Perioral Edema Research Articles

Improving Safety in Chondrolaryngoplasty.

Improving Safety in Chondrolaryngoplasty.

THE EVALUATION AND MANAGMENT OF DOCK8 DEFICIENCY

Introduction We present a case involving a child with a complex medical history who presented with a one-day history of fever, facial swelling, and skin rash. Case Description Our patient is a 2-year-old male with history of eczema, failure to thrive, chronic lymphadenopathy, developmental delay, and food allergies who presented with a one-day history of fever, facial swelling, and eczema. Physical examination was notable for an ill appearing child with coarse facial features. He had perioral edema, skin lichenification, diffuse eczematous rash, and chronic lymphadenopathy. Labs were notable for leukocytosis and elevated inflammatory markers. Skin and conjunctival cultures were positive for Herpes Simplex Virus. Wound cultures were positive for methicillin-resistant Staphylococcus aureus. He was treated with acyclovir, vancomycin, cefoxitin, and mupirocin ointment. The lab results from hospitalization were obtained. The hyper-IgE panel revealed a homozygous mutation in the DOCK8 gene which is located within one of the regions of homozygosity identified on SNP microarray diagnosing him with DOCK8 deficiency. Discussion This case demonstrates that DOCK8 deficiency should be included in the differential diagnosis involving children with eczema, failure to thrive, and cutaneous infections. Our patient had coarse facial features, however, this is not a common feature of DOCK8 deficiency. The study, Cutaneous Manifestations of DOCK8 Deficiency Syndrome, evaluated the clinical features of Job Syndrome and DOCK8 deficiency and found that in DOCK8 deficiency, coarse facial features were not present. Our case report shows that the presence of coarse facial features should not necessarily exclude DOCK8 deficiency.

Essential mixed cryoglobulinemia manifesting as recurrent perioral edema: report of a case

Cryoglobulins are immunoglobulins that precipitate in vitro at temperatures <37°C but that redissolve if heated; they are broadly divided into 3 classes. Cryoglobulinemia has a plethora of clinical manifestations, but it typically presents a vasculitic syndrome, most notably a triad of purpura, arthralgia, and weakness secondary to cryoglobulin deposition in small or medium-sized blood vessels. The highly variable clinical presentation and inconsistent laboratory isolation of cryoglobulins mean that the diagnosis may be one of exclusion or of retrospective review. We describe a case of a 67-year-old woman whose principal symptom was recurrent perioral edema, which was characteristically induced by cold. Raised cryoglobulins combined with a positive autoantibody screen suggested mixed cryoglobulinemia secondary to Sjögren syndrome. However, in the subsequent 6 years, she has remained generally well, having developed no autoimmune, neoplastic, or other disease, suggesting the diagnosis is one of "essential" mixed cryoglobulinemia. No treatment other than symptomatic analgesia has been required.

Angioedema after Treatment with Injectable Poly-L-Lactic Acid (Sculptra)

