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Allergy Grand Rounds. Sarbjit S. Saini, M.D. JHAAC December 3, 2004. Chief complaint . 13 yr old male referred in June 2004 for evaluation of severe chronic urticaria Referred by pediatric allergist in VA Significant illnesses: include Type I DM for 2.5 yrs ADHD mood disorder.

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allergy grand rounds

Allergy Grand Rounds

Sarbjit S. Saini, M.D.

JHAAC

December 3, 2004

chief complaint
Chief complaint
  • 13 yr old male referred in June 2004 for evaluation of severe chronic urticaria
  • Referred by pediatric allergist in VA
  • Significant illnesses:
    • include Type I DM for 2.5 yrs
    • ADHD
    • mood disorder
history of present illness i
History of Present Illness-I
  • June 02- lip swelling 1-2x/month, no Tx
  • Dec 02 -swelling of face, eyes, DIB and diffuse urticaria
    • Poor relief with fexofenadine & diphenhydramine
    • Required 4 prednisone tapers for control
  • March 03- seen by allergist in VA
    • Cetirizine, montelukast, ranitidine begun
    • PFTs within normal, FEV1 103%
    • Skin tests : + to dust mites, cats, dog, cockroach,trees, grasses, mold; +peanuts, nuts, garlic, shellfish, fish
    • Food RASTs all negative
history of present illness ii
History of Present Illness-II
  • June 03- 1 d hospitalization, anxiety?
  • July 03- 3 of last 6 mos on prednisone
    • Fexofenadine + to Cetirizine, montelukast, ranitidine
    • CBC,WESR, AST, ANA, thyroid antibodies, H. pylori
  • Sept 03- hydroxyzine added, zafirulukast bid
    • C3, C4, CH50 checked
    • Lesions-erythematous, pruritic, painful with occasional bruising
    • Duration of lesions-minutes to 1 day
history of present illness iii
History of Present Illness-III
  • Oct 03- cyclosporine 100 mg qd (2 mg/kg)
    • stopped all but prn H1
    • Insulin RAST drawn, baseline labs, BP 132/78
  • Nov 03-CsA helping, but hives still significant
    • increased to 100 mg BID CsA (4 mg/kg)
    • ER visit for lip swelling /DIB
    • 1 pred taper, prescribed an epipen
  • May 04 -2 pred courses, 3 uses of epipen due to DIB with urticaria
    • Restarted on fexofenadine,cetirizine, zafirlukast,
    • joint pains in knees with activity without morning stiffness and not steroid responsiveness
other atopic history
Other atopic history
  • No history of eczema or food allergy
  • Allergic rhinitis symptoms
  • Exercise-related asthma age 9 treated with prn albuterol prior to activity
  • Reported qhs cough, but denied wheezing
medications
Medications
  • Zoloft, 50 mg qd**
  • Oxcadazepine (Trileptal) 300 mg/600 mg **
  • Adderall 30 mg bid
  • Quetiapine (Seroquel) 200 mg qd
  • Fexofenadine 180 mg qd ( off 1 wk)
  • Cetirizine 10 mg qd ( off 1 week)
  • Cyclosporine 100 mg bid (off 1 wk)
  • Humulin 7 U/4 U, Humulin R 5 U/ 4 U
  • Epipen, Albuterol
past medical history
Past Medical History
  • Type I DM for 2.5 yrs
  • ADHD
  • Mood disorder, possible bipolar
    • exacerbated by steroids
    • suicidal ideation due to urticaria
  • Chicken pox as child
  • Salivary gland surgery
  • Normal birth history, negative history of other infections
past history
Family History

