Intralesional bleomycin injections (IBI) for the management of a huge life-threatening cervical cystic hygroma is superior to surgery. Surgery has a high rate of morbidity and even mortality. Recurrence and injury to major vessels, nerves, and lymphatics may be unavoidable. The authors report on a 3-day-old male infant who was diagnosed antenatally as a case of a huge cervical cystic hygroma. Postnatally, the patient required mechanical ventilation. The patient was treated successfully with IBI without complications. To our knowledge, this is the youngest case presenting acutely to be treated successfully with IBI without complications.Keywords: bleomycin, neonatal cystic hygroma, respiratory distres

Al Dumairy, Magdy A.

Alqahtani, Saad S.

Hader, Hamad A.

Ibrahim, Ashraf H.M.

English

AJOL - African Journals Online

Downloadedfromhttps://journals.lww.com/apsbyBhDMf5ePHKav1zEoum1tQfN4a+kJLhEZgbsIHo4XMi0hCywCX1AWnYQp/IlQrHD3iI+UqWbUAmSERZG9mMIIjf63deA2M12vp1Ht2+YscPo=on05/11/2018Downloadedfromhttps://journals.lww.com/apsbyBhDMf5ePHKav1zEoum1tQfN4a+kJLhEZgbsIHo4XMi0hCywCX1AWnYQp/IlQrHD3iI+UqWbUAmSERZG9mMIIjf63deA2M12vp1Ht2+YscPo=on05/11/2018Successful intralesional bleomycin injections for themanagement of a huge life-threatening cervicallymphangioma in a 3-day-old neonateSaad S. Alqahtania, Ashraf H.M. Ibrahimb, Hamad A. Haderb andMagdy A. Al DumairycIntralesional bleomycin injections (IBI) for the management ofa huge life-threatening cervical cystic hygroma is superior tosurgery. Surgery has a high rate of morbidity and evenmortality. Recurrence and injury to major vessels, nerves, andlymphatics may be unavoidable. The authors report on a3-day-old male infant who was diagnosed antenatally as acase of a huge cervical cystic hygroma. Postnatally, the patientrequired mechanical ventilation. The patient was treatedsuccessfully with IBI without complications. To ourknowledge, this is the youngest case presenting acutely to betreated successfully with IBI without complications. AnnPediatr Surg 14:92–94 © 2018 Annals of Pediatric Surgery.Annals of Pediatric Surgery 2018, 14:92–94Keywords: bleomycin, neonatal cystic hygroma, respiratory distressaDepartment of Interventional Radiology, bDepartment of Pediatric Surgery andcUS Unit, Department of Obstetrics/Gynecology, Armed Forces Hospital,Southern Region, Khamis Mushait, Saudi ArabiaCorrespondence to Ashraf H.M. Ibrahim, MD, FRCSI, DPS, Department ofPediatric Surgery, Armed Forces Hospital, Southern Region, PO Box 101,Khamis Mushait 61961, Saudi ArabiaTel: + 966 1725 00001 x22154; e-mail: ash_ib@hotmail.comReceived 7 July 2017 accepted 18 July 2017Case reportA 35 weeks’ gestational age boy was found to have a hugeneck mass antenatally on ultrasonography (US). Soon afterdelivery, the patient required mechanical ventilation due torespiratory distress. Clinically, the baby was 2 kg. Generalexamination was unremarkable. Locally, there was a hugecystic swelling mainly on the right side of the neck andextending to the left side (Fig. 1). Laboratory workup wasunremarkable. US of the neck at 1 day of age showed bilateralcystic lesions more sizable in the right side. The right and leftmasses were inseparable from each other. The diagnosis wasin favor of a lymphangioma or a cystic hygroma. A computedtomography of the neck with contrast confirmed theabove-mentioned findings (Fig. 2a and b). The cystic lesionsshowed multiple enhancing internal septations and multipleenhancing vascular lesions at the superior aspect. On the thirdday of life, the lesion in the neck was aspirated and theninjected with water-soluble bleomycin solution (0.5mg/1ml)under US guidance. The aspirate showed altered bloodin areas and serosanguinous fluid from other areas. A totalof 2ml (0.5mg/1ml/kg) was injected at different sites intoFig. 