En-bloc resection including SMV and duodenum in patient of locally advanced colon cancer.

Tumor could directly invade or is adherent to other organs, but superior mesentery vein (SMV) and duodenum invasion are very rare. A 62-year-old woman was diagnosed with abdominal pain for several months. Multiple erythematous brownish skin patches and palpable mass were found at epigastric area. Computed tomography imaging showed focal wall thickening at the transverse colon that invaded to the rectus muscle and anterior abdominal wall. On exploration, we identified tumor invaded or was adherent to the duodenum and superior mesenteric vein and performed en-bloc resection. After surgery, the patient received chemotherapy and was followed up without any recurrence for 16 months. Adhesion and invasion of tumor to surrounding organs can be unexpectedly found during surgery. In our case, we found duodenum and SMV invasion and achieved R0 resection by SMV and duodenum resection, which could improve the patient's prognosis.

Conservative Treatment in Two Patients With Spontaneous Hepatic Artery Dissection.

BACKGROUND: Spontaneous dissection of the hepatic artery is a rare finding for which there is no specific treatment algorithm. Even though endovascular treatment and surgery have been used to treat other visceral artery dissections, these approaches can be challenging in the case of hepatic artery dissection because it is difficult to access the hepatic artery. CASE SUMMARY: 2 patients with spontaneous dissection of the hepatic artery were admitted due to epigastric pain. Although the liver function test results were normal, and there were no complications such as bleeding, shock, or ischemia in other organs, a subsequent computed tomography scan directly revealed the spontaneous dissection of the hepatic artery in both patients. The patients were treated with anticoagulants without surgery. Because it is difficult to perform surgery or endovascular treatment on the hepatic artery, we focused on preventing the progression of hepatic artery dissection and the formation of a new thrombus in the dissection. CONCLUSION: Considering the risk of surgical treatment for the hepatic artery and the condition of the patients, medical treatment with anticoagulants may be considered as an initial treatment and provide more benefits than surgical treatment.

Huge adrenal schwannoma: a rare type of adrenal tumor, misconceived as adrenocortical carcinoma.

Schwannoma is a benign and uncommon neoplasm arising from the neural crest cells. The most common tumor sites are the skin and subcutaneous tissue of the head and neck. Retroperitoneal schwannomas account for 1-3% of all schwannomas and are predominantly female. In particular, adrenal schwannoma is very rare, and due to its large size at the time of diagnosis, adrenal schwannoma is frequently misdiagnosed as adrenocortical carcinoma or pheochromocytoma. Adrenal schwannoma is difficult to distinguish it from other adrenal diseases based on imaging findings alone. In this report, we introduce the case of a huge left adrenal schwannoma.

Bowel perforation surgery due to fecal impaction immediately after hernia mesh surgery (TAPP).

Inguinal hernia repair using prosthetic mesh is used as a standard treatment in most countries and considered superior to primary suture repair. Although prosthetic mesh has greatly reduced the risk of recurrence, the risk of mesh infection remains. A 71-year-old man was diagnosed with symptomatic bilateral inguinal hernias. He underwent successful laparoscopic transabdominal preperitoneal (TAPP) repair and was discharged the same day. After 3 days, he was diagnosed with small bowel perforation, and underwent emergency surgery. We found perforation of the distal ileum caused by the fecal impaction and severe intra-abdominal contamination. We performed subtotal colectomy and ileosigmoid anastomosis, but did not remove the prosthetic mesh because the previous TAPP site was intact. The patient recovered well post-operatively. Therefore, contaminated or dirty surgery immediately after the hernia mesh surgery could be a feasible treatment.

Initial experience with laparoscopic posterior retroperitoneal adrenalectomy in single tertiary center.

