Treatment Of Lymphatic Malformations Research Articles

Diagnosis and treatment of lymphatic malformations

More than seventy percent of lymphatic malformations (LMs) occur in head and neck region. The management of head and neck LMs is challenging,especially for diffuse and infiltrative cases.It can cause deformity and death in severe patients. Surgical resection is the main method to treat LMs in the past and sclerotherapy has also greatly improved the treatment out come of LMs over the past 20 years. However, it is still hardly to achieve satisfactory prognosis for the patients with extensively infiltrative lesions, regardless of surgical resection or sclerotherapy. In the past five years, some scholars have made a preliminary exploration of new treatment methods, such as oral medicine, to diversify the treatment options of head and neck LMs. This article will review the general situation, biological properties, clinical characteristics, diagnostic methods, and current treatment strategies for LMs as well as trends in management of LMs.

Clinical analysis of surgical treatment for head and neck lymphatic malformations in children: a series of 128 cases

Background: Lymphatic malformations (LMs) are caused due to abnormal lymphatic development, and mainly occur in neonates or young children. At present, the role of surgery in the treatment of head and neck LMs is still controversial, focusing mainly on surgical efficacy and indications.Objective: This study aimed to explore the effect and influential factors of surgical treatment in children with head and neck LMs, hoping to provide a basis for rational selection of surgical indications.Methods: This retrospective study enrolled 128 children with head and neck LMs and underwent surgical treatment in Beijing Children’s Hospital from May 2007 to June 2016. They were classified into three morphological groups: macrocystic, microcystic, and mixed. Based on de Serres staging, they were divided into five groups: stage I to V. The local lesion control rate, complication rate, and recurrence rate were summarized and analyzed.Results: The rate of completely controlled and almost completely controlled in cases with head and neck LMs was 71.1%. The postoperative complication rate was 13.3%, and the postoperative recurrence rate was 11.9%. Statistically significant difference was found for local lesion control and postoperative recurrence rates between different morphological and clinical staging groups. Furthermore, the complication rate showed a significant difference between different morphological groups, but not between clinical staging groups.Conclusions: Surgical resection in children with macrocystic, low-stage, or neck-limited LMs demonstrated better therapeutic effect, with fewer complications. However, the effect remained poor and had more complications for microcystic, diffused and high-stage patients. High stage and incomplete resection are considered as the main factors for postoperative recurrence. Current staging system for LMs has important predictive value in the prognosis of head and neck LMs. For LMs in posterior pharyngeal space, plasma ablation has certain advantages.

PIK3CA mutations in vascular malformations.

Recently, it has been discovered that a subset of vascular malformations, of the lymphatic and venous type, are caused by oncogenic mutations in the PIK3CA gene. Now, efforts have been focused in the understanding of the molecular and cellular consequences of these mutations and the opportunities for novel-targeted therapies for these diseases. Here, we review the latest findings in the biology of oncogenic PIK3CA mutations in the pathogenesis of vascular malformations. We focus on the recent development of in-vitro and in-vivo tools for the study of PIK3CA-mutant vascular malformations with special interest in preclinical models for drug testing. Also, we review the latest advances in phosphoinositide 3-kinase (PI3K) inhibitors in the clinic and their repurposing for the treatment of lymphatic malformations and venous malformations. Oncogenic mutations on PIK3CA causing lymphatic malformations and venous malformations are also frequently found in epithelial cancer. Thus, fundamental research done in the cancer field during the past decades might be applied to the understanding of lymphatic malformations and venous malformations. Likewise, repurposing PI3K pathway inhibitors that are currently in cancer clinical trials can be used as a novel strategy for the treatment of these diseases. Here, we also open a debate for the consideration of lymphatic malformations and venous malformations as developmental tumours.