1Initial presentation of the patient soon after delivery.Fig. 2(a) Computed tomography with contrast at 1 day of age showing a largemultilocular cystic mass more sizable at the right side. The right and leftmasses are connected together in the midline. There were multipleenhancing internal septations supporting the diagnosis of alymphangioma. (b) A coronal view.92 Case report1687-4137 © 2018 Annals of Pediatric Surgery DOI: 10.1097/01.XPS.0000522254.67407.36Copyright r 2018 Annals of Pediatric Surgery. Unauthorized reproduction of this article is prohibited.the swelling. This was repeated after 1 week. There was asignificant reduction in the swelling after the secondintralesional bleomycin injections (IBI) and the patient couldbe extubated safely. Thereafter, IBI was repeated every2 weeks under US guidance for up to six injection sessions.The dose was adjusted according to the weight. In the lasttwo sessions the dose of IBI was increased to 1mg/1ml/kg.The IBI was tolerated well except for local mild erythemaand oropharyngeal disorder requiring nasogastric tube feedingthat resolved spontaneously. The swelling markedlydecreased in size (Fig. 3a–c). There is no recurrence nowfor 7 months.DiscussionA lymphangioma or a cystic hygroma mainly affectsthe neck and axilla in 75 and 20% of cases, respectively.The lesion may grow and cause compression, infection,and hemorrhage [1]. It may present with clinical signs ofbronchiolitis [2,3]. Surgery is the main tool for therapyexcept in premature infants and when neurovascularstructures are involved [4]. The aim of surgery is toremove the swelling without complications. Thismight not be possible, especially in a huge cervical orcervicomediastinal lymphangioma. There is a mortalityrate of 2–10% and morbidities that include damageto neurovascular structures, lymphorrhea, chylothorax,recurrence, oropharyngeal disorders that may persistfor several months, and bad cosmetic appearance dueto heroic and mutilating surgery [4–7]. Ming et al. [8]successfully treated a case of bilateral diaphragmaticlymphangioma with IBI after a failed surgical excision.IBI is as effective as surgery. It causes regression in 88%of cystic hygromas and complete resolution in 44% [9].It can be used in primary or recurrent cases. However, itis claimed that it cannot be used in a huge cystichygromas involving the mediastinum, the abdomen, or inlife-threatening emergency situations [4,9]. After injec-tion, the lesion may enlarge in size and cause morecompression. This has been challenged by Ibrahim et al.[10] by treating a huge life-threatening cervicomediast-inal cystic hygroma using IBI. In such emergencysituations the tracheal intubation should continue duringIBI until the lesion regresses in size when extubation canbe tried with extreme caution.Side effects of IBI mentioned in the literature includefever, the risk for marked increase in size, local erythema,temporary oropharyngeal disorder, and pulmonary toxicity.Many authors denied pulmonary toxicity if IBI is givenlocally in proper doses [4,6,11]. The technique of IBIwhether soluble solution or fat emulsion is well reported inthe literature [4,9,11–13].Spontaneous resolution is uncommon but has beenreported in up to 15% of cases [14]. The authors of thepresent report do not recommend waiting for sponta-neous resolution to occur. This may end up withrespiratory distress or acute respiratory failure requiringtracheal intubation. A difficult intubation should beanticipated. Neuromuscular blockers should be avoided[15]. Alternative equipments should be available. Thisincludes light stylet, a bougie, a video laryngoscope, or alaryngeal mask airway. Rigid bronchoscopy, emergencycannula cricothyrotomy with transtracheal jet ventilation,surgical cricothyroidotomy, or tracheostomy may also berequired [16,17].In conclusion, IBI is a safe and effective alternativeto surgery. It