Brief Clinical Studies
The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
Recurrent Ptosis in a Patient With Blepharochalasis: Clinical and Histopathologic Findings Yasuhiro Takahashi, MD, PhD,* Xiaodong Zheng, MD, PhD,† Hidenori Mito, MD,‡ Kazunami Noma, MD, PhD,§ Hirohiko Kakizaki, MD, PhD* Abstract: A 37-year-old woman presented with right upper eyelid blepharochalasis with ptosis. Right upper eyelid edema had occurred 2 to 3 times per year by 30 years old, although the frequency decreased with age. The edema occurred spontaneously and resolved within 1 to 2 days. She underwent a right levator tucking surgery at 22 years old, and the ptosis recurred 2 years postoperatively. She again underwent ptosis surgery with skin excision at 37 years old. The intraoperative findings showed a thin levator aponeurosis. The white line was therefore advanced to the upper tarsal edge, resulting in an appropriate height and curvature. Three months later, the patient’s eyelid height was 1.5 mm higher with a little temporal peaking. The levator aponeurosis was histopathologically shown to contain many capillaries. The increased vascularity of the levator aponeurosis may contribute to recurrent bouts of edema resulting in stretching and disinsertion of the aponeurosis. Key Words: Blepharochalasis, edema, levator aponeurosis, temporal peaking, recurrent bouts
B
lepharochalasis is an uncommon disorder characterized by recurrent, nonpainful, nonerythematous episodes of eyelid edema.1 Multiple attacks of edema result in thinning, stretching, and atrophy of eyelid tissue.1 The eyelid skin becomes redundant, bronze discolored, and atrophic, appearing like wrinkled cigarette paper.1 Other sequelae included blepharoptosis, a pseudoepicanthal fold, and laxity of the lateral canthal tendon.1,2 Rounding of the lateral canthal angle is due to the lateral canthal tendon laxity.1,2 Common histopathologic findings are atrophy, fragmentation, and markedly decreased amounts of elastic fibers in the dermis as well as generally dilated capillaries that are increased in number.3 Although the condition of blepharochalasis is divided into active and quiescent stages, surgery is recommended to be deferred until the condition becomes quiescent, as edema attacks in an active stage decrease with age.1,2
From the *Department of Ophthalmology, Aichi Medical University, Nagakute, Aichi, Japan; †Department of Ophthalmology, Ehime University School of Medicine, Shitsukawa, Toon, Ehime, Japan; ‡Ide Eye Hospital, Kasumicho, Yamagata, Japan; and §Noma Eye Clinic, Naka, Hiroshima, Japan. Received March 10, 2014. Accepted for publication April 24, 2014. Address correspondence and reprint requests to Dr Hirohiko Kakizaki, Department of Ophthalmology, Aichi Medical University, Nagakute, Aichi 480–1195, Japan; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001081
e52
Here, we report a blepharochalasis patient with recurrent ptosis with emphasis on the clinical and histopathologic findings. Informed consent was obtained from the patient.
