidable challenge. Wet-adhesive materials are regarded as promising oral repair materials due to their unique advantages in easily overcoming physical and biological barriers in the oral cavity. This review first introduces the intricate wet-state environment prevalent in the oral cavity, meticulously explaining the fundamental physical and chemical adhesion mechanisms that underpin adhesive materials. It then comprehensively summarizes the diverse types of adhesives utilized in stomatology, encompassing polysaccharide, protein, and synthetic polymer adhesive materials. The review further evaluates the latest research advancements in utilizing these materials to treat various oral and maxillofacial soft tissue diseases, including oral mucosal diseases, periodontitis, peri-implantitis, oral and maxillofacial skin defects, and maxillofacial tumors. Finally, it also highlights the promising future prospects and pivotal challenges related to stomatology application of multifunctional adhesive materials.

Clinicopathologic findings in a cohort of metastases to the stomach

AimsMetastatic tumors to the stomach can mimic primary gastric adenocarcinoma or be subtle and difficult to identify. The current study aimed to characterize the clinicopathology of metastases to the stomach to aid in diagnosis. Methods and resultsForty-three metastatic tumors and 30 primary gastric adenocarcinoma cases were reviewed. Metastases originated from numerous primaries with the most common being mammary (n = 17) or melanoma (n = 9). The gastric metastasis represented the initial diagnosis for 9 (21%) cases without previous history of malignancy. The median age at diagnosis was similar (metastatic 66 years; primary 67.5 years; P = 0.42). The most common indication for procedure was abdominal pain (23%; P = 0.95) in metastases and melena (43%; P < 0.01) in primaries. Procedural findings suggestive of metastasis over primary adenocarcinoma were multiple lesions (23% versus 0%; P = 0.01), non-mass forming mucosal changes (30% versus 0%; P < 0.01), submucosal nodularity (14% versus 0%; P = 0.09), and absence of ulceration (9% versus 53%; P < 0.01). Histologic findings less commonly seen in metastasis were mucosal layer involvement (86% versus 100%; P = 0.09), ulceration (40% versus 70%; P = 0.02), surface epithelial involvement/colonization by tumor (12% versus 60%; P < 0.01), intestinal metaplasia (9% versus 53%; P < 0.01), background dysplasia (0% versus 30%; P < 0.01), and Helicobacter pylori infection (0% versus 20%; P < 0.01). Lymphovascular invasion had similar prevalence (metastatic 23%; primary 20%; P = 0.70). ConclusionsMetastasis to the stomach included a variety of primary sites and was not infrequently the initial diagnosis. Patient demographics were similar to primary adenocarcinoma. Multiple lesions, non-mass forming mucosal changes, and/or submucosal nodularity were more common in metastasis. Histologically, the absence of surface epithelial involvement, ulceration, intestinal metaplasia, background dysplasia, or H. pylori infection can raise suspicion for metastasis.

MALTectomy and psoriasis risk in women: A prospective study in the French E3N prospective cohort.

The involvement of mucosa associated lymphoid tissues (MALT) in the development of an autoimmune response in the skin is unclear and unstudied. In this study we sought to assess the relationship between removal of MALT tissues (MALTectomy) and the risk of development of psoriasis (overall or moderate-to-severe). We conducted a prospective observational study based on E3N, a French cohort composed of 98 995 women born between 1925 and 1950 and insured by the health insurance of the national education system (MGEN). The study population included the 90 119 women that completed the 1990 baseline questionnaire with available information on MALTectomy and a valid incident diagnostic date for psoriasis. During the 1990-2018 follow-up period 2 433 incident cases of psoriasis were identified through self-reports while during the period for which drug reimbursement data were available from the MGEN database (2004-2018), 120 cases of moderate-to-severe psoriasis were identified. Hazard ratios (HR) and their 95% confidence intervals were estimated by Cox proportional hazards models and adjusted for known or putative psoriasis risk factors. Appendectomy was associated with an increased risk of psoriasis both in the univariate [HR: 1.17 (95% CI: 1.08-1.27)] and multivariable models [HR: 1.14 (95% CI: 1.05-1.24)]. A suggestive association with appendectomy was observed for moderate-to-severe psoriasis risk [univariate HR: 1.40 (95%CI: 0.97-2.02); multivariable HR: 1.36 (95%CI: 0.94-1.96)]. No association was observed between tonsillectomy or adenoidectomy both for overall and moderate-to-severe psoriasis. The observed association between appendectomy and risk of psoriasis warrants further investigations as they may help to elucidate the disease etiology and improve risk prediction.