03:27 PM Abstract No. 396 Clinical response to image-guided sclerotherapy for treatment of lymphatic malformations in children

03:27 PM Abstract No. 396 Clinical response to image-guided sclerotherapy for treatment of lymphatic malformations in children

Use of Oral Sildenafil in Treatment of Pediatric Head and Neck Lymphatic Malformations

Background: Lymphatic malformation (LMs) is a low flow vascular malformation, with a morbidity rate of 1/2000 in neonates. Most of the LMs were found in the head and neck. Compared with the macrocrystic ones, the microcrystic LMs show stronger invasiveness and wider involved range, which leads to higher treatment difficulty and recurrence rate as well as a serious impact on patients´ health and quality of life. Aim: To evaluate the efficacy and safety of oral sildenafil in treatment of pediatric head and neck LMs. Methods: In total, 21 children diagnosed with LMs were given oral sildenafil and stopped at week 24. Therapeutic outcome was evaluated by comparing lesions' volume change with baseline. In addition, symptoms improvement and adverse effects were also recorded during the follow-up period. Results: Eight patients (38.0%) showed excellent improvement (>75%-100%), 1 patient (4.8%) had good (>50%-75%), 2 patients (9.6%) had fair (>25%-50%), 3 patients (14.3%) had minimal (0%-25%), and 7 patients (33.3%) had no improvement. The snoring or stridor completely disappeared in 2 subjects, although there was no apparent decrease in LMs size. Moreover, all 3 patients with macrocystic lesions displayed a positive response to sildenafil treatment, followed by mixed type (9/12, 75%). Only 2 out of 6 patients with microcystic lesions had minimal clearance. Only 4 patients showed transient mild diarrhea, there was no other adverse effect in this series. Conclusion: Our study showed that low-dose oral sildenafil citrate tablets are safe and effective to decrease the lesions volume in some LMs children, especially with macrocystic lesion or mixed type.

Treatment of Lymphatic Malformations with the mTOR Inhibitor Sirolimus: A Systematic Review.

Extensive lymphatic malformations are low-flow vascular malformations that can cause devastating complications. Treatment of these malformations is challenging. This systematic review presents current use of sirolimus in patients with extensive lymphatic malformations. MEDLINE and Google scholar search was conducted for studies on sirolimus treatment of lymphatic malformations up to July 2017. Search items included "lymphatic malformation," "lymphangioma," "cystic hygroma," "vascular malformation," "low-flow malformation," "sirolimus," "rapamycin," and "mTOR inhibitor." Twenty studies, including 71 patients receiving sirolimus, were included into this review. Forty-five patients had lymphatic malformations, eight patients venolymphatic malformations, and 19 patients capillary-lymphatico-venous malformations. Sirolimus led to a partial remission of disease in 60 patients, three patients had a progressive disease, and the outcome of eight patients was not reported. Dosing, target trough level, and duration of treatment differed between the studies. Common adverse effects were hyperlipidemia and neutropenia. Available literature indicated that sirolimus therapy might be effective for lymphatic malformations. However, further randomized controlled studies are required to analyze the efficacy and long-term adverse events and to clarify the potential role for sirolimus in the management of lymphatic malformations.

Use of percutaneous bleomycin sclerotherapy for orbital lymphatic malformations

ABSTRACTPurpose: Bleomycin sclerotherapy has been shown to be a viable treatment for lymphatic malformations. However, its use for these lesions confined to the orbit is becoming increasingly documented in the literature. In this study, we summarize the clinical manifestations and outcomes observed following percutaneous bleomycin sclerotherapy for orbital lymphatic malformation.Methods: A 5-year retrospective chart review of patients with clinical, radiographic, and/or biopsy-confirmed diagnoses of orbital lymphatic malformation that received bleomycin sclerotherapy was conducted at the Emory Hospital and Clinics. Data examined included patient demographics, patient history and symptoms, clinical findings, radiographic findings, route of bleomycin delivery, and outcome.Results: Of the 10 patients who met inclusion criteria, the median age of treatment was 7 years. The most common presenting symptoms included vision change and proptosis. Nine of 10 patients demonstrated macrocysts (>1 cm) on imaging. Seven of 10 patients had histories of prior interventions including resections, cyst drainage, and debulking. Because 2 of these 10 patients were lost to follow-up, 8 patients remained for post-procedural evaluation. Four of these eight showed improvement of visual acuity after post-bleomycin sclerotherapy. In seven of eight patients, extraocular motility either improved or remained stable. Pretreatment and posttreatment exophthalmometer measurements obtained in four patients revealed an average improvement in proptosis of 65% from their average pretreatment measurements.Conclusions: Our findings suggest that percutaneous bleomycin sclerotherapy is a viable option for treatment of orbital lymphatic malformations, with potentially greater benefit to those with macrocystic features.

