Successful retrieval of a foreign body in an infant's right pulmonary artery using the new boomerang loop-snare technique: A case report.

We report successful percutaneous retrieval of a foreign body located in an infant's right pulmonary artery using the new boomerang loop-snare technique. The case was an 18-month-old girl. A central venous catheter for chemotherapy was inserted from the right subclavian vein during treatment for myelodysplastic syndrome at another hospital. A postprocedural chest X-ray showed a foreign body in her right lung, and contrast-enhanced computed tomography confirmed the linear foreign body was located in the right pulmonary artery. The patient was transferred to our hospital to retrieve the foreign body. Under cooperation with pediatric cardiologists, a 6 Fr sheath was inserted via the right femoral vein, and a guiding catheter was advanced into the right pulmonary artery. Owing to the risk of vascular injury when using endoscopic forceps, we decided to use the loop-snare technique. We successfully crossed over the foreign body using a steerable microcatheter and a long microguidewire. The microguidewire was reinserted into the guiding catheter, and a loop was created by grasping the end of the wire using a microsnare catheter, which was inserted coaxially within the guiding catheter. By pulling the microsnare catheter, we were able to pull the foreign body into the guiding catheter and successfully retrieved it. There were no complications, such as pulmonary artery injuries or thrombi. The recovered foreign body was a piece of a guidewire. The boomerang loop-snare technique using a small-diameter system is useful for the retrieval of a foreign body in infants.

Analysis of cytokine profiles in sera of single and multiple infantile hemangioma.

Infantile hemangiomas (IH) are benign vascular tumors that are common in infancy. They vary in growth, size, location, and depth, and although most lesions are relatively small, approximately one fifth of patients have multiple lesions. Risk factors for IH include female sex, low birth weight, multiple gestation, preterm birth, progesterone therapy, and family history, but the mechanism that causes multiple lesions is unclear. We hypothesized that blood cytokines are involved as a cause of multiple IHs, and tried to prove this using sera and membrane arrays from patients with single and multiple IHs. Serum samples were obtained from five patients with multiple lesions and four patients with a single lesion, none of which had received any treatment. Serum levels of 20 cytokines were measured using human angiogenesis antibody membrane array. Four of the 20 cytokines (bFGF, IFN-γ, IGF-I, and TGF-ß1) were higher in the patients with multiple lesions than in those with single lesion, with statistically significant difference (p < 0.05). Notably, signal for IFN-γ was evident in all cases with multiple IHs, but was absent in cases with single IH. Although not significant, there was mild correlation between IFN-γ and IGF-I (r = 0.64, p = 0.065), and between IGF-I and TGF-ß1 (r = 0.63, p = 0.066). bFGF levels were strongly and significantly correlated with the number of lesions (r = 0.88, p = 0.0020). In conclusion, blood cytokines could act as a cause of multiple IHs. This is a pilot study with a small cohort, so further large-scale studies are necessary.

Analysis of onset and clinical characteristics in Japanese patients with infantile hemangioma.

Infantile hemangioma (IH) is a common benign tumor during infancy, although the detailed mechanism behind it has not been fully elucidated. Based on previous studies, we hypothesized that formation of hemangioma might be triggered by secondary physiological events (perinatal hypoxia or mechanical stress during delivery) in patients carrying germline risk mutations. We aimed to clarify the mechanism by evaluating whether head and neck lesions were more frequent in patients in who IH appeared after birth compared with those in who it was present at birth. Clinical data of 62 lesions in 51 patients with IH were collected. All patients were analyzed for correlation of onset with gender, localization, family histories, gestational age, birth weight, and clinical subtypes. Distribution of lesions on the head and neck was slightly more frequent in the after-birth IH group, compared with those with IH present at birth, but without significant difference (47.6% vs. 40.0%, p = 0.32). On the other hand, the ratio of superficial and deep type IH at birth was significantly altered compared with that in IH after birth (19:0 vs. 26:7, p = 0.039). In addition, IHs appearing after birth tended to more commonly have multiple lesions than those with IH present at birth, with statistically significant difference (25.8% vs. 0%, p = 0.0164). There may therefore be different triggers for IHs at birth and IH after birth. Further studies with greater number of patients are necessary to validate these findings.