Purpose: Laparoscopic posterior retroperitoneal adrenalectomy (LPRA) is a surgical method that accesses the adrenal gland through the back. The aim of this study was to report initial experience of LPRA and evaluate possibilities for surgical application. Methods: From March 2018 to December 2019, a total of 30 consecutive patients diagnosed with adrenal tumor underwent surgical treatment at Pusan National University Hospital were enrolled. Clinicopathologic features and various peri- and postoperative parameters were analyzed by retrospective medical record review. The mean age of the patients was 48.20±13.66 years. Results: The mean body mass index (BMI) was 25.50±4.30 kg/m2. Primary hyperaldosteronism was the most frequently preoperative diagnosed disease (n=13, 43.4%), followed by adrenal incidentaloma (n=8, 26.6%), Cushing syndrome (n=5, 16.6%) and pheochromocytoma (n=4, 13.3%). The mean size of postoperative adrenal tumor was 2.72±1.76 cm. The mean operating time was 162±58.14 minutes. Among the 30 patients, 28 patients underwent total adrenalectomy (93.3%) and two patients underwent cortical sparing adrenalectomy (6.7%). When LPRA was performed for patients with BMI >23.16 kg/m2, the operating time was longer than the average (P=0.016). Conclusion: LPRA was suitable and safe for patients with benign adrenal tumors. BMI, retroperitoneal fat density and postoperative adrenal weight may be related to the operating time, so they should be considered when deciding on a surgical method for adrenalectomy.

Influence of Infrapopliteal Runoff Vessels on Primary Patency after Superficial Femoral Artery Angioplasty with Stenting in Patients with Claudication.

Purpose: The number of infrapopliteal runoff vessels seems to be one of the factors influencing arterial patency in patients who had undergone superficial femoral artery (SFA) angioplasty with stenting. However, the effectiveness of infrapopliteal runoff vessels in predicting patency during SFA angioplasty remains unclear. This study aimed to determine whether the number and quality of infrapopliteal runoff vessels affect the primary patency after SFA angioplasty with stenting in patients with claudication. Materials and Methods: This study reviewed a retrospective database of patients with claudication who underwent SFA angioplasty with stenting between March 2011 and December 2016. The preoperative computed tomography findings of all patients were reviewed to assess infrapopliteal runoff vessels. The Trans-Atlantic Inter-Society (TASC) II classification and modified Society for Vascular Surgery (SVS) runoff score were used for subsequent analysis. Kaplan-Meier survival curves were constructed, and Fisher's exact and chi-square tests were used for data analysis. Results: A total of 153 limbs of 122 patients (88.2% male, mean age: 69.1 years) underwent SFA angioplasty with stenting. The overall primary patency rates of TASC II A/B and C/D cases were 77.1% and 31.2%, respectively, at 36 months (P<0.001). The primary patency rates at 36 months using the modified SVS runoff scoring system were 64.6% and 49.8% for the good-to-compromised (≤9 points) and poor (≥10 points) runoff groups, respectively (P=0.011). Conclusion: The modified SVS runoff scoring system is effective in predicting primary patency after SFA angioplasty with stenting in patients treated for claudication.

Application of minimal invasive technique for thyroidectomy without remote access in locally advanced thyroid carcinoma with gross extra-thyroidal extension.

INTRODUCTION: Minimally invasive thyroidectomy (MIT) is technically less damaging tissue and is better cosmetic results with small neck scar, decreasing postoperative pain and neck discomfort. We present the experience of MIT without remote access with case of grossly extra-thyroidal extension positive papillary thyroid carcinoma. PRESENTATION OF CASE: A 44-year-old, female presented 1.6 cm irregular hypoechoic nodule at upper pole in Right thyroid gland and diagnosed to papillary thyroid carcinoma. The tumor was suspicious to have extra-thyroidal extension. We performed MIT without remote access for this patient with 2 cm cervical incision. Postoperative course was uneventful and patient was discharged on the 3th postoperative day. At present, she is taking 100mcg levothyroxine and is free of disease 3 years post-surgery. CONCLUSION: If experienced surgeons can get sufficient resection margin and control bleeding of superior thyroid artery, MIT without remote access can be feasible method for thyroid carcinoma with gross extra-thyroidal extension, especially, upper pole carcinoma.