Percutaneous treatment of orofacial vascular malformations

The aim of this study was to evaluate the efficacy of fluoroscopy-guided percutaneous injection of bleomycin as the primary treatment for low-flow vascular malformations. A total of 34 patients (mean (range) age 24 (8–51) years) with orofacial vascular lesions were treated in the Department of Interventional Radiology and Maxillofacial Surgery. There were 20 low-flow venous malformations, 11 lymphatic malformations, and three of mixed type. All patients were treated by fluoroscopy-guided percutaneous injection of a mixture of bleomycin (mean (range) 15 (5–15)mg) and a radio-opaque agent (Ultravist® (iopromide), Bayer)/session. The number of sessions ranged from one to six. The clinical response was complete in 21 patients, obvious in nine, and of clinical benefit in four. Patients were reviewed within the first week, third week, and at three-month periods until 24 months. There were no serious complications such as pulmonary fibrosis. Fluoroscopy-guided intralesional injection of bleomycin should be considered as the first-line treatment for lymphatic malformations because it is effective and reliable with few complications.

CHOICE OF TREATMENT OPTIONS FOR CYSTIC LYMPHSTIC MALFORMATIONS IN THE HEAD AND NECK REGION: TREATMENT EXPERIENCE OF 81 CHILDREN

Background. Surgery has previously been the only treatment for lymphatic malformations (LMs), but in the head and neck region is challenging due to the risk of scarring, nerve damage, recurrence. Sclerotherapy may be a perfect alternative.Objective. The aim of the study is to determine the efficacy and safety of the OK-432 sclerotherapy in the children with craniofacial LMs.Methods. 81 children with head and neck LM between December 2010 and March 2017were involved into the study. The follow-up period was from 6 to 79 months. According to the size of cysts, LMs were classified into macrocystic, microcystic, and mixed. The result of the treatment of LMs was determined by the percentage of reduction in size as excellent (decrease by more than 90%), good (by 50%-89%), satisfactory (by 20%-49%) and none (by less than 20%).Results. The macrocystic LMs diagnosed in 41.97% of patients, microcystic - in 12.35%, and mixed – in 45.68% of children. OK-432 sclerotherapy only was performed for 83.9% of patients and in 12.3% in combination with surgery. The range of sclerotherapy sessions was from 1 to 11. An excellent result in 96.97% of cases was evidenced in the patients with macrocystic LM. Poor result was proved in the patients with microcystic LMs; the most of them (55.56%) had satisfactory result. In the patients with mixed LM, an excellent and good result was evidenced in 83.33%. After 198 sessions of OK-432 sclerotherapy, complications associated with the treatment occurred in 5 (2.52%) cases.Conclusions. OK-432 sclerotherapy is a safe and effective treatment of head and neck LMs in children. Macrocystic LMs proved the best response to OK-432 treatment.

Long-Term Follow-Up of Lymphatic Malformations in Children Treated with Sildenafil.

