Delayed-Type Hypersensitivity to Cow’s Milk Protein in Melkersson-Rosenthal Syndrome: Coincidence or Pathogenetic Role?

Abstract

Background: Intolerance to cow’s milk protein is frequently seen in children and rarely in adults. Non-IgE-mediated hypersensitivity to cow’s milk protein has been suspected in children based on in vitro evidence. Food intolerance may play a pathogenetic role in some cases of Melkersson-Rosenthal syndrome, which is often of unknown origin. Objective: We describe an adult female patient who developed a Melkersson-Rosenthal syndrome and at the same time was found to have in vivo and in vitro evidence of a delayed-type hypersensitivity to cow’s milk protein. Methods: Allergic and immunologic examinations, including skin prick tests, patch tests, serology, lymphocyte transformation tests and biopsies were performed. Results: A positive patch test to alpha-lactalbumin, a positive lymphocyte transformation test to whole cow’s milk and an immunohistology with infiltration of CD4+ and CD8+ T cells were found. Conclusions: There is a possible pathogenetic relation between delayed-type hypersensitivity to cow’s milk protein and Melkersson-Rosenthal syndrome in the patient presented.

The granulomatous reaction pattern

“…601 The presenting complaint is usually swelling of the lips, but adjacent areas of the cheek are frequently involved. 608 In one case, it was worsened by the cocoa in drinking chocolate. 602 Facial nerve palsy, usually unilateral, occurs in 13-50% of cases.…”

Anesthetic management of patients with Melkersson Rosenthal syndrome

“…Its etiology consists of infectious agents, allergic reactions to foods and supplements, and immunological and genetic factors [2][3][4][5]. Allergic reactions may appear as urticaria and angioedema.…”

“…Melkersson Rosenthal syndrome (MRS) is a rare granulomatous infl ammation, of unknown etiology, characterized by relapsing facial paralysis, persistent or recurrent orofacial edema, and lingua plicata [1][2][3][4][5]. MRS occurs more often in females than in males, with a ratio of nearly 3 : 1 [6].…”

Orofacial granulomatosis: review on aetiology and pathogenesis

“…However, the aetiology and pathogenesis of OFG, as a condition that is restricted to the orofacial region, remain to be elucidated, although numerous aetiological agents have been implicated. The available literature in relation to aetiology of OFG is linked to food substances, food preservatives, dental materials and microbiological agents (11)(12)(13)(14)(15)(16)(17)(18)(19)(20)(21)(22)(23)(24)(25)(26). Pathogenesis of OFG is shown to be due to delayed type hypersensitivity with some patients showing evidence of genetic predisposition.…”