Fournier's gangrene in a rectal cancer patient.

INTRODUCTION: Fournier's gangrene (FG) is caused by a variety of causes, but FG led by rectal cancer is rare. PRESENTATION OF CASE: A 62-year-old man presented with perineal pain for several days. Multiple black spots on the scrotum and perineum surrounded by erythema were found on a physical exam. Computed tomography showed diffuse air density with subcutaneous edema in the perineum, scrotum, anus, and left lower abdominal wall and showed 4.1 cm-size mass in anus. He was diagnosed with FG caused by rectal cancer. He underwent extensive debridement of the perineum, scrotum, medial buttocks, and diverting loop colostomy of transverse colon. After repeated debridement of some residual necrotic tissue, abdominal perineal resection was performed after 24 days after initial surgery. The patient received reconstruction surgery of the soft tissue defect and discharged on postoperative day 84. He is being followed up without any recurrence for 10 months. CONCLUSION: Prompt clinical diagnosis and urgent surgical management are crucial for patient's favorable outcome. The patient in our case study could be recovered by a combined modality therapy we provided.

Thyrotropin suppression therapy for papillary thyroid carcinoma with a huge recurred neck lymph node.

Differentiated thyroid cancer (DTC) originating from thyroid tissue is affected by thyrotropin (TSH). TSH suppression therapy is usually recommended after thyroidectomy in cases of DTC. A 57-year-old woman who harbored a very huge recurred lymph node underwent TSH suppression therapy because of the risk of surgical complications. After TSH suppression, the huge neck lymph node exhibited a response and decreased in size. She had been followed up for 144 months. TSH suppression therapy could be considered as an alternative treatment option in a recurred DTC patient with a high perioperative risk.

Mechanical intestinal obstruction following laparoscopic inguinal hernia repair in a patient with abdominal cocoon syndrome.

Abdominal cocoon syndrome (ACS) is a rare condition characterized by partial or complete encasement of small intestine by a thick fibro-collagenous membrane. A 65-year-old man presented to the surgical department with left inguinal. He underwent laparoscopic transabdominal preperitoneal inguinal hernia. When we inserted a trocar into the peritoneal cavity, the small intestine was injured and repaired immediately. We identified a fibrotic membrane covering the small intestine, which was found as ACS. Two weeks later after discharge, he presented to the emergency department with mechanical intestinal obstruction. Conservative treatment had no effect on the patient and membrane excision, adhesiolysis and small intestine resection with anastomosis were performed. Unfortunately, the patient was hospitalized for a long time with bowel leakage and discharged on postoperative day 48. The preoperative diagnosis is quite challenging and most cases are diagnosed intraoperatively. When finding the ACS during the operation, careful attention should be needed.

Oncologic outcomes in patients with 1-cm to 4-cm differentiated thyroid carcinoma according to extent of thyroidectomy.

