IHAPSH (Intrapapillary Hemorrhage with Adjacent Peripapillary Subretinal Hemorrhage)

Key Points at a Glance

Key Points of This Disease

• IHAPSH is a benign syndrome characterized by simultaneous optic disc hemorrhage and adjacent peripapillary subretinal hemorrhage.
• It is common in young myopic individuals (10–30s, predominantly female), and bilateral cases have been reported.
• Symptoms mainly include floaters and mild blurred vision; incidental asymptomatic findings are not uncommon.
• Visual field defects are often limited to enlarged blind spots or Bjerrum scotomas, and corrected visual acuity is generally preserved.
• The main pathophysiology is thought to involve a small scleral canal of the optic disc and vitreopapillary traction associated with posterior vitreous detachment (PVD).
• Coexistence with tilted disc syndrome and PHOMS has been reported, making differential diagnosis and long-term follow-up important.
• 多くは数週から数か月で自然吸収され、再発もまれである。経過観察が原則である。

1. 視神経乳頭出血（IHAPSH）とは

定義

Intrapapillary Hemorrhage with Adjacent Peripapillary Subretinal Hemorrhage（IHAPSH）は、視神経乳頭内（intrapapillary）の出血と、それに連続する乳頭周囲網膜下（peripapillary subretinal）の出血を同時に認める臨床像を指す。視神経乳頭浮腫様の所見を伴うこともあり、若年・近視眼に好発する良性の症候群として位置づけられる¹⁾。

歴史

乳頭出血と乳頭周囲網膜下出血を同一眼に認める病態は、1975年のCibisらによる後部硝子体剥離に伴う網膜出血の報告にさかのぼる²⁾。1995年にはKatzとHoytが、若年者で後部硝子体剥離が不完全な状態で生じる乳頭内・乳頭周囲出血を硝子体乳頭牽引（vitreopapillary traction）の所見として報告した³⁾。2004年、Kokameらが9例（女性7例・男性2例、平均年齢47歳、屈折は−1.00〜−9.50D）について「intrapapillary hemorrhage with adjacent peripapillary subretinal hemorrhage」として独立した症候群を提唱し、現在の名称が定着した¹⁾。

Epidemiology

The exact incidence is unknown, but case reports in Japan tend to be biased toward young, myopic women. In the initial 9 cases reported by Kokame et al., the majority were women, and refractive errors ranged from mild to high myopia ¹⁾. Bilateral cases are relatively rare, but there are reports of sequential onset in both eyes over a 2-year course in a 25-year-old woman ⁵⁾ and similar findings in both eyes at a 2-month interval in a 36-year-old woman ⁶⁾. Cases in teenagers are also increasing, with typical examples accumulating in an 11-year-old girl ⁷⁾, a 12-year-old ^9,10), a 17-year-old ¹¹⁾, and a 19-year-old ¹⁾.

Q How rare is IHAPSH?

A

The nationwide incidence is not known. Most reports are case-based, indicating it is clearly a rare condition. However, it is noted that it may be more common in populations with overlapping young age, myopia, and posterior vitreous detachment, and the actual frequency might be somewhat higher than recognized.

2. Main Symptoms and Clinical Findings

Wang Y, Chen H, Yuan L, et al. Intrapapillary hemorrhage with adjacent peripapillary subretinal hemorrhage of both eyes after COVID-19 infection: a case report. BMC Ophthalmol. 2024;24(1):101. Figure 1. PMCID: PMC10913657. License: CC BY 4.0.

Fundus photographs of both eyes. A dark red hemorrhage is seen within the optic disc, and along its margin, the hemorrhage extends continuously into the peripapillary subretinal space. The optic disc is small and tilted, reflecting the anatomical predisposition of tilted disc and small scleral canal characteristic of IHAPSH.

Subjective Symptoms

Typical subjective symptoms are floaters and mild blurred vision. Many cases are asymptomatic and discovered incidentally during routine eye exams or workup for other diseases. Visual acuity is often well preserved; in Kokame et al.’s initial 9 cases, 7 maintained visual acuity of 1.0 or better ¹⁾. In Japanese cases, a 12-year-old girl with corrected visual acuity of 0.9/1.2 at initial visit ¹⁰⁾, a 17-year-old maintaining 1.2 in both eyes ¹¹⁾, and an 11-year-old girl with 1.2/1.2 ⁷⁾ have been reported.