CLINICAL REPORT A 37-year-old woman sought consultation for right ptosis (Fig. 1A). History revealed that right upper eyelid recurrent edema had occurred until 5 years previously. The edema had occurred 2 to 3 times per year by 30 years old, but subsequently, the frequency decreased with age. The edema occurred spontaneously and resolved within 1 to 2 days. Allergy investigations showed negative results. The patient underwent a right levator tucking surgery when she was 22 years old. Although the results were good for 2 years after the operation, a right ptosis recurred. Without the diagnosis of edema, she did not receive any treatment. On examination, the patient showed right upper eyelid ptosis with a margin reflex distance (MRD) of 0 mm OD (+4.5 mm OS), an excellent levator function (12 mm OD and 13 mm OS), and a high upper eyelid crease consistent with levator dehiscence (Fig. 1A). An epinephrine test was performed resulting in no right upper eyelid elevation. The upper eyelid skin was redundant, thin, and wrinkled and had bronze discoloration. Atrophy of the nasal and preaponeurotic fat pads was noted with a prominent pseudoepicanthal fold. Laxity of the lateral canthal tendon and rounding of the lateral canthal angle were observed as well. A diagnosis of blepharochalasis syndrome was made. We performed surgery on the right ptosis and skin redundancy. The new skin crease was set at 8 mm from the eyelid margin, and the skin at 4 mm in height was removed. The levator aponeurosis was thin, and preaponeurotic fat was atrophic (Fig. 1B). The white line of the levator aponeurosis was fixed to the upper edge of the tarsal plate resulting in 3.5-mm MRD with an appropriate curvature (Fig. 1C). Three months later, the patient’s MRD was 5.0 mm, but a little temporal peaking was observed (Fig. 1D). The removed skin and aponeurosis were submitted for histopathologic examination using Masson trichrome and Elastica van Gieson staining. The skin showed many capillaries, and the reticular dermis demonstrated elastic fibers, but its volume was not much (Fig. 2A). The elastic fibers were fragmented (Fig. 2B). The levator aponeurosis showed many capillaries, several of which were dilated (Fig. 2C).
DISCUSSION We reported clinical and histopathologic findings in a blepharochalasis patient showing recurrent ptosis. The operative results were satisfactory, but some temporal peaking was demonstrated 3 months postoperatively. Surgical management is usually deferred until blepharochalasis is in a quiescent phase to avoid recurrent bouts of eyelid edema leading to further ptosis and eyelid atrophy.1–3 The patient in this study first
FIGURE 1. A, A right ptosis with blepharochalasis in a 37-year-old woman. B, The levator aponeurosis was thin, and the preaponeurotic fat was atrophic. C, Intraoperative photograph in the sitting position. The right upper MRD was 3.5 mm with an appropriate curvature. D, Three months after the operation, the patient’s right MRD was 5.0 mm with some temporal peaking.
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
FIGURE 2. A, The skin shows many capillaries. The reticular dermis demonstrates elastic fibers with a small volume; Elastica van Gieson, bar = 0.5 mm. B, The elastic fibers are fragmented. Several capillaries are seen in this figure; Elastica van Gieson, bar = 0.05 mm. C, The levator aponeurosis has many capillaries, several of which are dilated; Masson trichrome, bar = 0.5 mm.
underwent ptosis surgery in an active phase resulting in recurrence. A tucking procedure, which made a weak scar,4 may have accelerated the recurrence. A ptosis in blepharochalasis occasionally recurs even after a levator aponeurosis advancement procedure.2 Although an anterior approach has been recommended for ptosis surgery in blepharochalasis,1–3 a levator aponeurosis advancement should be adopted because of higher reliability against levator tucking.4 Results of ptosis surgery are usually unpredictable in patients with blepharochalasis.2 It has been speculated that a reason for this is a high incidence of lateral canthal tendon laxity, which increases the risk of excessive elevation of the lateral eyelid and temporal peaking when the aponeurosis is advanced.2 The patient in this study showed a lateral canthal tendon laxity as well. Although the upper eyelid curvature was acceptable during the operation, a little temporal peaking with higher MRD was shown 3 months later. As an upper eyelid moves laterally when it is opening,5 the peak position of the upper eyelid needs to be settled a little medially with a prospective estimation of a postoperative overcorrected tendency in blepharochalasis.2,3 A specific finding of the histopathologic examination was richness of capillaries in the levator aponeurosis. This is the first specimen of the levator aponeurosis tissue in blepharochalasis. Although an idiopathic angioedema is speculated as the pathology of blepharochalasis,2 increased vascularity of the levator aponeurosis may ease the extravasation of the fluid from vessels, which results in stretching and disinsertion of the aponeurosis.2,3 The patient in this report underwent ptosis surgery previously, but it was a tucking procedure with less influence on the aponeurosis tissue. The appearance of the skin was similar to the common findings of this condition,3 although the reticular dermis had a few elastic fibers. In conclusion, clinical and histopathologic findings in a blepharochalasis patient showing recurrent ptosis were reported. Surgery is recommended to be performed in a quiescent phase to avoid recurrent bouts of eyelid edema. The height and shape of the upper eyelid need to be settled with a prospective estimation of a postoperative overcorrected tendency of ptosis in blepharochalasis. Increased vascularity of the levator aponeurosis may contribute to recurrent bouts of eyelid edema resulting in stretching and disinsertion of the aponeurosis leading to ptosis recurrence.