BACKGROUND: Recent guidelines advocate unilateral thyroidectomy for low-risk 1-cm to 4-cm differentiated thyroid cancer (DTC). This study was designed to examine the association between the extent of thyroidectomy and oncologic outcomes in patients with 1-cm to 4-cm DTC. MATERIALS AND METHODS: From April 1978 to December 2011, 16 057 patients with DTC underwent thyroidectomy at Yonsei University Hospital. Among them, 5266 (32.8%) patients were classified as having 1-cm to 4-cm DTC and were enrolled in this study. Clinicopathologic features and prognostic results (disease-free survival [DFS] and disease-specific survival [DSS] rates) were analyzed by retrospective medical record review. The mean follow-up duration was 57.3 ± 58.1 months. RESULTS: Of tumor subtypes in the study group, papillary thyroid carcinoma was the most common (97.5%) and follicular thyroid carcinoma occurred at a rate of 2.5%. In this study, the mean tumor size was 1.84 ± 0.74 cm. Patients had extrathyroidal extension (69.3%), multiplicity (35.1%), bilaterality (26.4%), central lymph node metastasis (53.0%), and lateral neck node metastases (19.9%). Of the 5266 patients, 4292 (81.5%) underwent total thyroidectomy and 974 (18.5%) had lobectomies. Recurrence rates in the total thyroidectomy and lobectomy groups were 5.7% and 9.4%, respectively. The lobectomy group had lower DFS (P = .007) and higher DSS (P = .034) than the total thyroidectomy group. A multivariate analysis for DFS revealed that tumor size, N classification, and extent of thyroidectomy were independent risk factors. On multivariate analysis, independent risk factors for DSS were age, sex, tumor size, and M classifications. CONCLUSION: Although extent of thyroidectomy does not affect DSS, total thyroidectomy is beneficial for reducing recurrence in patients with 1-cm to 4-cm DTC. However, if such tumors have such low-risk features as being unifocal, intrathyroidal, and lymph node metastasis-negative, extent of thyroidectomy does not affect oncologic outcome and lobectomy may be sufficient.

Usefulness of dynamic risk stratification in pediatric patients with differentiated thyroid carcinoma.

PURPOSE: Recently, the American Thyroid Association (ATA) dynamic risk stratification (DRS) has been verified to be more valuable than the static anatomical staging system for predicting prognosis in patients with differentiated thyroid carcinoma (DTC). The purpose of this retrospective study was to compare the clinical usefulness of DRS, which is based on the response to initial treatment, with that of ATA initial risk stratification in pediatric patients. METHODS: A total of 144 pediatric patients underwent thyroid operation from August 1982 to December 2013 at Yonsei University Hospital (Seoul, Korea). Among them, 128 patients with complete clinical data were enrolled in this study. Clinicopathologic features and surgical outcomes were retrospectively analyzed by medical chart review. The mean follow-up duration was 11.5 years. RESULTS: The mean tumor size was 2.1 cm; 80.4% of patients were diagnosed with conventional papillary thyroid carcinoma, and 7.0% of patients were diagnosed with follicular thyroid carcinoma. Low-risk patients had the highest probability of an excellent response to initial treatment (66.6%). High-risk patients had the highest probability of a structural incomplete response (100%) and the lowest probability of an excellent response (11.1%). The ATA risk stratification and the DRS system were independent risk factors for disease-free survival (DFS) (P = 0.041 and P < 0.001, respectively). CONCLUSION: The DRS system, which is based on the response to initial treatment, can offer more useful prognostic information compared with ATA risk stratification in pediatric patients with DTC.

Long-term oncologic outcomes of papillary thyroid microcarcinoma according to the presence of clinically apparent lymph node metastasis: a large retrospective analysis of 5,348 patients.

PURPOSE: Active surveillance (AS) of low-risk papillary thyroid microcarcinoma (PTMC) may reduce the risk of overtreatment of clinically insignificant cancer. However, the absence of predictor for the progression of PTMC resulted in treatment delay and potentially compromising cure of aggressive disease. Therefore, to anticipate potential damage of delayed surgery, we investigated the oncologic outcomes of patients with low-risk PTMC initially eligible for AS except clinically apparent lymph node metastasis (LNM), imitating delayed surgery with neck dissection. MATERIALS AND METHODS: A total of 5,348 patients, enrolled between 1987 and 2016, with low-risk PTMC initially eligible for AS were included regardless of LNM. We classified our study patients into two groups: Group I, lobectomy with prophylactic central cervical node dissection; Group II, total thyroidectomy with modified radical neck dissection for LNM. In addition, we investigated the oncological outcomes of patients with second-wave surgery due to lateral lymph node recurrence (Group III, subgroup of Group I). RESULTS: Group I showed more favorable clinicopathological characteristics compared with Group II. In Group I, only 29 (0.58%) of 4,927 patients underwent second-wave surgery with neck dissection for lateral lymph node recurrences, whereas in Group II, all 22 (5.23%) of 421 patients underwent second-wave selective node dissection because of nodal recurrence. Disease-free survival rates were significantly different between Groups I and II (P<0.05). Of note, the recurrence rate of Group II was still significantly higher than that of Group III (5.2% vs 0%, respectively; P=0.021). In addition, Kaplan-Mayer survival analysis indicated poor disease-free survival rates in Group II compared with Group III (P<0.05). CONCLUSION: The long-term treatment outcome of PTMC without LNM was favorable even if the recurrence occurs during follow-up period compared with that of PTMC with LNM. It should be noted that AS might be able to cause poor prognosis due to clinically apparent LNM.