Sir:FigurePoly-L-lactic acid, marketed as Sculptra (Sanofi-Aventis, Bridgewater, N.J.), is a commonly used dermal filler for treatment of lipoatrophy and facial aging. Complications of poly-L-lactic acid injection are rare, with the most common being temporary bruising and swelling and the formation of subcutaneous papules.1,2 We report the case of a 59-year-old woman who presented with depleted malar volume and facial aging and who desired management with dermal fillers. The patient's medical history was significant for hypertension for which she was taking lisinopril, and she denied any history of medication allergies or sensitivities. Local anesthesia was achieved with 1% lidocaine with 1:100,000 epinephrine and topical lidocaine/prilocaine. Poly-L-lactic acid was injected into the bilateral malar areas, marionette lines, nasolabial folds, and periorally. Cross-linked hyaluronic acid plus lidocaine was used for lip augmentation. The patient returned to the clinic approximately 2 hours later with significant edema of the lips and perioral area (Fig. 1). She was transported immediately to the hospital, where she was admitted and treated with intravenous corticosteroids, famotidine, and diphenhydramine. The swelling resolved slowly, and she was discharged to home after 24 hours. The patient returned to the clinic 1 week later with satisfactory resolution of her edema (Fig. 2). The patient later had revision hyaluronic acid dermal injections with no adverse effect.Fig. 1: The patient showed lip and perioral edema 2 hours after injection.Fig. 2: The patient is shown 1 week postoperatively, with satisfactory resolution of the edema.In this patient with severe angioedema following the use of dermal fillers, there are two types of angioedema that must be considered: allergic and angiotensin-converting enzyme inhibitor related. Allergic angioedema is considered a type I or immediate hypersensitivity reaction and occurs following immunoglobulin E–mediated release of histamine, leukotrienes, and prostaglandins. This leads to vasodilation and extravasation of fluid from the vasculature into the subcutaneous tissue, resulting in edema, often in the head and neck.3 Angioedema caused by angiotensin-converting enzyme inhibitor is thought to occur by means of an accumulation of bradykinin in predisposed individuals using an angiotensin-converting enzyme inhibitor, which increases vascular permeability, resulting in edema of the face, lips, upper airway, and gastrointestinal tract.3 This can occur hours to years after starting an angiotensin-converting enzyme inhibitor.3,4 Local trauma can also induce a bradykinin response, which may be synergistic with the angiotensin-converting enzyme inhibitor, and angiotensin-converting enzyme inhibitor angioedema has been reported to occur after intubation, dental procedures, and lip biting.5 Immediate hypersensitivity reactions to dermal fillers are uncommon. To our knowledge, dermal fillers have not been described to induce an angiotensin-converting enzyme inhibitor angioedema, although certainly one would expect bradykinin release after a dermal injection. In the case of our patient, the fact that she received subsequent injections of hyaluronic acid without any adverse effect suggests that the angioedema may have been an effect of the poly-L-lactic acid itself. Patients undergoing injections with poly-L-lactic acid should be advised of a possible allergic reaction and monitored appropriately. In addition, practitioners administrating dermal fillers to patients on angiotensin-converting enzyme inhibitor therapy should be aware of the fact that the local trauma from injection could potentially induce angiotensin-converting enzyme inhibitor angioedema. This also has implications for the injection of dermal fillers by marginally or unsupervised aestheticians or injectors, as severe adverse reactions such as this can be fatal if not managed appropriately. Elizabeth Guardiani, M.D. Department of Otolaryngology–Head and Neck Surgery Steven P. Davison, M.D. Department of Otolaryngology–Head and Neck Surgery, Department of Plastic Surgery, Georgetown University Hospital, Washington, D.C. DISCLOSURE The authors have no financial interests to disclose. PATIENT CONSENT The patient provided written consent for the use of her images.

Amlodipine-Induced Bilateral Upper Extremity Edema

To report a case of bilateral upper extremity edema associated with amlodipine use in a child. A previously well and normotensive 6-year-old girl presented with a generalized vasculitis of unknown origin and severe hypertension. Large vessels predominantly affecting the neck, chest, and abdomen were found to be involved, resulting in abnormal arterial circulation and significant blood pressure differences between the upper and lower extremities. Multiple antihypertensive agents were initially required to control blood pressure. She was stabilized and discharged on amlodipine 10 mg each evening, atenolol 50 mg/day, and warfarin. Three days later she was noted to have facial and bilateral upper extremity pitting edema. Laboratory and radiologic assessments for possible etiologies were negative. Discontinuation of amlodipine resulted in resolution of edema. As of June 2007, there had been no cases of bilateral upper extremity edema associated with amlodipine use reported in the English literature. Adverse effects of amlodipine, a widely used antihypertensive, have been well reported. These include flushing, headache, and peripheral edema. Lower limb edema is the most common, while periocular and perioral edema have occurred less frequently. Anasarca edema has been described only once in the English literature. According to the Naranjo probability scale, amlodipine was a probable cause of bilateral upper extremity edema in this child. Bilateral upper extremity edema has been associated with amlodipine use in a child with an abnormal arterial circulation. The edema resolved upon discontinuation of the drug.