Change of serum cytokine profiles by propranolol treatment in patients with infantile hemangioma.

Infantile hemangioma is a benign cutaneous tumor, which sometimes rapidly enlarges, causes cosmetic problem, destroys normal tissue, and possibly threatens life. Dye lasers, steroid administration, and watchful waiting had been the treatment options for infantile hemangioma, but in recent years propranolol therapy has become available. The mechanism underlying the action of propranolol, however, is still unknown. We hypothesized that cytokines whose expressions change before and during the treatment are responsible for the efficacy of the drug. This study aims to prove the hypothesis using patients' sera and membrane array. In this study, the serum cytokine concentrations of five patients with infantile hemangioma were measured using membrane array of 20 angiogenic cytokines. We compared them before and during propranolol treatment to identify the cytokines responsible for the effect of propranolol. Signals for angiogenin, epidermal growth factor (EGF), platelet-derived growth factor-BB (PDGF-BB), regulated on activation, normal T-cell expressed and secreted chemokine (RANTES), tissue inhibitor of metalloproteinases 1 (TIMP-1), and tissue inhibitor of metalloproteinases 2 (TIMP-2) were evident in all five cases before treatment. Furthermore, PDGF-BB was the only cytokine of which concentration was decreased during treatment with statistically significant difference. This report is a pilot study with a small number of samples, and further detailed research with increased number of samples is necessary. Nonetheless, our results suggest that PDGF-BB may be involved in the action of propranolol. In addition, its serum concentration can be utilized as a potential marker of the therapeutic effect.

Distribution analysis of infantile hemangioma or capillary malformation on the head and face in Japanese patients.

Infantile hemangioma is one of the most common tumors in infancy. Delivery may be a clue for the trigger of infantile hemangioma formation in the head and face areas. In this study, we tried to plot localization of infantile hemangioma as well as capillary malformation on the head and face, and compared them to identify their characteristics and risk factors. The distribution of 104 lesions in 100 patients with infantile hemangioma was as follows: 32 lesions on the head, 12 on the forehead, 57 on the cheek and three in the jaw area. We could not find a statistically significant correlation of the distribution with three clinical subtypes (superficial, deep and mixed), sex or size of the lesions. However, the lesions in the jaw or chin areas were significantly less frequent than other areas (P = 0.0008 or 0.03, respectively). This tendency was not found in 40 patients with capillary malformation. Mechanical stress to jaw or chin areas may be less than other areas in normal cephalic delivery. Considering the emergence after birth and age-dependent involution of infantile hemangioma, we speculate that physiological events including perinatal hypoxia or mechanical stress during delivery as the trigger of hemangioma formation. Taken together, our results may reveal the contribution of mechanical stress to the trigger of infantile hemangioma, not capillary malformation, and may facilitate clinical differentiation between the two diseases by their localization. Further studies with an increased number of patients will be necessary to validate the finding.

Immunohistochemical expression of tumor necrosis factor-α in sepsis-induced lung injury.

Sepsis is asevere, systemic inflammatory disease caused by various kinds of microbes. In the present study, we immunohistochemically examined tumor necrosis factor (TNF)-α expression in sepsis-induced lung injury, and discuss its availability for the postmortem diagnosis of sepsis. Lung samples were obtained from different lung lobes of nine sepsis and eight control cases with postmortem intervals between 12 and 48 hours. Immunohistochemical analysis using anti-human TNF-α rabbit polyclonal antibodies was carried out. In sepsis and control groups, immunoreactivity for TNF-α was strongly detected in round-shaped mononuclear cells. The intensity of the immunohistochemical staining reaction was homogeneous in all lobes of the lungs examined. Furthermore, a double-color immunofluorescence analysis revealed that macrophages were a main cellular source of TNF-α in the lungs. To semiquantitatively evaluate the expression of TNF-α in the lungs, the ratios of the number of TNF-α-positive macrophages to total number of macrophages were calculated. Morphometrically, in lungs of the sepsis group, the ratio of TNF-α-positive macrophages was significantly higher, compared with the control group. TNF-α expression in the lungs can become a clue for the postmortem diagnosis of pulmonary inflammation, especially, TNF-α-positive ratios of 20% of more might suggest sepsis as the cause of death.