Ophthal Plast Reconstr Surg, Vol. 30, No. 4, 2014
Letters to the Editor
Severe Neutropenia Presenting With Candida Albicans Eyelid Abscess in a 1-Year-Old Child To the Editor: We wish to update the readership on the clinical course of the patient in the referenced article.1 The abscess recurred approximately 3 months after the second incision and drainage with positive cultures for Candida albicans and an absolute neutrophil count of 0.76 (normal range 1.19–7.21 K/μl). MRI findings showed a lacrimal sac diverticulum,2 and probing and irrigation revealed a patent nasolacrimal excretory system. Options discussed ranged from medical management to surgical excision of the diverticulum,3–7 and the parents elected medical management with daily fluconazole (4 mg/kg/d). The patient has tolerated fluconazole well and remained asymptomatic for the past 6 months, with no evidence of recurrence or amblyopia.
Bryan R. Costin, M.D. Elias I. Traboulsi, M.D. Jill A. Foster, M.D., F.A.C.S. Julian D. Perry, M.D. Correspondence: Bryan R. Costin, M.D., Cole Eye Institute, Cleveland Clinic, Cleveland, OH 44195 ([email protected])
REFERENCES 1. Costin BR, Costin CE, Wall PB, et al. Severe neutropenia presenting with Candida albicans eyelid abscess in a 1-year-old child. Ophthal Plast Reconstr Surg 2014;[ePub ahead of print, March 6, 2014]. 2. Polito E, Leccisotti A, Menicacci F, et al. Imaging techniques in the diagnosis of lacrimal sac diverticulum. Ophthalmologica 1995;209:228–32. 3. Kavanagh MC, Cahill KV. Congenital lacrimal system anomalies mimicking recurrent acute dacryocystitis. Ophthal Plast Reconstr Surg 2008;24:53–4. 4. Epley KD, Karesh JW. Lacrimal sac diverticula associated with a patent lacrimal system. Ophthal Plast Reconstr Surg 1999; 15:111–5. 5. Bullock JD, Goldberg SH. Lacrimal sac diverticuli. Arch Ophthalmol 1989;107:756. 6. Kim JH, Chang HR, Woo KI. Multilobular lacrimal sac diverticulum presenting as a lower eyelid mass. Korean J Ophthalmol 2012;26:297–300. 7. Sinnreich Z. Lacrimal diverticula. Orbit 1998;17:195–200.
Another Case of Neonatal Progeroid Syndrome and Bilateral Congenital Upper Eyelid Entropion To the Editor: After our report on a case of neonatal progeroid syndrome and bilateral congenital upper eyelid entropion was accepted for publication, we encountered a second case with similar features.1 This was a 28-day-old girl, born through a cesarean delivery, after an uneventful gestation of 38 weeks; her birth weight was 2,030 g. The parents of the infant were 26 years old, nonconsanguineous, and had no family history of similar disease; the mother was a primigravida. This patient had syndromic features similar to those of our previous patient: triangular face, macrocephaly, prominent scalp veins, mandibular hypoplasia, sparse
356
A 28-day-old girl with neonatal progeroid syndrome and bilateral congenital upper eyelid entropion. The patient is seen before (A) and 1 month after surgery (D). B, The iris refection is seen through the right upper eyelid. C, The levator-Müller muscle complex and tarsal plate were severely hypoplastic. The meibomian glands were few in number, thickened, and convoluted.
hair, low-set ears, subcutaneous lipodystrophy, and suprabuttock fat (Fig. A). Upper eyelid entropion was congenital, bilateral, and associated with lagophthalmos and purulent conjunctivitis. When the patient was 2 months old, the eyelids were examined and operated under a surgical microscope. Several abnormalities were noted, including visibility of the iris through the closed eyelids, pretarsal eyelid thickening, horizontal laxity, absence of the preaponeurotic fat tissue, hypoplasia of the tarsus, and levator-Müller muscle complex (Fig. C). In this case, the tarsus was narrower (1–2 mm) and the retractor muscles were much thinner than the previous case. The meibomian glands were convoluted and thick, and extended out of the tarsus. A striking finding was the absence of a tarsus-gland structure in the medial one-third of the upper eyelids (Fig. D). The patient was treated with horizontal eyelid shortening, anterior lamellar reposition, and retractor muscle advancement, as was done in the previous case.1 As a difference, however, the shortening procedure (the full-thickness pentagonal eyelid resection) was performed in the medial side of the eyelid, where the tarsal plate was defective. The retractor muscle layer over the conjunctiva and tarsus was very thin. Therefore, the 6.0 polyglactin sutures passing through these structures could grasp small amounts of tissues and may have slipped or formed a weak scar after surgery. Despite this concern, entropion resolved in both upper eyelids postoperatively and did not recur during the 3-month follow up (Fig. B).