Brief Clinical Studies
Immediate Reconstruction of the Maxillary Sinus After Resection of Preoperatively Misdiagnosed Unicystic Ameloblastoma With an Ectopic Third Molar Shin Hyuk Kang, MD, Tae Hui Bae, MD, PhD, Han Koo Kim, MD, PhD, Woo Seob Kim, MD, PhD, Mi Kyung Kim, MD, PhD Abstract: We report a case of unicystic ameloblastoma associated with an ectopic third molar in the right maxillary sinus, which was misdiagnosed as a dentigerous cyst on preoperative small incisional biopsy. Surgical enucleation of the cystic lesion was performed under general anesthesia with immediate reconstruction of the maxillary sinus using titanium mesh plate. The patient's postoperative recovery was uneventful, and there was no evidence of tumor recurrence during the 7-month follow-up period. Key Words: Unicystic ameloblastoma, ectopic teeth, maxillary sinus, dentigerous cyst, misdiagnose
A
n ameloblastoma is a locally invasive benign neoplasm that originates from the odontogenic epithelium and constitutes approximately 1% of all oral and maxillomandibular cysts and tumors.1 There are 4 distinct clinicopathologic subtypes: unicystic, solid/multicystic, peripheral, and malignant. The tumor has a predilection for the third molar-ramus area in the mandible and usually occurs in adults aged 30 to 50 years, with an almost equal distribution between men and women.2–4 It usually presents as an invasive odontogenic tumor, often asymptomatic and slow growing with no evidence of swelling. Although ameloblastoma has relative benign histologic characteristics, this neoplasm has a high local recurrence rate and possibility of malignant development. In this study, we present a case of a 49-year-old man with a 4.5-cm-diameter unicystic ameloblastoma in the maxillary sinus surrounding the crown of an unerupted ectopic third molar tooth, which was preoperatively misdiagnosed as a dentigerous cyst.
CLINICAL REPORT A 49-year-old man presented with a 2-month history of a painless swelling on the right cheek (Fig. 1). He reported no symptoms of bleeding, discharge, trismus, nasal obstruction, or paresthesia. Physical examination revealed a diffusely hard, nontender lesion over the right maxillary sinus. Trigeminal and facial nerve functions
REFERENCES 1. Bergin DJ, McCord CD, Berger T, et al. Blepharochalasis. Br J Ophthalmol 1988;72:863–867 2. Collin JRO. Blepharochalasis. A review of 30 cases. Ophthal Plast Reconstr Surg 1991;7:153–157 3. Koursh DM, Modjtahedi SP, Selva D, et al. The blepharochalasis syndrome. Surv Ophthalmol 2009;54:235–244 4. Anderson RL, Dixon RL. Aponeurotic ptosis Surgery. Arch Ophthalmol 1979;97:1123–1128 5. Kakizaki H, Zako M, Mito H, et al. A guide to making a natural eyelid margin curvature in blepharoptosis surgery. Acta Ophthalmol Scand 2004;82:240–241
From the Departments of Plastic and Reconstructive Surgery and Pathology, Chung-Ang University Hospital, Seoul, Korea. Received February 25, 2014. Accepted for publication April 24, 2014. Address correspondence and reprint requests to Dr Tae Hui Bae, Department of Plastic and Reconstructive Surgery, Chung-Ang University Hospital, 224-1, Heuksuk-Dong, Dongjak-Gu, Seoul, Korea; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001072
