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Successful treatment of crocodile tears by injection of botulinum toxin into the lacrimal gland: a case report.

OBJECTIVE: Pathologic lacrimation (crocodile tears) is a rare but stigmatizing symptom after facial nerve paralysis. The aim of this pilot study was to examine whether botulinum toxin injection into the lacrimal gland is effective in reducing pathologic tear secretion. DESIGN: Case report. INTERVENTION: One patient who had crocodile tears after a zoster oticus infection received a botulinum toxin injection (2.5 mouse units) into the lacrimal gland. TESTING: Before injection, 1 week, 1 month, and 6 months after injection, patient's lacrimation was assessed by a Schirmer test. RESULTS: The lacrimation of the injected eye was reduced after 1 week and equal after 1 month when compared to the healthy side. After 6 months, hyperlacrimation reoccurred. No side effects were observed. CONCLUSION: Intraglandular injection of botulinum toxin into the lacrimal gland may serve as a sufficient therapy for crocodile tears.

Botulinum Toxins, Type A↗

The Kaleff-Hollwich technique and results of external dacryocysto-rhinostomy operation.

After a review of the history of lacrimal surgery the author describes the techniques of dacryocystorhinostomy which are currently performed. The ideal method is that of Ohm (1921) and Dupuy-Dutemps (1921) with its totally closed mucosal anastomosis. Because of the technical problems demonstrated with this technique the author recommends that of Kaleff (1937), modified by Hollwich (1977). The advantages of this method are: 1. The almost closed mucosal cavity which permits primary would healing. 2. The easy strain-free practicability particularly in cases of small scarred sac and pre-positioned ethmoid cells. 3. The sure fixation of the mucosal roof at the subcutis particularly in cases of marked swelling. Finally a survey of long-term and short term observations on all 324 cases from 1964 to 1977 is given.

Anesthesia, Local↗

The distal lacrimal ductal system and traumatic epiphora.

While transient epiphora is well documented in the literature, the incidence of permanent epiphora associated with maxillofacial procedures (Le Fort I, II, III, osteotomies, nasal atrostomies and mid-face fractures) is rare. However, little attention has been paid to the distal lacrimal ductal system and its relationship to permanent epiphora. The distal nasolacrimal ductal system was studied in adult dry skulls and cadaver specimens. These findings were correlated by clinical observation. Specific areas of emphasis within this complex include the length, width, and angulation of this system. Clinical applications are discussed based on an anatomical rationale for the limitation of the superior repositioning of the maxilla as well as for proper placement of the antral window in nasal antrostomy procedures and what anticipated post-residual effects of mid-facial trauma are on the ductal system.

Adult↗

Thymus, kidney and craniofacial abnormalities in Six 1 deficient mice.

Six genes are widely expressed during vertebrate embryogenesis, suggesting that they are implicated in diverse differentiation processes. To determine the functions of the Six1 gene, we constructed Six1-deficient mice by replacing its first exon by the beta-galactosidase gene. We have previously shown that mice lacking Six1 die at birth due to thoracic skeletal defects and severe muscle hypoplasia affecting most of the body muscles. Here, we report that Six1(-/-) neonates also lack a kidney and thymus, as well as displaying a strong disorganisation of craniofacial structures, namely the inner ear, the nasal cavity, the craniofacial skeleton, and the lacrimal and parotid glands. These organ defects can be correlated with Six1 expression in the embryonic primordium structures as revealed by X-Gal staining at different stages of embryogenesis. Thus, the fetal abnormalities of Six1(-/-) mice appear to result from the absence of the Six 1 homeoprotein during early stages of organogenesis. Interestingly, these Six1 defects are very similar to phenotypes caused by mutations of Eya 1, which are responsible for the BOR syndrome in humans. Close comparison of Six1 and Eya 1 deficient mice strongly suggests a functional link between these two factors. Pax gene mutations also lead to comparable phenotypes, suggesting that a regulatory network including the Pax, Six and Eya genes is required for several types of organogenesis in mammals.

Animals↗

Pyogenic granulomas of the lacrimal sac.