However, when hemorrhage extends into the vitreous cavity or involves subretinal hemorrhage near the macula, blurred vision, visual field defects, and central scotoma may occur. A case of a 62-year-old woman with vitreous hemorrhage and peripapillary subretinal hemorrhage resulting in a corrected visual acuity of 0.01 in the right eye has been reported ⁴⁾.

Fundus Findings

Three characteristic hemorrhagic components are observed simultaneously or sequentially ^1,4).

• Intrapapillary hemorrhage: Red to dark red hemorrhage spreading radially on the optic disc surface.
• Peripapillary subretinal hemorrhage: Pale red to dark red hemorrhage extending from the disc margin to the nasal to inferonasal side, with less distinct borders than intraretinal hemorrhage.
• Vitreous hemorrhage: In mild cases, appears as thin hemorrhagic floaters in front of the disc; in severe cases, it may obscure fundus examination.

Peripapillary subretinal hemorrhage often occurs on the nasal side^4,7,8,10), which is thought to be related to the small scleral canal of the optic disc and the direction of vitreous traction. At onset, it may be accompanied by mild disc edema-like swelling, requiring differentiation from papilledema⁹⁾. Hwang and Lin summarized multilayered optic disc hemorrhages in adolescents, showing that three-layer hemorrhages (disc surface, subretinal, and vitreous) represent a continuous pathology explained by the same mechanism²¹⁾.

Visual Field Findings

Visual field changes are mild, presenting as enlargement of the Mariotte blind spot or Bjerrum scotoma, and are often asymptomatic. Goldmann or Humphrey perimetry may show arcuate sensitivity loss or nasal step corresponding to the hemorrhage site. After hemorrhage resolution, visual field defects usually improve^1,5).

Course and Prognosis

In principle, hemorrhage is naturally absorbed over a course of 6 weeks to several months without treatment. In Kokame et al.’s observation, 7 out of 9 cases recovered visual acuity of 1.2 or better, and no recurrence was observed ¹⁾. In typical cases in Japan, there are reports of a 12-year-old girl whose vitreous hemorrhage disappeared 6 weeks after the first visit and corrected visual acuity improved to 1.0 ¹⁰⁾, and a 36-year-old woman with bilateral involvement who recovered to 1.2 in the right eye and 1.0 in the left eye after 2 months ⁶⁾. Recurrence is rare, but there are cases where onset occurs at different times in both eyes, so follow-up of the contralateral eye is necessary even after unilateral onset ^5,6).

3. Causes and Risk Factors

Related Anatomical Factors

• Small optic disc and small scleral canal: Teng et al. reported that crowded optic nerve head and small scleral canal are associated with IHAPSH ¹³⁾. Dense nerve fibers within the disc and a small scleral canal opening may predispose to hemorrhage with minor mechanical stress.
• Disc tilt: Takahashi et al. used swept-source OCT to show that the disc tilt angle in IHAPSH eyes is larger than in control eyes ¹⁴⁾. A case in Japan with tilted disc syndrome, PHOMS, and IHAPSH also supports the association between tilt and this condition ¹⁵⁾.
• Myopia and axial elongation: Most reported cases involve mild to high myopia, and there is a case of a 12-year-old girl who developed the condition during the progression of myopia ⁹⁾. Axial elongation and weakening of peripapillary tissue are thought to be underlying factors.
• Association with PHOMS (peripapillary hyperreflective ovoid mass-like structures): PHOMS are ovoid hyperreflective structures observed on Bruch’s membrane around the optic disc and are thought to reflect axoplasmic flow stasis ^16,17). Several Japanese reports have described PHOMS in patients with IHAPSH ^15,18), suggesting common anatomical and mechanical predispositions. In children, PHOMS have been reported as a frequent cause of pseudopapilledema ²²⁾, and the prevalence of PHOMS is significantly higher in children with myopia ²³⁾.