Cutaneous manifestations and sequelae of Chikungunya fever: a single centre experience

Background: Chikungunya fever is a common viral illness in the tropical and subtropical areas. Various cutaneous manifestations can develop during the acute phase and post-febrile convalescent phase. Objective: To determine the frequency and types of cutaneous manifestations associated with chikungunya fever in acute and post-febrile phase. Methods: We conducted a prospective, observational study including 20 patients with laboratory-confirmed chikungunya fever. All the patients were followed up for 1 year to detect any cutaneous sequelae. Results: Pruritic maculopapular rash was the most common cutaneous manifestation during the acute febrile phase, noted in 60% of the patients. The rash usually appeared within 1–3 days of fever onset and spread in cephalo-caudal direction. Cutaneous hyperpigmentation was the most common dermatologic sequelae in the post-febrile period, occurring in 3 (15%) patients. In all patients, pigmentation appeared 3–4 weeks after resolution of fever, and started on the nose, gradually spreading to the forehead. The pigmentation improved significantly with topical hydroquinone after 4 weeks. Other additional manifestations of chikungunya fever included joint pain and joint swelling. None of our patients presented with mucosal or systemic involvement. Conclusion: Maculopapular skin rash was the most common manifestation during acute febrile phase of chikungunya fever, while cutaneous hyperpigmentation was the most common dermatologic sequelae after resolution of fever.

Kawasaki Disease: A Single-Center Study from a Tertiary Care hospital in Dhaka, Bangladesh

Background: Kawasaki Disease (KD) is recognized as the leading cause of acquired heart disease in children in developed countries. This is a pioneer study from Bangabandhu Sheikh Mujib Medical University (BSMMU), documenting the common epidemiological, clinical, laboratory and echocardiographic findings, including management and immediate outcome of KD children. Method: This retrospective study includes all patients with a diagnosis of KD, who attended the pediatric rheumatology division at BSMMU from January 2015 to December 2019. Data were collected from medical records at diagnosis and immediately after management. Relevant statistical tests were done and a p value &lt; 0.05 was considered as statistically significant. Result: A total of 129 cases were diagnosed with KD, among them complete KD cases were 62% and incomplete KD was 38%. The mean age at onset was significantly higher in incomplete KD than complete KD. The majority of the cases (67%) were between 1 to 5 years of age and male (72%) children were predominant. Fever was the most common presenting feature (100%), followed by bilateral conjunctivitis, mucosal involvement, rash, changes in extremities, cervical lymphadenopathy and others. Coronary artery dilatation was present in 30.5% cases with no significant differences between complete and incomplete KD cases. Aspirin was given to all the cases and IVIG was given to only 46.6% children. Rests of the children were treated with methyl prednisolone. All the cases improved after treatment and there was no immediate mortality. Conclusion: It seems that KD is not an uncommon disease and incomplete KD cases are relatively more prevalent. About 30% KD cases had coronary artery abnormalities. Immediate outcome of management is excellent but needs long term follow-up. BANGLADESH J CHILD HEALTH 2023; VOL 47 (1) : 9-16

Gastrointestinal manifestations and pathogenesis in childhood immunoglobulin A vasculitis.

Immunoglobulin A vasculitis (IgAV), previously known as Henoch-Schönlein purpura, is the most common form of systemic vasculitis in childhood. The primary organs involved are the skin, gastrointestinal (GI) tract, joints, and kidneys. The spectrum of GI involvement in IgAV ranges from being mild and self-limited to severe manifestations often requiring surgical intervention. Galactose-deficient IgA1 on the immunoglobulin hinge region and its immune complexes are thought to play a central pathogenetic role in IgAV, however, an association between such molecules and specific GI mucosal damage remains unclear. GI endoscopy (both upper and lower) shows a variety of mucosal findings, many of which are not specific for IgAV. In upper GI endoscopy, however, the mucosal features can be diagnostic when found localized in the more distal part of upper GI tract (second and/or third parts of the duodenum). Abdominal computed tomography and capsule endoscopy have demonstrated that the small intestine is most commonly involved in IgAV. The GI mucosal involvement when evaluated microscopically shows IgA deposition which is histologically diagnostic. Conversely, leukocytoclastic vasculitis is less useful. Since the 1960s, cases of duodenojejunitis, in which IgAV was suspected but evident purpura was not dermatologically present, have often been labeled as "idiopathic". In a pediatric case series, IgA enteropathy, without dermatological manifestations (i.e., purpura), was reported to have similar symptoms, as well as endoscopic characteristics and immunohistological findings as in IgAV. Subsequently, several case reports provide additional supportive evidence that IgA enteropathy must be a variant of IgAV. Thus, the immunologically driven auto-immune vasculitis results in the symptom complex dependent on the organ system involved, and the subsequent clinical features which are manifested. Present classification criteria are useful and universally available for diagnosing IgAV. However, based upon current knowledge including IgA enteropathy, minor modification of the IgAV criteria is proposed in the review.