Dynamic risk stratification in medullary thyroid carcinoma: Single institution experiences.

Recently, dynamic risk stratification has been found to be more valuable than static anatomic staging system in nonmedullary thyroid cancer and this strategy has also been accepted in medullary thyroid cancer (MTC). The present study was designed to compare the clinical usefulness of response to initial therapy stratification with a traditional anatomic staging system.From August 1982 to December 2012, a total of 144 MTC patients underwent thyroidectomy in Yonsei University Hospital. Among them, 117 (82.2%) patients with complete clinical data and sustained follow-up were enrolled in this study. Clinicopathological features and surgical outcomes were analyzed by retrospective medical chart review. Mean follow-up duration was 85.78 ±â€Š62.51 months.In this study, mean tumor size was 1.94 ±â€Š1.40 cm and 22 (18.9%) patients had hereditary MTC; 95 (81.1%) patients had sporadic MTC. Stage I patients had highest probability of excellent response to initial therapy (92.1%). Stage IV patients had highest probability of biochemical and structural incomplete response to initial therapy (57.5% and 30.3%) and lowest probability of excellent response to initial therapy (12.1%). Both response to initial therapy stratification and TNM staging system offered useful prognostic information in this study. The TNM staging system provided risk stratification pertaining to disease-free survival (DFS), disease-specific survival (DSS), and the probability of having no evidence of disease at final outcome, but did not provide risk stratification pertaining to the probability of having biochemical persistent/recurrence disease at final outcome. However, response to initial therapy stratification provided risk stratification pertaining to not only DFS, DSS, and the probability of having no evidence of disease at final outcome but also the probability of having biochemical persistent/recurrence disease at final outcome.In this study, we demonstrated that dynamic risk stratification with adjusted response to initial therapy system can offer more useful prognostic information than anatomic staging system in MTC.

Yonsei Experience of 5000 Gasless Transaxillary Robotic Thyroidectomies.

BACKGROUND: Since the use of robot systems in thyroid surgery was introduced in 2007, we have advanced a novel method of robotic thyroidectomy (RT) using a gasless transaxillary approach (TAA). We report our experience with this technique and detail the surgical outcome of 5000 robotic thyroidectomies. METHODS: From October 2007 to May 2016, we successfully performed 5000 robotic thyroidectomies using a gasless TAA at the Department of Surgery, Yonsei University Health System. The medical records of the patients are reviewed retrospectively, and the details of clinicopathologic characteristics, operation times, perioperative complications, and oncologic outcomes are analyzed. RESULTS: The 5000 patients with thyroid tumor (4804 with cancer and 196 with benign tumor) underwent RT using a gasless TAA. Mean operation time was 134.5 ± 122.0 min. The most common histologic subtype of thyroid cancer was papillary (98%), and the mean tumor size was 8.0 ± 6.0 mm. Stage I was found in 85.4% patients regarding tumor nodes metastasis staging. The 196 benign tumors consisted of 104 adenomatous hyperplasias (53.0%), 43 follicular adenomas (21.9%), 30 Graves' diseases (15.3%), and 19 others (9.7%). Postoperative complication occurred in 24.1% without any serious one, and overall morbidity tended to decrease over time. No disease-specific mortality was observed during the follow-up period. Locoregional recurrence was developed in 26 patients (0.5%). CONCLUSION: The authors have tried to improve RT technique using gasless TAA and achieved acceptable surgical outcomes. The rapid evolution of surgical robot technology and our constant effort to advance RT technique using gasless TAA would make it possible to reduce the perioperative morbidity and gain the best possible operative and oncologic outcomes.