Related mechanical factors

• Posterior vitreous detachment (PVD) and vitreopapillary traction: Cibis et al. reported that PVD can cause optic disc hemorrhage and retinal hemorrhage ²⁾, and Katz and Hoyt showed that vitreopapillary traction due to incomplete PVD can cause optic disc hemorrhage in young individuals ³⁾. In a 62-year-old Japanese case, PVD and rupture of superficial optic disc vessels were suspected as the bleeding mechanism ⁴⁾.
• Valsalva-like blood pressure fluctuations: Severe coughing, defecation, exercise, and other causes of sudden venous pressure elevation may trigger bleeding ¹²⁾.

Patient background as a risk factor

• Young women (10s to 30s)
• Myopia (mild to high)
• Tilted disc, small scleral canal, PHOMS
• Incomplete PVD, vitreopapillary adhesion
• Strong eye movements or Valsalva maneuver

4. Diagnosis and Examination Methods

Basics of Diagnosis

The starting point for diagnosis is the ophthalmoscopic confirmation of “intrapapillary hemorrhage” and “adjacent peripapillary subretinal hemorrhage” in the same eye. When findings such as nasally displaced subretinal hemorrhage, mild vitreous hemorrhage, and a background of young age, myopia, or female sex are present, IHAPSH should be strongly suspected ^1,4).

Key Points for Diagnosis

• The condition cannot be diagnosed with intrapapillary hemorrhage alone or peripapillary subretinal hemorrhage alone. Both must be present contiguously.
• Mild visual acuity loss and minimal visual field changes are useful for differentiating from other serious optic nerve diseases.
• Reversibility with spontaneous absorption over time also supports the diagnosis.

Imaging Tests

Wang Y, Chen H, Yuan L, et al. Intrapapillary hemorrhage with adjacent peripapillary subretinal hemorrhage of both eyes after COVID-19 infection: a case report. BMC Ophthalmol. 2024;24(1):101. Figure 2. PMCID: PMC10913657. License: CC BY 4.0.

Horizontal OCT sections of the optic disc in both eyes. A dome-shaped hyperreflective lesion within the optic disc is depicted as intrapapillary hemorrhage. A continuous hyperreflective layer in the subretinal space adjacent to the disc is captured as peripapillary subretinal hemorrhage. OCT noninvasively evaluates the location and layered structure of hemorrhage and is also used to assess concurrent PHOMS and optic disc tilt.

• Optical coherence tomography (OCT): Evaluates the layered structure of intrapapillary and peripapillary hemorrhage. Hemorrhage is captured as a hyperreflective image extending into the subretinal space, and the presence of PHOMS (oval hyperreflective lesions above the Bruch’s membrane opening) is also confirmed^10,15,16). Swept-source OCT is useful for observing optic disc tilt angle, small scleral canal, and PHOMS^14,15).
• OCT angiography (OCTA): Used to rule out choroidal neovascularization (CNV) and evaluate flow signals within PHOMS. In isolated IHAPSH, CNV is generally not observed^10,11).
• Fluorescein angiography (FA) / Indocyanine green angiography (IA): Evaluates the presence of CNV and dye leakage from the optic disc. In this condition, only fluorescence blockage at the hemorrhage site is observed, with no active CNV^1,12).
• 頭部MRI／MRA／MRV：うっ血乳頭・特発性頭蓋内圧亢進症（IIH）の鑑別に必要である。本邦の30代IIH 例では empty sella と PHOMS を認め、髄液圧 300 mmH₂O が確認された¹⁹⁾。IHAPSH 単独例では中枢神経画像に異常を認めない。

視野・電気生理検査

Goldmann 視野・Humphrey 視野で Mariotte 盲点の拡大や Bjerrum 型暗点の有無を評価する。本症単独では中心視野は保たれることが多い。視神経炎を疑う場合には visual evoked potential（VEP）や色覚検査を併用する。