Lymphatic malformations (LMs) are challenging to treat. Reports on the benefits of sildenafil for LM management have been mixed. This study evaluated long-term clinical outcomes of pediatric LM patients after sildenafil treatment. A retrospective chart review was performed on pediatric LM patients treated with sildenafil in the past 5years. Patients were also contacted to complete a survey of comprehensive questions about their LM after sildenafil and subsequent interventions. Of 12 patients identified, 10 (83.3%) participated in the follow-up survey. The average age was 8years (range 4-16yrs), median treatment duration was 9months, and the average time of follow-up after sildenafil was 4years; one patient is still taking sildenafil. Ten patients surveyed (83.3%) reported positive therapeutic response, with improvement in the size and compressibility of their LM during posttreatment clinical visits. Six received additional interventions (four sirolimus, one sclerotherapy, one surgery) after sildenafil was discontinued, with all but one reporting a positive response to subsequent interventions. Minor side effects at the time of sildenafil treatment included mild flushing, dizziness, and transient nausea, but no adverse effects were reported at the long-term follow-up. This is the first report of long-term follow-up of pediatric LM patients treated with sildenafil. Our findings suggest that sildenafil is beneficial for the symptomatic treatment of LMs. Additional analysis on the role of sildenafil as adjuvant therapy is necessary to optimize the use of this medication in the management of complex LMs.

Single center experience with intralesional bleomycin sclerotherapy for lymphatic malformations.

The purpose of our study was to evaluate the efficacy of percutaneous sclerotherapy using bleomycin in treatment of lymphatic malformations. Between January 2009 and January 2013, ten patients with lymphatic malformations who were admitted to the interventional radiology department were included in this retrospective study. Intralesional bleomycin was administered by percutaneous injection through 21-23 Gauge needles with a dose of 1mg/kg body weight. Patients were clinically and radiologically assessed at baseline and followed at first and third months after treatment. Response to treatment was measured visually by using photographs and by radiological images. Symptomatic improvement was also evaluated either by patients or parents. Excellent resolution was obtained visually in 80% of patients with lymphatic malformation. Significant resolution was achieved in 20% of patients. The percentage of radiographic resolution in size and the improvement in symptoms evaluated by patients or parents were similar with visual outcomes. No side effects were recorded except for fever in one patient and transient erythema in another patient. Intralesional bleomycin is a safe and effective treatment for patients with lymphatic malformations.

Diagnosis and surgical treatment of cervical macrocystic lymphatic malformations in infants.

The treatment of lymphatic malformations (LMs) represents a great clinical challenge. The present study reported on the treatment of 68 infants with cervical macrocystic LMs using surgical resection. The cases were retrospectively analyzed. All patients underwent pre-operative ultrasonography, computed tomography (CT), and magnetic resonance imaging (MRI) examinations. The surgery was performed under general anesthesia with endotracheal intubation. Ultrasonograms showed that 24 cases were monolocular, 44 were multilocular, 16 had no echo, 20 had a uniform low-level echo and 32 had a non-uniform low-level echo. CT showed non-enhancing low-attenuating cystic lesions and attenuation values of 10–45 HU. The magnetic resonance images of the LMs showed a low signal intensity on T1-weighted imaging (WI) and a high signal intensity on T2-WI. Complete resection was achieved in 56 patients, subtotal resection in eight and partial resection in four. Two complications were noted, including reversible paresis of the marginal mandibular branch of the facial nerve and a surgical-site infection. One patient in whom partial resection was achieved had recurrence at ~2 months after the surgery. Ultrasonography, CT and MRI clearly demonstrated the size, shape, extent and adjacent structures of LMs, which aided in surgical planning and assessment of potential risks. Surgical excision increased the chances of cure and was relatively safe for infants aged <1 year. Location and extent, rather than age, were determined to be the most important factors for successful surgical treatment.

Percutaneous ultrasound-guided sclerotherapy with polidocanol microfoam for lymphatic malformations.