Younger Sister with eczema

PGM with asthma

Paternal cousins with asthma

Environmental Hx

Apt dweller x 5 yrs

Dog since 1999

3 hamsters

Social Hx

7th grader

Lives with mom and sister

Past History
physical exam
Physical Exam
  • T-99.7, HR-121, BP-109/75, HT-5, WT-125,RR-22
  • General: no obvious pubertal signs,central obesity, moon facies
  • HEENT: “allergic shiners”,erythematous nasal mucosa, prominent turbinates
    • Normal TMs, oropharynx, neck
  • Resp: CTA, normal I:E ratio, CV: nl S1, S2 tachy
  • Abdomen: benign Ext: no joint swelling
  • Skin: urticaria on face, arms, feet, back, chest; no pigmentation
recent labs
Recent labs
  • CBC-WBC 7.3 HCT-41.2, Plts-331
  • HbA1C-8.6 (4-6) Jan 2004
  • Negative studies: ANA, H. Pylori Ab,anti-thyroid peroxidase antibodies, WESR
  • Normal C3, C4, CH50; TSH, thyroxine, T3 and T4
  • RASTS- negative for crab, lobster, fish garlic and insulin
cyclosporine related labs
Cyclosporine related labs
  • Jan 2004 reduced Hct-12.1 HB- 37.2
    • CsA: 37 ng/ml trough
  • March 2004 Normal studies
  • June 2004
    • CsA: 46 ng/ml trough
    • CBC, Mg, Cr, K normal
impression recommendations
Impression/ Recommendations
  • Severe CIU/angioedema h/o significant steroids requirements
    • No clear drug (insulin), food or systemic etiology
    • Avoiding NSAIDs
  • Consider alternate diagnoses:
    • Hx of autoimmunity with Type I DM
    • Rheumatologic?-joint symptoms, bruising
    • Obtain a skin Biopsy to verify urticaria vs. other
      • Consider immunofluorescence
follow up on recommendations
Follow-up on Recommendations
  • Rheum evaluation: Repeated ANA, RF, dsDNA, ANCA, Urine and SPEP- all normal
    • showed IgA of < 20, no other etiology for joints
  • October 04 -Csa 100 mg qd and fexofenadine with good control
  • Prednisone used only single day since 6/2004
  • No skin biopsy to date- attempted
  • Glucose under better control
cu in children association with thyroid autoimmunity
CU in children : association with thyroid autoimmunity
  • 187 CU pts (6- 18 yr) followed 7.5 yrs
  • Tests: CBC, sed, Chem, Antibodies to Hep B, HSV, EBV,CMV, mycoplasma, ASO, ANA, C3, C4, Thyroid function and antibodies,Ua, chest and sinus X-rays, food skin tests, ice cube test
  • Results: 8/187 antithyroid Ab (4.3%), all girls
    • 3x -1.27% rate seen in pediatric population
    • Much less than 14 to 33% range in adults
    • 5 +ANA, 4 + family Hx of autoimmunity

Levy, et al, Arch Dis Child 2003

cyclosporine in urticaria
Cyclosporine in Urticaria
  • CBC, Mg, K, renal function q 2 wks for first 3 months, CsA levels
  • Gingival hypertrophy
  • BP monitoring
  • Dose: 2-6 mg/kg/d similar to RA and psoriasis (2.5 mg/kg/day)
  • Tx dose 8 mg/kg/d; trough levels 100 ng/ml
csa and urticaria rdbct gratten br j dermatol 2000 143
CsA and Urticaria-RDBCTGratten Br J Dermatol 2000:143
  • 30 subjects, severe CIU unresponsive to H1 tx and positive ASST ( +HRA)
    • 4mg/kg CsA (n=20) or placebo (n=10) for 4 wks
    • All subjects followed for up to 20 wks, all on daily 20 mg cetirizine
  • Outcome: +< 25% of baseline UAS, relapse > 75% of UAS
  • Results: 8/19 + at week 4, 6 relapse wk 6
    • Noted reductions in HRA and ASST
csa in ciu adults
CsA in CIU:Adults
  • Open trial in 35 CIU with 3(0-3)
    • Low dose CsA 3 mos, 68% response (13/19) with few SEs1
  • DB trial :40 pts CsA 5 mg/kg x 8 wks, then 4 mg/kg x 8 wks vs. cetirizine 10 mg/d2
    • All cetirizine crossed to active CsA
    • 3 pts reduced CsA for Cr rise
    • On tx- 22 had relapse, 10 resolved spon 12 with H1
    • Off tx- 16/40 in remission at 9 mos

1Toubi,Allergy 1997; 2Di Giaccino Allergy Asthma Proc 2003

immunosuppression in adolescents cyclosporin
Immunosuppression in Adolescents: Cyclosporin
  • 80% of liver, kidney, cardiac Tx > 5 yr survivors on CsA
  • Nephrotoxicity: 4-5 % in cardiac and liver
    • 10% in RA dosed > 4 mg/kg avg 19 mos
  • HTN (20-30%)
  • Hyperlipedemia (10% of cardiac)
  • Post-tx lymphoproliferative disease:5-17%
  • Cosmetic-Gingival hyperplasia, hirsutism

Kelly, DA Pediatr Transplantation, 2002