鑑別診断

鑑別疾患	鑑別のポイント
Non-arteritic anterior ischemic optic neuropathy (NAION)	Elderly, acute vision loss with severe visual field defect, mainly optic disc edema
Optic neuritis	Pain on eye movement, central scotoma, relative afferent pupillary defect (RAPD)
Glaucomatous optic neuropathy	Chronic course, characteristic optic disc cupping and RNFL defect
Congestive optic disc / IIH	Bilateral papilledema, headache, pulsatile tinnitus, elevated CSF pressure
Optic disc drusen	Autofluorescence, OCT, ultrasound for calcification assessment
Tilted disc syndrome alone	Congenital optic disc dysplasia without hemorrhage
Terson syndrome	Preceded by central nervous system events such as subarachnoid hemorrhage
Traumatic optic disc hemorrhage	Clear history of trauma
Papillary hemorrhage related to blood disorders	Systemic findings such as severe anemia and thrombocytopenia20)

5. Standard Treatment

Observation as a Principle

IHAPSH is a benign disease that resolves spontaneously, and no definitive drug therapy or surgical treatment has been established. Many reports indicate that bleeding subsides within 6 weeks to a few months with observation alone ^1,6,10,11).

Key Points for Observation

• Initial evaluation: Combine visual acuity, refractive correction, intraocular pressure, visual field, OCT, and FA/OCTA to rule out CNV, optic neuritis, and papilledema.
• Observation interval: Initially every 1–2 weeks; once a reduction in bleeding is confirmed, extend to every 1–2 months. When visual acuity is stable, transition to regular checkups every 6 months.
• Lifestyle guidance: Advise avoiding strong straining or Valsalva maneuvers. It is safer to refrain from contact sports and extreme eye movements in the short term.
• Evaluation of the fellow eye: Given the possibility of bilateral onset, even in unilateral cases, OCT and visual field of the fellow eye should be assessed over time^5,6).

Exceptional interventions

Only when CNV is present or when bleeding extends to the macula and persistently affects visual function, anti-VEGF therapy or vitrectomy may be considered on a case-by-case basis. There is insufficient evidence to support the use of antithrombotic drugs or corticosteroids for this condition alone. In cases with persistent visual field defects or vision loss, reassess for concomitant chronic optic neuropathy or overlooked other diseases.

Caution

• Even after a diagnosis of IHAPSH, if visual field deterioration, optic disc pallor, or bilateral optic disc edema occurs, it is necessary to reconsider the coexistence or transition to glaucoma, optic neuritis, IIH, etc.
• Differentiation from optic disc drusen is performed using a combination of OCT, autofluorescence, and ultrasound. Be careful not to mistakenly introduce NAION treatment.

6. Pathophysiology and Detailed Mechanism of Onset

Mechanical Traction Hypothesis

The most supported mechanism is that nerve fibers and blood vessels densely packed in a small scleral canal of the optic disc are mechanically damaged by vitreopapillary traction associated with incomplete posterior vitreous detachment (PVD), leading to rupture of superficial capillaries^1,3,13). Hemorrhage can spread simultaneously into three layers: the optic disc surface, peripapillary subretinal space, and vitreous cavity.

Vascular and Tissue Vulnerability

In myopic axial elongation and tilted disc syndrome, distortion of the scleral canal morphology, thinning of the peripapillary choroid, and enlargement of the Bruch’s membrane opening occur, making the tissue vulnerable to vitreous traction and intraocular pressure fluctuations^14,15). In a Japanese case of combined tilted disc syndrome, PHOMS, and IHAPSH, it was suggested that the anatomical predisposition of the tilted disc promoted both PHOMS and IHAPSH¹⁵⁾.

Association with PHOMS

PHOMS are ovoid hyperreflective structures observed above the Bruch’s membrane opening on OCT. Initially, differentiation from optic disc drusen was a concern, but they are now understood to reflect axoplasmic flow stasis of retinal nerve fibers ^16,17). Borrelli et al. demonstrated blood flow signals within PHOMS on OCTA ¹⁷⁾. Tokuhisa et al. reported bilateral PHOMS in a 12-year-old girl with IHAPSH, suggesting that the coexistence of PHOMS and IHAPSH may not be coincidental ¹⁸⁾. Yamamoto et al. also presented an 11-year-old case with tilted disc syndrome accompanied by PHOMS and IHAPSH, discussing the possibility that PHOMS may predispose to the development of IHAPSH ¹⁵⁾.