PERITONEAL KERATIN GRANULOMAS MIMICKING PERITONEAL CARCINOMATOSIS: A POTENTIAL DIAGNOSTIC PITFALL

Abstract Introduction/Objective Keratin granulomas from a primary uterine cancer can present as peritoneal tumor nodules, mimicking peritoneal carcinomatosis. Endometrial adenocarcinomas with viable peritoneal tumor implants and/or involvement of bladder or bowel mucosa are categorized as stage IV disease. We present a rare case of endometrial adenocarcinoma of the uterus with squamous differentiation and multiple peritoneal nodules of keratin granulomas, masquerading as peritoneal carcinomatosis. Methods/Case Report A 68-year-old female with insignificant past medical history presented with complaints of postmenopausal bleeding for the past four months. Physical examination was unremarkable. Transvaginal ultrasound showed a 4.9 cm echogenic mass in the uterus. The endometrial biopsy showed squamous debris with atypical cells suspicious of malignancy. Endometrial curettings revealed endometrial adenocarcinoma, endometrioid type with squamous differentiation, FIGO Grade 1. Subsequently, she underwent hysterectomy with bilateral salpingo- oophorectomy. Intraoperatively, multiple suspicious nodules on the sigmoid colon and in the pelvis were identified, of which excisional biopsies and pelvic peritoneal washing were taken. Microscopic examination of the resection confirmed endometrial adenocarcinoma, FIGO Grade 1, nuclear grade 1, with extensive foreign body giant cell reaction to dead squamous cells (ghost cells). Cytologic examination of the peritoneal washing was unremarkable and excisions of the sigmoid serosal and peritoneal nodules showed pink eosinophilic laminated nodules surrounded by inflammatory cells including plasma cells, lymphocytes and multinucleated giant cells without any viable tumor cells. Results (if a Case Study enter NA) NA Conclusion Keratin granulomas are a foreign body reaction to keratin deposition from the squamous component of endometrioid adenocarcinoma. The pathogenesis of this foreign body response to keratin is considered to be a trans- tubal spread of keratin through retrograde shedding from the fallopian tubes. In the absence of viable tumor cells, it appears to have no negative prognostic significance. Careful microscopic examination of peritoneal keratin granulomas is imperative to avoid erroneous upstaging of the tumor and the subsequent advanced treatment.

Malakoplakia, a rare inflammatory condition presenting as a colon polyp

Abstract Introduction/Objective Malakoplakia is a rare histiocytic disease caused by defects in phagocytic or degradative functions of histiocytes in response to gram negative coliforms (E. coli or Proteus) that results in chronic inflammatory state, followed by intracellular deposition of iron and calcium (known as Michaelis-Gutmann bodies). It’s more common in immunocompromised patients and the mean age at diagnosis is fifth decade. It occurs in all organs, common in GU tract, particularly bladder, but also seen in gastrointestinal tract (most commonly colon, followed by stomach and duodenum), central nervous system and female genital tract. In gastrointestinal tract, it can present as a polyp with diffuse mucosal involvement, submucosal thickening, and even may cause mass. Methods/Case Report A 60 -year-old male smoker presented to our hospital with history of colon polyps; colonoscopy revealed a 6 mm sessile polyp at the hepatic flexure. A biopsy was performed which showed numerous epithelioid histiocytes with granular eosinophilic cytoplasm, abundant Michaelis-Gutmann bodies and increased lymphocytes. Immunohistochemical stains showed diffuse positivity for CD68; and PAS stain was positive for histiocytic cytoplasmic granules. Based on this data, the diagnosis of Malakoplakia was confirmed. Malakoplakia is treated with antibiotics that concentrate in macrophages (quinolones or trimethoprim-sulfamethoxazole), antibiotics directed against E. coli, surgery and possibly discontinuation of immunosuppressive drug therapy. Results (if a Case Study enter NA) Malakoplakia, in this case, was presented clinically as a sessile colon polyp and histologically showed numerous epithelioid histiocytes with granular eosinophilic cytoplasm, with Immunohistochemical stains (CD68; and PAS stain) the correct diagnosis was made. Other presentations of Malakoplakia include diffuse mucosal involvement, submucosal thickening, or mass. Histologically, it may rarely be mistaken as signet ring carcinoma. These variable presentations in GU tract or colon can be interpreted as malignancy with consequent unnecessary interventions. Hence, awareness of Malakoplakia with careful histopathologic examination and the use of immunohistochemical studies are advisable in that cases. Conclusion We would like to emphasize that Malakoplakia can be clinically and histologically misdiagnosed as a malignant condition; therefore histopathologic and immunohistochemical studies are needed to correctly diagnose this entity.

