An Egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature
نویسندگان
چکیده
INTRODUCTION Erythromelalgia is a rare clinical syndrome characterized by episodic erythema, warmth and intense burning pain, which commonly involves the extremities. For those affected, this disorder may lead to significant long-term morbidity. Unfortunately, to date, no definitive therapy is available. This case report describes an Egyptian child with primary erythromelalgia that manifested at an early age and showed partial response to therapy with cetirizine hydrochloride. This anecdotal case report may have a diagnostic value for clinicians who have not seen this disorder. CASE PRESENTATION A 34-month-old previously healthy right-handed Hamitic boy without any significant past medical history presented at the age of 2 years with episodic bilateral pain in his feet. His mother reported associated warmth and erythema localized to his feet that never extended beyond his ankle joints. This pain is triggered by exertion and/or warm temperature exposure and is relieved by cooling measures. The diagnosis of erythromelalgia was made based on the patient's medical history and a thorough physical examination during the episodes. No evidence of local or systemic infection was present. Other causes for the symptoms were excluded by a negative extensive diagnostic work-up. Our patient did not respond to ibuprofen (15mg/kg/dose) three times a day but partial improvement with the oral non-sedating antihistaminic cetirizine hydrochloride (2.5mg/kg/once daily) was observed. When the child stopped cetirizine hydrochloride for 1 month as a test, the symptoms became aggravated and were relieved when cetirizine therapy was restarted. Cetirizine hydrochloride had not previously been reported to have this effect in children with erythromelalgia. CONCLUSIONS Erythromelalgia is a clinical syndrome of which the etiology, diagnosis and management are controversial. We describe a case of a 34-month-old Egyptian child with primary erythromelalgia that manifested at an early age. We believe that this is the first Egyptian case report of this kind in the literature. Partial response of this patient to cetirizine hydrochloride may grant us a new clue to understanding this mysterious condition.
منابع مشابه
Treatment of a Child with Refractory Acute Myeloid Leukemia with Humanized Anti-CD33 Monoclonal Antibody: A Case Report and Review of Drug Development
Background: The induction chemotherapy regimen for acute myeloid leukemia has evolved as once induction is completed patients progress through the consolidation phase and achieve remission in 76% of cases. For patients with relapsed or refractory disease, alternative chemotherapy agents are available. Monoclonal antibody therapy with biological agents, such as the immunotoxin gemtuzumab ozog...
متن کاملUltrasound Findings of Appendico-cecal invagination: A Case Report and Literature Review
Introduction Appendicocecal intussusception is an uncommon condition which must be differentiated from other types of invagination due to different therapeutic approaches and its ultrasound findings are outspread and imperfectly presented in literature. We present a cas...
متن کاملPyoderma Vegetans: A Case Report in a Child Suspected to Primary Immunodeficiency and Review of the Literature
Pyoderma vegetans (PV) is a rare inflammatory disorder characterized by vegetating pustules and plaques affecting the skin and mucosal membranes. It is believed that this entity is mostly associated with inflammatory bowel disease (IBD), chronic malnutrition, human immunodeficiency virus (HIV), malignancies, and other immunocompromised states. Pyoderma vegetans occurs more commonly in young and...
متن کاملGiant cell tumor of patella: A case report and review of literature
Objective: Patella is a sesamoid bone which develops in the quadriceps tendon. It is an uncommon site for neoplasms. The most common primary tumors which involve patella are benign. These include chondroblastoma, giant cell tumor (GCT) and aneurysmal bone cyst. Malignant lesions are less common in patella. These encompass metastasis, osteosarcoma and hemangioendothelioma. The most common compla...
متن کاملProptosis as Initial Presentation of Acute Lymphoblastic Leukemia in an Otherwise Healthy Child: A Case Report and Review of The Literature
Acute lymphocytic leukemia (ALL) is one of the frequent malignancies in pediatrics and involves bone marrow and extramedullary sites. Proptosis as extramedullary involvement of leukemia usually present in acute and chronic myeloid leukemia. It is extremely rare for ALL to present initially as proptosis.Here, a-21-month-old boy was presented with proptosis without any associated symptoms except ...
متن کامل