Anasarca Edema with Amlodipine Treatment

To report a case of anasarca edema associated with amlodipine use. A 77-year-old woman with essential hypertension who had not been treated with any other drug was prescribed amlodipine 10 mg/day to control her blood pressure. She developed anasarca edema soon after amlodipine treatment was initiated. Laboratory test results for possible etiologies were negative. Discontinuation of amlodipine resulted in dramatic improvement. To our knowledge, as of February 3, 2005, there have been no other reports of amlodipine-related anasarca edema in the English literature, and only one case was described in the Japanese literature. Pretibial edema is the most common adverse effect of amlodipine. Periocular and perioral edema have occurred less frequently, but anasarca edema has not emerged as a problem. An objective causality assessment revealed amlodipine to be a probable cause of anasarca edema. In rare instances, amlodipine may cause generalized edema, which will resolve upon discontinuation of the drug.

Allergic reactions to isosulfan blue during sentinel node biopsy—a common event

Sentinel lymph node (SLN) dissection in the management of high-risk melanoma and other cancers, such as breast cancer, has recently increased in use. The procedure identifies an SLN by intradermal or intraparenchymal injection of an isosulfan blue dye, a radiocolloid, or both around the primary malignancy. At the time of selective SLN mapping, 3 to 5 mL of isosulfan blue was injected either intradermally or intraparenchymally around the primary malignancy. From October 1997 to May 2000, 267 patients underwent intraoperative lymphatic mapping with the use of both isosulfan 1% blue dye and radiocolloid injection. Five cases with adverse reactions to isosulfan blue were reviewed. We report 2 cases of anaphylaxis and 3 cases of "blue hives" after injection with isosulfan blue of 267 patients who had intraoperative lymphatic mapping by the procedure described above. The 2 patients with anaphylaxis experienced cardiovascular collapse, erythema, perioral edema, urticaria, and uvular edema. The blue hives in 3 patients resolved and transformed to blue patches during the course of the procedures. The incidence of allergic reactions in our series was 2.0%. As physicians expand the role of SLN mapping, they should consider the use of histamine blockers as prophylaxis and have emergency treatment readily available to treat the life- threatening complication of anaphylactic reaction.

Complicated emergent endovascular repair of a life-threatening bilateral internal jugular vein occlusion

A 62-year-old woman had painful facial swelling that progressed to extensive periorbital and perioral edema with loss of vision, hearing, and consciousness. Her past surgical history was significant for right radical neck dissection including internal jugular vein (IJV) resection, laryngectomy, partial esophagectomy, tracheoesophageal fistula repair, and tracheostomy for squamous cell carcinoma of the oropharynx. In addition, the patient had received radiation therapy to the neck. A venogram revealed occlusion of the left IJV. A guidewire from the femoral vein was passed through the occluded segment; however, attempts to introduce an angioplasty balloon failed. A percutaneous basilic vein approach allowed passage of a dilator sheath over a guidewire, thereby enabling Wallstent deployment across the IJV occlusion. A second Wallstent was inserted across a stenosis in the brachiocephalic vein; however, this second stent reoccluded the IJV. Surgical removal of the second Wallstent was required through a segmental claviculectomy and venotomy. Patency was restored in the IJV and the brachiocephalic vein with the return of baseline neurologic function. This case demonstrates a complicated emergent endovascular repair of a life-threatening IJV occlusion that required surgical salvage. (J Vasc Surg 2000;32:397-401.)

Anicteric Hepatitis And Polyarthritis

To the Editor.— The recent article by Stevens et al reported two patients with anicteric hepatitis presenting as polyarthritis (220:687, 1972). We would like to record a similar case we saw recently. Report of a Case.— A 29-year-old black woman was admitted to the hospital with rash, pruritus, and fever. She had been well until one day before admission when she noted perioral edema and diffuse arthritis associated with a vesiculopapular, erythematous rash over the knees and smaller joints of both hands. On the day of admission, she awoke with painful, tender metacarpal, phalangeal, and interphalangeal joints of both hands, shoulders, elbows, knees, ankles, and toes with obvious redness and warmth. Roentgenographic examination failed to reveal any abnormalities of the joints. The patient had received 2 units of whole blood and 2 units of plasma three months prior, during an admission for laparotomy for bowel obstruction. Results of a physical