Oral lymphoma with features of anaplastic large T‐cell lymphoma treated as an erosive and ulcerative stomatitis in a pony horse

AbstractLymphoma is the most common malignant haemolymphatic neoplasm in the horse, with a predominance of multicentric T‐cell‐rich large B‐cell lymphoma. Anaplastic large T‐cell lymphoma is a rare subtype of T‐cell lymphoma characterised by presence of atypical neoplastic lymphoid cells with kidney‐shaped or horseshoe‐shaped nuclei. This case report describes an unusual presentation of a lymphoma with morphological and immunohistological features of an anaplastic large T‐cell lymphoma with oral mucosal involvement in a horse that was originally presented with erosive to ulcerative stomatitis.

Ocular Manifestations in Paediatric Erythema Multiforme: A Case Report

Erythema multiforme (EM) is a type IV cytotoxic hypersensitivity reaction characterized by classic target lesions on the skin and mucosa, typically affecting the oral, genital, and rarely, ocular mucosa. We describe a 9-year-old girl with widespread rashes, blisters and target lesions on her trunkfor three days. She had eye redness, discomfort, a week-long fever, and a dry cough, without any medication. Ophthalmological examination revealed localized conjunctival injection in her right eye, with no visual impairment or corneal lesions. The diagnosis of EM major (EMM) was based on prior infection, characteristic skin lesions, and mucosal involvement, including ocular manifestation of conjunctival injection. Treatment requires addressing the underlying cause and ophthalmologic consultation for systemic or topical corticosteroids, antibiotics, lubricants, and antihistamines. This case highlights the diagnostic challenges of atypical EM presentations, emphasizing the need for comprehensive evaluation, early recognition, and interdisciplinary collaboration to prevent severe ocular complications and preserve vision.

Imaging approach for fungal sinusitis.

This article provides a comprehensive review of the computed tomography (CT) and magnetic resonance (MR) imaging findings of invasive fungal sinusitis with an emphasis on pattern recognition and approach to interpretation. Fungal sinusitis is categorized into invasive (acute, chronic, and granulomatous) and noninvasive forms (allergic fungal sinusitis and mycetoma). CT is superior for detecting bony erosion and hyperdense fungal elements, while MRI excels in evaluating soft tissue and mucosal involvement. Key radiologic signs such as bone destruction, sinus wall thickening, and 'black turbinate sign' aid in early diagnosis, especially in invasive cases. Early imaging signs can be subtle. Early detection is necessary, particularly in immunocompromised patients with acute invasive fungal sinusitis, where rapid intervention is critical. Pattern recognition and adequate interpretation of fungal sinusitis are possible using CT and MRI. Imaging can also help identify complications, aiding with reliable diagnosis and prompt intervention.

Case Report: Steven Johnson Syndrome (SIS) Induced By Antibiotics: A Detailed Analysis

Background: Stevens-Johnson Syndrome SJS is a rare but very serious mucocutaneous disease primarily caused by medications that includes antibiotics. This case report is about a 35-year-old male who suffered from SJS due to treatment for urinary tract infection using co-trimoxazole. Case Presentation: The patient began with symptoms of flu, later on followed by rapid progression of erythematous rash and painful mucosal involvement. On clinical examination, there were blisters, skin detachment involving 10% of the body surface area, and oral and ocular mucosal lesions. This reaction developed in a temporal association with co-trimoxazole therapy, making the drug a likely cause. She was systemically treated with corticosteroids and supportive care and showed marked recovery in two weeks from hospitalization. Conclusion: This case underlines the importance of early suspicion of SJS in patients receiving antibiotic treatment. Removal of the offending drug and timely initiation of multi-disciplinary management will only improve the outcome in such patients.