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Ophthal Plast Reconstr Surg, Vol. 30, No. 4, 2014
This second case supports the idea that in patients with neonatal progeroid syndrome, congenital entropion is not a coincidental finding. Some anatomic abnormalities in the upper eyelid may be a common characteristic in these patients. In the previous case, an undercorrection occurred in the right upper eyelid that manifested as medial segmental trichiasis. This may have resulted from a medial tarsal defect that was missed at the time of surgery. In such cases, it may be appropriate to perform the horizontal eyelid shortening on the side where the tarsus was defective. Upper eyelid entropion in patients with neonatal progeroid syndrome likely results from the defective tarsal plate and retractor muscles, and the activity of the orbicularis muscle.2 The surgical treatment should address these potential pathogenic processes.
Letters to the Editor
Bülent Yazıcı, M.D. Correspondence: Bülent Yazıcı, M.D., Department of Ophthalmology, Uludag University, Bursa, Turkey (byazici@ uludag.edu.tr)
REFERENCES 1. Yazıcı B, Toka F, Comez AT. Anatomical characteristics and surgical treatment of bilateral congenital upper eyelid entropion in an infant with neonatal progeroid syndrome. Ophthal Plast Recons Surg 2014; [Epub ahead of print, May 6, 2014]. 2. Mills DM, Meyer DR. Central lower eyelid thinning with trichiasis: characterization and management of a unique subset of entropion in elderly patients. Ophthal Plast Reconstr Surg 2009;25:445–9.
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
357
Letters to the Editor
Severe Neutropenia Presenting With Candida Albicans Eyelid Abscess in a 1-Year-Old Child To the Editor: We wish to update the readership on the clinical course of the patient in the referenced article.1 The abscess recurred approximately 3 months after the second incision and drainage with positive cultures for Candida albicans and an absolute neutrophil count of 0.76 (normal range 1.19–7.21 K/μl). MRI findings showed a lacrimal sac diverticulum,2 and probing and irrigation revealed a patent nasolacrimal excretory system. Options discussed ranged from medical management to surgical excision of the diverticulum,3–7 and the parents elected medical management with daily fluconazole (4 mg/kg/d). The patient has tolerated fluconazole well and remained asymptomatic for the past 6 months, with no evidence of recurrence or amblyopia.
Bryan R. Costin, M.D. Elias I. Traboulsi, M.D. Jill A. Foster, M.D., F.A.C.S. Julian D. Perry, M.D. Correspondence: Bryan R. Costin, M.D., Cole Eye Institute, Cleveland Clinic, Cleveland, OH 44195 ([email protected])
REFERENCES 1. Costin BR, Costin CE, Wall PB, et al. Severe neutropenia presenting with Candida albicans eyelid abscess in a 1-year-old child. Ophthal Plast Reconstr Surg 2014;[ePub ahead of print, March 6, 2014]. 2. Polito E, Leccisotti A, Menicacci F, et al. Imaging techniques in the diagnosis of lacrimal sac diverticulum. Ophthalmologica 1995;209:228–32. 3. Kavanagh MC, Cahill KV. Congenital lacrimal system anomalies mimicking recurrent acute dacryocystitis. Ophthal Plast Reconstr Surg 2008;24:53–4. 4. Epley KD, Karesh JW. Lacrimal sac diverticula associated with a patent lacrimal system. Ophthal Plast Reconstr Surg 1999; 15:111–5. 5. Bullock JD, Goldberg SH. Lacrimal sac diverticuli. Arch Ophthalmol 1989;107:756. 6. Kim JH, Chang HR, Woo KI. Multilobular lacrimal sac diverticulum presenting as a lower eyelid mass. Korean J Ophthalmol 2012;26:297–300. 7. Sinnreich Z. Lacrimal diverticula. Orbit 1998;17:195–200.