Eight cases of pyogenic granulomas of the lacrimal sac are reported. The clinical symptoms were consistent with chronic dacryocystitis which was unresponsive to previous probing and antibiotics. Extirpation of the sacs was done because of dacryocystocoele, pyocoele, or fistula formation through the skin. Trauma and inflammation were thought to have contributed to the growth of the pyogenic granulomas. The histopathological examination revealed nonspecific granulomas consisting of well vascularised, friable tissue of a myxomatous nature containing an infiltration of lymphocytes, plasma cells, and a few eosinophils. Over the granulomas an epithelial layer of cells is stretched showing no signs of dysplasia or neoplastic growth. The term 'lobular capillary hemangioma' has been suggested to describe lesions morphologically similar to those observed in the study. These masses made up 53.3% of a total of 15 lacrimal sac tumour biopsy specimens examined over a period of 10 years (1979-1989). The few reported cases of primary lacrimal sac tumour of non-epithelial origin suggest that they make up a large minority of the lacrimal sac tumours. A review shows that relatively few of these lesions have been described in recent English literature but these growths are probably not being as consistently reported as primary epithelial tumours.

Adult↗

Congenital lacrimal fistula.

Congenital lacrimal fistula is a rare developmental condition. Patients may be asymptomatic or have tearing from the fistula, the eye or both. The clinical features of 60 patients are reviewed, including presenting symptoms, presence of other lacrimal or systemic anomalies and morphology of the fistula. A detailed description is given of the surgical technique utilised in patients whose symptoms warrant surgical intervention. The results of this technique are presented and alternative treatment strategies are discussed. Theories of the aetiology of the condition are reviewed and morphological and histological evidence is presented to support our belief that congenital lacrimal fistulae represent aberrant canaliculi.

Adolescent↗

The role of nasolacrimal intubation in the management of childhood epiphora.

Nasolacrimal intubation has been advocated to obviate the need for dacryocystorhinostomy (DCR) for childhood epiphora which fails to resolve despite apparently successful probings. Twenty-eight intubations were attempted on children falling into this category. Of these, 25 were anatomically successful intubations (3 having had to be abandoned because of difficulties in retrieving the silicone tubes from the nose). Twenty patients (80%) had complete resolution of symptoms, 2 (8%) had improvement of symptoms such that no further intervention was necessary and 3 (12%) proceeded to DCR. A greater likelihood of a good outcome was seen if the tubes were left in situ for 6 months or more. We suggest that primary nasolacrimal intubation (that is nasolacrimal intubation without DCR) should be the next step in the management of childhood epiphora which fails to resolve after two probings. This approach may avoid a DCR in over 80% of children.

Child↗

Lacrimal surgery in patients with congenital cranial or facial anomalies.

A series of 26 patients with lacrimal dysfunction due to congenital cranial or facial anomalies are reviewed. The underlying anatomical abnormalities responsible for symptoms and their influence on surgical management and outcome are discussed and illustrated with representative cases. It is shown that by adaptation of standard surgical techniques a high success rate can be achieved.

Abnormalities, Multiple↗

A brief history of punctoplasty: the 3-snip revisited.

PURPOSE: To determine when and how to perform punctoplasty, review the history, and determine the efficacy of posterior ampullectomy. METHOD: Retrospective analysis of 53 cases of punctal stenosis, operated by a single surgeon by means of posterior ampullectomy via 3-snips. A review of the history of the procedure from papers found with a Medline search for 1-snip, 2-snip, 3-snip, and punctoplasty. RESULTS: In all, 102 sets of notes were reviewed. The age range was from 9 to 89 years with a mean age of 56 years. A total of 74% of patients were female. We excluded 22 patients who had additional surgery and 16 patients who elected not to have surgery. There was no comment as to success or failure in four sets of notes and seven notes could not be found; these cases were also excluded. Of the remaining 53 patients, success was documented in 49 cases, or 92%. CONCLUSIONS: The historical review helps explain the debate about the procedure. The retrospective review confirms that posterior ampullectomy via 3-snips is an effective procedure.

Adolescent↗

Endoscopic inferior dacryocystorhinostomy.

Conventional dacryocystorhinostomy (DCR) usually involves extensive removal of bone at the lacrimal fossa and hence risks disruption of the lacrimal pump mechanism. A physiological operation for nasal lacrimal blockage is described whereby only the inferior portion of the lacrimal sac and the adjacent duct are marsupialized into the nose. The operation is safe, quick and does not involve sophisticated instruments. Result of 81 consecutive endoscopic inferior DCRs reveals a success rate of over 90% which is maintained with time. Most patients had a wide lacrimal window with preserved lacrimal pump movement at the superior sac remnant.

Adult↗

An Epstein-Barr virus positive undifferentiated carcinoma in the lacrimal sac.