KINDLER’S SYNDROME − A loss of identity

Kindler’s syndrome (KS) is a rare inherited skin disease characterized by acral blistering, photosensitivity, cutaneous atrophy, and progressive poikiloderma along with different types of mucosal involvement. We hereby report KS in a north eastern female patient. The case is being reported for its rarity and for emphasizing the importance of considering this condition in the differential diagnosis of disorders that may cause blistering, cutaneous atrophy, and/or poikilodermatous skin changes. Besides, being a cause of acquired adermatoglyphia, it could lead to identification problems in the individuals by biometric technology, as was the scenario with our patient who faced difficulty in enrolling for Adhaar ID proof, making the case even more interesting.

Nodular Fasciitis of the Buccal Mucosa with a Novel USP6 Gene Rearrangement: A Case Report and Review of the Literature.

Nodular fasciitis is a rare but benign fibroblastic proliferation that typically presents as a solitary lesion with rapid growth and variable mitotic activity. The lesions usually occur on the extremities and occasionally in the head/neck region. Involvement of the buccal mucosa is extremely rare with only few reports in the literature; in this case report, we describe a 41 year old female who presented with a 6-month history of a stable intraoral lump at the junction of the upper and lower lip. Fine needle aspiration revealed an atypical spindle cell population with plump cells. The surgical excision demonstrated a well circumscribed tan-white firm nodule. Histologic examination revealed a spindle cell proliferation that grew in short, intersecting fascicles with focal storiform architecture. The lesion had a pushing border that was not overtly infiltrative and the stroma contained focal myxoid changes giving a "tissue culture" appearance to the cells. Immunohistochemical testing showed the tumor cells were vimentin (+), SMA (+), weakly Calponin (+), and desmin (-), cytokeratin (-), AE1/AE3 (-), S100 (-), ALK (-), STAT6 (-), and beta-catenin (-). Fluorescence in-situ hybridization (FISH) revealed a USP6 gene rearrangement with an atypical probe pattern. Next generation sequencing identified a novel SPTAN1::USP6 fusion gene confirming the diagnosis of buccal nodular fasciitis. Identification of the characteristic histologic features and USP6 gene rearrangements helped support the diagnosis. A review of the literature identified 25 cases of nodular fasciitis involving the buccal mucosa. The occurrence of this tumor in an unusual location may pose difficulties for diagnosis.

Clinical and immunoserological characteristics of anti-p200 pemphigoid: Comparisons of patients with epitope spreading and non-epitope spreading.

Anti-p200 pemphigoid is a rare autoimmune subepidermal blistering disease. Although the phenomenon of epitope spreading has been reported to be common in anti-p200 pemphigoid, the association between its clinical and immunoserological features has yet to be elucidated. Our aim was to compare the clinical and immunoserological characteristics of anti-p200 pemphigoid patients with and without epitope spreading. We performed a retrospective cohort study encompassing 30 patients with anti-p200 pemphigoid between January 2015 and December 2022. The clinical and immunoserological characteristics of anti-p200 pemphigoid were analyzed using combined immunoserological assays. Epitope spreading was observed in 11 of 30 patients (36.7%) with anti-p200 pemphigoid. Compared with patients in the non-epitope spreading group, patients in the epitope spreading group showed more heterogeneous clinical presentations (P = 0.018), a higher proportion of mucosal involvement (P = 0.003), higher Bullous Pemphigoid Disease Area Index (BPDAI) scores for skin erosions/blisters (P = 0.018), mucosal erosions/blisters (P = 0.001), activity (P = 0.017) and total scores (P = 0.022), and required a higher initial dose of prednisone for disease control (P = 0.040). This study supported the idea that anti-p200 pemphigoid was prone to epitope spreading. Anti-p200 pemphigoid patients with epitope spreading are more likely to present heterogeneous clinical phenotypes, frequent mucosal involvement, and a more severe and recalcitrant disease course.

Acute and chronic ocular outcomes in SJS/TEN patients treated with oral ciclosporin vs intravenous immunoglobulin.