Hemodynamic factors

Sudden venous pressure elevation similar to Valsalva maneuver, vigorous eye movements, and blood pressure fluctuations during exercise are thought to promote rupture of mechanically weakened superficial optic disc vessels ¹²⁾. Many cases have described an association between this condition and episodes of physical exertion or straining before onset.

Integrated model

In summary, IHAPSH can be understood as a multifactorial hemorrhage resulting from the combination of (1) anatomical predisposition of the optic disc (small scleral canal, tilted disc, PHOMS, myopia), (2) vitreopapillary traction (incomplete PVD), and (3) transient hemodynamic stress. After onset, traction is relieved as PVD progresses, and the hemorrhage is naturally absorbed ^1,3,13,14).

7. Latest Research and Future Prospects

Advances in Imaging Diagnostics

The widespread use of swept-source OCT and OCTA has made it possible in routine clinical practice to evaluate PHOMS, optic disc tilt, and three-dimensional understanding of the Bruch membrane opening ^14,17). Takahashi et al. quantified the large optic disc tilt in IHAPSH eyes ¹⁴⁾, and small case series in Japan have repeatedly shown a high rate of coexistence of tilted disc and PHOMS ^15,18).

Refinement of Disease Characteristics through Case Accumulation

Multiple case reports suggest a tendency for onset clustering in young women (teens) and middle-aged women (30s–40s), but the limited number of cases has not led to a definitive onset pattern ^7,9,10,11). Accumulation of bilateral cases and late-onset recurrences has also progressed, reaffirming the importance of surveillance of the fellow eye after unilateral onset ^5,6).

Development of a Differential Diagnosis Algorithm

Since PHOMS is also observed in IIH and demyelinating diseases, it is necessary to carefully rule out the coexistence of IIH or optic neuritis in IHAPSH with PHOMS. A Japanese case report of IIH with PHOMS in a 30-year-old patient highlights the importance of systematic evaluation when PHOMS findings are detected ¹⁹⁾. In the future, an algorithm that parameterizes the presence, size, and distribution of PHOMS to quantify the risk of developing IHAPSH is expected.

Prevention and Patient Education

Although clear preventive measures have not been established, controlling myopia progression, appropriate refractive correction, and avoiding strong Valsalva maneuvers may theoretically reduce mechanical stress on the optic disc. The intersection with management strategies for childhood myopia is a future challenge ⁹⁾.

For Patients: Please Read Carefully

• Optic disc hemorrhage (IHAPSH) is generally a relatively mild condition in which the bleeding is naturally absorbed and vision almost returns to normal.
• However, some diseases with similar appearances require prompt treatment. Be sure to undergo detailed examinations (OCT, visual field tests, and MRI if necessary) at an ophthalmology clinic.
• If you notice symptoms such as increased floaters, partial darkness in your vision, or sensitivity to light, do not self-diagnose; please see a doctor.
• Regular follow-up visits are very important to confirm absorption of bleeding and to quickly detect any abnormalities in the other eye. Keep to the scheduled appointment intervals.
• Avoid activities that suddenly increase blood pressure in the eye, such as straining, intense exercise, or lifting heavy objects, for the period instructed by your doctor.
• If your child or a family member with high myopia complains of similar symptoms, do not ignore them; seek an eye examination early.

8. References

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2. Cibis GW, Watzke RC, Chua J. Retinal hemorrhages in posterior vitreous detachment. Am J Ophthalmol. 1975;80(6):1043-1046. doi:10.1016/0002-9394(75)90334-7.
3. Katz B, Hoyt WF. Intrapapillary and peripapillary hemorrhage in young patients with incomplete posterior vitreous detachment. Signs of vitreopapillary traction. Ophthalmology. 1995;102(2):349-354. doi:10.1016/s0161-6420(95)31018-4.
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