We report a Chinese patient who presented with metastatic undifferentiated carcinoma in the cervical lymph node of unknown primary origin. Despite a raised IgA titre against Epstein-Barr virus capsid antigen, examination and biopsy of the nasopharynx were negative. Radiotherapy was given to the head and neck region with the orbit shielded. There was complete resolution of the metastatic lymph nodes. She developed proptosis of her left eye one year afterward when a large tumour was found in the lacrimal sac region. Review of the initial computerized tomography revealed a small soft tissue mass in the same region. The tumour was composed of undifferentiated carcinoma cells associated with dense lymphoid infiltrate. In situ hybridization for Epstein-Barr virus EBER RNA showed strong positive signals in the malignant cells. This is the first reported case of an Epstein-Barr virus positive undifferentiated carcinoma with lymphoid stroma in the lacrimal sac. In addition to the nasopharynx, salivary glands, nasal cavity and paranasal sinuses, the lacrimal sac should be considered as a potential primary site for Epstein-Barr virus positive metastatic undifferentiated carcinoma in the cervical lymph nodes.

Adult↗

Imaging of the lacrimal gland.

The lacrimal gland is situated superolateral to the eye and produces tears that moisten, lubricate, and protect the delicate corneal and conjunctival epithelium. Anatomically related to the orbit but embryologically and functionally more closely related to the salivary glands, radiological imaging has proven invaluable in delineation and differentiation of the unique range of pathological processes affecting the lacrimal gland. This article details traditional and new imaging techniques used in investigating such pathology and discusses the imaging findings and patterns of spread characteristic of various neoplastic, inflammatory, and structural processes ranging from benign adenomas, adenocarcinomas, and lymphomas to sarcoidosis, Mickulicz's syndrome, histiocytosis, and benign dacrocysts.

Diagnosis, Differential↗

[Angiolymphoid hyperplasia with eosinophilia with bilateral involvement of the lacrimal glands].

BACKGROUND: Angiolymphoid hyperplasia with eosinophilia (ALHE) and Kimura's disease are two rarely occurring disorders very similar to each other; however, they are individual nosological entities. For a long time they were considered as a single disease due to the frequency of similar characteristics. The majority of authors have recently considered these diseases as two individual entities with some reciprocal specifications, both clinical as well as morphological. PATIENT: We report on the case of a 76-year old male white European who had suffered for more than six years from subcutaneous tumour formations in different parts of the body. Eyelid oedema on the right side with palpable resistance under the upper temporal edge of the orbit initially occurred five years after the first symptoms of the disease. A similar finding occurred on the left side after eight months. The tumours on both sides were surgically removed and sent for histological analysis. A unilateral recurrence of the finding appeared after one year, followed by surgical intervention and histological examination. RESULTS: Tumour infiltrate in the first two orbita excisions was topically related to lacrimal gland structures; these structures were not found in the third excision. The case was histologically diagnosed as ALHE despite some similar characteristics with Kimura's disease. The diagnosis of ALHE was histologically supported, especially by the absence of lymphatic follicle formations and fibrotisation in the infiltrate, and the identification of the appearance of epitheloid up to histiocytoid of proliferating endothelium, which forms small lumens. The diagnosis of ALHE was additionally supported by the fact that the incidence of Kimura's disease in white Europeans is very rare. CONCLUSIONS: According to our findings, both entities overlap one another, even in some characteristics considered to be distinguishing. The question arises, therefore, whether the strict separation of these nosological entities can indeed be determined with one hundred percent accuracy.

Aged↗

[Idiopathic eosinophilic orbital pseudotumor of the lacrimal gland in a child].

BACKGROUND: Idiopathic orbital pseudotumor is a rare disease. 10 % of the cases occur in children. In most of these juvenile cases, the swelling is bilateral, and histologically, eosinophilic granulocytes are found. In contrast to adults, children very rarely develop an associated systemic disorder. The disease is generally treated with steroids. Little is known about the natural course of idiopathic orbital pseudotumors in childhood. PATIENT: A 2S-year old girl presented with a unilateral swelling of the right upper lid. Four weeks later, a comparable swelling of the left side was revealed. We took biopsies of the lacrimal gland on both sides. Histology displayed an eosinophilic pseudotumor of the lacrimal gland. Steroid treatment was started, but after 4 weeks the parents refused to continue the treatment. During the following year, the bilateral swelling remained unchanged. Surprisingly, the bilateral swelling disappeared 3 years later and no systemic disorder had occured. CONCLUSIONS: Our case shows that spontaneous resolution of a juvenile eosinophilic orbital pseudotumor is possible. Treatment with steroids does not appear to be compulsory in all cases.

Biopsy↗