Drug Allergy Testing in a Pediatric Patient with Diffuse Cutaneous Mastocytosis

Year 2024, Volume: 8 Issue: 3, 363 - 367, 30.12.2024

Öner Özdemir , Talha Dursunoğlu

Abstract

Mastocytosis represents a group of diseases characterized by excessive accumulation of mast cells in one or more tissues. It can affect
only the skin or have systemic involvement. It has a low prevalence and the prognosis in children is benign. Patients with pediatric mastocytosis
often require sedation or anesthesia for diagnostic and therapeutic procedures. Here, we report our experience with anesthesia in
a pediatric patient with diffuse cutaneous mastocytosis undergoing surgical intervention for circumcision. The aim was to determine the risk
of anesthetic drugs to be used for general anesthesia in a patient with diffuse cutaneous mastocytosis. A 5-year-old male patient who was
consulted with anesthesia for circumcision operation and diagnosed with diffuse cutaneous mastocytosis by skin biopsy was not operated
because of the risk of drug allergy and referred to the pediatric allergy and immunology department. Routine hemogram and biochemistry laboratory results were within normal limits. Occasional elevation of serum tryptase level was observed. Skin prick test with lidocaine did not
show any significant change compared to the negative control due to dermatographism. However, intradermal tests with lidocaine 1:1 and
1:10 dilutions were negative. For drug allergy provocation test, increasing doses of lidocaine were administered subcutaneously and no reaction
developed. Intradermal tests with midazolam and bupivacaine were also negative. There was no early or late reaction to any of these.
In conclusion, it has been presented in the literature which drugs can be used without any problem according to intradermal test results as in
our case. However, all precautions should be taken against the possibility of anaphylaxis in these patients. Re-consultation 24 hours before
surgery regarding the use of prophylactic drugs is appropriate

Keywords

Mastocytosis, anesthesia, drug, allergy

Project Number

Yok

Yaygın Kutanöz Mastositozlu Çocuk Hastada İlaç Alerjisi Testi

Abstract

Mastositoz, bir veya birden fazla dokuda aşırı mastosit birikimi ile karakterize bir grup hastalığı temsil eder. Sadece deriyi etkileyebileceği gibi
sistemik tutulumu da olabilir. Düşük bir prevalansa sahiptir ve çocuklarda prognoz iyi huyludur. Pediatrik mastositozlu hastalar sıklıkla tanısal
ve terapötik prosedürler için sedasyon veya anestezi gerektirir. Burada, sünnet için cerrahi girişim uygulanacak diffüz kutanöz mastositozlu
bir pediatrik hastadaki anestezi deneyimimizi sunmaktayız. Amaç, diffüz kutanöz mastositozlu bir hastada genel anestezi için kullanılacak
anestezik ilaçların riskini belirlemekti. Sünnet operasyonu için anestezi ile konsülte edilen, cilt biyopsisi ile diffüz kutanöz mastositoz tanısı
konulan 5 yaşındaki erkek hasta, ilaç alerjisi riski nedeniyle operasyona alınmayarak çocuk alerji ve immünoloji bölümüne yönlendirilmişti.
Rutin hemogram ve biyokimya laboratuvar sonuçları normal sınırlarda bulundu. Hastada zaman zaman serum triptaz düzeyinin yükseldiği
görüldü. Lidokain ile yapılan deri prick testi, dermatografizm oluşması nedeniyle negatif kontrole göre anlamlı bir değişiklik göstermedi.
Ancak lidokain 1:1 ve 1:10 dilüsyonları ile intradermal testler negatif bulundu. İlaç alerjisi provokasyon testi için artan dozda lidokain subkutan
uygulandı ve hastada herhangi bir reaksiyon gelişmedi. Midazolam ve bupivakain ile yapılan intradermal testler de negatifti. Bunlardan
hiçbirine erken veya geç reaksiyon oluşmadı. Sonuç olarak, olgumuzdaki gibi intradermal test sonuçlarına göre hangi ilaçların sorunsuz
kullanılabileceği literatürde sunulmuştur. Ancak bu hastalarda anafilaksi gelişme ihtimaline karşı tüm önlemler alınmalıdır. Ayrıca profilaktik
ilaçların kullanımı ile ilgili olarak ameliyattan 24 saat önce tekrar konsültasyon yapılması uygundur