Transaxillary robotic modified radical neck dissection: a 5-year assessment of operative and oncologic outcomes.

BACKGROUND: Robotic modified radical neck dissection (MRND) using a gasless transaxillary approach has been reported to be a safe and meticulous technique in patients with papillary thyroid carcinoma (PTC) and lateral neck node metastasis (N1b). Few studies, however, have attempted to assess the long-term oncologic outcomes of robotic MRND in these patients. This study aimed to compare perioperative and 5-year oncologic outcomes of robotic MRND with conventional open procedures in patients with N1b PTC. METHODS: Between September 2007 and February 2010, 193 patients with N1b PTC underwent total thyroidectomy and MRND by a single surgeon. Of these, 42 (21.8 %) underwent robotic procedures and 151 (78.2 %) underwent conventional open procedures. All patients received 3.7- to 5.5-GBq radioactive iodine (RAI) ablation, post-therapy whole-body scans (TxWBSs), and diagnostic WBS (DxWBSs) during follow-up. An exact 1:3 matching for age and stage was performed to minimize selection bias, and perioperative and 5-year oncologic outcomes were compared in the matched groups. RESULTS: The mean follow-up period was 66.0 months (range 60-90 months). Number of retrieved cervical lymph nodes (LNs) (p = .102) and postoperative ablation success rates (p = .864) were similar between the two groups. TSH-suppressed serum Tg concentrations after 5 years (0.7 ± 1.5 vs. 2.4 ± 14.1 ng/ml; p = .471) and recurrence rates in the robotic and open groups (1/41 [2.4 %] vs. 3/102 [2.9 %]; p = .864) were similar for the 5-year follow-up period. Four patients experienced recurrence: Three exhibited regional lymph node metastasis, and one showed bilateral lung metastases. CONCLUSION: The perioperative and 5-year oncologic outcomes were similar after robotic and conventional open MRND. Large, prospective randomized controlled trials with long-term follow-up data are needed to validate these results.

Optimal Cut-Off Values of Lymph Node Ratio Predicting Recurrence in Papillary Thyroid Cancer.

Regional lymph node (LN) metastasis has a significant impact for prediction of recurrence in patients with papillary thyroid cancers (PTC); however, the prognostic value of the lymph node ratio (LNR), which is defined as the ratio of the number of metastatic LNs to the total number of investigated LNs, is controversial. In this study, we determined the optimal cut-off values of LNRs for the prediction of recurrence in PTC patients.This large cohort study retrospectively evaluated 2294 patients who had undergone total thyroidectomy for PTC at a single institution from October 1985 to June 2009. The prediction probability of central LNR (cLNR, level VI) and total LNR (tLNR, levels II-VI) were estimated by binominal logistic regression analysis. Hazard ratios of the cut-off LNR values for cancer recurrence were calculated for relevant covariates using multivariate Cox regression analyses. Kaplan-Meier analyses were also utilized to assess the effects of estimated LNR cut-off values on recurrence-free survival (RFS).Of the 2294 patients, 138 (6.0%) presented cancer recurrence during the follow-up period (median duration = 107.1 months). The prediction probability indicated that LNRs of 0.4 and 0.5 for central LN and total LN, respectively, are optimal cut-off values for precise prediction with minimization of outliers. Multivariate Cox regression analyses revealed that cLNR ≥0.4 was independently predictive of recurrence in patients with N0 and N1a PTCs (hazard ratio [HR]: 7.016, 95% confidence interval [CI]: 3.72-12.986, P < 0.001) and that tLNR ≥0.5 indicated a significantly increased risk of recurrence in patients with N1b PTCs (HR: 2.372, 95% CI: 1.458-3.860, P < 0.001). In addition, Kaplan-Meier analyses clearly demonstrated that these LNR cut-off values are precisely operational in RFS estimation.The cut-off LNR values of 0.4 and 0.5 for cLNR and tLNR, respectively, were identified. Risk stratification combined with these LNR cut-off values may prove useful to determine treatment and follow-up strategies for PTC patients.