Lymphatic malformations (LMs) are low-flow congenital lesions that consist of cysts of varying size. Sclerotherapy with intralesional bleomycin and OK-432 has been reported to yield dramatically beneficial results for this disorder. However, inflammation-related symptoms are often seen after treatment with these sclerosing agents. On the other hand, polidocanol (POL) is reportedly associated with fewer allergic and inflammatory reactions. Up to now, however, very few reports have documented the use of POL microfoam for treatment of LMs. This study was performed to assess the efficacy and safety of POL microfoam sclerotherapy for LMs. Between 2003 and 2016, cases were identified from a prospectively compiled database on low-flow congenital vascular malformations before undertaking a retrospective electronic chart review. Patients were included if they had LMs that had been treated by POL microfoam sclerotherapy. The location, size, and type of LMs were assessed using ultrasound and magnetic resonance imaging. Twenty-gauge venous catheters were inserted into the lymphatic space under ultrasound visualization. The LMs were then fully aspirated if they were macrocystic in form. Microfoam composed of 3% POL was then injected under ultrasound guidance. Microcystic LMs were treated by direct injection with POL microfoam under ultrasound guidance. The outcome was assessed by clinical examination combined with findings of postsclerotherapy imaging using ultrasound and magnetic resonance imaging. During a 13-year period, 32 patients met the inclusion criteria. These were 11 (34%) male patients and 21 (66%) female patients with a mean age of 18years. The LMs were localized to the head and neck (47%), the trunk (38%), and the extremities (15%). The lesions were subdivided into macrocystic (56%), mixed macrocystic and microcystic (31%), and microcystic (13%) LMs. The average lesion size was 6.6× 4.6× 3.0cm. The mean number of treatment sessions was 2.8 (range, 1-15), with a mean foam volume of 4.6 (range, 1-10) mL. Excellent (47%) and moderate (41%) responses were seen in 88% of the patients. Notably, half of the patients achieved excellent or moderate resolution with a single treatment session. Intralesional hemorrhage occurred in four patients (13%) but resolved spontaneously. Only one patient with mixed macrocystic and microcystic LMs developed post-therapy infection. However, the other patients did not develop any post-therapy inflammation-related symptoms, including fever, pain, and marked swelling. Percutaneous sclerotherapy using POL microfoam appears to be safe and effective for treatment of LMs. Ultrasound-guided POL microfoam sclerotherapy should be considered for such lesions, particularly those that are exclusively macrocystic.

Multistage Sclerotherapy for Extensive Lymphatic Malformations With Airway Involvement in Infant: A Protocol to Prevent Tracheotomy

The management of extensive head and neck lymphatic malformations (LMs) in infants is challenging because of life-threatening upper airway compression. The aim of this study was to present a management protocol and evaluate the clinical outcomes for preventing tracheotomy in these patients. Fifteen infants with extensive head and neck LMs and airway involvement were enrolled from August 2010 through September 2015 at the Qilu Hospital of Shandong University (Jinan, China). According to various key factors associated with airway compression of patients in the perioperative period, different anesthesia types, treatment times, sclerosant concentrations, and sclerotherapy protocols were used. Multistage sclerotherapy was performed with bleomycin A5. All patients were followed at 1, 3, 6, and 12months. More extended follow-up was offered if patients had a residual lesion requiring supplementary sclerotherapy. Reviews on the site and size of the lesion, timesand durations of treatments, therapeutic response, airway complications, and conduction of tracheotomy were performed. LM lesions in the head and neck were located in the floor of the mouth, tongue, and neck. An overall average of 5 treatments was required; a lesion volume decrease of more than 50% was achieved in all patients. For efficacy, morphologic resolution was achieved in 3 of 15 cases (20%), and there was a substantial response in 12 of 15 cases (80%). Eight of 15 patients (53.3%) with microcystic LMs exhibited immediate swelling and had more serious upper airway symptoms than preoperatively, and 2 of 15 patients (13.3%) had feeding difficulty. No upper airway obstruction occurred and no tracheotomy was performed in the patients in this study. Multistage sclerotherapy with bleomycin A5 is a safe and effective treatment for extensive head and neck LMs in infants with airway involvement. A routine perioperative protocol is essential for decreasing airway complications.