Keywords

Mastositoz, anestezi, ilaç, alerji

Ethical Statement

Yok

Supporting Institution

Yok

Project Number

Yok

Thanks

Yok

References

• 1. Rydz A , Lange M , Ługowska-Umer H , Sikorska M , Nowicki RJ , Morales-Cabeza C , Alvarez-Twose I. Diffuse cutaneous mastocytosis: A current understanding of a rare disease. Int. J.Mol.Sci. 2024; 25: 1401.
• 2. Valent P, Akin C, Metcalfe DD. Mastocytosis: 2016 updated WHO classification and novel emerging treatment concepts. Blood. 2017; 16;129(11):1420-1427.
• 3. Arca E, Köse O, Taştan HB, Demiriz M, Gür AR. A case of diffuse cutaneous mastocytosis. Turkiye Klinikleri J Med Sci. 2004; 24(2):178-81.
• 4. Özdemir Ö, Savasan S. Cutaneous mastocytosis in childhood: An update from the literature. J Clin Pract Res. 2023; 45(4): 311-320.
• 5. Özdemir Ö. Çocukluk çağı mastositozunun tanı ve tedavisine güncel bakış. Klinik Tıp Pediatri Dergisi 2018; 10(2), 11-17.
• 6. Carter MC, Uzzaman A, Scott LM, Metcalfe DD, Quezado Z. Pediatric mastocytosis: routine anesthetic management for a complex disease. Anesth Analg. 2008;107(2):422-7.
• 7. Özdemir Ö. Çocukluk döneminde kutanöz mastositoz yönetimi: Literatürden güncelleme. Hitit Medical Journal 2024;6(1):84-90.
• 8. Tirel O, Chaumont A, Ecoffey C. [Circulatory arrest in the course of anesthesia for a child with mastocytosis] Ann Fr Anesth Reanim. 2001; 20:874–5.
• 9. Coleman MA, Liberthson RR, Crone RK, Levine FH. General anesthesia in a child with urticaria pigmentosa. Anesth Analg. 1980; 59:704–6.
• 10. Nelson LP, Savelli-Castillo I. Dental management of a pediatric patient with mastocytosis: a case report. Pediatr Dent. 2002; 24:343–6.
• 11. Brodier C, Guyot E, Palot M, David P, Rendoing J. [Anesthesia of a child with a cutaneous mastocytosis] Cah Anesthesiol. 1993; 41:77–9.
• 12. Castells M, Metcalfe DD, Escribano L. Diagnosis and treatment of cutaneous mastocytosis in children: practical recommendations. Am J Clin Dermatol. 2011;12(4):259-70.
• 13. Heide R, Beishuizen A, De Groot H, Den Hollander JC, Van Doormaal JJ, De Monchy JG, Pasmans SG, Van Gysel D, Oranje AP; Dutch National Mastocytosis Work Group. Mastocytosis in children: a protocol for management. Pediatr Dermatol. 2008;25(4):493-500.
• 14. Disli ZK, Karagöz AH, Öcal T, Basgül E. Anaesthetic approach to burned child with mastocytosis. Turk J Anaesth Reanim 2013; 41: 27-9.
• 15. James PD, Krafchik BR, Johnston AE. Cutaneous mastocytosis in children: anaesthetic considerations. Can J Anaesth. 1987; 34(5):522-4.
• 16. Dewachter P, Castells MC, Hepner DL, Mouton-Faivre C. Perioperative management of patients with mastocytosis. Anesthesiology. 2014;120(3):753-9.
• 17. Klein NJ, Misseldine S. Anesthetic considerations in pediatric mastocytosis: a review. J Anesth. 2013; 27(4):588-98.
• 18. Ahmad N, Evans P, Lloyd-Thomas AR. Anesthesia in children with mastocytosis--a case based review. Paediatr Anaesth. 2009; 19(2):97-107.
• 19. Mori F, Crisafulli G, Bianchi A, Bottau P, Caimmi S, Franceschini F, Liotti L, Paglialunga C, Saretta F, Caffarelli C. Drugs and Vaccines Hypersensitivity in Children with Mastocytosis. J Clin Med. 2022; 11(11):3153.
• 20. Lau S, Sprung J, Volcheck GW, Butterfield JH, Divekar RD, Weingarten TN. Perioperative management of mastocytosis. J Anesth. 2023; 37(5):741-748.