- Email: [email protected]
DRESS TO TEN: IS THIS A CONTINUUM?
Abstracts: Medically Challenging Cases / Ann Allergy Asthma Immunol 121 (2018) S63−S134
infrequently, a delayed type hypersensitivity. Various agents have been reported to cause CD including topical medications, surgical instruments, and surgical glue. We present a patient who developed rash around surgical wounds. Skin patch testing identified the causative agent of his CD. Case Description: A 15-year-old male presented with new onset of pruritic papulovesicular rash three weeks following Nuss procedure, a minimally invasive pectus excavatum repair using a metal bar. The rash initially appeared on upper extremities and chest wall around surgical wounds (Figure 1A). Later, new erythematous lesions occurred on foreskin of penis. Patient denied other systemic symptoms or prior history of metal or medication allergy. A delayed hypersensitivity to nickel or other components of stainless steel used in the pectus bar was suspected. Surgical removal of the bar was contemplated. A trial of oral prednisone 30mg BID led to resolution of the rashes (Figure 1B). Blood test for metal-lymphocyte proliferation assay was unremarkable. Skin patch test was negative to nickel and metal disc, and confirmed a reaction to 2-octyl cyanoacylate (surgical glue) (Figure 1C). Discussion: Delayed-type hypersensitivity to surgical glue should be considered in patients who develop rash around surgical wounds. Evaluation of surgery-related CD requires a thorough understanding of procedure steps and potential exposures to medical products. Patch test can aid in identification of the offending agent and allow a recommendation of alternative products to avoid recurrence.
S133
our patient was successfully treated with a combination of IVIG, steroids and Cyclosporine. Progression towards TEN.
Figure. A. Rash around surgical wounds Figure 1B. Improvement of rash after a 5-day course of prednisone 30 mg BID treatment Figure 1C. Positive patch test to 2-octyl cyanoacrylate (DermabondÒ surgical glue)
M511
M512
DRESS TO TEN: IS THIS A CONTINUUM? F. Khan*, J. Simonaire, N. Klaiber, S. Kumar, Richmond, VA
RECURRENT ERYTHEMA MULTIFORME, RESOLVED AFTER IMMULOGLOBULIN REPLACEMENT THERAPY IN A PATIENT WITH NORMAL IMMUNOGLOBULIN LEVELS A. McInerney*, S. Siegel, Valhalla, NY
Introduction: Drug Reaction with Eosinophilia and Systemic Symptoms (DRESS) is a potentially life-threatening severe cutaneous adverse drug reaction (SCAR) that can be triggered by medications. Toxic Epidermal Necrolysis (TEN) is also a type of SCAR. To the best of our knowledge, we present a first known clear case of DRESS which progressed to clinical TEN and was successfully treated. Case Description: A 17-year old female was started on Lamotrigine and Escitalopram for a mood disorder. One week later, she developed tender cervical lymphadenopathy, fevers, and abdominal discomfort. About 2.5 weeks after starting these two medications, she developed a raised, pruritic, erythematous rash on her arms. Although the medication was discontinued, she failed to improve and developed facial angioedema, hepatic failure, eosinophilia and atypical lymphocytosis - all concerning signs for DRESS. Skin biopsy confirmed perivascular lymphocytic infiltrate with scattered eosinophils supporting DRESS. She was treated with intravenous immunoglobulin and eventually discharged home with a resolving rash. As Prednisone was tapered, the patient was readmitted with concerns for relapsing DRESS. She went on to develop TEN with mucosal involvement as well as greater than 30 percent skin involvement. She was successfully treated with a combination of Cyclosporine and high dose Prednisone. Discussion: There is paucity of literature on SCAR overlap syndromes and we acknowledge the ambiguity and difficulty with this diagnosis given the overlapping presentation with SCAR syndromes. Although the mortality rate tends to be quite high for SCAR overlap syndromes,
Introduction: While immunoglobulin has been used for StevensJohnson syndrome (SJS), its use in erythema multiforme (EM) is limited. Our case describes recurrent EM after SJS with antibiotic exposure and Mycoplasma pneumoniae infection, with normal immunoglobulin levels, which failed standard treatment but resolved following immunoglobulin replacement therapy (IRT). Case Description: A 13-year-old boy developed fever, cough, and rash progressing to generalized sloughing bullae, with dyspnea that deteriorated to emphysematous lung changes. The lesions were targetoid with erythematous base, dusky center, including mucosa. Symptoms developed after treatment for presumed pneumonia with amoxicillin-clavulanate. He was admitted with SJS and Mycoplasma pneumoniae. Steroids, azithromycin, and acyclovir were given. Biopsy was consistent with EM. He was discharged on azithromycin, doxycycline, and valacyclovir for persistent lesions, although HSV titers and cultures were negative. Immune work-up was unremarkable. He lacked immunity to multiple pathogens, despite vaccination, but responded to re-immunization. Immunoglobulins remained normal. Lesions recurred frequently, with minimal response to steroids, and flares with weaning. IRT was administered during re-admission with near resolution. He was then started on IRT therapy. Steroids were weaned without recurrence.