Outcome of surgical excision of head and neck lymphatic malformations in children

BackgroundSurgical treatment of lymphatic malformations (LMs) in the head and neck is challenging. The aim of this study was to evaluate the outcome of surgical excision of head and neck LMs in children.Patients and methodsThis prospective study included cases of head and neck LMs treated primarily

Doxycycline Sclerotherapy Is Superior in the Treatment of Pediatric Lymphatic Malformations

PurposeTo evaluate efficacy of sclerotherapy with doxycycline versus sodium tetradecyl sulfate (STS) for treatment of macrocystic and mixed lymphatic malformations (LMs). Materials and MethodsThis single-center retrospective review identified 41 children (17 boys; 24 girls; age range, 1 month to 15.4 y) who underwent sclerotherapy with doxycycline (n = 32) or STS (n = 9) for macrocystic (n = 31) or mixed (n = 10) LMs. There were 114 treatments performed, averaging 2.8 treatments (range, 1–8 treatments) per patient. Average follow-up time was 10 months (range, 1–59 months). Clinical response was deemed excellent or moderate if > 90% or > 50% of LMs resolved based on visual estimate. ResultsWith doxycycline, 87% of patients (28 of 32) had excellent or moderate response with an average of 2.8 treatments (range, 1–7 treatments); 13% required subsequent resection. With 3% STS monotherapy, only 55% of patients (5 of 9) had excellent or moderate response with an average of 2.8 treatments (range, 1–8 treatments), and 33% required subsequent resection. Significantly fewer patients treated with STS responded well compared with patients treated with doxycycline (P = .03). Patients treated with STS had significantly longer follow-up than patients treated with doxycycline (27 months vs 6 months, P = .0001). ConclusionsDoxycycline monotherapy resulted in a high rate of excellent clinical outcomes after a few treatments without increased need for subsequent operative resection. These results support use of doxycycline sclerotherapy as primary treatment for macrocystic and mixed LMs in children.

РОЛЬ СКЛЕРОЗУВАЛЬНОЇ ТЕРАПІЇ У ЛІКУВАНН І СУДИННИХ МАЛЬФОРМАЦІЙ У ДІТЕЙ

&lt;p&gt;РЕЗЮМЕ. Сучасні підходи до лікування судинних мальформацій (СМ) – це використання хірургічних, консервативних та ендоваскулярних способів, де склерозувальна терапія відіграє важливу роль. Мета дослідження – визначення ефективності і безпечності різних способів склеротерапії периферійних СМ у дітей. У дослідження включено 96 пацієнтів віком від 1 до 17 років з СМ за період січень 2011 р. – березень 2016 р. Період спостереження складав від 3 до 63 місяців. Склерозування венозних мальформацій (ВМ) виконано у 10 дітей за показаннями: больовий синдром (n=7), повторні кровотечі (n=2), психологічний дискомфорт (n=1). Склерозування проводили полідоканолом та тетрадецилсульфатом натрію у вигляді «піни» в концентрації 0,5–1 %. «Пінне» склерозування артеріовенозних мальформацій (АВМ) виконано у 2 дітей після попередньої емболізації. Склерозування кістозних лімфатичних мальформацій (ЛМ) з використанням ОК-432 проведено у 84 пацієнтів, серед яких макрокістозні діагностовано у 34 (40,5 %) дітей, мікрокістозні – у 9 (10,7 %) та змішані ЛМ у 41 (48,8 %). Результатом склерозувальної терапії ВМ було припинення кровотеч (n=2), покращення естетичного вигляду (n=2), зменшення больового синдрому (n=7). Після емболізації та склерозування АВМ кровотеч не було. Результатом склерозування ВМ та АВМ є досягнення косметичного результату та зменшення симптомів, проте не одужання. У 84 пацієнтів з кістозними ЛМ виконано 194 сесії склеротерапії ОК-432. Результат лікування ЛМ оцінювали у відсотках зменшення об’єму ЛМ. Результат лікування макрокістозних ЛМ: відмінний – у 94,1 % пацієнтів, добрий – у 5,8 %; мікрокістозних – відмінний – 0, добрий – у 55,6 %, задовільний – у 33,3 %, відсутній – в 11,1 %; результат склеротерапії змішаних ЛМ: відмінний – у 19,5 %, добрий – у 61,0 %, задовільний – у 17,1 %, відсутній – у 2,4 %. Ускладнення – шкірна алергічна реакція – виникло в 1 пацієнта (1,1 %). Склерозувальна терапія є безпечним і ефективним способом лікування СМ у дітей. Для лікування ВМ і АВМ використовували детергенти у вигляді «піни», для лікування кістозних ЛМ використовували ОК-432. Відмінний і добрий результат склеротерапії кістозних ЛМ отримано у 85,7 % пацієнтів, у 2,4 % результат лікування був незадовільний. Використання склеротерапії у дітей з ВМ і АВМ приводить до досягнення контролю за симптомами, тоді як ЛМ з переважанням макрокістозного компонента можуть бути успішно вилікувані.&lt;/p&gt;&lt;p&gt; &lt;/p&gt;

