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Celecoxib as a Potential Treatment for Intractable Lymphatic Malformation.
Pediatrics 2019; 144(3)Ped

Abstract

Lymphatic malformation (LM) is a congenital disorder resulting from an abnormal development of lymphatic vessels. LM may result in problems of cosmesis and functional impairment, including airway compression. An 11-year-old girl was referred to our department with increasing dysphagia caused by a large left cervical LM with a long history of treatment. Because of the LM location, surgical resection was not an option, and various therapies, including use of picibanil, had proven ineffective. Celecoxib treatment (100 mg/day) was initiated for local pain management. Softening of the lesion was observed 2 weeks after treatment initiation, and the dose was increased to 200 mg/day with additional shrinking of the LM over the next 2 weeks. With parental consent, celecoxib was continued, with a 65% reduction in volume achieved at 6 months. The patient discontinued treatment at 12 months, and the LM volume increased. Control over the LM was achieved with resumption of celecoxib treatment. After 2 years of treatment, the LM persists, but the size of the malformation is significantly smaller. No adverse effects of celecoxib treatment were observed. The anti-cyclooxygenase-2 effect of celecoxib prevented lymphatic vessel growth through an inhibition of cyclooxygenase-2 activity in the conversion of prostaglandin to prostaglandin E2. In conclusion, celecoxib may be a promising therapeutic agent for LM management.

Authors+Show Affiliations

Departments of Pediatrics and.Departments of Pediatrics and okamoto@m2.kufm.kagoshima-u.ac.jp. Department of Pediatrics, Graduate School of Medical and Dental Sciences, Kagoshima University, Kagoshima, Japan.Departments of Pediatrics and. Department of Pediatrics, Graduate School of Medical and Dental Sciences, Kagoshima University, Kagoshima, Japan.Radiology, Ōshima Hospital, Amami, Japan; and.Departments of Pediatrics and.Departments of Pediatrics and.Department of Pediatrics, Graduate School of Medical and Dental Sciences, Kagoshima University, Kagoshima, Japan.

Pub Type(s)

Journal Article

Language

eng

PubMed ID

31462447

Citation

Imamura, Mari, et al. "Celecoxib as a Potential Treatment for Intractable Lymphatic Malformation." Pediatrics, vol. 144, no. 3, 2019.
Imamura M, Okamoto Y, Nishikawa T, et al. Celecoxib as a Potential Treatment for Intractable Lymphatic Malformation. Pediatrics. 2019;144(3).
Imamura, M., Okamoto, Y., Nishikawa, T., Yoneyama, T., Yamasaki, Y., Kawamura, J., & Kawano, Y. (2019). Celecoxib as a Potential Treatment for Intractable Lymphatic Malformation. Pediatrics, 144(3), doi:10.1542/peds.2019-0319.
Imamura M, et al. Celecoxib as a Potential Treatment for Intractable Lymphatic Malformation. Pediatrics. 2019;144(3) PubMed PMID: 31462447.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Celecoxib as a Potential Treatment for Intractable Lymphatic Malformation. AU - Imamura,Mari, AU - Okamoto,Yasuhiro, AU - Nishikawa,Takuro, AU - Yoneyama,Tomohide, AU - Yamasaki,Yuichi, AU - Kawamura,Junpei, AU - Kawano,Yoshifumi, PY - 2019/06/05/accepted PY - 2019/8/30/pubmed PY - 2019/8/30/medline PY - 2019/8/30/entrez JF - Pediatrics JO - Pediatrics VL - 144 IS - 3 N2 - Lymphatic malformation (LM) is a congenital disorder resulting from an abnormal development of lymphatic vessels. LM may result in problems of cosmesis and functional impairment, including airway compression. An 11-year-old girl was referred to our department with increasing dysphagia caused by a large left cervical LM with a long history of treatment. Because of the LM location, surgical resection was not an option, and various therapies, including use of picibanil, had proven ineffective. Celecoxib treatment (100 mg/day) was initiated for local pain management. Softening of the lesion was observed 2 weeks after treatment initiation, and the dose was increased to 200 mg/day with additional shrinking of the LM over the next 2 weeks. With parental consent, celecoxib was continued, with a 65% reduction in volume achieved at 6 months. The patient discontinued treatment at 12 months, and the LM volume increased. Control over the LM was achieved with resumption of celecoxib treatment. After 2 years of treatment, the LM persists, but the size of the malformation is significantly smaller. No adverse effects of celecoxib treatment were observed. The anti-cyclooxygenase-2 effect of celecoxib prevented lymphatic vessel growth through an inhibition of cyclooxygenase-2 activity in the conversion of prostaglandin to prostaglandin E2. In conclusion, celecoxib may be a promising therapeutic agent for LM management. SN - 1098-4275 UR - https://www.unboundmedicine.com/medline/citation/31462447/Celecoxib_as_a_Potential_Treatment_for_Intractable_Lymphatic_Malformation L2 - http://pediatrics.aappublications.org/cgi/pmidlookup?view=long&pmid=31462447 DB - PRIME DP - Unbound Medicine ER -