can be used as a primary therapy in ahuge cervical lymphangioma in a newborn even in acutelife-threatening emergency.Conflicts of interestThere are no conflicts of interest.References1 De Lorimier AA. Congenital malformation and neonatal problems of therespiratory tract. In: Welch KJ, Randolph JG, Ravitch MM, et al, editors.Pediatric surgery. Chicago, IL. 1986. pp. 631–644.Fig. 3The baby at 5 months of age in different positions. Note the discoloration at the right side due to intralesional bleomycin injections, which usuallydisappears with time. (a) Anterior view, (b) left side view, (c) right side view.Cervical cystic hygroma Alqahtani et al. 93Copyright r 2018 Annals of Pediatric Surgery. Unauthorized reproduction of this article is prohibited.2 Balkaran BN, Maharaj P, Pitt-Miller P. Intrathoracic cystic hygroma in aninfant with respiratory failure. West Indian Med J 1997; 46:128–129.3 Bosdure E, Mates M, Mely L, Guys JM, Devred P, Dubus JC. Cysticintrathoracic hemolymphangioma: a rare differential diagnosis of acutebronchiolitis in an infant. Arch Pediatr 2005; 12:168–172.4 Okada A, Kubota A, Fukazawa M, Imura K, Kamata S. Injection of bleomycinas a primary therapy of cystic lymphangioma. In: Fukazawa M, Imura K,Kamata S, editors. J Pediatr Surg 1992; 27:440–443.5 Grosfeld JL, Weber TR, Vane DW. One-stage resection for massivecervicomediastinal hygroma. Surgery 1982; 92:693–699.6 Tanaka K, Inomata Y, Utsunomiya H, Uemoto S, Asonuma K, Katayama T,et al. Sclerosing therapy with bleomycin emulsion for lymphangioma inchildren. Pediatr Surg Int 1990; 5:270–273.7 Cystic hygroma. Lancet 1990; 335:511–512.8 Ming YC, Wong KS, Wang CJ, Lai JY. Successful management of a bilateraldiaphragmatic lymphangioma. In: Wong KS, Wang CJ, Lai JY, editors. PediatrSurg Int 2007; 23:591–593.9 Orford J, Barker A, Thonell S, King P, Murphy J. Bleomycin therapy for cystichygroma. In: Barker A, Thonell S, King P, Murphy J, editors. J Pediatr Surg1995; 30:1282–1287.10 Ibrahim AHM, Kandeel A, Bazeed MF, Shaker M, Al Rawaf A. Successfulnon-surgical management of a huge life threatening cervicomediastinal cystichygroma. Case report and review of the literature. J Pediatr Surg Spec2009; 3:48–49.11 Muir T, Kristen M, Fourier P, Dippenaar N, Ionescu GO. IBI treatment forhemangiomas and congenital vascular malformation. Pediatr Surg Int 2004;19:766–773.12 Yura J. Bleomycin treatment for cystic hygroma in children. Arch Jap Chir1977; 46:607–614.13 Tanigawa N, Shimomatsuyz T, Takatrashi K, Inomata Y, Tanaka K, Satomura K,et al. Treatment of cystic hygroma and lymphangioma with the use ofbleomycin fat emulsion. Cancer 1987; 60:741–749.14 Alqahtani A, Nguyen LT, Flageole H, Shaw K, Laberge JM. 25 years'experience with lymphangiomas in children. J Pediatr Surg 1999;34:1164–1168.15 Fernandez-Alantud J, Hortelano-Arague A, Aranda-Romero C,Reinoso-Barbero F, Lahoz AI, Rabanal I. Undiagnosed cervical cystichygromas a cause of airway obstruction and difficult intubation in a child.Paediatr Anaesth 2008; 18:442–446.16 Sharma S, Aminuldin AG, Azlan W. Cystic hygroma: anaestheticconsiderations and review. Singapore Med J 1994; 35:529–531.17 Au CC, Chau SK, Chan HY, Tsoi NS. Cystic hygroma in the neck causingairway compromise: case report review of literature. J Paediatr Respirol CritCare 2015; 11:13–15.94 Annals of Pediatric Surgery 2018, Vol 14 No 2Copyright r 2018 Annals of Pediatric Surgery. Unauthorized reproduction of this article is prohibited.

Successful intralesional bleomycin injections for the management of a huge life-threatening cervical lymphangioma in a 3-day-old neonate

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Successful intralesional bleomycin injections for the management of a huge life-threatening cervical lymphangioma in a 3-day-old neonate

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