To evaluate differences in ocular complications of Stevens Johnson Syndrome (SJS)/Toxic Epidermal Necrolysis (TEN) patients receiving either systemic IVIG or Ciclosporin (CsA) as initial treatments. Retrospective review of consecutive patients admitted for SJS/TEN at the Singapore General Hospital (SGH) from 2011 to 2017 who received either IVIG or Ciclosporin at the onset of the disease and had ophthalmological follow-up of at least 6 months were included. Acute ocular severity of SJS/TEN was graded using the Gregory grading score; chronic ocular complications were graded using the Sotozono system. A total of 18 subjects were included for analysis, with eight in the IVIG group and 10 in the CsA group. There were no significant differences in acute Gregory severity grading between the two groups. The CsA group had a trend towards worse overall chronic Sotozono grading scores compared to the IVIG group (median [IQR]: 2 [0-3] vs. 1 [0-6.5], p = 0.27), with a higher incidence of acute severe cornea involvement (60% vs. 25%, p = 0.93) and chronic corneal and eyelid involvement in the former than the latter. SJS/TEN patients with worse acute ocular involvement were more likely to have TEN and perianal mucosal involvement (50% vs. 0, p = 0.01). Compared to those who received IVIG, SJS/TEN patients who received CsA at the acute disease stage, seemed to have worse acute corneal and chronic corneal and eyelid complications. Larger studies are needed to confirm this finding.

An Investigation of Mucosal Leishmaniasis in the Military Health System.

Leishmaniasis is a protozoal infection with an increased risk of transmission to those serving in the U.S. Military due to theaters of operation in endemic regions. There has, in recent decades, been robust experience with old-world leishmaniasis in the Military Health System (MHS); however, new-world leishmaniasis, which may result in mucosal leishmaniasis, has been less studied. A total of 88 patients from 2012 to 2022 with diagnosis codes for "mucocutaneous leishmaniasis" or "leishmaniasis, unspecified" were identified in the Military Data Repository and reviewed. Within this cohort, upon medical records review, there were 2 cases of leishmaniasis that met inclusion criteria. Case 1 was a 28-year-old active duty male with recent travel to Belize who presented with a mucosal lip lesion that was biopsied and had inconclusive species confirmation but was thought to be either L. braziliensis or L. mexicana. The second case involved a 30-year-old active duty male with a history of travel to French Guiana who had a cutaneous lesion on his left hand that was identified as L. guyanensis, a causative species for mucosal leishmaniasis. Neither had evidence of any further mucosal involvement on otolaryngologic evaluation, and both subsequently received systemic therapy with a good clinical response. Although only 2 cases were identified over an 11-year period, this disease remains an important medical consideration when conducting military operations within Central and South America, as both cases had recent military-specific travel to areas endemic for leishmaniasis.

Stevens–Johnson syndrome-toxic epidermal necrolysis overlap in a patient taking quetiapine and famotidine: a case report

BackgroundStevens–Johnson syndrome-toxic epidermal necrolysis (SJS-TNE) overlap is a rare skin disorder characterized by erythema, blisters, extensive exfoliation, epidermal detachment, the involvement of multiple mucosae, and positive Nikolsky’s sign. SJS-TEN has a high mortality rate. Our case involves a rare occurrence of drug-induced Stevens–Johnson syndrome-toxic epidermal necrolysis overlap with a delayed onset in the setting of quetiapine and famotidine therapy.Case presentationAn 82-year-old Taiwanese female was admitted to our hospital for decreased urine output, generalized edema, and multiple skin blisters and bedsores. With further spread of the lesions, multiple ruptured bullae with shallow erosions on the face, trunk, and limbs and mucosal involvement affected 20% of the total body surface area. Nikolsky’s sign was positive. A diagnosis of Steven–Johnson syndrome was highly suspected. One month prior, she had started famotidine and quetiapine. Intravenous methylprednisolone treatment was initiated, which ameliorated the skin lesions after 3 days. However, new lesions developed after only 1 day of methylprednisolone tapering. The patient died 12 days after admission.ConclusionStevens–Johnson syndrome-toxic epidermal necrolysis is a rare skin disorder. Although it is mainly acute and has a high mortality rate, delayed onset can still occur. Quetiapine and famotidine are generally safe and effective for treating geriatric and gastrointestinal problems, but rare drug hypersensitivity reactions can lead to debilitating consequences. Therefore, increased clinical awareness and the initiation of supportive care are imperative. Optimal management guidelines are still lacking, and confirmation of developed guidelines through randomized controlled trials is needed. Collaboration for better management strategies is warranted.