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e53
The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
Recurrent Ptosis in a Patient With Blepharochalasis: Clinical and Histopathologic Findings Yasuhiro Takahashi, MD, PhD,* Xiaodong Zheng, MD, PhD,† Hidenori Mito, MD,‡ Kazunami Noma, MD, PhD,§ Hirohiko Kakizaki, MD, PhD* Abstract: A 37-year-old woman presented with right upper eyelid blepharochalasis with ptosis. Right upper eyelid edema had occurred 2 to 3 times per year by 30 years old, although the frequency decreased with age. The edema occurred spontaneously and resolved within 1 to 2 days. She underwent a right levator tucking surgery at 22 years old, and the ptosis recurred 2 years postoperatively. She again underwent ptosis surgery with skin excision at 37 years old. The intraoperative findings showed a thin levator aponeurosis. The white line was therefore advanced to the upper tarsal edge, resulting in an appropriate height and curvature. Three months later, the patient’s eyelid height was 1.5 mm higher with a little temporal peaking. The levator aponeurosis was histopathologically shown to contain many capillaries. The increased vascularity of the levator aponeurosis may contribute to recurrent bouts of edema resulting in stretching and disinsertion of the aponeurosis. Key Words: Blepharochalasis, edema, levator aponeurosis, temporal peaking, recurrent bouts
B
lepharochalasis is an uncommon disorder characterized by recurrent, nonpainful, nonerythematous episodes of eyelid edema.1 Multiple attacks of edema result in thinning, stretching, and atrophy of eyelid tissue.1 The eyelid skin becomes redundant, bronze discolored, and atrophic, appearing like wrinkled cigarette paper.1 Other sequelae included blepharoptosis, a pseudoepicanthal fold, and laxity of the lateral canthal tendon.1,2 Rounding of the lateral canthal angle is due to the lateral canthal tendon laxity.1,2 Common histopathologic findings are atrophy, fragmentation, and markedly decreased amounts of elastic fibers in the dermis as well as generally dilated capillaries that are increased in number.3 Although the condition of blepharochalasis is divided into active and quiescent stages, surgery is recommended to be deferred until the condition becomes quiescent, as edema attacks in an active stage decrease with age.1,2
From the *Department of Ophthalmology, Aichi Medical University, Nagakute, Aichi, Japan; †Department of Ophthalmology, Ehime University School of Medicine, Shitsukawa, Toon, Ehime, Japan; ‡Ide Eye Hospital, Kasumicho, Yamagata, Japan; and §Noma Eye Clinic, Naka, Hiroshima, Japan. Received March 10, 2014. Accepted for publication April 24, 2014. Address correspondence and reprint requests to Dr Hirohiko Kakizaki, Department of Ophthalmology, Aichi Medical University, Nagakute, Aichi 480–1195, Japan; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001081
e52
Here, we report a blepharochalasis patient with recurrent ptosis with emphasis on the clinical and histopathologic findings. Informed consent was obtained from the patient.