Sclerotherapy for lymphatic malformations of the head and neck in the pediatric population

BackgroundSclerotherapy is one of the most commonly used minimally invasive interventions in the treatment of macrocystic lymphatic malformations (LMs). Several different sclerosing agents and injection protocols have been reported in...

Surgical Excision With Bleomycin Irrigation: A Better Primary Treatment Choice for Pediatric Submandibular Lymphatic Malformations

To compare the efficacy of surgical excision combined with intralesional bleomycin irrigation with that of needle aspiration with intralesional bleomycin sclerotherapy (IBS) in pediatric submandibular lymphatic malformations (LMs). The medical records of 32 patients were analyzed. Clinical response was categorized by improvement in size as determined by imaging studies and calculations of lesion volume. An excellent response was defined as a decrease in volume greater than 90%, a satisfactory response was defined as a decrease greater than 50%, and a poor response was defined as a decrease less than 50%. In addition, the authors developed a new scale to quantify treatment outcome. This scoring system is composed of 6 parts: size decrease, esthetic problem, functional problem (eg, breathing, eating, and speech), postoperative complications, necessity for further intervention, and times of intervention. Pearson χ2 and t tests were used to compare treatment outcome between the 2 groups. The average age at first treatment was 2years. Twenty-two patients underwent surgical excision with bleomycin irrigation as the primary treatment. Three of the 22 underwent 2 rounds of cyst aspiration with IBS during follow-up. IBS was performed as the primary treatment in 10 patients. All patients underwent a single surgery, and the number of IBS procedures per patient varied from 1 to 3 (mean, 1.8). In the surgical group, 72.7% of patients (16 of 22) showed an excellent response, 18.2% (4 of 22) showed a satisfactory response, and 9% (2 of 22) showed a poor response. The surgical excision group had a significantly higher excellent response rate than the sclerotherapy group (P < .05). According to the new scale, there was a marked difference between the mean values of the 2 groups (14.2 ± 2.1 vs 9.8 ± 1.8), and surgical excision with bleomycin irrigation showed better efficacy than IBS. Surgical excision with bleomycin irrigation is a better primary treatment for pediatric submandibular LMs. Cyst aspiration with IBS is a good method to treat localized recurrence.

Treatment of deep-seated facial microcystic lymphatic malformations with intralesional injection of pingyangmycin.

Treatment of microcystic lymphatic malformations (LMs) is still a great challenge to physicians in the field of managing vascular anomalies. Several kinds of treatment have been proposed for microcystic LMs, but the responses to these treatment modalities vary considerably among individuals. The aim of the study was to investigate the safety and efficacy of intralesional injection of pingyangmycin for microcystic LMs located in the deep facial region.Twenty-one consecutive patients with deep-seated facial microcystic LMs were treated with intralesional injection of pingyangmycin between March 2010 and April 2015. The patients received 2 to 8 injections, and the average session was 3.7. The therapeutic efficacy was accessed on the basis of the imaging findings and clinical measurements.Among the 21 patients, the clinical responses were excellent in 7 patients (33.33%), good in 9 patients (42.86%), fair in 3 patients (14.29%), and poor in 2 patients (9.52%). No severe side effects were encountered. Furthermore, therapeutic outcomes were significantly associated with lesion location (P = 0.006) and number of injections (P = 0.003).Our study supports that sclerotherapy with pingyangmycin is safe and effective for the treatment of deep-seated facial microcystic LMs.