infrequently, a delayed type hypersensitivity. Various agents have been reported to cause CD including topical medications, surgical instruments, and surgical glue. We present a patient who developed rash around surgical wounds. Skin patch testing identified the causative agent of his CD. Case Description: A 15-year-old male presented with new onset of pruritic papulovesicular rash three weeks following Nuss procedure, a minimally invasive pectus excavatum repair using a metal bar. The rash initially appeared on upper extremities and chest wall around surgical wounds (Figure 1A). Later, new erythematous lesions occurred on foreskin of penis. Patient denied other systemic symptoms or prior history of metal or medication allergy. A delayed hypersensitivity to nickel or other components of stainless steel used in the pectus bar was suspected. Surgical removal of the bar was contemplated. A trial of oral prednisone 30mg BID led to resolution of the rashes (Figure 1B). Blood test for metal-lymphocyte proliferation assay was unremarkable. Skin patch test was negative to nickel and metal disc, and confirmed a reaction to 2-octyl cyanoacylate (surgical glue) (Figure 1C). Discussion: Delayed-type hypersensitivity to surgical glue should be considered in patients who develop rash around surgical wounds. Evaluation of surgery-related CD requires a thorough understanding of procedure steps and potential exposures to medical products. Patch test can aid in identification of the offending agent and allow a recommendation of alternative products to avoid recurrence.
S133
our patient was successfully treated with a combination of IVIG, steroids and Cyclosporine. Progression towards TEN.
Figure. A. Rash around surgical wounds Figure 1B. Improvement of rash after a 5-day course of prednisone 30 mg BID treatment Figure 1C. Positive patch test to 2-octyl cyanoacrylate (DermabondÒ surgical glue)
M511
M512
DRESS TO TEN: IS THIS A CONTINUUM? F. Khan*, J. Simonaire, N. Klaiber, S. Kumar, Richmond, VA
RECURRENT ERYTHEMA MULTIFORME, RESOLVED AFTER IMMULOGLOBULIN REPLACEMENT THERAPY IN A PATIENT WITH NORMAL IMMUNOGLOBULIN LEVELS A. McInerney*, S. Siegel, Valhalla, NY
Introduction: Drug Reaction with Eosinophilia and Systemic Symptoms (DRESS) is a potentially life-threatening severe cutaneous adverse drug reaction (SCAR) that can be triggered by medications. Toxic Epidermal Necrolysis (TEN) is also a type of SCAR. To the best of our knowledge, we present a first known clear case of DRESS which progressed to clinical TEN and was successfully treated. Case Description: A 17-year old female was started on Lamotrigine and Escitalopram for a mood disorder. One week later, she developed tender cervical lymphadenopathy, fevers, and abdominal discomfort. About 2.5 weeks after starting these two medications, she developed a raised, pruritic, erythematous rash on her arms. Although the medication was discontinued, she failed to improve and developed facial angioedema, hepatic failure, eosinophilia and atypical lymphocytosis - all concerning signs for DRESS. Skin biopsy confirmed perivascular lymphocytic infiltrate with scattered eosinophils supporting DRESS. She was treated with intravenous immunoglobulin and eventually discharged home with a resolving rash. As Prednisone was tapered, the patient was readmitted with concerns for relapsing DRESS. She went on to develop TEN with mucosal involvement as well as greater than 30 percent skin involvement. She was successfully treated with a combination of Cyclosporine and high dose Prednisone. Discussion: There is paucity of literature on SCAR overlap syndromes and we acknowledge the ambiguity and difficulty with this diagnosis given the overlapping presentation with SCAR syndromes. Although the mortality rate tends to be quite high for SCAR overlap syndromes,
Introduction: While immunoglobulin has been used for StevensJohnson syndrome (SJS), its use in erythema multiforme (EM) is limited. Our case describes recurrent EM after SJS with antibiotic exposure and Mycoplasma pneumoniae infection, with normal immunoglobulin levels, which failed standard treatment but resolved following immunoglobulin replacement therapy (IRT). Case Description: A 13-year-old boy developed fever, cough, and rash progressing to generalized sloughing bullae, with dyspnea that deteriorated to emphysematous lung changes. The lesions were targetoid with erythematous base, dusky center, including mucosa. Symptoms developed after treatment for presumed pneumonia with amoxicillin-clavulanate. He was admitted with SJS and Mycoplasma pneumoniae. Steroids, azithromycin, and acyclovir were given. Biopsy was consistent with EM. He was discharged on azithromycin, doxycycline, and valacyclovir for persistent lesions, although HSV titers and cultures were negative. Immune work-up was unremarkable. He lacked immunity to multiple pathogens, despite vaccination, but responded to re-immunization. Immunoglobulins remained normal. Lesions recurred frequently, with minimal response to steroids, and flares with weaning. IRT was administered during re-admission with near resolution. He was then started on IRT therapy. Steroids were weaned without recurrence.