CLINICAL REPORT A 37-year-old woman sought consultation for right ptosis (Fig. 1A). History revealed that right upper eyelid recurrent edema had occurred until 5 years previously. The edema had occurred 2 to 3 times per year by 30 years old, but subsequently, the frequency decreased with age. The edema occurred spontaneously and resolved within 1 to 2 days. Allergy investigations showed negative results. The patient underwent a right levator tucking surgery when she was 22 years old. Although the results were good for 2 years after the operation, a right ptosis recurred. Without the diagnosis of edema, she did not receive any treatment. On examination, the patient showed right upper eyelid ptosis with a margin reflex distance (MRD) of 0 mm OD (+4.5 mm OS), an excellent levator function (12 mm OD and 13 mm OS), and a high upper eyelid crease consistent with levator dehiscence (Fig. 1A). An epinephrine test was performed resulting in no right upper eyelid elevation. The upper eyelid skin was redundant, thin, and wrinkled and had bronze discoloration. Atrophy of the nasal and preaponeurotic fat pads was noted with a prominent pseudoepicanthal fold. Laxity of the lateral canthal tendon and rounding of the lateral canthal angle were observed as well. A diagnosis of blepharochalasis syndrome was made. We performed surgery on the right ptosis and skin redundancy. The new skin crease was set at 8 mm from the eyelid margin, and the skin at 4 mm in height was removed. The levator aponeurosis was thin, and preaponeurotic fat was atrophic (Fig. 1B). The white line of the levator aponeurosis was fixed to the upper edge of the tarsal plate resulting in 3.5-mm MRD with an appropriate curvature (Fig. 1C). Three months later, the patient’s MRD was 5.0 mm, but a little temporal peaking was observed (Fig. 1D). The removed skin and aponeurosis were submitted for histopathologic examination using Masson trichrome and Elastica van Gieson staining. The skin showed many capillaries, and the reticular dermis demonstrated elastic fibers, but its volume was not much (Fig. 2A). The elastic fibers were fragmented (Fig. 2B). The levator aponeurosis showed many capillaries, several of which were dilated (Fig. 2C).
DISCUSSION We reported clinical and histopathologic findings in a blepharochalasis patient showing recurrent ptosis. The operative results were satisfactory, but some temporal peaking was demonstrated 3 months postoperatively. Surgical management is usually deferred until blepharochalasis is in a quiescent phase to avoid recurrent bouts of eyelid edema leading to further ptosis and eyelid atrophy.1–3 The patient in this study first
FIGURE 1. A, A right ptosis with blepharochalasis in a 37-year-old woman. B, The levator aponeurosis was thin, and the preaponeurotic fat was atrophic. C, Intraoperative photograph in the sitting position. The right upper MRD was 3.5 mm with an appropriate curvature. D, Three months after the operation, the patient’s right MRD was 5.0 mm with some temporal peaking.
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
FIGURE 2. A, The skin shows many capillaries. The reticular dermis demonstrates elastic fibers with a small volume; Elastica van Gieson, bar = 0.5 mm. B, The elastic fibers are fragmented. Several capillaries are seen in this figure; Elastica van Gieson, bar = 0.05 mm. C, The levator aponeurosis has many capillaries, several of which are dilated; Masson trichrome, bar = 0.5 mm.
underwent ptosis surgery in an active phase resulting in recurrence. A tucking procedure, which made a weak scar,4 may have accelerated the recurrence. A ptosis in blepharochalasis occasionally recurs even after a levator aponeurosis advancement procedure.2 Although an anterior approach has been recommended for ptosis surgery in blepharochalasis,1–3 a levator aponeurosis advancement should be adopted because of higher reliability against levator tucking.4 Results of ptosis surgery are usually unpredictable in patients with blepharochalasis.2 It has been speculated that a reason for this is a high incidence of lateral canthal tendon laxity, which increases the risk of excessive elevation of the lateral eyelid and temporal peaking when the aponeurosis is advanced.2 The patient in this study showed a lateral canthal tendon laxity as well. Although the upper eyelid curvature was acceptable during the operation, a little temporal peaking with higher MRD was shown 3 months later. As an upper eyelid moves laterally when it is opening,5 the peak position of the upper eyelid needs to be settled a little medially with a prospective estimation of a postoperative overcorrected tendency in blepharochalasis.2,3 A specific finding of the histopathologic examination was richness of capillaries in the levator aponeurosis. This is the first specimen of the levator aponeurosis tissue in blepharochalasis. Although an idiopathic angioedema is speculated as the pathology of blepharochalasis,2 increased vascularity of the levator aponeurosis may ease the extravasation of the fluid from vessels, which results in stretching and disinsertion of the aponeurosis.2,3 The patient in this report underwent ptosis surgery previously, but it was a tucking procedure with less influence on the aponeurosis tissue. The appearance of the skin was similar to the common findings of this condition,3 although the reticular dermis had a few elastic fibers. In conclusion, clinical and histopathologic findings in a blepharochalasis patient showing recurrent ptosis were reported. Surgery is recommended to be performed in a quiescent phase to avoid recurrent bouts of eyelid edema. The height and shape of the upper eyelid need to be settled with a prospective estimation of a postoperative overcorrected tendency of ptosis in blepharochalasis. Increased vascularity of the levator aponeurosis may contribute to recurrent bouts of eyelid edema resulting in stretching and disinsertion of the aponeurosis leading to ptosis recurrence.
