Laryngeal papillomatosis in an infant leading to fatal outcome - Maternal vulvar condylomata acuminata as a possible cause of intrapartum infection
Sandra Jerkovic Gulin1, Zlatko Hrgovic2* and Sanja Jaska2
1Sibenik General Hospital, Sibenik, Croatia
2Johann Wolfgang Goethe University, Frankfurt am Main, Germany
*Address for Correspondence: Zlatko Hrgovic, Johann Wolfgang Goethe University, Frankfurt am Main, Germany, Tel: +385 49 69 293000; Email: firstname.lastname@example.org
Dates: Submitted: 05 April 2019; Approved: 24 April 2019; Published: 25 April 2019
How to cite this article: Gulin SJ, Hrgovic Z, Jaska S. Laryngeal papillomatosis in an infant leading to fatal outcome - Maternal vulvar condylomata acuminata as a possible cause of intrapartum infection. Ann Dermatol Res. 2019; 3: 001-002. DOI: 10.29328/journal.adr.1001006
Copyright: © 2019 Gulin SJ, et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Death due to laryngeal papillomatosis is unfrequently described . According to the literature, sudden death due to respiratory failure was described in adults and children (mainly above 7 years of age) but not in infants , so we present this case as a curiosity.
A 31-year-old tripara delivered a female eutrophic newborn baby with precipitous vaginal labour (3100 g/52 cm; Apgar score 10/10). The course of pregnancy, with an exception of cigarette smoking approximately ten cigarettes daily, was unremarkable. According the medical documentation, the Pap smear from previous pregnancy (three years prior to this pregnancy) revealed moderate cervical intraepithelial neoplasia (CIN II). Proposed HPV typing was rejected by the patient. Multiple condylomata acuminata of various sizes were observed in external genital and perineal area. Before giving birth, surgical removal of genital warts was proposed but rejected by the patient. Pap smear taken during this pregnancy did not show any abnormalities. Spontaneous precipitous labour as well as early neonatal period, were unremarkable. Both mother and baby were discharged from the hospital with recommendation for breastfeeding. Two weeks after delivery baby started to lose weight and breastfeeding was disabled with frequent choking. Consequently, the mother began bottle-formula-feeding. Because of stridorous breathing, the baby was examined by a pediatrician and an otorhinolaryngologist. Laryngoscopy examination revealed papillomatous lesions on both vocal cords. The infant was referred to the specialized pediatric clinic where the fulguration of the lesions was performed. The breathing normalized for the next two weeks, followed again by severe breathing. ENT specialist observed recurrent lesions obstructing rima glottides. The lesions were surgically removed, but due to the apparent recurrence of the aggressive papillomatosis lesion, the tracheostomy was performed.
Discussion and Conclusion
During the prolonged hospitalization for next three months the infant was fed through the nasogastric tube. Surgical excision and fulguration of papillomatous lesions were repeatedly performed with regular tracheostomy care. At the end of the third month of life, the child presented clinical and laboratory signs of severe infection (sepsis). The primary focus was bacterial and mycotic pneumonia resistant to the aggressive antibiotic and antimycotic therapy. Unfortunately, due to severe inflammatory response and multi-organ failure, the child died at the age of 3.5 months. The autopsy finding indicated the supravocal and infravocal progression of papillomatosis lesions with severe tracheomalacia, massive bacterial and mycotic pneumonia with hepatization of lungs HPV type 11.
Laryngeal papillomatosis is caused by the human papilloma virus (HPV), mostly associated with type 6 and 11, and rarely 16, 18, 31, 45 [2,3]. In children this condition is characterised by massive growth, frequently multifocal lesions with a tendency to recur and spread throughout the respiratory tract (recurrent respiratory papillomatosis) . HPV shows high affinity for the junction of ciliary and squamous epithelia. Therefore, papillomas are frequently found on the false vocal folds, the upper and lower margins of the ventricle, and the lower surface of the true vocal folds . Although spontaneous remission may occur, pulmonary spread and malignant transformation to squamous cell carcinoma of the lung have been reported . Surgical excisions may secure an adequate airway, albeit repeated recurrences are common and frequently leading to multiple procedures and serious complications . In patients who underwent tracheotomy, lesions are commonly revealed at the stoma . Moreover, tracheotomy is considered a cause of an accelerated dissemination of papillomas throughout the respiratory tract . Younger age at the time of diagnosis is associated with more aggressive disease behavior . Genital warts during pregnancy are considered the strongest risk factor for acquiring laryngeal papillomatosis by vertical HPV transmission from mother to child. Furthermore, if genital warts are present at the time of delivery, a cesarean section must be performed in order to prevent possible serious laryngeal papillomatosis .
We found in the literature cases of children’s death due to chronic respiratory insufficiency after multiple interventions for recurrent respiratory papillomatosis , as well as due to pulmonary carcinoma , but the case of severe, recurrent, massive and fast growing laryngeal papillomas in a newborn leading to a death after just 3,5 months has not been reported.
- Zawadzka-Głos L, Chmielik M, Bielicka A. Laryngeal papillomatosis in children and their surgical treatment. New Med. 2002; 2: 64-66. Ref.: https://tinyurl.com/y2mxtrs5
- Venkatesan NN, Pine HS, Underbrink MP. Recurrent respiratory papillomatosis. Otolaryngol Clin North Am. 2012; 45: 671-694. Ref.: https://tinyurl.com/yyvmzbux
- Omland T, Lie KA2, Akre H1, Sandlie LE2, Jebsen P, et al. Recurrent respiratory papillomatosis: HPV genotypes and risk of high-grade laryngeal neoplasia. PLoS One. 2014; 9: e99114. Ref.: https://tinyurl.com/y6r6nq7z
- Simma B, Burger R, Uehlinger J, Ghelfi D, Hof E, et al. Squamous-cell carcinoma arising in a non-irradiated child with recurrent respiratory papillomatosis. Eur J Pediatr. 1993;152: 776-778. Ref.: https://tinyurl.com/y3c7crvl
- Stamataki S, Nikolopoulos TP, Korres S, Felekis D, Tzangaroulakis A, et al. Juvenile recurrent respiratory papillomatosis: Still a mystery disease with difficult management. Head Neck. 2007; 29: 155-162. Ref.: https://tinyurl.com/y2nnrbwr
- Cole RR, Myer CM, Cotton RT. Tracheotomy in children with recurrent respiratory papillomatosis. Head Neck. 1989; 11: 226-230. Ref.: https://tinyurl.com/y6k5kha3
- Derkay CS, Wiatrak B. Recurrent Respiratory Papillomatosis: A Review. Laryngoscope. 2008; 118: 1236-1247. Ref.: https://tinyurl.com/y2p5zwgf
- Silverberg MJ, Thorsen P, Lindeberg H, Grant LA, Shah KV. Condyloma in pregnancy is strongly predictive of juvenile-onset recurrent respiratory papillomatosis. Obstet Gynecol. 2003; 101: 645-652. Ref.: https://tinyurl.com/y3xpd5t6