Another Case of Neonatal Progeroid Syndrome and Bilateral Congenital Upper Eyelid Entropion To the Editor: After our report on a case of neonatal progeroid syndrome and bilateral congenital upper eyelid entropion was accepted for publication, we encountered a second case with similar features.1 This was a 28-day-old girl, born through a cesarean delivery, after an uneventful gestation of 38 weeks; her birth weight was 2,030 g. The parents of the infant were 26 years old, nonconsanguineous, and had no family history of similar disease; the mother was a primigravida. This patient had syndromic features similar to those of our previous patient: triangular face, macrocephaly, prominent scalp veins, mandibular hypoplasia, sparse
356
A 28-day-old girl with neonatal progeroid syndrome and bilateral congenital upper eyelid entropion. The patient is seen before (A) and 1 month after surgery (D). B, The iris refection is seen through the right upper eyelid. C, The levator-Müller muscle complex and tarsal plate were severely hypoplastic. The meibomian glands were few in number, thickened, and convoluted.
hair, low-set ears, subcutaneous lipodystrophy, and suprabuttock fat (Fig. A). Upper eyelid entropion was congenital, bilateral, and associated with lagophthalmos and purulent conjunctivitis. When the patient was 2 months old, the eyelids were examined and operated under a surgical microscope. Several abnormalities were noted, including visibility of the iris through the closed eyelids, pretarsal eyelid thickening, horizontal laxity, absence of the preaponeurotic fat tissue, hypoplasia of the tarsus, and levator-Müller muscle complex (Fig. C). In this case, the tarsus was narrower (1–2 mm) and the retractor muscles were much thinner than the previous case. The meibomian glands were convoluted and thick, and extended out of the tarsus. A striking finding was the absence of a tarsus-gland structure in the medial one-third of the upper eyelids (Fig. D). The patient was treated with horizontal eyelid shortening, anterior lamellar reposition, and retractor muscle advancement, as was done in the previous case.1 As a difference, however, the shortening procedure (the full-thickness pentagonal eyelid resection) was performed in the medial side of the eyelid, where the tarsal plate was defective. The retractor muscle layer over the conjunctiva and tarsus was very thin. Therefore, the 6.0 polyglactin sutures passing through these structures could grasp small amounts of tissues and may have slipped or formed a weak scar after surgery. Despite this concern, entropion resolved in both upper eyelids postoperatively and did not recur during the 3-month follow up (Fig. B).
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Ophthal Plast Reconstr Surg, Vol. 30, No. 4, 2014
This second case supports the idea that in patients with neonatal progeroid syndrome, congenital entropion is not a coincidental finding. Some anatomic abnormalities in the upper eyelid may be a common characteristic in these patients. In the previous case, an undercorrection occurred in the right upper eyelid that manifested as medial segmental trichiasis. This may have resulted from a medial tarsal defect that was missed at the time of surgery. In such cases, it may be appropriate to perform the horizontal eyelid shortening on the side where the tarsus was defective. Upper eyelid entropion in patients with neonatal progeroid syndrome likely results from the defective tarsal plate and retractor muscles, and the activity of the orbicularis muscle.2 The surgical treatment should address these potential pathogenic processes.
Letters to the Editor
Bülent Yazıcı, M.D. Correspondence: Bülent Yazıcı, M.D., Department of Ophthalmology, Uludag University, Bursa, Turkey (byazici@ uludag.edu.tr)
REFERENCES 1. Yazıcı B, Toka F, Comez AT. Anatomical characteristics and surgical treatment of bilateral congenital upper eyelid entropion in an infant with neonatal progeroid syndrome. Ophthal Plast Recons Surg 2014; [Epub ahead of print, May 6, 2014]. 2. Mills DM, Meyer DR. Central lower eyelid thinning with trichiasis: characterization and management of a unique subset of entropion in elderly patients. Ophthal Plast Reconstr Surg 2009;25:445–9.
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
357