Brief Clinical Studies
Immediate Reconstruction of the Maxillary Sinus After Resection of Preoperatively Misdiagnosed Unicystic Ameloblastoma With an Ectopic Third Molar Shin Hyuk Kang, MD, Tae Hui Bae, MD, PhD, Han Koo Kim, MD, PhD, Woo Seob Kim, MD, PhD, Mi Kyung Kim, MD, PhD Abstract: We report a case of unicystic ameloblastoma associated with an ectopic third molar in the right maxillary sinus, which was misdiagnosed as a dentigerous cyst on preoperative small incisional biopsy. Surgical enucleation of the cystic lesion was performed under general anesthesia with immediate reconstruction of the maxillary sinus using titanium mesh plate. The patient's postoperative recovery was uneventful, and there was no evidence of tumor recurrence during the 7-month follow-up period. Key Words: Unicystic ameloblastoma, ectopic teeth, maxillary sinus, dentigerous cyst, misdiagnose
A
n ameloblastoma is a locally invasive benign neoplasm that originates from the odontogenic epithelium and constitutes approximately 1% of all oral and maxillomandibular cysts and tumors.1 There are 4 distinct clinicopathologic subtypes: unicystic, solid/multicystic, peripheral, and malignant. The tumor has a predilection for the third molar-ramus area in the mandible and usually occurs in adults aged 30 to 50 years, with an almost equal distribution between men and women.2–4 It usually presents as an invasive odontogenic tumor, often asymptomatic and slow growing with no evidence of swelling. Although ameloblastoma has relative benign histologic characteristics, this neoplasm has a high local recurrence rate and possibility of malignant development. In this study, we present a case of a 49-year-old man with a 4.5-cm-diameter unicystic ameloblastoma in the maxillary sinus surrounding the crown of an unerupted ectopic third molar tooth, which was preoperatively misdiagnosed as a dentigerous cyst.
CLINICAL REPORT A 49-year-old man presented with a 2-month history of a painless swelling on the right cheek (Fig. 1). He reported no symptoms of bleeding, discharge, trismus, nasal obstruction, or paresthesia. Physical examination revealed a diffusely hard, nontender lesion over the right maxillary sinus. Trigeminal and facial nerve functions
REFERENCES 1. Bergin DJ, McCord CD, Berger T, et al. Blepharochalasis. Br J Ophthalmol 1988;72:863–867 2. Collin JRO. Blepharochalasis. A review of 30 cases. Ophthal Plast Reconstr Surg 1991;7:153–157 3. Koursh DM, Modjtahedi SP, Selva D, et al. The blepharochalasis syndrome. Surv Ophthalmol 2009;54:235–244 4. Anderson RL, Dixon RL. Aponeurotic ptosis Surgery. Arch Ophthalmol 1979;97:1123–1128 5. Kakizaki H, Zako M, Mito H, et al. A guide to making a natural eyelid margin curvature in blepharoptosis surgery. Acta Ophthalmol Scand 2004;82:240–241
From the Departments of Plastic and Reconstructive Surgery and Pathology, Chung-Ang University Hospital, Seoul, Korea. Received February 25, 2014. Accepted for publication April 24, 2014. Address correspondence and reprint requests to Dr Tae Hui Bae, Department of Plastic and Reconstructive Surgery, Chung-Ang University Hospital, 224-1, Heuksuk-Dong, Dongjak-Gu, Seoul, Korea; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001072
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e53