Long-term oncologic outcome of robotic versus open total thyroidectomy in PTC: a case-matched retrospective study.

PURPOSE: The role of the robot in thyroid surgery remains uncertain, and it is unclear whether robotic total thyroidectomy (R-TT) can be justified as a standard treatment for patients with thyroid cancer. This study compared the long-term operative results and oncologic outcomes of R-TT and conventional open TT (O-TT) after propensity score matching of the cohorts. METHODS: This study retrospectively evaluated patients with papillary thyroid cancer (PTC) who underwent TT with central compartment node dissection (CCND) by a single surgeon in tertiary medical center. Of the 833 patients, 94 (11.3 %) were lost to follow-up. 245 (33.2 %) underwent R-TT, and 494 (66.8 %) underwent O-TT. The mean follow-up duration was 74 (range 61-91) months. Propensity score matching in age, gender, tumor size, extrathyroidal invasion, multiplicity, bilaterality, and TNM stage identified 206 pairs of patients. The long-term oncologic outcomes were assessed in the R-TT and O-TT groups before and after adjustment for baseline covariates. RESULTS: After adjustment for baseline covariates, serum thyroglobulin (Tg) (p = 0.746) and anti-thyroglobulin antibody (TgAb) (p = 0.394) concentrations were similar in the two groups 5 years after surgery. Nine patients experienced locoregional recurrence, six in the O-TT and three in the R-TT group, with all recurrences in regional LNs. Disease-free survival (DFS) was similar in the R-TT and O-TT groups before matching (p = 0.890) and after adjustment for baseline covariates (p = 0.882). CONCLUSION: This represents the first report of 5-year surgical outcomes in patients who underwent R-TT for thyroid cancer. Long-term oncologic quality was similar after R-TT and O-TT.

Intralesional injection of OK-432 for vision-threatening orbital lymphangioma.

BACKGROUND: Surgical excision of orbital lymphangiomas is difficult, and almost always incomplete due to the diffusely infiltrative pattern of these tumors. The present report describes the successful use of intralesional OK-432 administration to treat two patients with intractable hemorrhagic proptosis due to orbital lymphangiomas. METHODS: A 3-year-old girl (case 1) presented with aggressive proptosis and ptosis, and a 1-year-old boy (case 2) presented with massive proptosis and exposure keratopathy, associated with recurrent intracystic bleeding of an orbital lymphangioma. In case 1, 0.02 mg OK-432 was intracystically injected in a volume of 2 ml of physiologic saline. Due to a lack of therapeutic response, a second injection of 0.05 mg OK-432 in 1 ml was administered. In case 2, a single dose of 0.05 mg OK-432 in 1 ml was administered. RESULTS: In both cases, intracystic administration of 0.05 mg of OK-432 in a 1-ml volume resulted in a successful outcome. The adverse effects were minor (mild transient fever and lid swelling), and rebleeding and intraocular pressure elevation did not occur. Proptosis and eyelid swelling gradually improved over 1 month, and completely resolved within 3 months of treatment. CONCLUSIONS: Intralesional administration of 0.05 mg/ml OK-432 (delivered in 1 ml) resulted in the successful treatment of two cases of orbital lymphangioma. Although this drug concentration is higher than in previous reports, there were no major adverse effects.